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- Adverse anthropometric risk profile in biochemically controlled acromegalic patients: comparison with an age- and gender-matched primary care population
Abstract GH and IGF-1 play an important role in the regulation of metabolism and body composition. In patients with uncontrolled acromegaly, cardiovascular morbidity and mortality are increased but are supposed to be normalised after biochemical control is achieved. We aimed at comparing body composition and the cardiovascular risk profile in patients with controlled acromegaly and controls. A cross-sectional study. We evaluated anthropometric parameters (height, weight, body mass index (BMI), waist and hip circumference, waist to height ratio) and, additionally, cardiovascular risk biomarkers (fasting plasma glucose, HbA1c, triglycerides, total cholesterol, HDL, LDL, and lipoprotein (a), in 81 acromegalic patients (58% cured) compared to 320 age- and gender-matched controls (ratio 1:4), sampled from the primary care patient cohort DETECT. The whole group of 81 acromegalic patients presented with significantly higher anthropometric parameters, such as weight, BMI, waist and hip circumference, but with more favourable cardiovascular risk biomarkers, such as fasting plasma glucose, total cholesterol, triglycerides and HDL levels, in comparison to their respective controls. Biochemically controlled acromegalic patients again showed significantly higher measurements of obesity, mainly visceral adiposity, than age- and gender-matched control patients (BMI 29.5 ± 5.9 vs. 27.3 ± 5.8 kg/m2; P = 0.020; waist circumference 100.9 ± 16.8 vs. 94.8 ± 15.5 cm; P = 0.031; hip circumference 110.7 ± 9.9 vs. 105.0 ± 11.7 cm; P = 0.001). No differences in the classical cardiovascular biomarkers were detected except for fasting plasma glucose and triglycerides. This effect could not be attributed to a higher prevalence of type 2 diabetes mellitus in the acromegalic patient group, since stratified analyses between the subgroup of patients with acromegaly and controls, both with type 2 diabetes mellitus, revealed that there were no significant differences in the anthropometric measurements. Biochemically cured acromegalic patients pertain an adverse anthropometric risk profile, mainly because of elevated adiposity measurements, such as BMI, waist and hip circumference, compared to an age- and gender-matched primary care population.
- Content Type Journal Article
- DOI 10.1007/s11102-010-0218-7
- Authors
- C. Dimopoulou, Max Planck Institute of Psychiatry, Department of Endocrinology Munich Germany
- C. Sievers, Max Planck Institute of Psychiatry, Department of Endocrinology Munich Germany
- H. U. Wittchen, Institute of Clinical Psychology and Psychotherapy, Technical University Dresden Dresden Germany
- L. Pieper, Institute of Clinical Psychology and Psychotherapy, Technical University Dresden Dresden Germany
- J. Klotsche, Institute of Clinical Psychology and Psychotherapy, Technical University Dresden Dresden Germany
- J. Roemmler, Ludwig-Maximilians-University of Munich Department of Endocrinology Munich Germany
- J. Schopohl, Ludwig-Maximilians-University of Munich Department of Endocrinology Munich Germany
- H. J. Schneider, Max Planck Institute of Psychiatry, Department of Endocrinology Munich Germany
- G. K. Stalla, Max Planck Institute of Psychiatry, Department of Endocrinology Munich Germany
- Journal Pituitary
- Online ISSN 1573-7403
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- Management of prolactinomas in Brazil: an electronic survey
Abstract Dopamine agonists are the treatment of choice for prolactinomas. However, there are still controversies concerning dose, treatment duration and criteria for drug withdrawal in different clinical situations. The aim of this study was to assess diagnostic and therapeutic approaches to prolactinomas among members of the Brazilian Society of Endocrinology and Metabolism (SBEM). SBEM members answered a questionnaire sent by e-mail that included 18 questions related to controversial issues about the management of prolactinomas. Among SBEM members, 721 (approximately 24% of total) answered the questionnaire. Concerning the diagnosis, 38% of the respondents stated that prolactin levels < 100 ng/ml would exclude the presence of a prolactinoma. Most of them favored the screening for macroprolactin in asymptomatic individuals instead of a routine screening (74% vs. 26%). Regarding the treatment, 70% of the respondents chose cabergoline as the drug of choice to treat macroprolactinomas whereas similar proportions advised cabergoline or bromocriptine as the best treatment for microprolactinomas (52% vs. 48%). Only 20% and 34% of respondents favored treatment withdrawal 2?3 years after prolactin normalization in patients with macroprolactinomas and microprolactinomas, respectively. In case of pregnancy, only 58 and 70% of respondents advocated discontinuation of treatment with dopamine agonists in patients with macroprolactinomas and microprolactinomas, respectively. Finally, only 36% would allow breast-feeding without restriction, 44% would restrict it to patients with microprolactinomas and 20% would not recommend it for women with prolactinomas There are several points of disagreement among SBEM members regarding the management of prolactinomas.
- Content Type Journal Article
- DOI 10.1007/s11102-010-0217-8
- Authors
- Lucio Vilar, Pernambuco Federal University Division of Endocrinology, Hospital das Clinicas Rua Clovis Silveira Barros, 84/1202 Boa Vista, Recife PE CEP 50.050-270 Brazil
- Luciana Ansaneli Naves, Brasilia University Division of Endocrinology, Hospital Universitario Brasilia Brazil
- Luiz Augusto Casulari, Brasilia University Division of Endocrinology, Hospital Universitario Brasilia Brazil
- Monalisa Ferreira Azevedo, Brasilia University Division of Endocrinology, Hospital Universitario Brasilia Brazil
- José Luciano Albuquerque, Pernambuco Federal University Division of Endocrinology, Hospital das Clinicas Rua Clovis Silveira Barros, 84/1202 Boa Vista, Recife PE CEP 50.050-270 Brazil
- Fabiano Marcel Serfaty, Rio de Janeiro Federal University Division of Endocrinology, Hospital Universitario Clementino Fraga Filho Rio de Janeiro Brazil
- Flavia R. Pinho Barbosa, Rio de Janeiro Federal University Division of Endocrinology, Hospital Universitario Clementino Fraga Filho Rio de Janeiro Brazil
- Antonio Ribeiro de Oliveira, Minas Gerais Federal University Division of Endocrinology, Hospital das Clinicas Belo Horizonte Brazil
- Renan Magalhães Montenegro, Ceara Federal University Division of Endocrinology, Hospital Universitario Walter Cantidio Fortaleza Brazil
- Renan Magalhães Montenegro, Ceara Federal University Division of Endocrinology, Hospital Universitario Walter Cantidio Fortaleza Brazil
- Alberto José Santos Ramos, Campina Grande Federal University Division of Endocrinology, Hospital Universitario Alcides Carneiro Campina Grande Brazil
- Manuel dos Santos Faria, Maranhão Federal University Division of Endocrinology, Hospital Universitario Sao Luiz Brazil
- Nina Rosa C. Musolino, São Paulo University Medical School Unit of Neuroendocrinology, Division of Neurosurgery, Hospital das Clinicas São Paulo Brazil
- Monica R. Gadelha, Rio de Janeiro Federal University Division of Endocrinology, Hospital Universitario Clementino Fraga Filho Rio de Janeiro Brazil
- Cesar Luiz Boguszewski, Parana Federal University Division of Endocrinology and Metabolism, Hospital das Clinicas Curitiba Brazil
- Marcello D. Bronstein, Sao Paulo University Medical School Unit of Neuroendocrinology, Hospital das Clinicas São Paulo Brazil
- Journal Pituitary
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- Effect of transsphenoidal surgery on decreased visual acuity caused by pituitary apoplexy
Abstract We treated 12 patients with pituitary apoplexy out of 103 patients with pituitary tumors from August 1994 to March 2008 in the Nishi-Kobe Medical Center. The male to female ratio was 1:2 and the average age was 43 years old, ranging from 19 to 73. The symptoms on presentation were a decrease of visual acuity in nine, headache in seven, endocrinological disturbance in six, visual field defect in seven, a febrile state in six, vomiting in four, oculomotor disturbance in two, abducens palsy in one, and transient altered consciousness in one. All patients underwent transsphenoidal surgery and, in four of these, surgery was conducted within 7 days after onset. All nine patients with a decrease in the visual acuity recovered (100%) and, in addition, complete or near-complete vision recovery was noted in six out of eight patients (75%), excluding one patient whom we were unable to examine accurately. Emergent surgery was performed for only two patients with an acute deterioration of the visual acuity, with one finally developing complete blindness. Based on this study, we conclude that decompressive surgery is very useful for decreased visual acuity caused by pituitary apoplexy, but it is not necessary to perform emergent surgery for pituitary apoplexy in the absence of severe visual deterioration.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0216-9
- Authors
- Naoya Takeda, Nishi-Kobe Medical Center Department of Neurosurgery 5-7-1 Kouji-dai, Nishi-ku Kobe City Japan
- Katsuzo Fujita, Nishi-Kobe Medical Center Department of Neurosurgery 5-7-1 Kouji-dai, Nishi-ku Kobe City Japan
- Shigenori Katayama, Nishi-Kobe Medical Center Department of Neurosurgery 5-7-1 Kouji-dai, Nishi-ku Kobe City Japan
- Nobuyuki Akutu, Nishi-Kobe Medical Center Department of Neurosurgery 5-7-1 Kouji-dai, Nishi-ku Kobe City Japan
- Shigeto Hayashi, Nishi-Kobe Medical Center Department of Neurosurgery 5-7-1 Kouji-dai, Nishi-ku Kobe City Japan
- Eiji Kohmura, Kobe University Graduate School of Medicine Department of Neurosurgery Kobe City Japan
- Journal Pituitary
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- Pituitary function in subjects with mild traumatic brain injury: a review of literature and proposal of a screening strategy
Abstract Traumatic brain injury (TBI) is an important public health problem all over the world. The level of consciousness of the patients and the severity of the brain injury is commonly evaluated by the Glascow Coma Scale as mild, moderate and severe TBI. When we consider the high frequency of mild TBI (MTBI) among the all TBI patients the burden of the pituitary dysfunction problem in this group could not be ignored. However, one of the most important and still unresolved questions is which patients with MTBI should be screened for hypopituitarism? Another type of head trauma which could be considered as the subgroup of MTBI is sports related chronic repetitive head trauma. Therefore, in this review we will discuss the frequency, characteristics and current management of pituitary dysfunction in patients with MTBI including the subjects exposed to sports related chronic mild head trauma.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0215-x
- Authors
- Fatih Tanriverdi, Erciyes University Medical School Department of Endocrinology 38039 Kayseri Turkey
- Kursad Unluhizarci, Erciyes University Medical School Department of Endocrinology 38039 Kayseri Turkey
- Fahrettin Kelestimur, Erciyes University Medical School Department of Endocrinology 38039 Kayseri Turkey
- Journal Pituitary
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- The endocrine tumor summit 2008: appraising therapeutic approaches for acromegaly and carcinoid syndrome
Abstract The Endocrine Tumor Summit convened in December 2008 to address 6 statements prepared by panel members that reflect important questions in the treatment of acromegaly and carcinoid syndrome. Data pertinent to each of the statements were identified through review of pertinent literature by one of the 9-member panel, enabling a critical evaluation of the statements and the evidence supporting or refuting them. Three statements addressed the validity of serum growth hormone (GH) and insulin-like growth factor-I (IGF-I) concentrations as indicators or predictors of disease in acromegaly. Statements regarding the effects of preoperative somatostatin analog use on pituitary surgical outcomes, their effects on hormone and symptom control in carcinoid syndrome, and the efficacy of extended dosing intervals were reviewed. Panel opinions, based on the level of available scientific evidence, were polled. Finally, their views were compared with those of surveyed community-based endocrinologists and neurosurgeons.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0210-2
- Authors
- Anne Klibanski, Massachusetts General Hospital, Harvard Medical School 55 Fruit Street Boston MA 02114 USA
- Shlomo Melmed, Cedars-Sinai Medical Center, Academic Affairs Room #2015, 8700 Beverly Boulevard Los Angles CA 90048 USA
- David R. Clemmons, University of North Carolina School of Medicine 8024 Burnette Womack, CB 7170, Bowles Building Chapel Hill NC 27599-7170 USA
- Annamaria Colao, University Federico II of Naples Department of Molecular and Clinical Endocrinology and Oncology Via S. Pansini 5 Naples 80131 Italy
- Regina S. Cunningham, The Cancer Institute of New Jersey, Robert Wood Johnson Medical Center 195 Little Albany Street New Brunswick NJ 08903-2681 USA
- Mark E. Molitch, Northwestern University Feinberg School of Medicine 645 N. Michigan Avenue Suite 530 Chicago IL 60611 USA
- Aaron I. Vinik, Eastern Virginia Medical School, Strelitz Diabetes Center Department of Internal Medicine 855 West Brambleton Ave. Norfolk VA 23510 USA
- Daphne T. Adelman, Northwestern University Feinberg School of Medicine 645 N. Michigan Avenue Suite 530 Chicago IL 60611 USA
- Karen J. P. Liebert, Massachusetts General Hospital Neuroendocrine Unit Bulfinch 457 B, 55 Fruit Street Boston MA 02114 USA
- Journal Pituitary
- Online ISSN 1573-7403
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- The risk for breast cancer is not evidently increased in women with hyperprolactinemia
Abstract The question has been raised whether hyperprolactinemia in humans is associated with an excess risk for breast cancer. We aimed to assess the risk of breast cancer in a previously defined large cohort of patients treated for idiopathic hyperprolactinemia or prolactinomas. Based on the pattern of drug prescriptions we identified 11,314 subjects in the PHARMO network with at least one dispensing of dopamine agonists between 1996 and 2006. Of these, 1,607 subjects were considered to have dopamine agonist?treated hyperprolactinemia based on the prescribing pattern. For the present analysis, we included only women (n = 1,342). Patients with breast cancer were identified by hospital discharge codes. Data on breast cancer incidence in the Netherlands were derived from the Dutch cancer registry. Standardized mortality ratio (SMR) was the measure of outcome to assess the association between hyperprolactinemia and breast cancer. The 1,342 patients accounted for a total of 6,576 person years. Eight patients with breast cancer during follow-up were identified. Indirect standardization with incidence proportions from the general Dutch population revealed a 7.47 expected cases. The calculated SMR for breast cancer risk in patients treated hyperprolactinemia was 1.07 (95% confidence interval 0.50?2.03). In conclusion, there is no clear evidence for increased breast cancer risk in female patients treated for either idiopathic hyperprolactinemia or prolactinomas. The uncertainty about the exact risk that is due to the relatively low number of breast cancer cases, should be overcome by pooling results in a future meta-analysis.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0214-y
- Authors
- O. M. Dekkers, Leiden University Medical Center Department of Endocrinology and Metabolic Diseases P.O. Box 9600 2300 RC Leiden The Netherlands
- J. A. Romijn, Leiden University Medical Center Department of Endocrinology and Metabolic Diseases P.O. Box 9600 2300 RC Leiden The Netherlands
- A. de Boer, Utrecht University Division of Pharmacoepidemiology and Pharmacotherapy, Utrecht Institute for Pharmaceutical Sciences Utrecht The Netherlands
- J. P. Vandenbroucke, Leiden University Medical Center Department of Clinical Epidemiology P.O. Box 9600 2300 RC Leiden The Netherlands
- Journal Pituitary
- Online ISSN 1573-7403
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- A heritable predisposition to pituitary tumors
Abstract Pituitary tumors are prevalent in the general population, with a frequency of nearly 1 in 5. The cause of most pituitary tumors remains unknown, although a genetic contribution is recognized for some. We analyzed the Utah Population Data Base (UPDB), a resource combining a computerized genealogy of the Utah population with a statewide tumor registry, to investigate familial clustering of pituitary tumors. We analyzed the genetic relationships among 741 individuals diagnosed with benign or malignant pituitary tumors who had Utah genealogy data. To test for evidence of genetic contribution to predisposition, we compared average relatedness between all pairs of individuals with pituitary tumors with the expected relatedness in this population. We also estimated relative risks (RRs) for pituitary tumors in close and distant relatives of cases by comparing observed and expected numbers of cases among relatives. Relative risks for first- and third-degree relatives were significantly elevated (RR = 2.83 and 1.63, respectively), while relative risk for second-degree relatives was not significantly different from 1.0 (RR = 0.83). The average pairwise relatedness of pituitary tumor cases was significantly higher than expected, even when close relationships were ignored. The significantly elevated risks to relatives as well as the significant excess distant relatedness observed in cases provide strong support for a genetic contribution to predisposition to pituitary tumors. Multiple high-risk pedigrees can be identified in the UPDB, and study of such pedigrees might allow identification of the gene(s) responsible for our observations. Recognizing genetic contribution to the disease may also help with counseling family members of affected individuals.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0212-0
- Authors
- William T. Couldwell, University of Utah School of Medicine Department of Neurosurgery 175 North Medical Drive East Salt Lake City UT 84132 USA
- Lisa Cannon-Albright, University of Utah School of Medicine Department of Biomedical Informatics Salt Lake City UT USA
- Journal Pituitary
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- Evaluation of antioxidant systems in pituitary-adrenal axis diseases
Abstract The role of adrenal steroids in antioxidant regulation is not known. Previously, we demonstrated some Coenzyme Q10 (CoQ10) alterations in pituitary diseases, which can induce complex pictures due to alterations of different endocrine axes. Therefore we determined CoQ10 and Total Antioxidant Capacity (TAC) in pituitary-dependent adrenal diseases: 6 subjects with ACTH-dependent adrenal hyperplasia (AH); 19 with secondary isolated hypoadrenalism (IH), 19 with associated hypothyroidism (multiple pituitary deficiencies, MPH). CoQ10 was assayed by HPLC; TAC by the system metmyoglobin-H2O2, which, interacting with the chromogenous 2,2I-azinobis-(3-ethylbenzothiazoline-6-sulphonate), generates a spectroscopically revealed radical compound after a latency time (Lag) proportional to the antioxidant content. CoQ10 levels were significantly lower in IH than AH and MPH, with a similar trend when adjusted for cholesterol. Also TAC was lower in IH than in AH and MPH, suggesting that adrenal hormones can influence antioxidants. However, since thyroid hormones modulate CoQ10 levels and metabolism, when thyroid deficiency coexists it seems to play a prevalent influence.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0213-z
- Authors
- A. Mancini, Catholic University of the Sacred Heart Chair of Endocrinology Largo A. Gemelli 8 00168 Rome Italy
- E. Leone, Catholic University of the Sacred Heart Chair of Endocrinology Largo A. Gemelli 8 00168 Rome Italy
- A. Silvestrini, Catholic University of the Sacred Heart Institute of Biochemistry and Clinical Biochemistry Rome Italy
- R. Festa, ?Università Politecnica delle Marche? School of Medicine Department of Molecular Pathology Ancona Italy
- V. Di Donna, Catholic University of the Sacred Heart Chair of Endocrinology Largo A. Gemelli 8 00168 Rome Italy
- L. De Marinis, Catholic University of the Sacred Heart Chair of Endocrinology Largo A. Gemelli 8 00168 Rome Italy
- A. Pontecorvi, Catholic University of the Sacred Heart Chair of Endocrinology Largo A. Gemelli 8 00168 Rome Italy
- G. P. Littarru, ?Università Politecnica delle Marche? School of Medicine Institute of Biochemistry Ancona Italy
- E. Meucci, Catholic University of the Sacred Heart Institute of Biochemistry and Clinical Biochemistry Rome Italy
- Journal Pituitary
- Online ISSN 1573-7403
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- Temozolomide (Temodar®) and capecitabine (Xeloda®) treatment of an aggressive corticotroph pituitary tumor
Abstract Only rarely do corticotroph pituitary tumors become invasive leading to symptoms caused by compression of cranial nerves and other local structures. When aggressive pituitary neuroendocrine tumors do develop, conventional treatment options are of limited success. A 50-year-old man developed a giant invasive corticotroph pituitary tumor 2 years after initial presentation. His tumor and symptoms failed to respond to maximal surgical, radio-surgical, radiation and medical therapy and a bilateral adrenalectomy was done. He subsequently developed rapid growth of his tumor leading to multiple cranial nerve deficits. He was administered salvage chemotherapy with capecitabine and temozolomide (CAPTEM), a novel oral chemotherapy regimen developed at our institution for treatment of neuroendocrine tumors. After two cycles of CAPTEM, his tumor markedly decreased in size and ACTH levels fell by almost 90%. Despite further decreases in ACTH levels, his tumor recurred after 5 months with increased avidity on PET scan suggesting a transformation to a more aggressive phenotype. Temozolomide had been reported to be effective against other pituitary tumors and this case adds to this literature demonstrating its use along with capecitabine (CAPTEM) against a corticotroph tumor. Further evaluation of the CAPTEM regimen in patients with pituitary neuroendocrine tumors which fail to respond to classic treatments is warranted.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0211-1
- Authors
- Marie S. Thearle, Columbia University College of Physicians and Surgeons Department of Medicine, Division of Medical Oncology 650 West 168th St. BB 20-05 New York NY 10032 USA
- Pamela U. Freda, Columbia University College of Physicians and Surgeons Department of Medicine, Division of Medical Oncology 650 West 168th St. BB 20-05 New York NY 10032 USA
- Jeffrey N. Bruce, Columbia University College of Physicians and Surgeons Department of Neurosurgery, Division of Medical Oncology New York NY 10032 USA
- Steven R. Isaacson, Columbia University College of Physicians and Surgeons Department of Radiation Oncology, Division of Medical Oncology New York NY 10032 USA
- Yoomi Lee, Columbia University College of Physicians and Surgeons Experimental Therapeutics Program, Division of Medical Oncology New York NY 10032 USA
- Robert L. Fine, Columbia University College of Physicians and Surgeons Department of Medicine, Division of Medical Oncology 650 West 168th St. BB 20-05 New York NY 10032 USA
- Journal Pituitary
- Online ISSN 1573-7403
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- Effectiveness of cabergoline in monotherapy and combined with ketoconazole in the management of Cushing?s disease
Abstract The expression of dopamine receptor subtypes has been reported in corticotroph adenomas, and this finding support the possibility for medical treatment of Cushing?s disease (CD) with dopamine agonists when conventional treatment has failed. The aim of this study was to evaluate the effectiveness of cabergoline (at doses of up 3 mg/week), alone or combined with relatively low doses of ketoconazole (up to 400 mg/day), in 12 patients with CD unsuccessfully treated by transsphenoidal surgery. After 6 months of cabergoline therapy, normalization of 24 h urinary free cortisol (UFC) levels occurred in three patients (25%) at doses ranging from 2?3 mg/week, whereas reductions ranging from 15.0 to 48.4% were found in the remaining. The addition of ketonocazole to the nine patients without an adequate response to cabergoline was able to normalize UFC excretion in six patients (66.7%) at doses of 200 mg/day (three patients), 300 mg/day (two patients) and 400 mg/day (one patient). In the remaining patients UFC levels did not normalize but a significant reduction ranging from to 44.4 to 51.7% was achieved. In two of the six responsive patients to combination therapy, the weekly dose of cabergoline could be later reduced from 3 to 2 mg. Our findings demonstrated that cabergoline monotherapy was able to reverse hypercortisolism in 25% of patients with CD unsuccessfully treated by surgery. Moreover, the addition of relatively low doses of ketoconazole led to normalization of UFC in about two-thirds of patients not achieving a full response to cabergoline.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0209-8
- Authors
- Lucio Vilar, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- Luciana A. Naves, Hospital Universitário de Brasilia Division of Endocrinology Brasilia Brazil
- Monalisa F. Azevedo, Hospital Universitário de Brasilia Division of Endocrinology Brasilia Brazil
- Maria Juliana Arruda, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- Carla M. Arahata, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- Lidiane Moura e Silva, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- Rodrigo Agra, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- Lisete Pontes, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- Larissa Montenegro, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- José Luciano Albuquerque, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- Viviane Canadas, Federal University of Pernambuco Division of Endocrinology, Hospital das Clínicas Recife Brazil
- Journal Pituitary
- Online ISSN 1573-7403
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- Diagnostic value of 18F-dihydroxyphenylalanine positron emission tomography for growth hormone-producing pituitary adenoma
Diagnostic value of 18F-dihydroxyphenylalanine positron emission tomography for growth hormone-producing pituitary adenoma
- Content Type Journal Article
- DOI 10.1007/s11102-009-0208-9
- Authors
- Takafumi Taguchi, Kochi University Kochi Medical School Kohasu, Oko-cho Nankoku Japan
- Toshihiro Takao, Kochi University Kochi Medical School Kohasu, Oko-cho Nankoku Japan
- Yasumasa Iwasaki, Kochi University Kochi Medical School Kohasu, Oko-cho Nankoku Japan
- Kenichi Oyama, Toranomon Hospital Department of Hypothalamic & Pituitary Surgery Minato-ku Tokyo Japan
- Shozo Yamada, Toranomon Hospital Department of Hypothalamic & Pituitary Surgery Minato-ku Tokyo Japan
- Mari Inoue, Kochi University Kochi Medical School Kohasu, Oko-cho Nankoku Japan
- Yoshio Terada, Kochi University Kochi Medical School Kohasu, Oko-cho Nankoku Japan
- Journal Pituitary
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- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Acromegaly with negative pituitary MRI and no evidence of ectopic source: the role of transphenoidal pituitary exploration?
Abstract Growth hormone (GH) producing adenomas of the pituitary gland are usually macroadenomas (>10 mm in size). Often these adenomas are locally invasive by the time of diagnosis. Acromegaly secondary to a very small pituitary microadenoma not visualized on pituitary magnetic resonance (MR) imaging is rare. We report a patient with acromegaly and an unremarkable pituitary MR imaging who had negative work up for ectopic growth hormone-releasing hormone (GHRH) or GH secreting tumors. Transsphenoidal pituitary exploration revealed a pituitary adenoma located on the left side of the sella against the medial wall of the cavernous sinus extending posteriorly along the floor of the sella all the way to the right side. The acromegaly was treated with resection of the pituitary adenoma and normalization of serum insulin-like growth factor 1 (IGF-1) and GH levels. In a patient with acromegaly and unremarkable pituitary MR imaging, with no evidence of ectopic GH and GHRH production, transsphenoidal pituitary exploration is a reasonable approach and may result in clinical improvement and biochemical cure in the hand of experienced surgeon. This approach may avoid long term medical treatment with its associated cost.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0205-z
- Authors
- Sameera Daud, 452 Old Hook Road Emerson NJ 07630 USA
- Amir H. Hamrahian, The Endocrinology and Metabolism Institute, Cleveland Clinic The Department of Endocrinology 9500 Euclid Ave, Desk A53 Cleveland OH 44195 USA
- Robert J. Weil, The Neurological Institute, Cleveland Clinic The Brain Tumor and Neuro-Oncology Center, Department of Neurosurgery Cleveland OH USA
- Marwan Hamaty, The Endocrinology and Metabolism Institute, Cleveland Clinic The Department of Endocrinology 9500 Euclid Ave, Desk A53 Cleveland OH 44195 USA
- Richard A. Prayson, Cleveland Clinic Department of Pathology and Laboratory Medicine Cleveland OH USA
- Leann Olansky, The Endocrinology and Metabolism Institute, Cleveland Clinic The Department of Endocrinology 9500 Euclid Ave, Desk A53 Cleveland OH 44195 USA
- Journal Pituitary
- Online ISSN 1573-7403
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- Effectiveness of self- or partner-administration of an extended-release aqueous-gel formulation of lanreotide in lanreotide-naïve patients with acromegaly
Abstract Surgical resection is often not curative in patients with acromegaly and long-acting somatostatin analogues (lanreotide or octreotide) are often needed. This study assessed the efficacy and safety of self- or partner-administration of lanreotide in patients with acromegaly. This was a six-month, single-arm, open-label study conducted at 13 endocrinology clinics. Fifty-nine patients received deep subcutaneous lanreotide injections every 28 days. Twelve patients started on 120 mg lanreotide and forty-seven started on 90 mg lanreotide. At week 16, the dose was adjusted to 60, 90 or 120 mg based on insulin-like growth factor-1 (IGF-1) levels at week 12. Fifty-nine patients with acromegaly either switched from long-acting octreotide (switch; n = 33) or were somatostatin analogue treatment-naïve or not currently taking long-acting octreotide (?other?; n = 26). The key endpoints included the percentage of patients/partners able to self- or partner-inject lanreotide and those with normal IGF-1 or growth hormone (GH) levels at week 24/early termination. 100% of patients/partners correctly self- (n = 41) or partner-injected (n = 18) lanreotide by week 4. By week 24/early termination, IGF-1 levels were controlled in 93.7% of switch and 46.2% of ?other? patients, while GH levels were controlled in 76.9% and 39.1% of patients, respectively. Both IGF-1 and GH were controlled in 73.1% of switch and 30.4% of ?other? patients. Most switch patients (81%) reported they preferred lanreotide over long-acting octreotide for future use (P = 0.0001). Self- or partner-administration of lanreotide is generally well tolerated and associated with IGF-1 and GH control in many lanreotide-naïve patients with acromegaly.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0207-x
- Authors
- Roberto Salvatori, Johns Hopkins University School of Medicine Division of Endocrinology 1830 East Monument street #333 Baltimore MD 21287 USA
- Lisa B. Nachtigall, Massachusetts General Hospital Boston MA USA
- David M. Cook, Oregon Health & Science University Portland OR USA
- Vivien Bonert, Cedars-Sinai Medical Center Los Angeles CA USA
- Mark E. Molitch, Northwestern University Chicago IL USA
- Sandra Blethen, Tercica, Inc. a subsidiary of the Ipsen Group Brisbane CA USA
- Stephen Chang, Tercica, Inc. a subsidiary of the Ipsen Group Brisbane CA USA
- The SALSA Study Group
- Journal Pituitary
- Online ISSN 1573-7403
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- Management of acromegaly in Latin America: expert panel recommendations
Abstract Although there are international guidelines orienting physicians on how to manage patients with acromegaly, such guidelines should be adapted for use in distinct regions of the world. A panel of neuroendocrinologists convened in Mexico City in August of 2007 to discuss specific considerations in Latin America. Of major discussion was the laboratory evaluation of acromegaly, which requires the use of appropriate tests and the adoption of local institutional standards. As a general rule to ensure diagnosis, the patient?s GH level during an oral glucose tolerance test and IGF-1 level should be evaluated. Furthermore, to guide treatment decisions, both GH and IGF-1 assessments are required. The treatment of patients with acromegaly in Latin America is influenced by local issues of cost, availability and expertise of pituitary neurosurgeons, which should dictate therapeutic choices. Such treatment has undergone profound changes because of the introduction of effective medical interventions that may be used after surgical debulking or as first-line medical therapy in selected cases. Surgical resection remains the mainstay of therapy for small pituitary adenomas (microadenomas), potentially resectable macroadenomas and invasive adenomas causing visual defects. Radiotherapy may be indicated in selected cases when no disease control is achieved despite optimal surgical debulking and medical therapy, when there is no access to somatostatin analogues, or when local issues of cost preclude other therapies. Since not all the diagnostic tools and treatment options are available in all Latin American countries, physicians need to adapt their clinical management decisions to the available local resources and therapeutic options.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0206-y
- Authors
- Ariel Barkan, The University of Michigan Ann Arbor MI USA
- Marcello D. Bronstein, University of São Paulo Medical School Hospital das Clínicas São Paulo Brazil
- Oscar D. Bruno, UBA Hospital de Clínicas José de San Martín Buenos Aires Argentina
- Alejandro Cob, Hospital San Juan de Dios San José Costa Rica
- Ana Laura Espinosa-de-los-Monteros, Hospital de Especialidades del Centro Médico Nacional Siglo XXI, del Instituto Mexicano del Seguro Social (IMSS) Endocrinology Unit Aristóteles # 68, Col. Polanco CP 115560 Mexico City Mexico
- Monica R. Gadelha, Universidade Federal do Rio de Janeiro Rio de Janeiro Brazil
- Gloria Garavito, Instituto Nacional de Cancerología Bogotá Colombia
- Mirtha Guitelman, Hospital General de Agudos Carlos G. Durand División Endocrinología Buenos Aires Argentina
- Ruth Mangupli, Hospital Universitario Caracas Venezuela
- Moisés Mercado, Hospital de Especialidades del Centro Médico Nacional Siglo XXI, del Instituto Mexicano del Seguro Social (IMSS) Endocrinology Unit Aristóteles # 68, Col. Polanco CP 115560 Mexico City Mexico
- Lesly Portocarrero, Instituto Nacional de Neurologia y Neurocirugia Mexico City Mexico
- Michael Sheppard, The University of Birmingham Birmingham UK
- Journal Pituitary
- Online ISSN 1573-7403
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- Hypopituitarism due to sports related head trauma and the effects of growth hormone replacement in retired amateur boxers
Abstract Traumatic brain injury (TBI) has been recently recognized as a leading cause of pituitary dysfunction. Current data clearly demonstrated that sports related head trauma due to boxing, kickboxing, and soccer might results in pituitary hormone deficiencies, isolated growth hormone (GH) deficiency in particular. In the present report physiologic dose GH replacement therapy (GHRT) was performed in two GH deficient retired amateur boxers for the first time. The boxers received recombinant GH for 6 months. After 6 months of GHRT there were substantial improvements, but not complete normalization, in the body composition parameters, lipid profiles and quality of life scores in both boxers. These preliminary results suggest that GHRT may have beneficial effects in retired boxers with severe isolated GH deficiency due to sports related head trauma. But more data with higher number of boxers and longer GHRT duration are warranted.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0204-0
- Authors
- F. Tanriverdi, Erciyes University Medical School Department of Endocrinology 38039 Kayseri Turkey
- K. Unluhizarci, Erciyes University Medical School Department of Endocrinology 38039 Kayseri Turkey
- Z. Karaca, Erciyes University Medical School Department of Endocrinology 38039 Kayseri Turkey
- F. F. Casanueva, Santiago de Compostela University Department of Medicine, School of Medicine Santiago de Compostela Spain
- F. Kelestimur, Erciyes University Medical School Department of Endocrinology 38039 Kayseri Turkey
- Journal Pituitary
- Online ISSN 1573-7403
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- Apoplexy in non functioning pituitary adenoma after one dose of leuprolide as treatment for prostate cancer
Abstract We report the case of a 60 year old male who complained of headache and blurry vision?that progressed to left ophthalmoplegia and ptosis?after receiving a dose of leuprolide for Prostate cancer therapy. Imaging showed a hemorrhagic sellar mass. The patient underwent transsphenoidal debulking, and the tissue obtained demonstrated immunohistochemical staining for LH. A literature review revealed nine previously reported cases of pituitary apoplexy after GnRH agonist therapy for prostate cancer. In most cases, the sellar tissues stained for LH, consistent with a gonadotropinoma. The pathophysiology of these events is unclear, but recent animal models suggest possible explanations. The predominance of gonadotropinomas is important because they do not usually present with hypersecretory symptoms. Particular attention to clinical findings suggestive of a non functioning pituitary tumor in patients receiving GnRH agonist therapy is critical as routine screening with MRI is not practical.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0202-2
- Authors
- Yannis Guerra, John H. Stroger Jr., Hospital of Cook County/Rush University Medical Center Chicago IL USA
- Evelyn Lacuesta, John H. Stroger Jr., Hospital of Cook County/Rush University Medical Center Chicago IL USA
- Francisco Marquez, John H. Stroger Jr., Hospital of Cook County Chicago IL USA
- P. B. Raksin, John H. Stroger Jr., Hospital of Cook County/Rush University Medical Center Chicago IL USA
- Manuel Utset, John H. Stroger Jr., Hospital of Cook County Chicago IL USA
- Leon Fogelfeld, John H. Stroger Jr., Hospital of Cook County/Rush University Medical Center Chicago IL USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Outcome of gonadotropin therapy for male infertility due to hypogonadotrophic hypogonadism
Abstract Data on the management of male infertility secondary to hypogonadotrophic hypogonadism (HH) are limited. We report our extensive experience with intramuscular injections of gonadotropins, one of the two methods used for this purpose. Eighty-seven married men (median age, 28 years) with either congenital (47 men) or acquired (40 men) HH were treated for a median of 26 months (range, 6?57) with intramuscular injections of gonadotropins (HCG/HMG) three times weekly for the purpose of achieving fertility. The outcome was assessed by achievement of one or more pregnancies. Of the 151 courses of HCG/HMG treatment administered to 87 patients, 85 courses (56.3%) were successful, resulting in 85 pregnancies (median pregnancy rate 2, range 1?10) in 35 patients (40%) while 52 cases did not achieve pregnancy. Responders had larger pretherapy testicular volume (9 ± 3.6 cc) compared to non-responders [(5.7 ± 2.0 cc), P < 0.0001], but there was no difference in age, LH, FSH or testosterone levels or doses of HCG/HMG used. The pregnancy rate was similar in those with congenital (51.4%) and acquired causes (48.6%) of HH (P = 0.83). Only testicular size was predictive of conception (P < 0.001, odds ratio 1.5, 95% CI 1.21?1.92) while age, pretherapy levels of testosterone, LH, FSH and doses of HCG/HMG did not predict the success of pregnancy. Gonadotropins are moderately effective in achieving one to several pregnancies in HH. Only testicular size is predictive of success in achieving pregnancy. There is no difference in success between those with congenital and acquired causes of HH.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0203-1
- Authors
- Rafif Farhat, King Faisal Specialist Hospital & Research Centre Department of Medicine Riyadh 11211 Saudi Arabia
- Fatma Al-zidjali, King Faisal Specialist Hospital & Research Centre Department of Medicine Riyadh 11211 Saudi Arabia
- Ali S. Alzahrani, King Faisal Specialist Hospital & Research Centre Department of Medicine Riyadh 11211 Saudi Arabia
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Cushing disease as possible cause of persistent growth failure despite growth hormone therapy in a small for gestational age male
Abstract Growth hormone (GH) therapy in children with small for gestational age (SGA) has been shown to be of significant therapeutic benefit. We report the case of an 11-year-old Caucasian male who developed early adrenarche, hypertension and insulin resistance on GH therapy for SGA and profound short stature (ht ?5 SD). This patient demonstrated a poor response to GH therapy and developed physical and biochemical findings of insulin resistance responsive to metformin therapy. He remained hypertensive, however, and continued to have elevated serum dehydroepiandrosterone sulfate levels. Urinary free cortisol excretion was subsequently found to be elevated. The diagnosis of Cushing?s disease was confirmed with inferior petrosal sinus sampling and pituitary MRI. The patient underwent partial adenohypophysectomy with resulting normalization of plasma cortisol levels and associated symptoms. Our patient?s diagnosis of Cushing?s disease was complicated by his past history of poor growth since birth and history of SGA. The signs of Cushing?s disease did not overtly appear until GH therapy was initiated to help treat severe short stature. It is possible that the metabolic effects of GH therapy unmasked the presence of underlying Cushing?s disease.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0201-3
- Authors
- MaryKathleen Heneghan, Medical College of Wisconsin Section of Pediatric Endocrinology and Diabetes, Department of Pediatrics 8701 Watertown Plank Road Milwaukee WI 53226-0509 USA
- Ramin Alemzadeh, Medical College of Wisconsin Section of Pediatric Endocrinology and Diabetes, Department of Pediatrics 8701 Watertown Plank Road Milwaukee WI 53226-0509 USA
- Journal Pituitary
- Online ISSN 1573-7403
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- Inferior petrosal sinus ACTH and prolactin responses to CRH in ACTH-dependent Cushing?s syndrome: a single centre experience from the United Kingdom
Abstract Inferior petrosal sinus sampling (IPSS) of ACTH with CRH stimulation helps distinguish pituitary ACTH-dependent Cushing?s syndrome from the ectopic ACTH syndrome (EAS). The usefulness of the paradoxical response of other pituitary hormones including prolactin to CRH remains controversial. Data from 33 IPSS procedures carried out at the Walton Centre for Neurology and Neurosurgery in Liverpool were analyzed. Patients were selected for this procedure if they had been diagnosed with ACTH dependent Cushing?s syndrome and the majority had no obvious pituitary adenoma on Magnetic Resonance Imaging. Satisfactory simultaneous bilateral catheterization was accomplished in 23/33 (success rate 70%). The diagnostic sensitivity of a basal central/peripheral ACTH ratio >2.0 and >3 post-CRH was 94%. In two patients with subsequently confirmed EAS the maximal central/peripheral ACTH ratio was <2.0 on basal samples and did not change following CRH. The maximal central/peripheral prolactin ratio was noted at 5 min post-CRH, coinciding with the maximal central/peripheral ACTH ratio. The intersinus gradient (ISG) of ACTH was paralleled by a consistent ISG of prolactin and in 7 out of 9 patients (with successful bilateral IPSS and unilateral adenomas) the ISG of prolactin correctly lateralized the microadenoma whereas the ISG of ACTH correctly lateralized in 8 out of 9 patients. Neither of the patients with EAS achieved a central/peripheral prolactin ratio >2 in the basal state and >3 post-CRH. Bilateral catheterization of inferior petrosal sinuses can be successful in up to 70% of cases. Prolactin measurements do not have superior lateralizing capability compared with ACTH but may be useful in the differential diagnosis of pituitary-driven from EAS.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0200-4
- Authors
- Christina Daousi, Clinical Sciences Center, University Hospital Aintree Diabetes & Endocrinology Clinical Research Group, Department of Endocrinology and Diabetes Lower Lane, Liverpool L9 7AL UK
- Thomas Nixon, The Walton Centre for Neurology and Neurosurgery Department of Neuroradiology Liverpool UK
- Mohsen Javadpour, The Walton Centre for Neurology and Neurosurgery Department of Neurosurgery Liverpool UK
- Katharine Hayden, University Hospital Aintree Department of Clinical Biochemistry Liverpool L9 7AL UK
- Ian A. MacFarlane, Clinical Sciences Center, University Hospital Aintree Diabetes & Endocrinology Clinical Research Group, Department of Endocrinology and Diabetes Lower Lane, Liverpool L9 7AL UK
- Journal Pituitary
- Online ISSN 1573-7403
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- Sellar collision tumor involving pituitary gonadotroph adenoma and chondroma: a potential clinical diagnosis
Abstract We report on a 74-year-old male patient who presented with progressive neuroophthalmologic symptoms soon after the administration of a long-acting gonadotropin-releasing hormone agonist for treatment of a prostate cancer. Imaging revealed a destructively growing and extensively calcified sellar mass inconsistent with a pituitary adenoma. A transseptal transsphenoidal tumor mass reduction yielded a histological diagnosis of a collision tumor comprised of a gonadotroph adenoma intermingled with osteochondroma. We discuss a potential causal relationship between the administration of the long-acting gonadotropin-releasing hormone agonist and the sudden appearance of the previously unsuspected sellar lesion. Although the association of these two tumors is very likely coincidental, the possibility of causal relationship is addressed.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0199-6
- Authors
- Rahel Sahli, Bern University Hospital and University of Bern Department of Endocrinology, Diabetes and Clinical Nutrition 3010 Bern Switzerland
- Emanuel Christ, Bern University Hospital and University of Bern Department of Endocrinology, Diabetes and Clinical Nutrition 3010 Bern Switzerland
- Dominique Kuhlen, Bern University Hospital and University of Bern Department of Neurosurgery 3010 Bern Switzerland
- Olivier Giger, University of Bern Institute of Pathology 3010 Bern Switzerland
- Istvan Vajtai, University of Bern Institute of Pathology 3010 Bern Switzerland
- Journal Pituitary
- Online ISSN 1573-7403
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- Overview of vascular complications of pituitary surgery with special emphasis on unexpected abnormality
Abstract Arterial bleeding during transsphenoidal surgery for pituitary adenoma is known complication. This usually happens due to rupture of intracavernous carotid or delayed hemorrhage due to the carotico-cavernous fistula and/or pseudoaneurysm. There is also evidence that cavernous carotid aneurysms may occur with pituitary tumors, yet largest series failed to demonstrate any link between aneurysm formation and pituitary tumors. Usually such an aneurysm rupture results in formation of carotico-cavernous fistula. However, pituitary apoplexy and even epistaxis have been reported. In this paper we present a patient with recurrent pituitary adenoma and cavernous carotid artery aneurysm, which caused significant hemorrhage during the surgery. Although retrospective analysis of MRI disclosed that the patient had the aneurysm before the first surgery, it remained silent until the second operation. Therefore neurosurgeons should be very susceptive to any signal changes on preoperative MR images, especially in recurrent cases, where normal anatomical relations are disturbed by fibrotic tissue. Also, we reviewed the vascular complication of pituitary surgery based on the literature.
- Content Type Journal Article
- Category Review Paper
- DOI 10.1007/s11102-009-0198-7
- Authors
- Mustafa Berker, Hacettepe School of Medicine Department of Neurosurgery Ankara Turkey
- Kamran Aghayev, Hacettepe School of Medicine Department of Neurosurgery Ankara Turkey
- Isil Saatci, Hacettepe School of Medicine Department of Neuroradiology Ankara Turkey
- Selçuk Palao?lu, Hacettepe School of Medicine Department of Neurosurgery Ankara Turkey
- Metin Önerci, Hacettepe School of Medicine Department of Otorhinolaryngology Ankara Turkey
- Journal Pituitary
- Online ISSN 1573-7403
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- Diagnostic efficacy of midnight cortisol and midnight ACTH in the diagnosis and localisation of Cushing?s syndrome
Abstract Classical tests for diagnosis of Cushing?s syndrome (CS) like urine free cortisol and dexamethasone suppression tests have limitations in various clinical settings. This study evaluated the usefulness of sleeping midnight serum cortisol (SMNC) as a diagnostic test for hypercortisolemia. A simultaneously done midnight plasma ACTH level was used to classify the disease as ACTH dependent or independent. Standard biochemical tests, SMNC, midnight plasma ACTH and appropriate imaging evaluated patients with a clinical suspicion of Cushing?s syndrome. We evaluated 43 patients with CS comprising of 34 patients with Cushing?s disease (CD), 2 patients with thymic carcinoid producing ectopic CS, 5 patients with adrenal carcinoma and 2 with adrenal adenoma. Thirteen patients with clinical suspicion were also evaluated with the above tests and CS was ruled out. SMNC, midnight plasma ACTH and dexamethasone suppressed cortisol was collected from patients with a suspicion of CS. SMNC was evaluated against histopathology as the gold standard. SMNC achieved 100% sensitivity in the diagnosis of endogenous CS at cut offs of 138 nmol/l and below. Raising the cut off to 207 nmol/l resulted in a test sensitivity of 90.5%. At a cut off of 1.65 pmol/l, midnight plasma ACTH could distinguish ACTH independent causes of CS with 100% sensitivity. We concluded that a single midnight collection could identify all patients with CS and classify the ACTH status at the proposed cut offs.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0197-8
- Authors
- Mathew John, Seth Gordhandas Sunderdas Medical College & King Edward Memorial Hospital Department of Endocrinology Parel, Mumbai 400012 India
- Anurag R. Lila, Seth Gordhandas Sunderdas Medical College & King Edward Memorial Hospital Department of Endocrinology Parel, Mumbai 400012 India
- Tushar Bandgar, Seth Gordhandas Sunderdas Medical College & King Edward Memorial Hospital Department of Endocrinology Parel, Mumbai 400012 India
- Padma S. Menon, Seth Gordhandas Sunderdas Medical College & King Edward Memorial Hospital Department of Endocrinology Parel, Mumbai 400012 India
- Nalini S. Shah, Seth Gordhandas Sunderdas Medical College & King Edward Memorial Hospital Department of Endocrinology Parel, Mumbai 400012 India
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Primary sellar lymphoma: intravascular large B-cell lymphoma diagnosed as a double cancer and improved with chemotherapy, and literature review of primary parasellar lymphoma
Abstract Lymphoma is one of the causative factors of hypothalamus?pituitary dysfunction, and intravascular large B-cell lymphoma (IVLBCL) is a subtype of primary extranodal neoplasm. A 69-year-old woman visited our hospital because of general fatigue. We diagnosed her with presumable non-functional primary pituitary adenoma and subsequent dysfunction. Eight months after, the patient revisited our hospital because of dyspnea. Though we conducted systemic investigations including chest and abdomen enhanced computer tomography, transbronchial lung biopsy, and bone marrow biopsy, the diagnosis was not confirmed. Inadvertently, a breast cancer was found, and the surgical specimen proved that the patient had double cancer?adenocarcinoma and IVLBCL. Rituximab, cyclophosphamide, adriamycin, vincristine, and prednisolone regimen was initiated, and complete remission was achieved. Notably, the sellar mass returned to normal size with improved function. We reviewed 32 patients with primary parasellar lymphoma. In affected sites, both sellar and pituitary stalk (6.7%), both hypothalamus and pituitary stalk (6.7%), only sellar (63.3%), only pituitary stalk (6.7%), only hypothalamus (13.3%), and only clivus (3.3%) were observed. In hypothalamus?pituitary dysfunction, both anterior and posterior dysfunction (20.7%), only anterior dysfunction (58.6%), only posterior dysfunction (3.4%), and no dysfunction (17.2%) were observed. It seemed that hypothalamic lesion is related to both anterior and posterior dysfunction, while sellar lesion is related to mainly anterior dysfunction. In cranial nerve dysfunction, 2nd nerve dysfunction (45.2%) and 6th nerve dysfunction (35.5%) were frequently observed. It seemed that sellar lesion is related to both 2nd and 6th nerve dysfunction, while hypothalamic lesion is related to mainly 2nd nerve dysfunction.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0196-9
- Authors
- Mutsuko Yasuda, Tokyo Metropolitan Bokutoh Hospital Department of Internal Medicine 4-23-15, Koutoh-bashi, Sumida-ku 130-8575 Tokyo Japan
- Nobu Akiyama, Tokyo Metropolitan Bokutoh Hospital Department of Internal Medicine 4-23-15, Koutoh-bashi, Sumida-ku 130-8575 Tokyo Japan
- Sachio Miyamoto, Tokyo Metropolitan Bokutoh Hospital Department of Surgery Tokyo Japan
- Masahiro Warabi, Tokyo Metropolitan Bokutoh Hospital Department of Pathology Tokyo Japan
- Yumiko Takahama, Tokyo Metropolitan Bokutoh Hospital Department of Surgery Tokyo Japan
- Mari Kitamura, Tokyo Metropolitan Bokutoh Hospital Department of Internal Medicine 4-23-15, Koutoh-bashi, Sumida-ku 130-8575 Tokyo Japan
- Fumiatsu Yakushiji, Tokyo Metropolitan Bokutoh Hospital Department of Internal Medicine 4-23-15, Koutoh-bashi, Sumida-ku 130-8575 Tokyo Japan
- Hiroyuki Kinoshita, Tokyo Metropolitan Bokutoh Hospital Department of Internal Medicine 4-23-15, Koutoh-bashi, Sumida-ku 130-8575 Tokyo Japan
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Endoscopic endonasal transsphenoidal surgery: surgical results of 228 pituitary adenomas treated in a pituitary center
Abstract Pituitary tumors are challenging tumors in the sellar region. Surgical approaches to the pituitary have undergone numerous refinements over the last 100 years. The introduction of the endoscope have revolutionized pituitary surgery. The aim of this study is to report the results of a consecutive series of patients undergoing pituitary surgery using a pure endoscopic endonasal approach and to evaluate the efficacy and safety of this procedure. We reviewed the data of 228 consecutive patients who underwent endonasal transsphenoidal adenoma removal over an 10-year period. Pre- and post-operative hormonal status (at least 3 months after surgery) were analyzed and compared with clinical parameters presented by the patients. Tumor removal rate, endocrinological outcomes, and complications were retrospectively assessed in 228 patients with pituitary adenomas who underwent 251 procedures between December 1998 and December 2007. There were 93 nonfunctioning adenomas, 58 growth hormone-secreting, 41 prolactin-secreting, 28 adrenocorticotropin hormone secreting, 7 FSH-LH secreting and 1 thyroid-stimulating hormone-secreting adenomas. Gross total removal was achieved in 79.3% of the cases after a median follow-up of 61.5 months. The remission results for patients with nonfunctioning adenomas was 83% and for functioning adenomas were 76.3% (70.6% for GH hormone-secreting, 85.3% for prolactin hormone-secreting, 71.4% for ACTH hormone-secreting, 85.7% for FSH-LH hormone-secreting and 100% for TSH hormone-secreting), with no recurrence at the time of the last follow-up. Post-operative complications were present in 35 (13.9%) cases. The most frequent complications were temporary and permanent diabetes insipidus (six and two cases, respectively), syndrome of inappropriate antidiuretic hormone secretion (two cases) and CSF leaks (eight cases). There was no death related to the procedure in this series. The endoscopic endonasal approach for resection of pituitary adenomas, provides acceptable results representing a safe alternative procedure to the microscopic approach. This less invasive method, associated with a small number of complications, provides excellent tumor removal rates and represents an important tool for the achievement of good results in the pituitary surgery, mainly for the complete removal of large adenomas.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0195-x
- Authors
- Jackson A. Gondim, General Hospital of Fortaleza Department of Neurosurgery Fortaleza Brazil
- Michele Schops, Federal University of Ceara School of Medicine Fortaleza Brazil
- João Paulo C. de Almeida, General Hospital of Fortaleza Department of Neurosurgery Fortaleza Brazil
- Lucas Alverne F. de Albuquerque, General Hospital of Fortaleza Department of Neurosurgery Fortaleza Brazil
- Erika Gomes, General Hospital of Fortaleza Department of Ear, Nose and Throat Surgery Fortaleza Brazil
- Tânia Ferraz, General Hospital of Fortaleza Department of Endocrinology Fortaleza Brazil
- Francisca Andréa C. Barroso, General Hospital of Fortaleza Department of Endocrinology Fortaleza Brazil
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Possible role of a radiation-induced p53 mutation in a Nelson?s syndrome patient with a fatal outcome
Abstract Nelson?s syndrome (NS) is characterized by the appearance and/or progression of ACTH-secreting pituitary macroadenomas in patients who had previously undergone bilateral adrenalectomy for the treatment of Cushing?s disease. Such corticotroph macroadenomas respond poorly to currently available therapeutic options which include surgery, radiotherapy and chemotherapy. P53 protein accumulation may be detected by immunohistochemistry in pituitary corticotroph adenomas and it has been suggested that it might be causally related to tumor development. Wild type P53 protein plays an important role in the cellular response to ionizing radiation and other DNA damaging agents and is mutated in many human tumors. In this study we report an adult male patient with NS who underwent both transsphenoidal and transcranial pituitary surgeries, conventional and stereotaxic radiotherapy and brachytherapy. Despite of the efforts to control tumor mass and growth, this macroadenoma displayed relentless growth and aggressive behavior. DNA extracted from the first two surgical samples, as well as DNA from peripheral blood leukocytes disclosed normal p53 sequence. DNA extracted from tumor samples obtained at surgeries performed after pituitary irradiation carried a somatic heterozygous mutation, consisting of a deletion of four cytosines between nucleotides 12,144?12,149 in exon 4 of the p53 gene. This frameshift mutation creates a stop codon in exon 4 excluding the expression of a functional protein from the defective allele. These data demonstrate a possible association between the P53 protein loss of function induced by radiotherapy and the aggressive course of the disease in this patient.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0194-y
- Authors
- Emilia Modolo Pinto, Faculdade de Medicina da Universidade de São Paulo Unidade de Endocrinologia do Desenvolvimento, Laboratório de Hormônios e Genética Molecular LIM/42, Disciplina de Endocrinologia e Metabologia, Hospital das Clínicas São Paulo Brazil
- Sheila A. C. Siqueira, Faculdade de Medicina da Universidade de São Paulo Divisão de Anatomia Patológica, Hospital das Clínicas São Paulo Brazil
- Priscilla Cukier, Faculdade de Medicina da Universidade de São Paulo Unidade de Endocrinologia do Desenvolvimento, Laboratório de Hormônios e Genética Molecular LIM/42, Disciplina de Endocrinologia e Metabologia, Hospital das Clínicas São Paulo Brazil
- Maria C. B. V. Fragoso, Faculdade de Medicina da Universidade de São Paulo Unidade de Endocrinologia do Desenvolvimento, Laboratório de Hormônios e Genética Molecular LIM/42, Disciplina de Endocrinologia e Metabologia, Hospital das Clínicas São Paulo Brazil
- Chin Jia Lin, Faculdade de Medicina da Universidade de São Paulo Laboratório de Patologia Molecular, LIM22, Departamento de Patologia Av. Dr. Arnaldo, 455, sala 1144 São Paulo 01246-903 Brazil
- Berenice Bilharinho de Mendonca, Faculdade de Medicina da Universidade de São Paulo Unidade de Endocrinologia do Desenvolvimento, Laboratório de Hormônios e Genética Molecular LIM/42, Disciplina de Endocrinologia e Metabologia, Hospital das Clínicas São Paulo Brazil
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Anti-CTLA-4 antibody therapy associated autoimmune hypophysitis: serious immune related adverse events across a spectrum of cancer subtypes
Abstract Anti-cytotoxic T-lymphocyte antigen-4 (CTLA-4) therapies represent a novel approach to cancer treatment via disruption of immune tolerance to antigens located on tumor cells. Disruption of immune tolerance, however, may occur at a cost. A host of immune related adverse events (IRAEs) are associated with anti-CTLA-4 therapy. Autoimmune hypophysitis has been reported in up to 17% of patients with melanoma and renal cell carcinoma treated with this therapy. Familiarity with the spectrum of IRAEs connected to these therapies is paramount for endocrinologists, oncologists and those involved in the care of these subjects. We review here key aspects of diagnosis and treatment of anti-CTLA-4 antibody therapy resultant IRAEs. We describe the first two cases of hypopituitarism in prostate cancer subjects undergoing experimental therapy with ipilimumab. The clinical evidence strongly suggests that the prostate cancer subjects developed autoimmune hypophysitis as a consequence of anti-CTLA-4 treatment. High dose glucocorticoid treatment resulted in markedly improved symptoms, and resolution of focal symptoms and diabetes insipidus. One subject recovered pituitary-thyroid axis function after 9 months; however, both continue to require GC replacement. These cases highlight the importance of early screening and treatment for hypopituitarism in all subjects undergoing treatment with anti-CTLA-4 therapy to prevent a potentially fatal outcome from secondary adrenal insufficiency, a readily treatable disease. We recommend mandatory long term follow-up to monitor the development of other hormonal deficits.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0193-z
- Authors
- Troy Dillard, Oregon Health & Science University Department of Medicine, Division of Endocrinology, Diabetes and Clinical Nutrition Portland OR USA
- Chris G. Yedinak, Oregon Health & Science University Department of Neurological Surgery, Oregon Health & Science University Portland OR USA
- Joshi Alumkal, Oregon Health & Science University Department of Medicine, Division of Hematology and Oncology Portland OR USA
- Maria Fleseriu, Oregon Health & Science University Department of Medicine, Division of Endocrinology, Diabetes and Clinical Nutrition Portland OR USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Rapid and sustained reduction of serum growth hormone and insulin-like growth factor-1 in patients with acromegaly receiving lanreotide Autogel® therapy: a randomized, placebo-controlled, multicenter study with a 52 week open extension
Abstract The study was designed to evaluate the long-term efficacy and safety of the 28-day prolonged-release Autogel formulation of the somatostatin analogue lanreotide (Lan-Autogel) in unselected patients with acromegaly. The study comprised four phases: washout; a double-blind comparison with placebo, at a single randomized dose (60, 90 or 120 mg) of Lan-Autogel; a single-blind, fixed-dose phase for four injections (placebo group was re-allocated to active treatment); and eight injections with doses tailored according to biochemical response. Serum samples were assessed for growth hormone (GH) and insulin-like growth factor-1 (IGF-1) levels, at weeks 4, 13, 14, 15, 16, 32 and 52. 108 patients were enrolled and 99 completed 52 weeks? treatment. Four weeks after the first injection, serum GH levels decreased by >50% from baseline in 63% of patients receiving Lan-Autogel compared with 0% receiving placebo (P < 0.001). After four injections, 72% of patients had a >50% reduction in GH levels; 49% patients achieved GH levels ? 2.5 ng/ml; 54% had normalized IGF-1; and 38% achieved the combined criterion of GH level ? 2.5 ng/ml and normalized IGF-1. The corresponding proportions by week 52 were 82, 54, 59 and 43%, respectively. In patients not requiring dose escalation to 120 mg, 85% achieved biochemical control (combined criterion). Treatment was well tolerated by all patients. In conclusion, Lan-Autogel was effective in controlling GH and IGF-1 hypersecretion in patients with acromegaly and showed a rapid onset of action.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0191-1
- Authors
- Shlomo Melmed, Cedars-Sinai Medical Center Department of Medicine Los Angeles CA USA
- David Cook, Oregon Health & Science University Department of Medicine Portland OR USA
- Jochen Schopohl, Medizinische Klinik Innenstadt Division of Endocrinology Munich Germany
- Miklos I. Goth, National Health Center Division of Endocrinology, 2nd Department of Medicine Budapest Hungary
- Karen S. L. Lam, University of Hong Kong Department of Medicine Hong Kong China
- Josef Marek, Charles University 3rd Department of Medicine, 1st School of Medicine Prague Czech Republic
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Rathke?s cleft cyst presenting as bilateral abducens nerve palsy
Abstract We present a patient with a Rathke?s cleft cyst who presented with rapidly progressive bilateral 6th nerve palsy. A 20-year-old woman with a history of cleft palate, hypertension, and hydronephrotic kidneys presented with a one month history of headache, associated with dizziness and diplopia on horizontal gaze. Examination was significant for profound bilateral 6th nerve palsies. Magnetic resonance imaging showed a hypodense mass that filled the sella and compressed the right cavernous sinus without contacting the optic chiasm. Pituitary function was normal. An endoscopic, transnasal transsphenoidal resection of the lesion was performed; microscopic examination revealed a Rathke?s cleft cyst. Surgical excision resulted in near complete resolution of the bilateral 6th nerve palsy. Rathke?s cleft cysts are an unusual cause of bilateral sixth nerve palsy and represent a potential cause of cranial neuropathy.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0192-0
- Authors
- Vinni Grover, Cleveland Clinic Department of Endocrinology, Diabetes and Metabolism, Endocrinology and Metabolism Institute 9500 Euclid Avenue Cleveland OH 44195 USA
- Amir H. Hamrahian, Cleveland Clinic Department of Endocrinology, Diabetes and Metabolism, Endocrinology and Metabolism Institute 9500 Euclid Avenue Cleveland OH 44195 USA
- Richard A. Prayson, Cleveland Clinic The Department of Pathology and Laboratory Medicine Cleveland OH USA
- Robert J. Weil, Cleveland Clinic The Brain Tumor and Neuro-Oncology Center, Department of Neurosurgery, The Neurological Institute 9500 Euclid Avenue Cleveland OH 44195 USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Collision sellar lesions: experience with eight cases and review of the literature
Abstract The concomitant presence of a pituitary adenoma with a second sellar lesion in patients operated upon for pituitary adenoma is an uncommon entity. Although rare, quite a great variety of lesions have been indentified coexisting with pituitary adenomas. In fact, most combinations have been described before, but an overview with information on the frequency of combined pathologies in a large series has not been published. We present a series of eight collision sellar lesions indentified among 548 transsphenoidally resected pituitary adenomas in two Neurosurgical Departments. The histological studies confirmed a case of sarcoidosis within a non-functioning pituitary adenoma, a case of intrasellar schwannoma coexisting with growth hormone (GH) secreting adenoma, two Rathke?s cleft cysts combined with pituitary adenomas, three gangliocytomas associated with GH-secreting adenomas, and a case of a double pituitary adenoma. The pertinent literature is discussed with emphasis on pathogenetic theories of dual sellar lesions. Although there is no direct evidence to confirm the pathogenetic relationship of collision sellar lesions, the number of cases presented in literature makes the theory of an incidental occurrence rather doubtful. Suggested hypotheses about a common embryonic origin or a potential interaction between pituitary adenomas and the immune system are presented.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0190-2
- Authors
- Maria Koutourousiou, G. Gennimatas Athens General Hospital Department of Neurosurgery 154 Messogion Ave 115 27 Athens Greece
- George Kontogeorgos, G. Gennimatas Athens General Hospital Department of Pathology Athens Greece
- Pieter Wesseling, UMC St Radboud Department of Pathology Nijmegen The Netherlands
- Andre J. Grotenhuis, UMC St Radboud Department of Neurosurgery Nijmegen The Netherlands
- Andreas Seretis, G. Gennimatas Athens General Hospital Department of Neurosurgery 154 Messogion Ave 115 27 Athens Greece
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Immunohistochemistry of COUP-TFI: an adjuvant diagnostic tool for the identification of corticotroph microadenomas
Abstract Cushing?s disease is caused by an ACTH-producing pituitary tumor, and accounts for 10?15% of pituitary tumors. The majority of corticotroph tumors are microadenomas (<10 mm), and accurate histologic identification of these tumors can be challenging because of their small size and the presence of nests of normal corticotroph cells in the anterior pituitary. Retinoic acid has been shown to inhibit ACTH production and induce apoptosis in corticotroph tumor cells. The expression of the orphan nuclear receptor COUP-TFI antagonizes retinoic acid signaling and has been shown to be expressed in normal corticotroph cells, but absent in corticotroph tumor cell lines. We analyzed 34 corticotroph tumor specimens by immunohistochemistry using a goat polyclonal IgG antibody with epitope mapping to the N-terminus of human COUP-TFI. Segments of normal pituitary in each of the 34 specimens demonstrate COUP-TFI immunoreactivity in normal corticotroph cells. Twenty-nine of 34 ACTH producing tumors were immunonegative for COUP-TFI. All of the tumors measuring less than 5 mm by preoperative MRI were COUP-TFI immunonegative. Two tumors, measuring 9 and 11 mm, showed consistent (>90%) expression of COUP-TFI, and three adenomas (5, 11, and 18 mm) showed heterogenous (20?80%) expression of COUP-TFI. Immunohistochemistry of COUP-TFI may be a useful adjuvant diagnostic tool in distinguishing corticotroph microadenomas from nests of normal corticotroph cells in the anterior pituitary. Furthermore, this study identifies two unique corticotroph tumor populations which differ in their expression of COUP-TFI, the presence of which occurs more frequently in macroadenomas.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0189-8
- Authors
- Zachary M. Bush, University of Virginia Health System Division of Endocrinology and Metabolism P.O. Box 801408 Charlottesville VA 22908-1408 USA
- Maria-Beatriz S. Lopes, University of Virginia Health System Department of Pathology Charlottesville VA 22908-1408 USA
- Isa M. Hussaini, University of Virginia Health System Department of Pathology Charlottesville VA 22908-1408 USA
- John A. Jane, University of Virginia Health System Department of Neurosurgery Charlottesville VA 22908-1408 USA
- Edward R. Laws, Brigham and Women?s Hospital Department of Neurosurgery Boston Massachusetts 02115 USA
- Mary Lee Vance, University of Virginia Health System Division of Endocrinology and Metabolism P.O. Box 801408 Charlottesville VA 22908-1408 USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 13
- Journal Issue Volume 13, Number 1 / March, 2010
- Limited effects of growth hormone replacement in patients with GH deficiency during long-term cure of acromegaly
Abstract The aim of this study was to assess the effects of replacement with recombinant human growth hormone (rhGH) in patients with GH deficiency (GHD) after treatment of acromegaly. Intervention study. Sixteen patients (8 men, age 56 years), treated for acromegaly by surgery and radiotherapy, with an insufficient GH response to insulin-induced hypoglycaemia, were treated with 1 year of rhGH replacement. Study parameters were assessed at baseline and after 1 year of rhGH replacement. Study parameters were cardiac function, body composition, bone mineral density (BMD), fasting lipids, glucose, bone turnover markers, and Quality of Life (QoL). During rhGH replacement IGF-I concentrations increased from ?0.4 ± 0.7 to 1.0 ± 1.5 SD (P = 0.001), with a mean daily dose of 0.2 ± 0.1 mg in men and 0.3 ± 0.2 mg in women. Nonetheless, rhGH replacement did not alter cardiac function, lipid and glucose concentrations, body composition or QoL. Bone turnover markers (PINP and ? crosslaps) levels increased (P = 0.005 and P = 0.021, respectively), paralleled by a small, but significant decrease in BMD of the hip. The beneficial effects of rhGH replacement in patients with GHD during cure from acromegaly are limited in this study.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0186-y
- Authors
- Agatha A. van der Klaauw, Leiden University Medical Center Department of Endocrinology and Metabolic Diseases C4-R P.O. Box 9600 2300 RC Leiden The Netherlands
- Jeroen J. Bax, Leiden University Medical Center Department of Cardiology P.O. Box 9600 2300 RC Leiden The Netherlands
- Ferdinand Roelfsema, Leiden University Medical Center Department of Endocrinology and Metabolic Diseases C4-R P.O. Box 9600 2300 RC Leiden The Netherlands
- Marcel P. M. Stokkel, Leiden University Medical Center Department of Nuclear Medicine P.O. Box 9600 2300 RC Leiden The Netherlands
- Gabe B. Bleeker, Leiden University Medical Center Department of Cardiology P.O. Box 9600 2300 RC Leiden The Netherlands
- Nienke R. Biermasz, Leiden University Medical Center Department of Endocrinology and Metabolic Diseases C4-R P.O. Box 9600 2300 RC Leiden The Netherlands
- Johannes W. A. Smit, Leiden University Medical Center Department of Endocrinology and Metabolic Diseases C4-R P.O. Box 9600 2300 RC Leiden The Netherlands
- Johannes A. Romijn, Leiden University Medical Center Department of Endocrinology and Metabolic Diseases C4-R P.O. Box 9600 2300 RC Leiden The Netherlands
- Alberto M. Pereira, Leiden University Medical Center Department of Endocrinology and Metabolic Diseases C4-R P.O. Box 9600 2300 RC Leiden The Netherlands
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Pituitary and systemic autoimmunity in a case of intrasellar germinoma
Abstract Germinomas arising in the sella turcica are difficult to differentiate from autoimmune hypophysitis because of similar clinical and pathological features. This differentiation, nevertheless, is critical for patient care due to different treatments of the two diseases. We report the case of an 11-year-old girl who presented with diabetes insipidus and growth retardation, and was found to have an intra- and supra-sellar mass. Initial examination of the pituitary biopsy showed diffuse lymphocytic infiltration of the adenohypophysis and absent placental alkaline phosphatase expression, leading to a diagnosis of hypophysitis and glucocorticoid treatment. Because of the lack of clinical and radiological response, the pituitary specimen was re-examined, revealing this time the presence of scattered c-kit and Oct4 positive germinoma cells. The revised diagnosis prompted the initiation of radiotherapy, which induced disappearance of the pituitary mass. Immunological studies showed that the patient?s serum recognized antigens expressed by the patient?s own germinoma cells, as well as pituitary antigens like growth hormone and systemic antigens like the Sjögren syndrome antigen B and alpha-enolase. The study first reports the presence of pituitary and systemic antibodies in a patient with intrasellar germinoma, and reminds us that diffuse lymphocytic infiltration of the pituitary gland and pituitary antibodies does not always indicate a diagnosis of autoimmune hypophysitis.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0187-x
- Authors
- Angelika Gutenberg, Georg August University Göttingen Department for Neurosurgery Göttingen Germany
- Jennifer J. Bell, Texas Children?s Hospital Department for Pediatrics Houston TX USA
- Isabella Lupi, University of Pisa Department of Endocrinology Pisa Italy
- Shey-Cherng Tzou, Johns Hopkins University Department for Pathology Baltimore MD USA
- Melissa A. Landek-Salgado, Johns Hopkins University Department for Pathology Baltimore MD USA
- Hiroaki Kimura, Johns Hopkins University Department for Pathology Baltimore MD USA
- Jack Su, Texas Children?s Hospital Department for Pediatrics Houston TX USA
- Lefkothea P. Karaviti, Texas Children?s Hospital Department for Pediatrics Houston TX USA
- Roberto Salvatori, Johns Hopkins University Division of Endocrinology, Department of Medicine Baltimore MD USA
- Patrizio Caturegli, Johns Hopkins University Department for Pathology Baltimore MD USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Prevalence and clinical demographics of cerebral salt wasting in patients with aneurysmal subarachnoid hemorrhage
Abstract Hyponatremia is a frequent complication following subarachnoid hemorrhage (SAH), and is commonly attributed either to the syndrome of inappropriate antidiuretic hormone secretion (SIADH) or cerebral salt wasting syndrome (CSW). The object of this study is to elucidate the clinical demographics and sequelae of hyponatremia due to CSW in subjects with aneurysmal SAH. Retrospective chart review of patients >18 years with aneurysmal SAH admitted between January 2004 and July 2007 was performed. Subjects with moderate to severe hyponatremia (serum sodium <130 mmol l?1) were divided into groups consistent with CSW and SIADH based on urine output, fluid balance, natriuresis, and response to saline infusion. Clinical demographics were compared. Of 316 subjects identified, hyponatremia (serum sodium <135 mmol l?1) was detected in 187 (59.2%) subjects and moderate to severe hyponatremia in 48 (15.2%). Of the latter group, 35.4% were categorized with SIADH and 22.9% with CSW. Compared to eunatremic subjects, hyponatremia was associated with significantly longer hospital stay (15.7 ± 1.9 vs. 9.6 ± 1.1 days, p < 0.001). Subjects with CSW had similar mortality and duration of hospital stay vs. those with SIADH. Though less common than SIADH, CSW was detected in approximately 23% of patients with history of aneurysmal SAH and was not clearly associated with enhanced morbidity and mortality compared to subjects with SIADH. Further studies regarding the pathogenesis and management, along with the medical consequences, of CSW are important.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0188-9
- Authors
- Lily Kao, Stanford University School of Medicine Department of Medicine Stanford CA 94305 USA
- Zahraa Al-Lawati, Stanford University School of Medicine Department of Medicine Stanford CA 94305 USA
- Joli Vavao, Stanford University School of Medicine Department of Neurosurgery Stanford CA 94305 USA
- Gary K. Steinberg, Stanford University School of Medicine Department of Neurosurgery Stanford CA 94305 USA
- Laurence Katznelson, Stanford University School of Medicine Department of Medicine Stanford CA 94305 USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Pituicytoma and isolated ACTH deficiency
Pituicytoma and isolated ACTH deficiency
- Content Type Journal Article
- Category Letter
- DOI 10.1007/s11102-009-0185-z
- Authors
- John J. Orrego, Kaiser Permanente Department of Endocrinology and Metabolism 280 Exempla Circle Lafayette CO 80026 USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Evaluation of insulin sensitivity in hyperprolactinemic subjects by euglycemic hyperinsulinemic clamp technique
Abstract The background and aim of the study is to evaluate insulin sensitivity in hyperprolactinemic subjects via euglycemic hyperinsulinemic clamp technique. Sixteen hyperprolactinemic subjects and 12 healthy subjects were included in the study. HOMA-B and HOMA-IR values of groups were calculated. Euglycemic hyperinsulinemic clamp technique was performed in both groups, and the M value of the groups was defined. Mann?Whitney U and chi-square tests were used in statistical analysis. Basal insulin level of hyperprolactinemic patients were higher than the control group (6.85 ± 4.68; 3.66 ± 0.88 ?U/ml respectively; P < 0.05). Mean HOMA-IR and HOMA-B values of patients were higher than control group (1.49 ± 1.30; 0.78 ± 0.27 respectively; P = 0.02 and 136.28 ± 72.53; 64.77 ± 23.31, respectively, P < 0.001). M values of the patients were statistically lower than the control group (5.64 ± 2.36; 7.05 ± 1.62 kg/mg/min respectively; P < 0.05). (1) Hyperprolactinemic patients were more insulin resistant than control subjects. (2) Insulin resistance in hyperprolactinemic patients is not associated with obesity or anthropometric parameters such as fat content, waist circumference and BMI.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0183-1
- Authors
- Alpaslan Tuzcu, Dicle University School of Medicine Department of Endocrinology 21280 Diyarbakir Turkey
- Serkan Yalaki, Dicle University School of Medicine Department of Internal Medicine Diyarbakir Turkey
- Senay Arikan, Dicle University School of Medicine Department of Endocrinology 21280 Diyarbakir Turkey
- Deniz Gokalp, Dicle University School of Medicine Department of Endocrinology 21280 Diyarbakir Turkey
- Mithat Bahcec, Dicle University School of Medicine Department of Endocrinology 21280 Diyarbakir Turkey
- Sadiye Tuzcu, Dicle University School of Medicine Department of Nuclear Medicine Diyarbakir Turkey
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Utility of single luteinizing hormone determination 3 h after depot leuprolide in monitoring therapy of gonadotropin-dependent precocious puberty
Abstract To determine utility of luteinizing hormone (LH) estimation, post intramuscular (IM) depot leuprolide in comparison with subcutaneous leuprolide stimulation test. Test for monitoring therapy in patients with gonadotropin dependent precocious puberty (GDPP). In seven patients of GDPP, who were treated with 11.25 mg depot leuprolide, the LH peak after subcutaneous (sc) test was compared with LH at hourly interval for 4 h after IM depot leuprolide for 13 tests and 3rd hour value for next ten tests. These two values were compared both before and after therapy. Before therapy, the mean ± SD LH peak after subcutaneous leuprolide stimulation test was 20.6 ± 7.85 IU/l (range 9.64?30.4 IU/l), and it was 27.3 ± 12.21 IU/l 3 h after the first depot leuprolide injection (range 10.5?45.4 IU/l). During therapy, the mean ± SD of LH peak after sc stimulation test was 1.96 ± 0.75 IU/l (range 1.1?3.1 IU/l), and it was 2.58 ± 0.54 IU/l (range 1.4?3.4 IU/l) 3 h after depot leuprolide injection. Three-hour LH value following IM depot leuprolide injection (11.25 mg) can be used for monitoring therapy in patients with GDPP because of its convenience and cost effectiveness.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0184-0
- Authors
- Shrikrishna V. Acharya, K E M Hospital Department of Endocrinology Parel Mumbai-12 Maharashta India
- Raju A. Gopal, K E M Hospital Department of Endocrinology Parel Mumbai-12 Maharashta India
- Joe George, K E M Hospital Department of Endocrinology Parel Mumbai-12 Maharashta India
- Tushar R. Bandgar, K E M Hospital Department of Endocrinology Parel Mumbai-12 Maharashta India
- Padma S. Menon, K E M Hospital Department of Endocrinology Parel Mumbai-12 Maharashta India
- Nalini S. Shah, K E M Hospital Department of Endocrinology Parel Mumbai-12 Maharashta India
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Ghrelin and GHRP-6-induced ACTH and cortisol release in thyrotoxicosis
Abstract Thyrotoxicosis might alter the hypothalamic-pituitary-adrenal (HPA) axis. We evaluated the effects of ghrelin and GHRP-6 on the HPA axis in 20 hyperthyroid patients and in 9 controls. Mean basal cortisol (?g/dl) and ACTH (pg/ml) levels were higher in hyperthyroidism (cortisol: 10.7 ± 0.7; ACTH: 21.5 ± 2.9) compared to controls (cortisol: 8.1 ± 0.7; ACTH: 13.5 ± 1.8). In thyrotoxicosis ∆ AUC cortisol values (?g/dl.90 min) after ghrelin (484 ± 80) and GHRP-6 (115 ± 63) were similar to controls (ghrelin: 524 ± 107; GHRP-6: 192 ± 73). A significant increase in ∆ AUC ACTH (pg/ml.90 min) after ghrelin was observed in thyrotoxicosis (4,189 ± 1,202) compared to controls (1,499 ± 338). ∆ AUC ACTH values after GHRP-6 were also higher, although not significantly (patients: 927 ± 330; controls: 539 ± 237). In summary, our results suggest that ghrelin-mediated pathways of ACTH release might be activated by thyroid hormone excess, but adrenocortical reserve is maintained.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0181-3
- Authors
- Sergio Oliva Nascif, Federal University of São Paulo, UNIFESP/EPM Division of Endocrinology Pedro de Toledo Street, 910 São Paulo SP 04039-002 Brazil
- Patrícia Molica, Federal University of São Paulo, UNIFESP/EPM Division of Endocrinology Pedro de Toledo Street, 910 São Paulo SP 04039-002 Brazil
- Silvia Regina Correa-Silva, Federal University of São Paulo, UNIFESP/EPM Division of Endocrinology Pedro de Toledo Street, 910 São Paulo SP 04039-002 Brazil
- Marcos Roberto Silva, Federal University of São Paulo, UNIFESP/EPM Division of Endocrinology Pedro de Toledo Street, 910 São Paulo SP 04039-002 Brazil
- Ana-Maria Judith Lengyel, Federal University of São Paulo, UNIFESP/EPM Division of Endocrinology Pedro de Toledo Street, 910 São Paulo SP 04039-002 Brazil
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Effects of 5 years of growth hormone (GH) replacement therapy on cardiac parameters and physical performance in adults with GH deficiency
Abstract The purpose of this study was to evaluate the effects of 5 years of GH substitution on cardiac structure and function, physical work capacity and blood pressure levels in adults with GH deficiency (GHD). Fourteen patients were clinically assessed every 3 months for 5 years. Transthoracic echocardiography and exercise test were performed at baseline, 24, 48 and 60 months. Blood pressure (BP) was measured by means of ambulatory monitoring of blood pressure at baseline, 6, 12, 24 and 60 months. Left ventricular mass and its index increased progressively during the 5 years of GH substitution (P = 0.008 and 0.007, respectively). There were no significant changes in all others cardiac parameters evaluated. It was observed a significant improve in functional capacity (P < 0.001) and maximal oxygen uptake (P = 0.006) during the treatment. Diurnal systolic BP increased by 15 mmHg (P = 0.024) and diurnal diastolic BP by 4.5 mmHg (P = 0.037). There was no change in dirnal systolic pressure load but a considerable but non-statistically significant reduction in diurnal diastolic pressure load was observed during the study. During the night diastolic BP increased by 4 mmHg (P = 0.012) despite a substantial but non-statistically significant reduction in diastolic pressure load. We observed an increase in the proportion of persons with a non-physiological nocturnal fall (non-dippers) throughout the study (from 36.4% at baseline to 54.6% after 60 months of therapy). We concluded that 5 years of GH replacement promoted positive effects on exercise capacity and maximum oxygen uptake in spite of a modest increase in BP levels and left ventricular mass. Continuous monitoring is mandatory to arrive at further conclusions concerning the effects of GH substitution in adults on cardiovascular parameters with respect to possible unfavorable long term effects.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0182-2
- Authors
- Maria Claudia Peixoto Cenci, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Débora Vieira Soares, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Luciana Diniz Carneiro Spina, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Rosane Resende de Lima Oliveira Brasil, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Priscila Marise Lobo, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Vera Aleta Mansur, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Jaime Gold, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Eduardo Michmacher, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Mario Vaisman, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Flávia Lúcia Conceição, Federal University of Rio de Janeiro Service of Endocrinology, Clementino Fraga Filho University Hospital Rio de Janeiro Brazil
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- The diagnostic value of fused positron emission tomography/computed tomography in the localization of adrenocorticotropin-secreting pituitary adenoma in Cushing?s disease
Abstract Despite the high resolution of magnetic resonance imaging (MRI) of the pituitary gland, up to 40% of cases of Cushing?s disease (CD) have normal MRI. Fused images of positron emission tomography and computed tomography (PET-CT) may have a potential diagnostic role in CD in general and in such cases in particular. Objective of this study is to explore the diagnostic potential of PET-CT for localization of adrenocorticotropin-secreting pituitary adenomas in CD. PET-CT was performed in 12 cases with de novo (7 cases) or persistent CD (5 cases) that were proven to have CD on biochemical, radiological and/or histopathological findings. These cases had a definite CD confirmed on histopathological and immunostaining examination of the subsequent transphenoidal surgical specimens (10 cases) and/or bilateral inferior petrosal sinus sampling (IPSS, 4 cases). PET-CT was positive in 7 of the 12 cases of CD (58%) showing a focal area of uptake in the pituitary gland. In these seven cases, MRI was positive in six (85.7%) but negative in one case (14.3%). In the other five cases with negative PET-CT, MRI was positive in two and negative in three cases. Of four cases with negative MRI, PET-CT was positive in one case (25%). We conclude that PET-CT is positive in around 60% of the cases of CD. Although the majority of cases with positive PET-CT had positive MRI, PET-CT may detect some cases with negative MRI and thus provides important diagnostic information. If these findings are confirmed in larger studies, PET-CT might become an important diagnostic technique, especially when the more invasive and technically demanding procedure of IPSS is not available or inconclusive.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0180-4
- Authors
- Ali S. Alzahrani, King Faisal Specialist Hospital and Research Centre Department of Medicine (MBC-46) P.O. Box 3354 Riyadh 11211 Saudi Arabia
- Rafif Farhat, King Faisal Specialist Hospital and Research Centre Department of Medicine (MBC-46) P.O. Box 3354 Riyadh 11211 Saudi Arabia
- Abdullah Al-Arifi, King Faisal Specialist Hospital and Research Centre Department of Medicine (MBC-46) P.O. Box 3354 Riyadh 11211 Saudi Arabia
- Nora Al-Kahtani, King Faisal Specialist Hospital and Research Centre Department of Medicine (MBC-46) P.O. Box 3354 Riyadh 11211 Saudi Arabia
- Imad Kanaan, King Faisal Specialist Hospital and Research Centre Department of Neuroscience Riyadh Saudi Arabia
- Mohei Abouzied, King Faisal Specialist Hospital and Research Centre Department of Radiology Riyadh Saudi Arabia
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Brain and optic chiasmal herniations into sella after cabergoline therapy of giant prolactinoma
Abstract Optic chiasmal herniation following dopamine agonist therapy is a rare complication in patients with giant prolactinomas. But there are a few case reports of brain and chiasmal herniation following medical therapy in such cases. We report a young man who developed secondary visual loss and seizures after 6 months of medical treatment with cabergoline for giant prolactinoma. Magnetic resonance imaging of hypothalamic pituitary region revealed optic chiasmal and frontal lobe herniation into sella. There was marginal improvement in his vision after cabergoline dose reduction. The present case report highlights frontal lobe herniation in conjunction with optic chiasmal herniation as a very rare complication of medical therapy of giant prolactinoma. Different treatment options of this condition are being discussed.
- Content Type Journal Article
- Category CASE REPORT
- DOI 10.1007/s11102-009-0179-x
- Authors
- Dinesh Kumar Dhanwal, Maulana Azad Medical College Department of Medicine and Division of Endocrinology 115, B.L. Taneja Block, Bahadur Shah Zafar Marg New Delhi 110002 India
- Ashok Kumar Sharma, Maulana Azad Medical College Department of Radiology, G.B. Pant Hospital New Delhi India
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Effects of physical training on serum and pituitary growth hormone contents in diabetic rats
Abstract The present study investigated the effects of moderate physical training on some of the parameters in the GH-IGF axis in experimental diabetic rats. Male Wistar rats were allocated into the following groups: sedentary control, trained control, sedentary diabetic, trained diabetic. Diabetes was induced by alloxan (32 mg/kg, b.w. iv). The physical training protocol consisted of 1 h swimming session/day, 5 days/week for 8 weeks supporting a load corresponding to 90% of maximal lactate steady state. After the experimental period, blood was collected to measure serum glucose, insulin, triglycerides, albumin, insulin-like growth factors-I (IGF-I), and growth hormone (GH). Pituitary gland was removed for GH quantification. Diabetes increased blood glucose and triglycerides and decreased insulin, IGF-I, serum and pituitary GH. Physical training decreased glucose and triglycerides, and also counteracted the reduction of serum IGF-I in diabetic rats. In conclusion, physical training recovered serum IGF-I showing no alteration of serum or pituitary GH levels.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0178-y
- Authors
- José Alexandre Curiacos de Almeida Leme, São Paulo State University (UNESP) Department of Physical Education Avenida 24A no. 1515 Caixa Postal 199 Bela Vista, Rio Claro São Paulo CEP 13506-900 Brazil
- Michel Barbosa de Araújo, São Paulo State University (UNESP) Department of Physical Education Avenida 24A no. 1515 Caixa Postal 199 Bela Vista, Rio Claro São Paulo CEP 13506-900 Brazil
- Leandro Pereira de Moura, São Paulo State University (UNESP) Department of Physical Education Avenida 24A no. 1515 Caixa Postal 199 Bela Vista, Rio Claro São Paulo CEP 13506-900 Brazil
- Ricardo José Gomes, São Paulo State University (UNESP) Department of Physical Education Avenida 24A no. 1515 Caixa Postal 199 Bela Vista, Rio Claro São Paulo CEP 13506-900 Brazil
- Rodrigo Ferreira de Moura, São Paulo State University (UNESP) Department of Physical Education Avenida 24A no. 1515 Caixa Postal 199 Bela Vista, Rio Claro São Paulo CEP 13506-900 Brazil
- Gustavo Puggina Rogatto, São Paulo State University (UNESP) Department of Physical Education Avenida 24A no. 1515 Caixa Postal 199 Bela Vista, Rio Claro São Paulo CEP 13506-900 Brazil
- Maria Alice Rostom de Mello, São Paulo State University (UNESP) Department of Physical Education Avenida 24A no. 1515 Caixa Postal 199 Bela Vista, Rio Claro São Paulo CEP 13506-900 Brazil
- Eliete Luciano, São Paulo State University (UNESP) Department of Physical Education Avenida 24A no. 1515 Caixa Postal 199 Bela Vista, Rio Claro São Paulo CEP 13506-900 Brazil
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Multiple head and neck tumors following treatment for craniopharyngioma
Abstract To report a unique case of a patient with multiple head and neck tumors following treatment for craniopharyngioma. Retrospective review of the medical record. There is an association between radiation treatment for primary intracranial malignancies and the subsequent development of other intracranial tumors. At the same time, reports regarding the association between thyroid cancer and radiation exposure are abundant within the literature. We present a unique case of a young female who underwent resection and radiation treatment for a craniopharyngioma in 1988. With the exception of bitemporal visual loss and panhypopituitarism requiring hormone replacement therapy, the patient had a relatively uneventful course until 2005. At that time, she developed hearing loss and incapacitating vertigo, which was ultimately attributed to a temporal bone Langerhans histiocytosis. A year and a half later, she was noted to have a thyroid nodule, the pathology of which revealed papillary carcinoma. Seven months after that, she developed new auditory and vestibular symptoms attributable to an acoustic neuroma. This case is remarkable because neither Langerhans histiocytosis following radiation of a craniopharyngioma nor this distinctive combination of head and neck tumors in general have ever been previously reported.
- Content Type Journal Article
- Category Case report
- DOI 10.1007/s11102-009-0177-z
- Authors
- Alyson Dobracki, Crozer Chester Medical Center Department of Internal Medicine 3 East, One Medical Center Blvd. Upland PA 19013 USA
- Paul Woolf, Crozer Chester Medical Center Department of Internal Medicine 3 East, One Medical Center Blvd. Upland PA 19013 USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Hematologic neoplasias and acromegaly
Abstract We report a 59-year-old acromegalic woman, who presented with generalized bone pain, weakness, fatigue and foamy urine, who was found to have multiple myeloma (MM); and a 60-year-old acromegalic woman with dizziness, vomiting and abdominal pain, high blood pressure and splenomegaly that was posteriorly diagnosed as having Waldenstrom?s macroglobulinemia (WM). Acromegaly is an uncommon disease and epidemiological studies have provided increasingly debated evidence that elevated IGF-I levels might enhance the neoplastic risk, and that cancers constitute the third leading cause of mortality in acromegaly. It is known that GH and IGF-I can activate B cell lymphocytes, and that IGF-I receptor is universally expressed in MM cells. Although the complication of acromegaly with WM or MM in patients has rarely been reported until now, we described two case reports of acromegalic patients with those hematological neoplasias, which allow a discussion about this controversial issue.
- Content Type Journal Article
- Category Case Report
- DOI 10.1007/s11102-009-0176-0
- Authors
- Flavia Regina P. Barbosa, Hospital Universitário Clementino Fraga Filho/Universidade Federal do Rio de Janeiro Division of Endocrinology, Department of Internal Medicine Rua Nascimento Silva, 555/101 Ipanema, Rio de Janeiro 22421-020 Brazil
- Leonardo Vieira Neto, Hospital Universitário Clementino Fraga Filho/Universidade Federal do Rio de Janeiro Division of Endocrinology, Department of Internal Medicine Rua Nascimento Silva, 555/101 Ipanema, Rio de Janeiro 22421-020 Brazil
- Giovanna Aparecida B. Lima, Hospital Universitário Clementino Fraga Filho/Universidade Federal do Rio de Janeiro Division of Endocrinology, Department of Internal Medicine Rua Nascimento Silva, 555/101 Ipanema, Rio de Janeiro 22421-020 Brazil
- Luiz Eduardo Wildemberg, Hospital Universitário Clementino Fraga Filho/Universidade Federal do Rio de Janeiro Division of Endocrinology, Department of Internal Medicine Rua Nascimento Silva, 555/101 Ipanema, Rio de Janeiro 22421-020 Brazil
- Rodrigo Portugal, Hospital Universitário Clementino Fraga Filho/Universidade Federal do Rio de Janeiro Division of Hematology, Department of Internal Medicine Rio de Janeiro Brazil
- Monica R. Gadelha, Hospital Universitário Clementino Fraga Filho/Universidade Federal do Rio de Janeiro Division of Endocrinology, Department of Internal Medicine Rua Nascimento Silva, 555/101 Ipanema, Rio de Janeiro 22421-020 Brazil
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Acromegaly: correlation between expression of somatostatin receptor subtypes and response to octreotide-lar treatment
Abstract About one-third of acromegalics are resistant to the clinically available somatostatin analogs (SA). The resistance is related to density reduction or different expression of somatostatin receptor subtypes (SSTR). This study analyzes SSTR?s expression in somatotrophinomas, comparing to SA response, hormonal levels, and tumor volume. We analyzed 39 somatotrophinomas; 49% were treated with SA. The most expressed SSTR was SSTR5, SSTR3, SSTR2, SSTR1, and SSTR4, respectively. SSTR1 and SSTR2 had higher expression in patients that had normalized GH and IGF-I. SSTR3 was more expressed in patients with tumor reduction. There was a positive correlation between the percentage of tumor reduction and SSTR1, SSTR2 and SSTR3 expression. Also, a positive correlation between SSTR2 mRNA expression and the immunohistochemical reactivity of SSTR2 was found. Our study confirmed the association between the SA response to GH and IGF-I and the SSTR2. Additionally, this finding was also demonstrated in relation to SSTR1.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0175-1
- Authors
- Ana Paula M. Casarini, University of Sao Paulo Medical School Neuroendocrine Unit, Division of Endocrinology and Metabolism Av. 9 de Julho 3858 01406-100 Sao Paulo SP Brazil
- Raquel S. Jallad, University of Sao Paulo Medical School Neuroendocrine Unit, Division of Endocrinology and Metabolism Av. 9 de Julho 3858 01406-100 Sao Paulo SP Brazil
- Emília M. Pinto, University of Sao Paulo Medical School Endocrine and Development Unit Laboratory of Hormone and Molecular Genetics (LIM42) Sao Paulo SP Brazil
- Iberê C. Soares, University of Sao Paulo Medical School Department of Pathology, Hospital of Clinicas Sao Paulo SP Brazil
- Suely Nonogaki, University of Sao Paulo Medical School Department of Pathology, Hospital of Clinicas Sao Paulo SP Brazil
- Daniel Giannella-Neto, University of Sao Paulo Medical School Laboratory Cellular and Molecular Endocrinology (LIM25) Sao Paulo SP Brazil
- Nina R. Musolino, University of Sao Paulo Medical School Division of Neurosurgery Sao Paulo SP Brazil
- Venâncio A. F. Alves, University of Sao Paulo Medical School Department of Pathology, Hospital of Clinicas Sao Paulo SP Brazil
- Marcello D. Bronstein, University of Sao Paulo Medical School Neuroendocrine Unit, Division of Endocrinology and Metabolism Av. 9 de Julho 3858 01406-100 Sao Paulo SP Brazil
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Differential diagnosis of ACTH-dependent hypercortisolism: imaging versus laboratory
Abstract Differential diagnosis of ACTH-dependent Cushing?s syndrome often presents major difficulties. Diagnostic troubles are increased by suboptimal specificity of endocrine tests, the rarity of ectopic ACTH secretion and the frequent incidental discovery of pituitary adenomas. A 43-year-old female reported with mild signs and symptoms of hypercortisolism, and initial hormonal tests and results of pituitary imaging (7-mm adenoma) were suggestive for Cushing?s disease. However, inadequate response to corticotrophin-releasing hormone and failure to suppress after 8 mg dexamethasone pointed towards an ectopic source. Total body CT scan visualized only a small, non-specific nodule in the right posterior costophrenic excavation. Inferior petrosal sinus sampling revealed an absent center:periphery ACTH gradient but octreoscan and 18F-FDG-PET-CT failed to detect abnormal tracer accumulation. We weighed results of the laboratory with those of imaging and decided to remove the lung nodule. Pathology identified a typical, ACTH-staining carcinoid and the diagnosis was confirmed by postsurgical hypoadrenalism. In conclusion, imaging may prove unsatisfactory or even misleading for the etiologial diagnosis of ACTH-dependent Cushing?s syndrome and should therefore be interpreted only in context with results of hormonal dynamic testing.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0174-2
- Authors
- Massimiliano Andrioli, University of Milan Chair of Endocrinology, Ospedale San Luca IRCCS, Istituto Auxologico Italiano Via Spagnoletto 3 20149 Milan Italy
- Francesca Pecori Giraldi, University of Milan Chair of Endocrinology, Ospedale San Luca IRCCS, Istituto Auxologico Italiano Via Spagnoletto 3 20149 Milan Italy
- Martina De Martin, University of Milan Chair of Endocrinology, Ospedale San Luca IRCCS, Istituto Auxologico Italiano Via Spagnoletto 3 20149 Milan Italy
- Agnese Cattaneo, University of Milan Chair of Endocrinology, Ospedale San Luca IRCCS, Istituto Auxologico Italiano Via Spagnoletto 3 20149 Milan Italy
- Chiara Carzaniga, University of Milan Chair of Endocrinology, Ospedale San Luca IRCCS, Istituto Auxologico Italiano Via Spagnoletto 3 20149 Milan Italy
- Francesco Cavagnini, University of Milan Chair of Endocrinology, Ospedale San Luca IRCCS, Istituto Auxologico Italiano Via Spagnoletto 3 20149 Milan Italy
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Efficacy of long-term lanreotide treatment in patients with acromegaly
Abstract We investigated the effectiveness of lanreotide for the treatment of active acromegaly in a retrospectively multicenter case series including 53 patients (24 male, 29 female; mean age at diagnosis, 49.5 ± 13.9 years) with acromegaly treated with lanreotide in nine different centers. Mean tumor diameter was 20 ± 13 mm; mean basal levels of growth hormone (GH) and insulin-like growth factor I (IGF-I) were 21.3 ± 26.3 and 579 ± 177 ?g/l, respectively. The primary mode of treatment was surgery in 70% of patients. Twenty-nine patients received only lanreotide (Prolonged Release, Autogel), whereas 24 subjects were also treated with octreotide at another treatment stage. Primary therapy with lanreotide was administered in five patients. Maximal monthly dose of lanreotide Autogel (n = 44) was 60 mg in 45%, 90 mg in 26%, 120 mg in 21% and 180 mg in 8%. During 36 months of lanreotide treatment, mean IGF-I levels decreased from 443 ± 238 to 276 ± 147 ?g/l (P < 0.001), and mean GH levels, from 5.2 ± 6.4 to 3.2 ± 3.0 ?g/l (P < 0.001). IGF-I levels normalized in 51% of patients and decreased by >50% towards normal in 32%; the normalization rate was higher in women (65%) than men (33%, P = 0.04). Safe random GH levels (?2 ?g/l) were achieved in 49% of patients. Both IGF-I normalization and safe GH levels were reached in 32% of the cohort. Lanreotide is an effective treatment for active acromegaly. Female sex was associated with higher rates of IGF-I normalization.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0172-4
- Authors
- Yoel Toledano, Hillel Yaffe Medical Center Unit of Endocrinology & Diabetes Hadera Israel
- Liat Rot, Rabin Medical Center Institute of Endocrinology Beilinson Campus 49100 Petah Tiqva Israel
- Yona Greenman, Sourasky Medical Center Institute of Endocrinology, Metabolism and Hypertension Tel Aviv Israel
- Sophia Orlovsky, Maccabi Health Services Haifa Israel
- Yulia Pauker, Clalit Health Services Haifa Israel
- David Olchovsky, Sheba Medical Center Department of Medicine A Tel Hashomer Israel
- Achia Eliash, Maccabi Health Services Bnei-Brak Israel
- Orit Bardicef, Clalit Health Services Haifa Israel
- Ofa Makhoul, Clalit Health Services Jerusalem Israel
- Gloria Tsvetov, Rabin Medical Center Institute of Endocrinology Beilinson Campus 49100 Petah Tiqva Israel
- Michal Gershinsky, Clalit Health Services Haifa Israel
- Odile Cohen-Ouaqnine, Clalit Health Services Jerusalem Israel
- Rosane Ness-Abramof, Sapir Medical Center Endocrine Unit Kfar Saba Israel
- Zaina Adnan, Rambam Medical Center Institute of Endocrinology, Diabetes and Metabolism Haifa Israel
- Jacob Ilany, Chaim Sheba Medical Center Institute of Endocrinology Tel Hashomer Israel
- Hadassah Guttmann, Maccabi Health Services Haifa Israel
- Mazal Sapir, Clalit Health Services Tel Aviv Israel
- Carlos Benbassat, Rabin Medical Center Institute of Endocrinology Beilinson Campus 49100 Petah Tiqva Israel
- Ilan Shimon, Rabin Medical Center Institute of Endocrinology Beilinson Campus 49100 Petah Tiqva Israel
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 4 / December, 2009
- Self-limited acute hepatotoxicity caused by pegvisomant
Abstract We present a case of acute severe hepatitis in a patient with acromegaly receiving combination therapy with somatostatin analogs and pegvisomant. Hepatitis resolved completely 18 weeks after diagnosis of hypertransaminasemia without discontinuation of therapy and with a close clinical and biochemical follow-up. In this case, despite the severity of the hepatitis, therapy could be continued as hypertransaminasemia was gradually decreasing after the maximum peak. We also review the literature on toxic hepatitis associated to pegvisomant therapy analyzing the etiology, clinical predisposing factors and natural evolution.
- Content Type Journal Article
- Category Case Report
- DOI 10.1007/s11102-009-0173-3
- Authors
- A. Soto Moreno, Virgen del Rocio University Hospital Department of Endocrinology and Nutrition, Instituto de Biomedicina de Sevilla (IBIS) Manuel Siurot S/N Avenue 41013 Seville Spain
- R. Guerrero Vázquez, Virgen del Rocio University Hospital Department of Endocrinology and Nutrition, Instituto de Biomedicina de Sevilla (IBIS) Manuel Siurot S/N Avenue 41013 Seville Spain
- E. Venegas Moreno, Virgen del Rocio University Hospital Department of Endocrinology and Nutrition, Instituto de Biomedicina de Sevilla (IBIS) Manuel Siurot S/N Avenue 41013 Seville Spain
- S. Palma Milla, Virgen del Rocio University Hospital Department of Endocrinology and Nutrition, Instituto de Biomedicina de Sevilla (IBIS) Manuel Siurot S/N Avenue 41013 Seville Spain
- J. P. Castaño, University of Córdoba Department of Cell Biology, Physiology and Immunology Córdoba Spain
- A. Leal Cerro, Virgen del Rocio University Hospital Department of Endocrinology and Nutrition, Instituto de Biomedicina de Sevilla (IBIS) Manuel Siurot S/N Avenue 41013 Seville Spain
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Simultaneous above and below approach to giant pituitary adenomas: surgical strategies and long-term follow-up
Abstract Introduction Giant pituitary adenomas of excessive size, fibrous consistency or unfavorable geometric configuration may be unresectable through conventional operative approaches. We present our select case series for operative resection and long-term follow-up for these unusual tumors, employing both a staged procedure and a combined transsphenoidal-transcranial above and below approach. Method A retrospective chart review was performed on patients operated via the staged, and combined approaches by the senior author (J.N·B.). Preoperative characteristics and postoperative outcomes were reviewed. A detailed description of the operative technique and perioperative management is provided. Results Between 1993 and 1996, two patients harboring giant pituitary adenomas underwent an intentionally staged resection, and between 1997 and 2006, nine patients harboring giant pituitary adenomas underwent surgery via a single-stage above and below approach. Nine patients (82%) presented with non-secreting adenomas and two patients (18%) presented with prolactinomas refractory to medical management. Gross total resection was achieved in six patients (55%), near total resection in 1 (9%), and subtotal removal in 4 (36%). Seven patients (64%) experienced visual improvement postoperatively and no major complications occurred. Long-term follow-up averaged 51.6 months. Panhypopituitarism was observed in four patients, partial hypopituitarism in four, persistent DI in two, and persistent SIADH in one. Conclusions The addition of a transcranial component to the transsphenoidal approach offers additional visualization of critical neurovascular structures during giant pituitary adenoma resection. Complications rates are similar to other series in which complex pituitary adenomas are resected by other means. The above and below approach is both safe and effective and the immediate and long-term advantages of a single-stage approach justify its utility in this select group of patients.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0171-5
- Authors
- Anthony L. D?Ambrosio, Columbia University Medical Center Department of Neurological Surgery, New York Presbyterian Hospital New York NY USA
- Omar N. Syed, Columbia University Medical Center Department of Neurological Surgery, New York Presbyterian Hospital New York NY USA
- Bartosz T. Grobelny, Columbia University Medical Center Department of Neurological Surgery, New York Presbyterian Hospital New York NY USA
- Pamela U. Freda, Columbia University Medical Center Department of Medicine ? Endocrinology, New York Presbyterian Hospital New York NY USA
- Sharon Wardlaw, Columbia University Medical Center Department of Medicine ? Endocrinology, New York Presbyterian Hospital New York NY USA
- Jeffrey N. Bruce, Columbia University Medical Center Department of Neurological Surgery, New York Presbyterian Hospital New York NY USA
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Journal Volume Volume 12
- Journal Issue Volume 12, Number 3 / September, 2009
- Recurrent spindle cell oncocytoma of the pituitary, a case report and review of literature
Abstract Spindle cell oncocytoma (SCO) is a rare non-functioning tumour of the pituitary which has just been formally recognized as a distinct entity by the 2007 WHO classification of brain tumours. We report a case of SCO who presented with symptoms of visual blurring, weight loss, intermittent vomiting and excessive tiredness of several months duration. Investigations revealed a bitemporal visual field defect, a panhypopituitary hormonal profile and a large pituitary tumour with suprasellar extension. He underwent a successful trans-sphenoidal resection of the pituitary tumour but it subsequently recurred twice at 9 months interval which required further two debulking procedures. A diagnosis of SCO was made based on its unique histologic and staining properties. To date there are only ten reported cases of SCO in total with only two of these cases being recurrent. Our case displayed the most aggressive clinical course despite having a low Ki-67 index contrary to the previously reported cases of recurrent SCO.
- Content Type Journal Article
- DOI 10.1007/s11102-009-0170-6
- Authors
- Yared N. Demssie, Lancashire Teaching Hospitals NHS Foundation Trust Department of Endocrinology Sharoe Green Lane, Fullwood Preston PR2 9HT UK
- Jacob Joseph, Lancashire Teaching Hospitals NHS Foundation Trust Department of Neuropathology Sharoe Green Lane, Fullwood Preston PR2 9HT UK
- Timothy Dawson, Lancashire Teaching Hospitals NHS Foundation Trust Department of Neuropathology Sharoe Green Lane, Fullwood Preston PR2 9HT UK
- Gareth Roberts, Lancashire Teaching Hospitals NHS Foundation Trust Department of Neurosurgery Sharoe Green Lane, Fullwood Preston PR2 9HT UK
- John de Carpentier, Lancashire Teaching Hospitals NHS Foundation Trust Department of Neurosurgery Sharoe Green Lane, Fullwood Preston PR2 9HT UK
- Simon Howell, Lancashire Teaching Hospitals NHS Foundation Trust Department of Endocrinology Sharoe Green Lane, Fullwood Preston PR2 9HT UK
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X
- Secondary resistance to cabergoline therapy in a macroprolactinoma: a case report and literature review
Abstract Primary resistance to dopamine agonists occurs in 10?15% of prolactinomas but secondary resistance following initial biochemical and anti-proliferative response is very rare and has only been hitherto described in four previous cases, two with bromocriptine and two with cabergoline. We describe a case of a 57-year-old woman who presented with a large macroprolactinoma with suprasellar extension. She was initially treated with bromocriptine therapy with a resolution of symptoms, marked reduction in prolactin concentration and complete tumour shrinkage; a response which was subsequently maintained on cabergoline. After 8 years of dopamine agonist therapy, her prolactin concentration began to rise and there was symptomatic recurrence of her tumour despite escalating doses of cabergoline up to 6 mg weekly. Non-compliance was outruled by observed inpatient drug administration. The patient underwent surgical debulking followed by radiotherapy with good response. This case adds to the previous two cases of secondary resistance to cabergoline therapy in prolactinomas a marked initial response. While the mechanism of secondary resistance remains unknown and not possible to predict, close observation of prolactinoma patients on treatment is necessary.
- Content Type Journal Article
- Category Case Report
- DOI 10.1007/s11102-009-0168-0
- Authors
- L. A. Behan, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- M. S. Draman, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- C. Moran, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- T. King, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- R. K. Crowley, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- E. P. O?Sullivan, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- D. Smith, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- C. J. Thompson, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- A. Agha, Beaumont Hospital and RCSI Medical School Department of Academic Endocrinology and Diabetes Dublin 9 Ireland
- Journal Pituitary
- Online ISSN 1573-7403
- Print ISSN 1386-341X