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ABSTRACT Objective: Data regarding rare FPAs from India, a resource limited setting, are limited. We describe a case series of rare FPAs from a single center in western India. Materials and methods: This was a retrospective case record review of patients diagnosed between January 2010 and July 2022. The diagnosis was based on biochemical (inappropriately elevated serum FSH/LH) and pathologic (positive immunostaining for FSH/LH) features in patients with FGA, and elevated serum thyroid hormones and normal/elevated TSH in patients with TSHomas. Results: We identified 11 patients with a total of six FGAs (median age 43.5 years, five men, one FGA cosecreting TSH, median largest dimension 40 mm, range 33-60 mm) and six TSHomas (median age 34.5 years, four women, two TSHomas cosecreting GH, median largest dimension 42.5 mm, range 13-60 mm). Symptoms of sellar mass effects led to pituitary imaging in most patients with FGA. Patients with TSHomas had symptoms of excess hormone secretion (GH/TSH) or sellar mass effects. The TSHomas that cosecreted GH/FSH were larger than those secreting only TSH. Transsphenoidal resection was the most common first-line therapy but significant residual disease was frequent (3 out of 6 FGAs and 4 out of 5 TSHomas). Conclusion: This is the first and second case series of FGAs and TSHomas, respectively, from India. In this study, TSHomas presented at younger age, were larger and had low surgical cure rates.
RESUMO
BACKGROUND/AIMS: Thyrotropin (TSH)-secreting pituitary adenoma (TSHoma) is rare and represents 1~2% of all pituitary adenomas. TSHoma should be distinguished from the thyroid hormone resistance syndrome. Patients with TSHoma may be misdiagnosed with primary hyperthyroidism and often receive inappropriate thyroid gland treatment. METHODS: We assessed the clinical characteristics of patients with TSHoma who presented to Severance Hospital at the Yonsei University College of Medicine, Seoul, Korea between 2005 and 2009. RESULTS: Of 484 patients who underwent pituitary tumor resection, eight (1.65%; five women and three men) were found to have TSHoma. The mean age was 40.6+/-8.9 years at diagnosis (range, 28~55 years). The median duration from onset of symptoms to diagnosis was 17 months (range, 4~60 months). Four patients had overt symptoms of hyperthyroidism and two had visual field defect. Six patients had elevated free thyroxine (FT4) levels with elevated or inappropriately normal TSH levels, and two patients had symptoms associated with Hashimoto's thyroiditis. The serum levels of free alpha-subunit measured in two patients were elevated. Six of the tumors were macroadenomas (>10 mm) and two were microadenomas. Complete tumor removal was achieved in all patients. Five patients had preoperative anterior pituitary dysfunction; three patients recovered from this after surgery. Three patients were lost to follow up and five patients showed no evidence of recurrence or hyperthyroidism in the follow-up period (mean, 30.8 months, range, 3~57). CONCLUSIONS: Early diagnosis and complete removal of the tumor mass may improve the neurological and endocrine deficits.