Follicle-stimulating Hormone-secreting Pituitary Adenoma Accompanied by Painful Thyroiditis

Ichijo, Masashi; Tsuchiya, Kyoichiro; Kasai, Tsuyoshi; Inoshita, Naoko; Yoshimoto, Haruko; Yamada, Shozo; Kitamura, Kenichiro

doi:10.2169/internalmedicine.3667-19

Cited by 3 publications

(2 citation statements)

References 17 publications

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“…In the present case, the results of IHC for pituitary transcription factors and gonadotropins were compatible with FSH-producing PitNETs. Consistent with a previous report by Ichijo et al (12), the tumor exhibited high SSTR2A and low SSTR5 expression. 111 In-pentetreotide is a radiolabeled somatostatin analog used in somatostatin receptor scintigraphy.…”

Section: Discussionsupporting

confidence: 93%

“…Therefore, in patients with FSH-producing pituitary tumors, increased serum estradiol concentrations can suppress the hypothalamus-anterior pituitary gland axis by a negative feedback mechanism, reducing excessive FSH production to normal levels. Moreover, the serum LH concentration could be reduced below the lower limit of the normal range by the negative feedback mechanism or by compression of the normal pituitary gland by the tumor (3,12). Another hypothesis is that alterations in the alpha and beta chains of the heterodimer could potentially lead to increased bioactivity of FSH (13).…”

Section: Discussionmentioning

confidence: 99%

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FSH-producing pituitary neuroendocrine tumor as a cause of ovarian hyperstimulation syndrome

Kitamura,

Nanba,

Doi

et al. 2024

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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Summary Functioning gonadotroph tumors are rare neoplasms that can cause ovarian hyperstimulation syndrome (OHSS) in women of reproductive age. Here, we present a case of a follicle-stimulating hormone (FSH)-producing pituitary neuroendocrine tumor (PitNET) with irregular menstrual cycles and OHSS in a Japanese woman. A 34-year-old woman with bilateral multi-cystic ovarian mass was referred to our hospital for ovarian surgery. The imaging feature of magnetic resonance imaging (MRI) of the ovary and elevated estradiol levels with normal FSH and low luteinizing hormone (LH) levels led us to suspect the presence of a functioning gonadotroph PitNET. MRI revealed a 19-mm pituitary tumor, and increased tracer uptake was observed in the pituitary lesion on 111In-pentetreotide scintigraphy. Transsphenoidal tumor resection resulted in the resolution of the ovarian enlargement, normalization of her menstrual cycles, and spontaneous pregnancy. Immunohistochemistry (IHC) of the resected tumor for pituitary transcription factors, including steroidogenesis factor 1 (SF1) and estrogen receptor alpha, demonstrated positive immunoreactivity, whereas IHC for pituitary-specific positive transcription factor 1 was negative, suggesting that the tumor belonged to the SF1 lineage of PitNETs (gonadotroph tumor). The tumor cells showed positive expression of FSHβ, while LHβ was mostly negative. Consistent with the high pituitary tumor uptake observed on 111In-pentetreotide scintigraphy, the pituitary tumor showed positive expression of somatostatin receptor 2A. Detailed clinical and histological evaluations will provide useful information to understand these rare functioning gonadotroph tumors better. Learning points Functioning gonadotroph tumors are very rare neuroendocrine tumors of pituitary origin. Women of reproductive age presenting with bilateral multi-cystic ovarian enlargement, irregular menstrual cycles, and hyperestrogenemia under unsuppressed follicle-stimulating hormone (FSH) levels should be evaluated for FSH-producing tumor. Raising awareness of OHSS due to functioning gonadotroph tumors is crucial to prevent unnecessary ovarian surgery. Comprehensive histological analysis may provide useful information to better understand the characteristics of functioning gonadotroph tumors.

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Section: Discussionsupporting

confidence: 93%

Section: Discussionmentioning

confidence: 99%

FSH-producing pituitary neuroendocrine tumor as a cause of ovarian hyperstimulation syndrome

Kitamura,

Nanba,

Doi

et al. 2024

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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Functioning gonadotroph adenomas in premenopausal women: clinical and molecular characterization and review of the literature

et al. 2022

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Local symptoms of Hashimoto’s thyroiditis: A systematic review

Yuan

Qi²,

Zhang³

et al. 2023

Front. Endocrinol.

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ObjectiveHashimoto’s thyroiditis (HT) is the most common type of thyroid disease and can cause many different manifestations. The local symptoms of HT are an under-studied area of research. Therefore, the purpose of this study was to investigate the local symptoms of HT and their prevalence.MethodsA systematic review was performed to find articles in PubMed that discuss the local symptoms of HT. Relevant vocabulary terms and key terms included: autoimmune thyroid disease (AITD), hyperthyroidism, hypothyroidism, neck, throat, pharynx, airway, esophagus, breathe, swallow, globus, sleep apnea, symptoms, and quality of life. Two investigators independently screened the eligible studies.ResultsA total of 54 articles fulfilled the inclusion criteria. Of these, 25 were clinical studies, 24 were case reports, and five were reviews. These clinical studies and case reports included a total of 2660 HT patients. There were eight local symptoms related to HT: neck pain (0.02%~16%), voice changes (7%~30%), throat discomfort (20%~43.7%), shortness of breath (28%~50%), dysphagia (29%), goiter-related symptoms (69.44%), sleep apnea, and generally defined compressive symptoms. Due to the use of different outcome measures among all the studies, a meta-analysis of the data could not be performed.ConclusionGoiter symptoms, which are an item on the ThyPRO scales, are the most frequent local symptoms in HT patients, and include neck pain, voice changes, throat discomfort, and dysphagia. These local symptoms should be identified in the clinic and included in the early diagnosis and management of HT, as well as evaluated further to understand their relevance in the pathogenesis of HT.

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Follicle-stimulating Hormone-secreting Pituitary Adenoma Accompanied by Painful Thyroiditis

Cited by 3 publications

References 17 publications

FSH-producing pituitary neuroendocrine tumor as a cause of ovarian hyperstimulation syndrome

FSH-producing pituitary neuroendocrine tumor as a cause of ovarian hyperstimulation syndrome

Functioning gonadotroph adenomas in premenopausal women: clinical and molecular characterization and review of the literature

Local symptoms of Hashimoto’s thyroiditis: A systematic review

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