Ullah Mohammad T, Lopes M Beatriz S, Jane John A, Hong Gregory K, Love Kaitlin M
Department of Endocrinology and Metabolism, University of Virginia Health System, Charlottesville, Virginia.
Department of Pathology and Neurological Surgery, University of Virginia Health System, Charlottesville, Virginia.
AACE Clin Case Rep. 2022 Nov 5;9(1):5-9. doi: 10.1016/j.aace.2022.11.001. eCollection 2023 Jan-Feb.
BACKGROUND/OBJECTIVE: Functional gonadotroph adenomas (FGAs) are adenomas producing active gonadotropins, follicle-stimulating hormone or luteinizing hormone. Double pituitary adenomas are 2 distinct adenomas occurring in an individual. This report aimed to present an extremely rare case of an FGA, itself an uncommon disorder, co-occurring with a lactotroph adenoma.
A 33-year-old woman presented with menorrhagia and was found to have ovarian enlargement, large uterine leiomyomas, and bitemporal hemianopsia. Initially, the levels of follicle-stimulating hormone, luteinizing hormone, estradiol, and prolactin were 73.3 mIU/mL (midcycle peak, 2.3-20.9 mIU/L), 3.74 mIU/L (midcycle peak, 8.7-76.3 mIU/L), 1071 pg/mL (midcycle peak 38-649 pg/mL), and 402 ng/mL (2-30 ng/mL), respectively. Pituitary magnetic resonance imaging demonstrated a single sellar mass (2.0 × 2.2 cm). Two months of cabergoline did not reverse visual field deficits; therefore, transsphenoidal resection was performed. Diagnosis of 2 separate adenomas, a gonadotroph and lactotroph adenoma, was confirmed on pathology.
In this case, gonadotropins did not suppress in response to hyperprolactinemia. Although marked hyperprolactinemia has been associated with functional and clinically silent gonadotroph adenomas in prior cases, this is the first case to confirm an FGA co-occurring with a lactotroph adenoma.
In patients who present with elevated gonadotropin levels despite hyperprolactinemia, we suggest considering FGA. Further research is needed to clarify whether there is underdiagnosis of lactotroph adenomas co-occurring with gonadotroph adenomas.
背景/目的:功能性促性腺激素腺瘤(FGAs)是分泌活性促性腺激素、促卵泡生成素或促黄体生成素的腺瘤。双垂体腺瘤是指个体中出现的两种不同腺瘤。本报告旨在呈现一例极为罕见的FGAs病例,FGAs本身就是一种罕见疾病,且该病例同时合并催乳素瘤。
一名33岁女性因月经过多就诊,检查发现卵巢增大、子宫有大的平滑肌瘤以及双颞侧偏盲。最初,促卵泡生成素、促黄体生成素、雌二醇和催乳素水平分别为73.3 mIU/mL(月经周期中期峰值为2.3 - 20.9 mIU/L)、3.74 mIU/L(月经周期中期峰值为8.7 - 76.3 mIU/L)、1071 pg/mL(月经周期中期峰值为38 - 649 pg/mL)和402 ng/mL(2 - 30 ng/mL)。垂体磁共振成像显示蝶鞍区有一个单发病灶(2.0×2.2 cm)。服用两个月的卡麦角林未能改善视野缺损,因此进行了经蝶窦切除术。病理检查确诊为两种独立的腺瘤,即促性腺激素腺瘤和催乳素瘤。
在该病例中,促性腺激素并未因高催乳素血症而受到抑制。尽管在之前的病例中,明显的高催乳素血症与功能性及临床无症状的促性腺激素腺瘤有关,但这是首例证实FGAs与催乳素瘤同时存在的病例。
对于尽管存在高催乳素血症但促性腺激素水平升高的患者,我们建议考虑FGAs。需要进一步研究以明确促性腺激素腺瘤合并催乳素瘤是否存在诊断不足的情况。