Olmedilla Yoko, Khan Shoaib, Young Victoria, Joseph Robin, Cudlip Simon, Ansgorge Olaf, Grossman Ashley, Pal Aparna
Endocrinology and Nutrition Service, Gregorio Marañón General Universitary Hospital, Madrid, Spain.
Oxford Centre for Endocrinology, Diabetes and Metabolism, Churchill Hospital, Oxford, UK.
Endocrinol Diabetes Metab Case Rep. 2019 Mar 21;2019. doi: 10.1530/EDM-18-0130.
A 21 year-old woman was found to have a pituitary macroadenoma following an episode of haemophilus meningitis. Biochemical TSH and GH excess was noted, although with no clear clinical correlates. She was treated with a somatostatin analogue (SSA), which restored the euthyroid state and controlled GH hypersecretion, but she re-presented with a further episode of cerebrospinal fluid (CSF) leak and recurrent meningitis. Histology following transsphenoidal adenomectomy revealed a Pit-1 lineage plurihormonal adenoma expressing GH, TSH and PRL. Such plurihormonal pituitary tumours are uncommon and even more unusual to present with spontaneous bacterial meningitis. The second episode of CSF leak and meningitis appears to have been due to SSA therapy-induced tumour shrinkage, which is not a well-described phenomenon in the literature for this type of tumour. Learning points: Pit-1 lineage GH/TSH/PRL-expressing plurihormonal pituitary adenomas are uncommon. Moreover, this case is unique as the patient first presented with bacterial meningitis. Inmunohistochemical plurihormonality of pituitary adenomas does not necessarily correlate with biochemical and clinical features of hormonal hypersecretion. Given that plurihormonal Pit-1 lineage adenomas may behave more aggressively than classical pituitary adenomas, accurate pathological characterization of these tumours has an increasing prognostic relevance. Although unusual, a CSF leak and meningitis may be precipitated by SSA therapy of a pituitary macroadenoma via tumour shrinkage.
一名21岁女性在患嗜血杆菌性脑膜炎后被发现患有垂体大腺瘤。虽然没有明确的临床相关表现,但生化检查显示促甲状腺激素(TSH)和生长激素(GH)分泌过多。她接受了生长抑素类似物(SSA)治疗,该治疗恢复了甲状腺功能正常状态并控制了GH分泌过多,但她再次出现脑脊液(CSF)漏和复发性脑膜炎。经蝶窦腺瘤切除术后的组织学检查显示为一种表达GH、TSH和催乳素(PRL)的Pit-1谱系多激素腺瘤。这种多激素垂体肿瘤并不常见,更罕见的是伴有自发性细菌性脑膜炎。第二次脑脊液漏和脑膜炎发作似乎是由于SSA治疗导致肿瘤缩小所致,而这种现象在这类肿瘤的文献中并未得到充分描述。学习要点:表达Pit-1谱系GH/TSH/PRL的多激素垂体腺瘤并不常见。此外,该病例独特之处在于患者最初表现为细菌性脑膜炎。垂体腺瘤的免疫组织化学多激素性不一定与激素分泌过多的生化和临床特征相关。鉴于多激素Pit-1谱系腺瘤可能比经典垂体腺瘤行为更具侵袭性,对这些肿瘤进行准确的病理特征描述对预后的相关性越来越大。虽然不常见,但垂体大腺瘤的SSA治疗通过肿瘤缩小可能引发脑脊液漏和脑膜炎。