Kovacs K, Horvath E, Thorner M O, Rogol A D
Virchows Arch A Pathol Anat Histopathol. 1984;403(1):77-86. doi: 10.1007/BF00689340.
An 11-year-old girl, with the McCune-Albright syndrome, exhibited fibrous dysplasia of several bones, skin pigmentation, precocious puberty, growth hormone hypersecretion, acromegaly and hyper-prolactinemia. Histologic, immunocytologic and ultrastructural investigation of the surgically-removed pituitary showed massive mammosomatotroph hyperplasia. Since no adenoma was found, the abundance of these bihormonal cells, capable of producing both growth hormone and prolactin, was implicated in the causation of growth hormone and prolactin excess. Somatoliberin overproduction and/or somatostatin and dopamine deficiency could not account for the hypophysial abnormality, since changes in secretory rates of these hypothalamic hormones would lead to proliferation of mature somatotrophs and lactotrophs, rather than mammosomatotrophs. In our patient, a congenital hypothalamic malfunction might have been accompanied by hypersecretion of an unidentified releasing factor, resulting in pathologic differentiation of the pituitary and mammosomatotroph hyperplasia. Alternatively, mammosomatotroph hyperplasia may have been due to an inherent genetic or embryonic defect affecting primarily the pituitary. According to this interpretation, the pituitary lesion represented yet another developmental error in the setting of the McCune-Albright syndrome.
一名患有McCune - Albright综合征的11岁女孩,表现出多骨纤维发育不良、皮肤色素沉着、性早熟、生长激素分泌过多、肢端肥大症和高泌乳素血症。对手术切除的垂体进行组织学、免疫细胞学和超微结构研究显示,有大量的泌乳生长激素细胞增生。由于未发现腺瘤,这些能够产生生长激素和泌乳素的双激素细胞数量过多被认为与生长激素和泌乳素分泌过多的病因有关。生长激素释放素分泌过多和/或生长抑素及多巴胺缺乏不能解释垂体异常,因为这些下丘脑激素分泌率的变化会导致成熟的生长激素细胞和催乳素细胞增生,而不是泌乳生长激素细胞增生。在我们的患者中,先天性下丘脑功能障碍可能伴有一种未明释放因子的分泌过多,导致垂体的病理分化和泌乳生长激素细胞增生。或者,泌乳生长激素细胞增生可能是由于主要影响垂体的内在遗传或胚胎缺陷。根据这一解释,垂体病变代表了McCune - Albright综合征背景下的又一种发育异常。