Cuttler L, Jackson J A, Saeed uz-Zafar M, Levitsky L L, Mellinger R C, Frohman L A
Department of Pediatrics, University of Chicago, Illinois 60637.
J Clin Endocrinol Metab. 1989 Jun;68(6):1148-54. doi: 10.1210/jcem-68-6-1148.
Acromegaly and hyperprolactinemia have been reported in association with the McCune-Albright syndrome, but the pathophysiology of the GH and PRL hypersecretion that occurs in patients with this disorder has not been defined. We studied GH and PRL secretory dynamics in three patients with McCune-Albright syndrome and hypersecretion of these hormones. Each patient had excessive linear growth, glucose-non-suppressible plasma GH concentration, and GH responsiveness to TRH and GHRH. In response to exogenous GHRH, plasma GH concentrations rose approximately 2-fold in all three patients. Plasma GHRH levels were 20-40 ng/L (normal, less than 30). Study of the spontaneous GH secretory pattern in two patients indicated nocturnal augmentation of GH release. Bromocriptine therapy failed to reduce plasma GH in all patients; in one patient treatment with octreotide, a long-acting somatostatin analog, partially suppressed plasma GH and insulin-like growth factor I levels. These results suggest that hypersecretion of GH in the McCune-Albright syndrome is not due to ectopic GHRH production or autonomous somatotroph function. The results are similar to those described in classic acromegaly due to GH-secreting pituitary tumors. However, the lack of radiographic pituitary enlargement, the variable pituitary pathology reported in similar patients, and frequent concordance of GH and PRL excess suggest that the pathogenesis of this disorder may differ fundamentally from other forms of acromegaly or gigantism. The pathophysiology may reflect abnormal hypothalamic regulation and/or an embryological defect in pituitary cellular differentiation and function.
已有报告称肢端肥大症和高泌乳素血症与McCune-Albright综合征相关,但该疾病患者出现的生长激素(GH)和泌乳素(PRL)分泌过多的病理生理学机制尚未明确。我们研究了3例患有McCune-Albright综合征且这些激素分泌过多的患者的GH和PRL分泌动力学。每位患者均有过度的线性生长、葡萄糖不能抑制的血浆GH浓度,以及GH对促甲状腺激素释放激素(TRH)和生长激素释放激素(GHRH)的反应性。对外源性GHRH的反应中,所有3例患者的血浆GH浓度均升高约2倍。血浆GHRH水平为20 - 40 ng/L(正常范围,低于30)。对2例患者的自发性GH分泌模式研究表明存在夜间GH释放增加。溴隐亭治疗未能降低所有患者的血浆GH;在1例患者中,使用长效生长抑素类似物奥曲肽治疗可部分抑制血浆GH和胰岛素样生长因子I水平。这些结果表明,McCune-Albright综合征中GH分泌过多并非由于异位GHRH产生或生长激素细胞自主功能所致。这些结果与因分泌GH的垂体肿瘤导致的经典肢端肥大症中所描述的结果相似。然而,缺乏影像学上垂体增大、类似患者中报告的垂体病理变化多样,以及GH和PRL过多常同时出现,提示该疾病的发病机制可能与其他形式的肢端肥大症或巨人症有根本不同。其病理生理学机制可能反映下丘脑调节异常和/或垂体细胞分化及功能的胚胎学缺陷。