Adrenal Medullary Hyperplasia: A Systematic Review and Meta-analysis

医学 嗜铬细胞瘤 内科学 儿茶酚胺 内分泌学 背景(考古学) 肾上腺髓质 肾上腺切除术 甲状腺髓样癌 胃肠病学 泌尿科 甲状腺 生物 甲状腺癌 古生物学
作者
Rafal Ganni,David J. Torpy,Henrik Falhammar,R. Louise Rushworth
出处
期刊:The Journal of Clinical Endocrinology and Metabolism [Oxford University Press]
卷期号:108 (9): e885-e892 被引量:1
标识
DOI:10.1210/clinem/dgad121
摘要

Abstract Context Adrenal medullary hyperplasia (AMH) is a rare, incompletely described disorder of the adrenal medulla that is associated with catecholamine excess. Objective To increase knowledge about AMH by reviewing the reported cases of this disorder. Design Systematic review and meta-analysis of the genotype/phenotype relationship in all reported cases of AMH. Setting Literature review and analysis. Patients or Other Participants All cases of AMH published to date. Main Outcome Measure(s) Characteristics of AMH cases and genotype-phenotype relationships. Results A total of 66 patients, median age of 48 years, were identified from 29 reports. More than one-half were male (n = 39, 59%). The majority had unilateral (73%, n = 48) disease; 71% (n = 47) were sporadic and 23% (n = 15) were associated with the MEN2. Most (91%, n = 60) displayed signs and symptoms of excess catecholamine secretion, particularly hypertension. Elevated catecholamine concentrations (86%, n = 57) and adrenal abnormalities on imaging were common (80%, n = 53). More than one-half (58%, n = 38) had concurrent tumors: pheochromocytoma (42%, n = 16/38); medullary thyroid cancer (24%, n = 9/38); and adrenocortical adenoma (29%, n = 11/38). Most (88%, n = 58) underwent adrenalectomy with 45/58 achieving symptom resolution. Adrenalectomy was less common in patients under 40 years and those with bilateral disease (both P < .05). Conclusion AMH may be sporadic or associated with MEN2, most have catecholamine excess and imaging abnormalities. Unilateral involvement is more common. Most reported patients have been treated with adrenalectomy, which is usually curative with regard to catecholamine hypersecretion.

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