2nd Department of Internal Medicine, Hamamatsu University School of Medicine, Japan.
Department of Internal Medicine, Futagawa Hospital, Japan.
Intern Med. 2024 Nov 15;63(22):3071-3076. doi: 10.2169/internalmedicine.3084-23. Epub 2024 Apr 9.
17α-hydroxylase deficiency is a type of congenital adrenocortical hyperplasia that is typically diagnosed in childhood or adolescence. It manifests as hypertension with gonadal dysfunction as the primary symptom. We herein report 17α-hydroxylase/17,20-lyase deficiency (17OHD) diagnosed at the age of 45 years. The patient presented with hypertension, irregular menstruation, and hyperaldosteronism. The clinical manifestations of 17OHD vary based on the specific variant pattern of CYP17A1. In this case, the variant was c.157_159 TCC del p. Phe53del, which has been frequently reported in Japan. The enzymatic deficiency due to this variant is partial, leading to a delay in making a correct diagnosis.
17α-羟化酶缺陷是一种先天性肾上腺皮质增生症,通常在儿童或青少年时期被诊断出来。它以高血压和性腺功能障碍为主要症状表现。本文报告了一例在 45 岁时被诊断为 17α-羟化酶/17,20-裂合酶缺陷(17OHD)的病例。该患者表现为高血压、月经不规律和醛固酮增多症。17OHD 的临床表现因 CYP17A1 的具体变异模式而异。在这种情况下,变异为 c.157_159TCCdel p. Phe53del,在日本经常被报道。由于这种变异导致的酶缺乏是部分的,导致正确诊断的时间延迟。