Siklar Zeynep, Berberoğlu Merih, Adiyaman Pelin, Salih Mustafa, Tükün Ajlan, Cetinkaya Ergun, Aycan Zehra, Evliyaoğlu Olcay, Ergur Ayça T, Oçal Gönül
Ankara University, School of Medicine, Department of Pediatric Endocrinology, Ankara, Turkey.
Pediatr Endocrinol Rev. 2007 Mar;4(3):210-7.
The most complicated group of sexual differentiation disorders is that of gonadal development. Disorders of gonadal development form a wide clinical, cytogenetic and histopathological spectrum. There are still some unsolved difficulties of diagnosis, development of malignancy and the sex rearing of these patients. We reviewed 23 cases of gonadal developmental disorders among 169 patients with ambiguous genitalia or delayed puberty. Among 169 patients, 87 patients were 46,XY disorders of sex development (DSD), 59 patients were 46,XX DSD without disorders of gonadal development and the remaining 23 patients had disorders of gonadal development. Nine of these 23 patients were diagnosed as 46,XY gonadal dysgenesis, 7 patients had ovotesticular DSD, 5 patients had 45,X/46,XY mixed gonadal dysgenesis. Fourteen patients with disorders of gonadal development had genital ambiguity, 5 patients had a female genital phenotype with a palpable gonad and/or delayed puberty. Four patients had the male genital phenotype. Disorder of gonadal development is a very important clinical problem with different aspects of diagnosis, treatment, rearing sex and prophylaxis. Each patient should be evaluated individually employing a multidiciplinary approach.
性分化障碍中最复杂的一组是性腺发育障碍。性腺发育障碍形成了广泛的临床、细胞遗传学和组织病理学谱。这些患者在诊断、恶性肿瘤发生以及性别抚养方面仍存在一些未解决的难题。我们回顾了169例生殖器模糊或青春期延迟患者中的23例性腺发育障碍病例。在这169例患者中,87例为46,XY性发育障碍(DSD),59例为46,XX DSD且无性腺发育障碍,其余23例有性腺发育障碍。这23例患者中,9例被诊断为46,XY性腺发育不全,7例患有卵睾性DSD,5例患有45,X/46,XY混合性腺发育不全。14例性腺发育障碍患者有生殖器模糊,5例有女性生殖器表型且可触及性腺和/或青春期延迟。4例有男性生殖器表型。性腺发育障碍是一个非常重要的临床问题,涉及诊断、治疗、性别抚养和预防等不同方面。每个患者都应采用多学科方法进行个体化评估。