Pasquini Neto Ricardo, Carnielli Tebet Maria Letícia, Ivanski Dória de Vasconcelos Ohana, Faucz Munhoz da Cunha Mariana, Figueroa Magalhães Maria Cristina
School of Medicine, Pontifícia Universidade Católica do Paraná, Curitiba, BRA.
School of Medicine, Faculdade Evangélica Mackenzie do Paraná, Curitiba, BRA.
Cureus. 2023 Apr 26;15(4):e38149. doi: 10.7759/cureus.38149. eCollection 2023 Apr.
The 46,XY disorder of sexual development (DSD) is a rare congenital condition characterized by a 46,XY karyotype associated with complete or disturbed female gonadal development and a non-virilized phenotype. The presence of Y chromosome material in these patients' karyotypes increases the risk of germ cell tumor development. The present study reports a unique case of a 16-year-old phenotypically female patient presenting with primary amenorrhea, who was later diagnosed with 46,XY DSD. After bilateral salpingo-oophorectomy, the patient was diagnosed with stage IIIC dysgerminoma. The patient received four cycles of chemotherapy and showed a good response. The patient is currently alive and well, with no evidence of disease after the residual lymph node resection.
46,XY性发育障碍(DSD)是一种罕见的先天性疾病,其特征为46,XY核型,伴有完全或紊乱的女性性腺发育以及非男性化表型。这些患者核型中Y染色体物质的存在增加了生殖细胞肿瘤发生的风险。本研究报告了一例独特病例,一名16岁表型为女性的患者因原发性闭经就诊,后来被诊断为46,XY DSD。双侧输卵管卵巢切除术后,患者被诊断为IIIC期无性细胞瘤。患者接受了四个周期的化疗,反应良好。患者目前健在,残余淋巴结切除术后无疾病证据。