Manne Sriharibabu, Veeraabhinav C H, Jetti Mounica, Himabindu Yalamanchali, Donthu Kiranmai, Badireddy Mutyalarayudu
Department of Medicine, G.S.L. Medical College and General Hospital, Rajahmundry, Andhra Pradesh, India.
Department of Obstetrics and Gynecology, G.S.L. Medical College and General Hospital, Rajahmundry, Andhra Pradesh, India.
J Hum Reprod Sci. 2016 Oct-Dec;9(4):263-266. doi: 10.4103/0974-1208.197694.
46,XX gonadal dysgenesis is a rare genetically heterogeneous disorder characterized by underdeveloped ovaries with consequent, impuberism, primary amenorrhea, and hypergonadotropic hypogonadism. Mullerian agenesis or Mayer-Rokitansky-Kuster-Hauser (MRKH) syndrome is characterized by congenital aplasia of the uterus and the upper part (2/3) of the vagina in a woman with normal development of secondary sexual characteristics and a normal 46,XX karyotype. The phenotypic manifestations of MRKH syndrome may sometimes overlap with various other syndromes and require accurate delineation. The coexistence of both these disorders is extremely rare. Here, we report a case of 46,XX gonadal dysgenesis and MRKH syndrome with anatomically dispersed congenital anomalies unique among reported cases.
46,XX性腺发育不全是一种罕见的遗传性异质性疾病,其特征为卵巢发育不全,继而出现青春期发育延迟、原发性闭经和高促性腺激素性性腺功能减退。苗勒管发育不全或 Mayer-Rokitansky-Kuster-Hauser(MRKH)综合征的特征是子宫和阴道上2/3先天性发育不全,而女性的第二性征发育正常且核型为正常的46,XX。MRKH综合征的表型表现有时可能与其他各种综合征重叠,需要准确鉴别。这两种疾病同时存在极为罕见。在此,我们报告一例46,XX性腺发育不全合并MRKH综合征的病例,其先天性异常在解剖学上呈分散状态,在已报道的病例中独一无二。