Lee Hong Jun, Yeom Jung-Sook, Park Ji Sook, Park Eun Sil, Seo Ji-Hyun, Lim Jae Young, Park Chan-Hoo, Woo Hyang-Ok, Youn Hee-Shang
Department of Pediatrics, Gyeongsang National University School of Medicine, Jinju, Korea.
Ann Pediatr Endocrinol Metab. 2014 Jun;19(2):100-3. doi: 10.6065/apem.2014.19.2.100. Epub 2014 Jun 30.
There is a wide variety of genital abnormalities observed in patients with Denys-Drash syndrome (DDS). WT1 is thought to influence the genes related to genital development and mutations in this gene have been associated with DDS. DDS should be considered in the differential diagnosis of newborns with genital anomalies. In contrast to other conditions with 46,XY disorders of sex development, individuals with DDS often have duplicated genital organs (a double vagina, cervix or uterus). A double uterus has not yet been reported with 1390G>A (Arg464 Asn) mutation. However, duplicated genitals have been reported with other genetic mutations in patients with DDS. The duplicated genitals in DDS may be associated with low anti-Mullerian hormone (AMH) secretion. Measurement of the AMH levels may add to our understanding of variations in genital development and their abnormalities in disorders such as DDS. In conclusion, this is first case of low level of AMH and double uterus in 1390G>A (Arg464 Asn) mutations of DDS male.
在患有迪尼-德拉斯综合征(DDS)的患者中观察到多种生殖器异常。WT1被认为会影响与生殖器发育相关的基因,该基因的突变与DDS有关。对于患有生殖器异常的新生儿进行鉴别诊断时应考虑DDS。与其他46,XY性发育障碍的情况不同,患有DDS的个体通常有重复的生殖器官(双阴道、宫颈或子宫)。尚未有关于1390G>A(Arg464 Asn)突变导致双子宫的报道。然而,已有报道称患有DDS的患者因其他基因突变出现重复生殖器。DDS中重复的生殖器可能与抗苗勒管激素(AMH)分泌减少有关。测量AMH水平可能有助于我们了解生殖器发育的变异及其在DDS等疾病中的异常情况。总之,这是首例DDS男性1390G>A(Arg464 Asn)突变导致AMH水平降低和双子宫的病例。