Hur Mina, Cho Hyoun Chan, Lee Kyu Man, Park Hyokhan, Lee So Yeon, Kim Kwang Nam, Kim Sun Hee, Ki Chang Suk
Department of Laboratory Medicine, Konkuk University School of Medicine, Seoul, Korea.
Cleft Palate Craniofac J. 2009 Sep;46(5):555-7. doi: 10.1597/07-149.1. Epub 2009 Feb 20.
Variant Klinefelter syndrome with 48,XXXY/46,XY mosaicism has been rarely reported, and its phenotypic features, compared with those of the classic type, have not been well delineated. We describe a newborn baby with phenotypic abnormalities, including cleft palate and low-set ears. The cytogenetic analysis of peripheral blood lymphocytes showed a karyotype of 48,XXXY[36]/46,XY[4]. To the best of our knowledge, this is the first case in which 48,XXXY/46,XY mosaicism was related to the congenital anomaly of cleft palate. This case underscores that cytogenetic analysis should be a mandatory workup for the patient with cleft palate and that cleft palate may be a rare clinical presentation of the variant Klinefelter syndrome.
具有48,XXXY/46,XY嵌合体的变异型克兰费尔特综合征鲜有报道,与经典型相比,其表型特征尚未得到很好的描述。我们描述了一名患有包括腭裂和低位耳在内的表型异常的新生儿。外周血淋巴细胞的细胞遗传学分析显示核型为48,XXXY[36]/46,XY[4]。据我们所知,这是第一例48,XXXY/46,XY嵌合体与先天性腭裂相关的病例。该病例强调细胞遗传学分析对于腭裂患者应是一项强制性检查,并且腭裂可能是变异型克兰费尔特综合征的一种罕见临床表现。