Coppin B, Moore I, Hatchwell E
Wessex Clinical Genetics Service, The Princess Anne Hospital, Southampton, UK.
Clin Genet. 1997 Jun;51(6):375-8. doi: 10.1111/j.1399-0004.1997.tb02494.x.
We present the case of a male infant, born prematurely (at 33 weeks gestation) with macrosomia, disproportionate macrocephaly, facial dysmorphism, short penis and a small umbilical defect. He had a large ASD and was ventilated from birth for respiratory distress syndrome. He died at 12 hours of age despite neonatal ITU care. Post-mortem examination showed highly lobulated kidneys with nodules of blastema and foci of hamartomatous change in the medulla. Prominence of pancreatic islet cells and expansion of hepatic portal tracts were also noted. His mother has minor cervical spine abnormalities. We discuss the differential diagnosis and the difficulty in confidently assigning a diagnosis to this patient, as considerable overlap is becoming evident between Simpson-Golabi-Behmel syndrome and Perlman syndrome.
我们报告一例男性婴儿病例,该婴儿早产(孕33周),患有巨大儿、不成比例的巨头畸形、面部畸形、阴茎短小和脐部小缺损。他有一个大的房间隔缺损,出生后因呼吸窘迫综合征接受通气治疗。尽管在新生儿重症监护病房接受了治疗,他仍在12小时龄时死亡。尸检显示肾脏高度分叶,有胚芽结节和髓质错构瘤样改变灶。还注意到胰岛细胞突出和肝门管扩张。他的母亲有轻微的颈椎异常。我们讨论了鉴别诊断以及对该患者进行明确诊断的困难,因为辛普森-戈拉比-贝梅尔综合征和佩尔曼综合征之间的显著重叠正变得明显。