Haldar A, Sharma A K, Phadke S R, Jain A, Agarwal S S
Department of Medical Genetics, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, India.
Am J Med Genet. 1994 Oct 15;53(1):21-3. doi: 10.1002/ajmg.1320530105.
We report on a 31-week fetus with hydrocephalus, hypertelorism, microtia, short neck, vertebral and rib defects, scoliosis, omphalocele, exstrophy of bladder, absent external genitalia and pubic rami, imperforate anus, diaphragmatic hernia, defective lobulation of lungs, single kidney, bicornuate uterus, and flexion deformities of the limbs. Similar extensive anomalies in the rostral and caudal regions were described by Russell et al. [Pediatrics, 67:176-182, 1981] and Stewart et al. [Am J Med Genet, 45:426-429, 1993]. The patients described by them had a combination of the oculo-auriculo-vertebral sequence (OAV) and caudal deficiency sequence, whereas the patient reported here can best be described as a combination of OAV and OEIS (omphalocele, exstrophy of bladder, imperforate anus, spinal defects) complexes. The widespread malformations seen in our patient may be the result of an error during blastogenesis.
我们报告一例31周大的胎儿,其患有脑积水、眼距过宽、小耳畸形、短颈、脊椎和肋骨缺陷、脊柱侧弯、脐膨出、膀胱外翻、外生殖器和耻骨支缺如、肛门闭锁、膈疝、肺叶分叶缺陷、单肾、双角子宫以及肢体屈曲畸形。Russell等人[《儿科学》,67:176 - 182, 1981]和Stewart等人[《美国医学遗传学杂志》,45:426 - 429, 1993]描述了头侧和尾侧区域类似的广泛异常情况。他们所描述的患者具有眼耳脊椎序列(OAV)和尾侧发育不全序列的组合,而此处报告的患者最好描述为OAV和OEIS(脐膨出、膀胱外翻、肛门闭锁、脊柱缺陷)复合体的组合。我们患者中所见的广泛畸形可能是胚胎发育过程中出错的结果。