Lammens M, Moerman P, Fryns J P, Lemmens F, van de Kamp G M, Goemans N, Dom R
Stichting PAMM, Catharinaziekenhuis, Eindhoven, The Netherlands.
Neuropediatrics. 1997 Apr;28(2):116-9. doi: 10.1055/s-2007-973683.
Nine patients with the characteristic signs of fetal akinesia sequence (polyhydramnion, multiple joint contractures and lung hypoplasia) are described. In 8 of the 9 patients nemaline myopathy could be demonstrated with histology. The ninth patient presented the same phenotype as his 4 affected siblings in whom the nemaline myopathy could be histologically proven. Seven of the patients belonged to 2 families; the other 2 patients were isolated cases. In one fetal case nemaline myopathy was documented at week 22 of gestation. These observations demonstrate that nemaline myopathy can cause the fetal akinesia sequence, with onset of first symptoms as early as the beginning of the second trimester of pregnancy.
本文描述了9例具有胎儿运动不能序列征(羊水过多、多关节挛缩和肺发育不全)典型体征的患者。9例患者中有8例经组织学检查证实为杆状肌病。第9例患者与其4名受影响的兄弟姐妹表现出相同的表型,后者经组织学检查证实患有杆状肌病。其中7例患者来自2个家族;另外2例为散发病例。在1例胎儿病例中,妊娠22周时记录到杆状肌病。这些观察结果表明,杆状肌病可导致胎儿运动不能序列征,最早在妊娠中期开始时出现首发症状。