Bongers E M H F, Knoers N V A M
Universitair Medisch Centrum St Radboud, afd. Antropogenetica, Postbus 9101, 6500 HB Nijmegen.
Ned Tijdschr Geneeskd. 2003 Jan 11;147(2):67-9.
Nail-patella syndrome (NPS) is an autosomal dominant hereditary disorder characterised by nail dysplasia, patellar apoplasia/hypoplasia, iliac horns, elbow dysplasia, and frequently primary open angle glaucoma and progressive nephropathy. The gene underlying NPS, LMX1B on chromosome 9q34.1, is a transcription factor involved in the normal dorsoventral patterning of the limb and normal development of the glomerular basement membrane in the kidney. Recent studies suggest a role for LMX1B in the regulation of collagen IV expression and in the transcriptional regulation of podocyte specification and differentiation. At present, no evidence for a correlation between the presence and severity of the clinical anomalies and the LMX1B genotype has been found.
指甲-髌骨综合征(NPS)是一种常染色体显性遗传性疾病,其特征为指甲发育异常、髌骨发育不全/发育不良、髂骨角、肘部发育异常,且常伴有原发性开角型青光眼和进行性肾病。NPS的致病基因是位于9号染色体q34.1区域的LMX1B,它是一种转录因子,参与肢体正常的背腹模式形成以及肾脏肾小球基底膜的正常发育。最近的研究表明,LMX1B在IV型胶原蛋白表达的调控以及足细胞特化和分化的转录调控中发挥作用。目前,尚未发现临床异常的存在及严重程度与LMX1B基因型之间存在相关性的证据。