Pediatrics II, University Children's Hospital Essen, 45122 Essen, Germany.
Am J Hum Genet. 2011 Nov 11;89(5):668-74. doi: 10.1016/j.ajhg.2011.10.007.
Urinary bladder malformations associated with bladder outlet obstruction are a frequent cause of progressive renal failure in children. We here describe a muscarinic acetylcholine receptor M3 (CHRM3) (1q41-q44) homozygous frameshift mutation in familial congenital bladder malformation associated with a prune-belly-like syndrome, defining an isolated gene defect underlying this sometimes devastating disease. CHRM3 encodes the M3 muscarinic acetylcholine receptor, which we show is present in developing renal epithelia and bladder muscle. These observations may imply that M3 has a role beyond its known contribution to detrusor contractions. This Mendelian disease caused by a muscarinic acetylcholine receptor mutation strikingly phenocopies Chrm3 null mutant mice.
与膀胱出口梗阻相关的膀胱畸形是儿童进行性肾衰竭的常见原因。我们在此描述了一种常染色体显性遗传的先天性膀胱畸形伴梅干腹样综合征,该综合征与 M3 型毒蕈碱乙酰胆碱受体(CHRM3)(1q41-q44)纯合移码突变有关,确定了该疾病的一个孤立的基因缺陷。CHRM3 编码 M3 型毒蕈碱乙酰胆碱受体,我们发现其存在于发育中的肾上皮和膀胱肌肉中。这些观察结果可能表明,M3 除了对逼尿肌收缩的已知作用外,还有其他作用。这种由毒蕈碱乙酰胆碱受体突变引起的孟德尔疾病与 Chrm3 基因缺失突变小鼠的表型惊人地相似。