Génétique Médicale, Faculté de Médecine, Strasbourg, France.
Génétique Médicale, Faculté de Médecine, Strasbourg, France.
Eur J Med Genet. 2022 Nov;65(11):104612. doi: 10.1016/j.ejmg.2022.104612. Epub 2022 Sep 20.
Associated congenital anomalies may be observed in cases with achondroplasia. The prevalence reported in the literature and the types of co-occurring congenital anomalies are variable between the reported studies. The aim of this study was to establish the prevalence and to describe the associated anomalies in cases with achondroplasia. This study included 25 cases ascertained from our registry of congenital anomalies including all terminations of pregnancy, stillbirths and live births between 1979 and 2007 in 387,067 consecutive births (the prevalence of achondroplasia was 6.4 per 100,000 births), and 223 cases ascertained from the French Little People organization built on the model of LPA (Little People of America, Inc.). Out of these 248 cases of achondroplasia 37 (14.9%) had associated anomalies including 4 (1.6%) cases with chromosomal abnormalities (2 trisomies 21, one 22 q11.2 deletion, and one 47, XXX), 2 (0.8%) cases with recognizable non-chromosomal conditions (one Moebius syndrome and one Pierre Robin sequence) and 31(12.5%) cases with MCA (multiple congenital anomalies). The 31 cases with MCA had 45 anomalies. Anomalies in the urogenital system (24.4%), the cardiovascular system (20.0%), the musculoskeletal system (15.5%), the central nervous system (11.1%), the eye (11.1%), and the orofacial system (8.8%) were the most common MCA. The overall prevalence of associated anomalies shows that the individuals with achondroplasia need a careful screening for other congenital anomalies.
在软骨发育不全病例中可能会观察到相关的先天性异常。文献中报道的患病率和同时发生的先天性异常类型在报道的研究之间存在差异。本研究的目的是确定软骨发育不全病例的患病率并描述相关的异常。本研究包括从我们的先天性异常登记处确定的 25 例病例,这些病例包括 1979 年至 2007 年间 387,067 例连续出生中所有的终止妊娠、死产和活产(软骨发育不全的患病率为每 10 万出生 6.4 例),以及从法国小人生组织(基于美国小人生组织的模型)确定的 223 例病例。在这 248 例软骨发育不全病例中,有 37 例(14.9%)伴有相关异常,包括 4 例(1.6%)染色体异常(21 三体、22q11.2 缺失、47,XXX),2 例(0.8%)可识别的非染色体疾病(1 例 Moebius 综合征和 1 例 Pierre Robin 序列)和 31 例(12.5%)伴有 MCA(多发性先天性异常)。31 例 MCA 病例有 45 种异常。泌尿生殖系统(24.4%)、心血管系统(20.0%)、肌肉骨骼系统(15.5%)、中枢神经系统(11.1%)、眼(11.1%)和口面系统(8.8%)是最常见的 MCA。相关异常的总体患病率表明,软骨发育不全患者需要仔细筛查其他先天性异常。