Hanna E J, Nevin N C, Nelson J
Department of Medical Genetics, Royal Victoria Hospital, Belfast, Northern Ireland.
J Med Genet. 1994 Nov;31(11):858-63. doi: 10.1136/jmg.31.11.858.
Congenital heart defects are a major congenital abnormality and are assuming increasing importance. A study was undertaken to estimate the incidence of congenital heart defects in Northern Ireland over a five year period (1974-1978), to determine the age at diagnosis and to assess the risk of recurrence in sibs. An incidence rate of 7.3 per 1000 total births was found. This reduced to 3.1 per 1000 total births if only invasive methods of diagnosis (catheter studies, surgery, or necropsy) were considered. The overall risk of recurrence for sibs (excluding index patients with chromosomal abnormalities and syndromes) was 3.1%. In addition, excluding families with an affected parent and child gave a recurrence risk of 2.6%. By 6 weeks of age 63% of index patients had been diagnosed and this figure had risen to 88% by 1 year. This has important implications for studies which include only congenital heart defects diagnosed in the first year of life. Of 388 patients with a congenital heart defect confirmed by invasive criteria, 96 (24.7%) were found to have an extracardiac abnormality (ECA). Excluding those with epilepsy or mental handicap as the sole ECA left 91 (23.5%) with an ECA. This highlights the importance of looking for other abnormalities in a child with a congenital heart defect. The 388 index patients had 952 sibs of whom 72 (7.6%) had an ECA. Excluding those with minor abnormalities (inguinal hernias, undescended testes) as the sole ECA left 62 (6.5%) with a major ECA. In addition, excluding those with epilepsy or mental handicap as the sole ECA left 51 (5.4%) with a major ECA. Since parents are often reassured after the birth of a child with a congenital heart defect that their risk of having a child with a noncardiac abnormality is no greater than the general population this finding has important implications for genetic counselling.
先天性心脏缺陷是一种主要的先天性异常,且其重要性日益增加。开展了一项研究,以估计北爱尔兰五年期间(1974 - 1978年)先天性心脏缺陷的发病率,确定诊断年龄,并评估同胞中复发的风险。发现总出生率中先天性心脏缺陷的发病率为每1000例7.3例。如果仅考虑侵入性诊断方法(心导管检查、手术或尸检),这一数字降至每1000例总出生数3.1例。同胞中复发的总体风险(不包括患有染色体异常和综合征的索引患者)为3.1%。此外,排除父母和子女均患病的家庭后,复发风险为2.6%。到6周龄时,63%的索引患者已被诊断,到1岁时这一数字已升至88%。这对于仅包括出生后第一年内诊断出的先天性心脏缺陷的研究具有重要意义。在388例经侵入性标准确诊患有先天性心脏缺陷的患者中,96例(24.7%)被发现有心脏外异常(ECA)。排除那些以癫痫或智力障碍作为唯一ECA的患者后,剩下91例(23.5%)有ECA。这凸显了在患有先天性心脏缺陷的儿童中寻找其他异常的重要性。388例索引患者有952名同胞,其中72例(7.6%)有ECA。排除那些以轻微异常(腹股沟疝、隐睾)作为唯一ECA的患者后,剩下62例(6.5%)有主要ECA。此外,排除那些以癫痫或智力障碍作为唯一ECA的患者后,剩下51例(5.4%)有主要ECA。由于父母在患有先天性心脏缺陷的孩子出生后通常会放心,认为他们生育患有非心脏异常孩子的风险不高于一般人群,这一发现对遗传咨询具有重要意义。