Ethnic variations in the childhood prevalence of Hirschsprung disease in New Zealand

IF 1.5 4区医学 Q3 SURGERY

ANZ Journal of Surgery Pub Date : 2018-10-03 DOI:10.1111/ans.14857

Kiarash Taghavi, Lucy Goddard, Stephen M. Evans, Andrew Hobson, Spencer W. Beasley, Sasikumar Sankaran, Askar Kukkady, Jonathan Stevenson, Mark D. Stringer

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引用次数: 5

Abstract

Background

Epidemiological studies have suggested that there may be ethnic variations in the prevalence of Hirschsprung disease (HD) but no study has systematically investigated this issue or potential ethnic variations in the extent of aganglionosis in HD. This study aimed to investigate this in a childhood population in New Zealand.

Methods

A multicentre national retrospective review was undertaken of all newly diagnosed cases of HD at each of the four paediatric surgical centres in New Zealand over a 16-year period (January 2000 to December 2015). Original histological, radiological and operative reports were obtained and analysed. Self-identified ethnicity was recorded from admission documents. Birth statistics were obtained from Statistics New Zealand.

Results

A total of 246 cases of HD were identified. The prevalence of HD was 1:3790 live births for European, 1:6610 among Māori, 1:1834 among Pacific Peoples, 1:3847 among Asian and 1:5694 among Middle Eastern. The prevalence of HD was statistically significantly greater in Pacific Peoples (P < 0.0005). The proportion of children with long-segment HD was also significantly greater in Pacific and Asian populations than others (P = 0.04). These findings were not due to differences in the proportion of familial cases of HD among the different populations.

Conclusions

The prevalence and phenotype of HD varies significantly between different ethnic groups within New Zealand. This may well be related to variations in the frequencies of HD-associated gene mutations within these populations.

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新西兰儿童先天性巨结肠病患病率的种族差异

流行病学研究表明，巨结肠病(HD)的患病率可能存在种族差异，但没有研究系统地调查这一问题或HD中神经节病程度的潜在种族差异。这项研究的目的是在新西兰的儿童人群中调查这一点。方法对新西兰4个儿科外科中心16年间(2000年1月至2015年12月)所有新诊断的HD病例进行多中心全国回顾性分析。获得原始的组织学、放射学和手术报告并进行分析。从录取文件中记录自我认同的种族。出生统计数据来自新西兰统计局。结果共检出HD患者246例。欧洲人活产HD患病率为1:37 790,Māori人群为1:6610，太平洋人群为1:18 834，亚洲人群为1:38 847，中东人群为1:56 694。太平洋人种的HD患病率有统计学意义(P < 0.0005)。太平洋和亚洲人群中患有长段HD的儿童比例也显著高于其他地区(P = 0.04)。这些发现不是由于不同人群中HD家族病例比例的差异。结论新西兰不同种族人群HD患病率和表型差异显著。这很可能与这些人群中hd相关基因突变频率的变化有关。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

ANZ Journal of Surgery 医学-外科

CiteScore

2.50

自引率

11.80%

发文量

720

审稿时长

2 months

期刊介绍： ANZ Journal of Surgery is published by Wiley on behalf of the Royal Australasian College of Surgeons to provide a medium for the publication of peer-reviewed original contributions related to clinical practice and/or research in all fields of surgery and related disciplines. It also provides a programme of continuing education for surgeons. All articles are peer-reviewed by at least two researchers expert in the field of the submitted paper.