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探究额外先天性畸形对腭裂患儿五岁时言语结果的影响。

Investigating the Impact of Additional Congenital Malformations on Speech Outcomes at age Five in Children with a Cleft Palate.

作者信息

Butterworth Sophie, Fitzsimons Kate J, Britton Lorraine, Van Eeden Stephanie, Medina Jibby, van der Meulen Jan, Russell Craig J H

机构信息

Cleft Registry and Audit Network, Clinical Excellence Unit, The Royal College of Surgeons of England, London, UK.

Trent Regional Cleft Network, Nottingham University Hospital NHS Trust, Nottingham, UK.

出版信息

Cleft Palate Craniofac J. 2024 Oct 9:10556656241287759. doi: 10.1177/10556656241287759.

DOI:10.1177/10556656241287759
PMID:39380396
Abstract

OBJECTIVE

To determine whether children born with a cleft palate ± lip (CP ± L) and additional congenital differences (ACDs - including 'Congenital malformations and deformations' as coded in ICD-10), are less likely to meet the three national speech outcome standards at age five compared to children with CP ± L and no ACDs.

DESIGN

An observational study, utilizing national data from the UK Cleft Registry and Audit NEtwork (CRANE) Database linked to national administrative data of hospital admissions.

SETTING

National Health Service, England.

PATIENTS

2191 children (993 female, 1198 male) with CP ± L, born 2006-2012 in England, with complete speech data and consent to data linkage.

OUTCOME MEASURE

Perceptual speech analysis utilised the Cleft Audit Protocol for Speech - Augmented (CAPS-A) rating and United Kingdom National Speech Outcome Standards (Speech Standard 1 (SS1), Speech Standard 2a (SS2a) and Speech Standard 3 (SS3)).

RESULTS

Of 2191 children, 759 (35%) had at least one ACD. Presence of one ACD did not significantly impact speech outcomes but two or more ACDs reduced the odds of achieving all three speech standards: SS1 aOR 0.602 (CI 0.45-0.82,  = .002), SS2a aOR 0.563 (CI 0.41-0.77,  = .001), SS3 aOR 0.606 (0.43-0.84,  = .003). When exploring ACDs by ICD-10 groupings, congenital malformations of the 'Eye, ear, face and neck', 'Circulatory system', 'Digestive system', 'Musculoskeletal system' and 'Other congenital malformations' reduced a child's odds of achieving the speech standards.

CONCLUSIONS

ACDs, in the absence of a known syndrome, have a significant impact on speech outcome at age five. Incorporating these factors into risk-adjustment models for service level outcome reporting is recommended.

摘要

目的

确定与患有唇腭裂(CP±L)但无其他先天性差异(ACD,包括国际疾病分类第十版(ICD - 10)中编码的“先天性畸形和变形”)的儿童相比,患有CP±L及其他先天性差异的儿童在五岁时达到三项国家言语结果标准的可能性是否更低。

设计

一项观察性研究,利用来自英国腭裂登记与审计网络(CRANE)数据库的国家数据,并与医院入院的国家行政数据相链接。

背景

英国国家医疗服务体系,英格兰。

患者

2006年至2012年在英格兰出生的2191名患有CP±L的儿童(993名女性,1198名男性),拥有完整的言语数据且同意进行数据链接。

结果测量

感知言语分析采用言语增强腭裂审计协议(CAPS - A)评分以及英国国家言语结果标准(言语标准1(SS1)、言语标准2a(SS2a)和言语标准3(SS3))。

结果

在2191名儿童中,759名(35%)至少有一项ACD。存在一项ACD对言语结果没有显著影响,但两项或更多项ACD降低了达到所有三项言语标准的几率:SS1的调整后比值比(aOR)为0.602(95%置信区间为0.45 - 0.82,P = 0.002),SS2a的aOR为0.563(95%置信区间为0.41 - 0.77,P = 0.001),SS3的aOR为0.606(95%置信区间为0.43 - 0.84,P = 0.003)。当按ICD - 10分组探究ACD时,“眼、耳、面和颈部”“循环系统”“消化系统”“肌肉骨骼系统”以及“其他先天性畸形”的先天性畸形降低了儿童达到言语标准的几率。

结论

在无已知综合征的情况下,ACD对五岁时的言语结果有显著影响。建议将这些因素纳入服务水平结果报告的风险调整模型中。

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