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

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, P = .002), SS2a aOR 0.563 (CI 0.41-0.77, P = .001), SS3 aOR 0.606 (0.43-0.84, P = .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.