Rethinking speech sound disorder (SSD) in non-syndromic cleft lip and palate: The importance of recognizing phonological and language difficulties.

Background: Children born with cleft palate ± lip (CP ± L) are at risk of speech sound disorder (SSD). Up to 40% continue to have SSD at age 5-6 years. These difficulties are typically described as articulatory in nature and often include cleft speech characteristics (CSC) hypothesized to result from structural differences. In non-CP ± L SSD comorbidity with language difficulties is often reported. There is growing evidence of concomitant language difficulties in children with CP ± L and of a higher prevalence of developmental speech errors in children compared with non-CP ± L peers. The impact of underlying phonological and language skills on speech production in children with CP ± L is poorly understood.

Aims: To investigate language outcomes in children with CP ± L and the relationship to speech production, by answering the following research questions: (1) Does the profile of language skills in children with CP ± L differ from normative samples? (2) Do children with CP ± L and SSD have poorer language skills than those with typically developing speech? (3) Is there an association between language skills and speech profile in children with CP ± L at age 5-8 years?

Methods & procedures: In this prospective cross-sectional, observational study, 95 participants were recruited from regional cleft lip and palate services in the UK. They were aged 5;0-7;11 with non-syndromic CP ± L. Those with a syndromic diagnosis, global learning disability, sensorineural hearing loss and first language other than English were excluded. Assessments of speech (Diagnostic Evaluation of Articulation and Phonology-DEAP) and language (Clinical Evaluation of Language Fundamentals-5th UK edition-CELF) were completed. Language outcomes were analysed and compared with normative samples and according to speech error analysis.

Outcomes & results: Average language scores were within the expected range. For those presenting with SSD, language scores were significantly lower than those with typically developing speech. Analysis of speech errors showed four distinct speech profiles: typical speech, CSC only, developmental speech characteristics (DSC), and combined CSC + DSC. Language scores were lower for participants with DSC (±CSC). A significant association was found between the presence of CSC + DSC and expressive language outcomes (odds ratio (OR) = 10.82; 95% confidence interval (CI) = 2.42, 48.32, p = 0.002).

Conclusions & implications: An association between language skills and speech production was observed. The distribution of speech errors in children with CP ± L varied with a high level of DSC as well as CSC. Those with CSC + DSC had significantly lower language scores than those with typically developing speech or CSC only. Speech and language therapists working with this caseload should be alerted to potential ongoing phonological and language difficulties in children presenting with this profile.

What this paper adds: What is already known on this subject Children born with CP ± L are known to be at risk of SSD. There is also evidence of language delay in the early years. Evidence for persistent language difficulties is equivocal but some studies have shown a higher than average prevalence of developmental phonological errors in addition to cleft articulation errors in speech production. What this paper adds to the existing knowledge This study investigates links between speech development and language skills in children aged 5-8 years with non-syndromic CP ± L. It adds to our understanding of the nature of SSD in children with CP ± L and in particular the relationship between language skills and speech production. It shows that children with CP ± L have varied speech profiles and that those presenting with delayed phonological processes are also at risk of language difficulties. What are the potential or actual clinical implications of this work? Speech and language therapists working with children with CP ± L should be aware of the varied nature of SSD in this population. They should be alert to the need for additional assessment of language for those presenting with delayed phonological processes and the implications this has for educational attainment.