TY - JOUR

T1 - Influence of timing of delayed hard palate closure on articulation skills in 3-year-old Danish children with unilateral cleft lip and palate

AU - Willadsen, Elisabeth

AU - Boers, Maria

AU - Schops, Antje

AU - Kisling-Møller, Mia

AU - Bogh Nielsen, Joan

AU - Jørgensen, Line Dahl

AU - Andersen, Mikael

AU - Bolund, Stig

AU - Andersen, Helene Søgaard

PY - 2017/7/25

Y1 - 2017/7/25

N2 - Background Differing results regarding articulation skills in young children with cleft palate (CP) have been reported and often interpreted as a consequence of different surgical protocols. Aims To assess the influence of different timing of hard palate closure in a two-stage procedure on articulation skills in 3-year-olds born with unilateral cleft lip and palate (UCLP). Secondary aims were to compare results with peers without CP, and to investigate if there are gender differences in articulation skills. Furthermore, burden of treatment was to be estimated in terms of secondary surgery, hearing and speech therapy. Methods & Procedures A randomized controlled trial (RCT). Early hard palate closure (EHPC) at 12 months versus late hard palate closure (LHPC) at 36 months in a two-stage procedure was tested in a cohort of 126 Danish-speaking children born with non-syndromic UCLP. All participants had the lip and soft palate closed around 4 months of age. Audio and video recordings of a naming test were available from 113 children (32 girls and 81 boys) and were transcribed phonetically. Recordings were obtained prior to hard palate closure in the LHPC group. The main outcome measures were percentage consonants correct adjusted (PCC-A) and consonant errors from blinded assessments. Results from 36 Danish-speaking children without CP obtained previously by Willadsen in 2012 were used for comparison. Outcomes & Results Children with EHPC produced significantly more target consonants correctly (83%) than children with LHPC (48%; p < .001). In addition, children with LHPC produced significantly more active cleft speech characteristics than children with EHPC (p < .001). Boys achieved significantly lower PCC-A scores than girls (p = .04) and produced significantly more consonant errors than girls (p = .02). No significant differences were found between groups regarding burden of treatment. The control group performed significantly better than the EHPC and LHPC groups on all compared variables. What this paper adds What is already known on the subject Quite different results have been reported on articulation skills in young children with CP as a consequence of different surgical protocols, or different timings of hard palate closure in two-stage procedures. Comparisons between studies are often impossible due to substantial differences in methodologies and methodological flaws. Most often results on articulation only include cleft speech characteristics that makes it difficult to understand the overall articulation proficiency of the participants. What this paper adds to existing knowledge This study of 113 Danish children with UCLP shows that leaving the hard palate unrepaired until 36 months, as opposed to closing it at 12 months of age, has a negative impact on development of articulation skills. An overall measure of articulation proficiency considering all consonant errors was included to provide a better understanding of the articulation skills of the participants. Inclusion of a control group of children without CP allowed comparison between articulation skills of 3-year-olds with and without CP. What are the potential or actual clinical implications of this work? From a linguistic point of view, it is not recommendable to postpone hard palate closure in two-stage procedures until 36 months of age due to the impact on acquisition of speech sounds. Results of the present study suggest that it is important to pay attention to all consonant errors in young children with CP, not only those directly related to the cleft condition. Results also indicate that young boys with CP have poorer articulation than girls do, but further investigation is needed to draw conclusions on clinical implications.

AB - Background Differing results regarding articulation skills in young children with cleft palate (CP) have been reported and often interpreted as a consequence of different surgical protocols. Aims To assess the influence of different timing of hard palate closure in a two-stage procedure on articulation skills in 3-year-olds born with unilateral cleft lip and palate (UCLP). Secondary aims were to compare results with peers without CP, and to investigate if there are gender differences in articulation skills. Furthermore, burden of treatment was to be estimated in terms of secondary surgery, hearing and speech therapy. Methods & Procedures A randomized controlled trial (RCT). Early hard palate closure (EHPC) at 12 months versus late hard palate closure (LHPC) at 36 months in a two-stage procedure was tested in a cohort of 126 Danish-speaking children born with non-syndromic UCLP. All participants had the lip and soft palate closed around 4 months of age. Audio and video recordings of a naming test were available from 113 children (32 girls and 81 boys) and were transcribed phonetically. Recordings were obtained prior to hard palate closure in the LHPC group. The main outcome measures were percentage consonants correct adjusted (PCC-A) and consonant errors from blinded assessments. Results from 36 Danish-speaking children without CP obtained previously by Willadsen in 2012 were used for comparison. Outcomes & Results Children with EHPC produced significantly more target consonants correctly (83%) than children with LHPC (48%; p < .001). In addition, children with LHPC produced significantly more active cleft speech characteristics than children with EHPC (p < .001). Boys achieved significantly lower PCC-A scores than girls (p = .04) and produced significantly more consonant errors than girls (p = .02). No significant differences were found between groups regarding burden of treatment. The control group performed significantly better than the EHPC and LHPC groups on all compared variables. What this paper adds What is already known on the subject Quite different results have been reported on articulation skills in young children with CP as a consequence of different surgical protocols, or different timings of hard palate closure in two-stage procedures. Comparisons between studies are often impossible due to substantial differences in methodologies and methodological flaws. Most often results on articulation only include cleft speech characteristics that makes it difficult to understand the overall articulation proficiency of the participants. What this paper adds to existing knowledge This study of 113 Danish children with UCLP shows that leaving the hard palate unrepaired until 36 months, as opposed to closing it at 12 months of age, has a negative impact on development of articulation skills. An overall measure of articulation proficiency considering all consonant errors was included to provide a better understanding of the articulation skills of the participants. Inclusion of a control group of children without CP allowed comparison between articulation skills of 3-year-olds with and without CP. What are the potential or actual clinical implications of this work? From a linguistic point of view, it is not recommendable to postpone hard palate closure in two-stage procedures until 36 months of age due to the impact on acquisition of speech sounds. Results of the present study suggest that it is important to pay attention to all consonant errors in young children with CP, not only those directly related to the cleft condition. Results also indicate that young boys with CP have poorer articulation than girls do, but further investigation is needed to draw conclusions on clinical implications.

U2 - 10.1111/1460-6984.12331

DO - 10.1111/1460-6984.12331

M3 - Journal article

JO - International Journal of Language and Communication Disorders

JF - International Journal of Language and Communication Disorders

SN - 1368-2822

ER -