Speech Outcome in Cleft Palate

Cleft lip and palate (CP±L) occurs in Sweden in about one child per 500 born. However, children with CLP are a heterogeneous group with several subgroups and differences between them. This applies to the type of cleft, size and cooccurrence of other malformations or disabilities.

Illustration av May Johansson

Primary surgical repair of the cleft palate is primarily to provide the best conditions for speech development, occlusion and appearance. The treatment is multidisciplinary and also includes the speech and palatal function, the function of the ear and hearing, which are important contributing components for speech and language development, as well as dental development and growth in the face, which are important contributing components for appearance.

Early identification of children who are at risk of being delayed or deviating in speech development is necessary to be able to develop care programs and routines to prevent poor health in terms of speech disorders that make speech different and difficult to understand and that affect the ability for communicative activity and participation.

Speech deviations related to CP±LG

Today there is rather good knowledge of the occurrence of speech deviations related to cleft palate as well as about differences depending on the type of cleft. International studies show relatively consistent results, although these vary. In summary, up to 50 percent of the children treated for CP±L still seem to have speech and / or nasality problems in preschool or early school age, and some support that there is a higher incidence of deviations in speech and language abilities in the case of a major defect or in the presence of other malformations/syndromes. 

Furthermore, that peers perceive deviations in speech which could have a negative impact on participation. Data on ear and hearing problems are still scarce, but provide support for the fact that otitis media with effusion (OME) with associated fluctuating mild hearing loss occurs in virtually all children with cleft palate. However, there is still no support for any surgical protocol to providing the best conditions for speech development. In addition, there is insufficient knowledge of people's own perception of their speech and the need for and effect of speech intervention.

Ongoing projects

The group investigates the effect of the method for surgery of the cleft palate in children born with cleft lip, jaw and / or cleft palate (CP±L) on how speech develops, how people perceive their own speech and the effect of speech therapy.

The Scandcleft Trials project, which started in 1997, consists of three concurrent randomized trials of primary surgery for infants born with complete unilateral cleft lip and palate (UCLP). The project was developed and executed by 10 North European cleft teams: Århus/Copenhagen (Denmark), Helsinki (Finland), Bergen/Oslo (Norway), Gothenburg/Linköping/ Stockholm (Sweden), and Manchester/Belfast (UK). One surgical protocol was defined to serve as a common method in each trial, against which a local protocol was compared. Recruitment of 448 infants took place over a 9-year period, with high subsequent retention of participants in each trial. The 5- and 10-year outcome has been published. Longitudinal analyses are ongoing as well as follow-up at young adulthood. 

Trial registration number ISRCTN29932826.

PI Speech: Anette Lohmander 2001 – 2016, Christina Persson 2017 –

The primary objective of the TOPS-trial is to determine whether surgery for cleft palate performed at 6 or 12 months of age is most beneficial for speech outcomes. Approximately 550 children from five countries (all centres Denmark, Norway and Sweden, the Bauru center in Brazil and around 10 in the UK).

The primary outcome is perceived velopharyngeal function at 5 years of age. Secondary outcomes at 12 and 36 months of age will be reported as well.

Trial registration number NCT00993551.

PI Stockholm Center:  Anette Lohmander.

Fluctuating hearing loss due to otitis media with effusion (OME) is very common in children born with cleft palate (CP±L). In the TUTH project, early development of speech and hearing is studied in a controlled group study of children with CP±L and children with OME, respectively. 

Subprojects include hearing, perception and production of consonant sounds, consonant inventory and proficiency, velopharyngeal function, intelligibility, vocabulary and language comprehension up to 3 years of age.

PI: Anette Lohmander
Postdoc-project: Åsa Okhiria

In Sweden, children born with cleft palate are operated on at 6 different university hospitals. There are mainly two methods of cleft palate surgery that are used; closure in one session at 9-12 months of age or closure in two sessions where the soft palate is closed at around 6 months and the cleft in the hard palate at around 2 years of age (earlier in preschool or early school age). The evidence regarding the speech result after various surgical methods has been insufficient both in terms of a specific age and longitudinally. The purpose of a series of projects is partly to contribute generally to the basis for choosing a surgical treatment method by evaluating how speech develops in children born with LKG cleft, and partly to specifically investigate the impact of speech abnormalities on the environment's perception of the individual's speech and communication. Three project parts have been completed:

  • Speech outcome in children born with cleft lip and palate: aspects of function and environment (doctoral student Jill Nyberg, 2016, PhD).
  • Orofacial function, articulation proficiency and intelligibility in 5-year old children born with cleft lip and palate (doctoral student Ann Malmenholt, 2020: PhD)
  • Speech outcome and self-reported communicative ability in young adults born with unilateral cleft lip and palate: comparing long-term results after two different surgical methods for palatal repair (doctoral student Petra Peterson, 2022; PhD)

The speech of a group of children born with UCLP and treated with a two-stage palate repair are examined longitudinally from 1-7 years and is compared with the results in a previous group treated with one-stage palate repair. Age-relevant speech variables are assessed perceptually, and the results are compared between the groups. Both studies include measures of intelligibility, that is, how well the environment perceives / understands the speech, and the individuals' own perception of their speech.

Doctoral student: Emilie Hagberg

Speech disorders in children born with cleft palate are treated with speech therapy, but treatment studies of speech disorders related to cleft palate are still few and with great methodological variation. The project includes a systematic review and meta-analysis.

Furthermore, two treatment studies where both are controlled case series (Controlled single subject series design) in collaboration with speech and language pathologists in the Stockholm Region. In the second, distance treatment is planned.

Postdoctoral project: Anders Sand
Doctoral student project: Emilie Hagberg

These and the other projects are described on the respective researcher's profile page.

Collaborators

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Emilie Hagberg

Karolinska Institutet, Karolinska Universitetssjukhuset

Karolinska Institutet

Petra Peterson

Karolinska Universitetssjukhuset
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Ann Malmenholt

Karolinska Universitetssjukhuset, Karolinska Institutet

Jill Nyberg

Mentor

Åsa Okhiria

Affiliated to Research

Christina Persson

Göteborgs universitet

Kristina Klintö

Malmö Universitetssjukhus/Lunds universitet

Dissertations

Petra Peterson, PhD, KI, 2022: A multidisciplinary long-term evaluation of patients born with unilateral cleft lip and palate treated with different surgical protocols.

Birgitta Tengroth, Med Lic, KI, 2020: Hearing in children with cleft palate.

Ann Malmenholt, PhD, KI, 2020: Speech and language in 5-year-old children with Childhood Apraxia of Speech or Cleft Palate.

Jill Nyberg, PhD, KI, 2016: Speech outcome in children with cleft palate treated with one-stage palate repair: Aspects of function and environment.

Kristina Klintö, PhD, KI, 2014: Speech and expressive language in Swedish-speaking children with unilateral cleft lip and palate.

Selected publications

Scandcleft Project

Persson C, Davies J, Havstam C, Søgaard H, Bowden M, Boers M, Nielsen JB, Alaluusua S, Lundeborg Hammarström I, Emborg BK, Sand A, Lohmander A. Speech outcome at 5 and 10 years in unilateral cleft lip and palate: influence of speech therapy and secondary velopharyngeal surgery. Cleft Palate-Craniofac J. Accepted Dec 20, 2023.

Willadsen E, Jørgensen L, Alalusuua S, Pedersen NH, Nielsen J, Hölttä E……Lohmander A, Persson C. Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: speech proficiency at 10 years of age. Int J Lang Comm Dis. 2023;58:892-909 

Persson C, Pedersen NH, Hayden C, Bowden M, Aukner R, Vindenes H, Åbyholm F, Whitby D, Willadsen E, Lohmander A. Scandcleft Project Trial 3: Comparison of speech outcome in relation to sequence in two-stage palatal repair procedures in 5-year-olds with UCLP. Cleft Palate-Craniofac J. 2020;57:352-363. 

Hammarström IL, Nyberg J, Alaluusua S, Rautio J, Neovius E, Berggren A, Willadsen E, Persson C, Lohmander A. Scandcleft Project Trial 2: Comparison of speech outcome in one and two-stage palatal closure in 5-year-olds with UCLP. Cleft Palate-Craniofac J. 2020;57:458-469

Willadsen, E., Lohmander, A., Persson, C., Boers, M., Kisling-Møller, M., Havstam, C., . . . Andersen, M. (2019). Scandcleft Project, Trial 1: Comparison of Speech Outcome in Relation to Timing of Hard Palate Closure in 5-Year-Olds With UCLP. Cleft Palate Craniofac J, 2019;56:1276-1286. 

Lohmander A, Persson C, Willadsen E, Lundeborg I, Alaluusua S, Aukner R, et.al. Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 4. Speech outcomes in 5-year-olds - velopharyngeal competency and hypernasalityJ Plast Surg Hand Surgery, 2017;51:26-36. 

Willadsen E, Lohmander A, Persson C, Lundeborg I, Alaluusua S, Aukner R, et.al. Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 5. Speech outcomes in 5-year-olds - consonant production and errorsJ Plast Surg Hand Surgery, 2017;51:37-50.

Lohmander A, Hagberg E, Persson C. Willadsen E, LundeborgI, Davies J, Havstam C, Boers M, Kisling-Møller M, Alaluusua S, AuknerR, Pedersen NH, Tuurunen L, NybergJ. Validity of auditory perceptual assessment of velopharyngeal function and dysfunction: the VPC-Sum and the VPC-Rate. Clinical Linguistics & Phonetics, 2017;31:589-597. 

Billaud Feragen K, Semb G, Heliövaara A, Lohmander A, Johannessen EC, Boysen BM, et al. Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 10. Parental perceptions of appearance and treatment outcomes in their 5-year-old childJ Plast Surg Hand Surg, 2017;51:80-86. 

Semb G, Enemark H, Friede H, Paulin G, Lilja J, Rautio J, Andersen M, Åbyholm F, Lohmander A, ….Worthington W. Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 1. Planning and managementJ Plast Surg Hand Surg, 2017;51:1-12.  

Lohmander A, Willadsen E, Bowden M, Henningsson G, Persson C, Hutters B. Methodology for Speech Assessment in the Scandcleft Project – an international randomised clinical trial on palatal surgery: experiences from a pilot study. Cleft Palate-Craniofac J. 2009; 46:347-362. 

TOPS-project

Gamble C, Persson C, Willadsen E, ………..Lohmander A, ………..Nyberg J, ……….Westberg LR, ……..Munro K, Walsh T, Shaw W. Timing Of Primary Surgery (TOPS) for Cleft Palate Repair. New England Journal of Medicine, 2023;389(9):795-807. 

Shaw W, Semb G, Lohmander A, Persson C, Willadsen E, Clayton-Smith J, Trindade IK, Munro KJ, Gamble C, Harman NH, Conroy EJ, Weichart D, Williamson P. Timing Of Primary Surgery for cleft palate (TOPS): Protocol for a randomised trial of palate surgery at 6 months versus 12 months of ageBritish Medical Journal Open, 2019;9:e029780.  

Speech outcome and communicative ability in individuals born with CP±L - comparing results after different methods for palatal repair in Sweden

Peterson P, Nyberg J, Persson C, Mark H, Lohmander A. Speech outcome and self-reported communicative ability in young adults born with UCLP: comparing results after two methods for palatal repair. Cleft Palate-Craniofac J. 2022;59(6):751–764. 

Klintö K, Brunnegård K, Havstam C, Appelqvist M, Hagberg E, Taleman A-S, Lohmander A. Speech in 5-year-olds born with unilateral cleft lip and palate – a prospective Swedish intercenter studyJ Plast Surg Hand Surgery, 2019;53:309-315. 

Malmenholt, A, McAllister A, Lohmander A. Orofacial function, articulation proficiency and intelligibility in 5-year-old children born with cleft lip and palateCleft Palate-Craniofacial J. 2019;56:321-330. 

Nyberg J, Neovius E, Lohmander A.  Speech outcomes after one-stage palatal repair with muscle reconstruction in children with isolated cleft palate – a longitudinal perspective. J Plastic Surgery Hand Surgery, 2018;52:20-29. 

Nyberg J, Peterson P, Lohmander A. Speech outcomes at age 5 and 10 years in unilateral cleft lip and palate after one-stage palatal repair with Minimal Incision Technique - a longitudinal perspectiveInt J Pediatric Otorhinolaryngology, 2014;78:1662-1670.

Lohmander A, Friede H, Lilja J. Long-term, longitudinal follow-up of individuals with unilateral cleft lip and palate after the Gothenburg primary early veloplasty and delayed hard palate closure protocol: Speech outcomeCleft Palate-Craniofac J, 2012;49:657-671. 

Lohmander A, Persson C. A longitudinal study of speech production in Swedish children with cleft palate and two-stage palatal repairCleft Palate-Craniofacial J, 2008;45:32-41. 

Lohmander A, Friede H, Elander A, Persson C, Lilja J. Speech development in patients with unilateral cleft lip and palate treated with different delays of hard palate closure after early velar repair: a longitudinal perspectiveScand J Plast Reconstr Surg Hand Surgery, 2006;46:267-274

Early Development of Speech and Hearing – TUTH (and related)

Lohmander A, Raud Westberg L, Olsson S, Tengroth B, Flynn T. Canonical babbling and early consonant development related to hearing in children with otitis media with effusion with or without cleft palate. Cleft Palate-Craniofac J. 2021;58:894-905. 

Tengroth B, Lohmander A, Hederstierna C. Hearing thresholds concurrent with OME in young children with and without cleft palateCleft Palate Craniofac J. 2020;57:616-623. 

Flynn T, Lohmander A. A longitudinal study of hearing and middle ear status in individuals with UCLPOtology & Neurology, 2014;35:989-996. 

Flynn T, Möller C, Jönsson R, Lohmander A. The high prevalence of otitis media with effusion in children with cleft lip and palate as compared to children without clefts. Int J of Pediatric Otorhinolaryngology, 2009;73:1441-1446

Effect of speech therapy of LKG-related speech disorders

Hagberg E, Andersson Stockhaus U, Fagius T, Hartstein M, Raud Westberg L, Nyberg J, Sand A, Lohmander A: Speech therapy in children with cleft palate: speech outcome from multiple single subject casesIJSLP Under Review 2024.

Sand A, Hagberg E, Lohmander A. On the benefits of speech-language therapy for individuals born with cleft palate: a systematic review and meta-analysis of individual participant dataJ Speech Lang Hear Res. 2022;65:555-573.

Financing

Forte
Grants from the Swedish government under the ALF agreement
NIH, National Institute for Dental and Craniofacial research (NIH-NIDCR)
Stiftelsen Frimurare Barnhuset i Stockholm
Aina Börjesons Research Foundation
Queen Silvia's Jubilee Fund

AL
Content reviewer:
05-11-2024