Inter-centre comparison of data on surgery and speech outcomes at 5 years of age based on the Swedish quality registry for patients born with cleft palate with or without cleft lip

(2022) 22:303 Klintö et al. BMC Pediatrics https://doi.org/10.1186/s12887-022-03367-2 Open Access RESEARCH Inter‑centre comparison of data on surgery and speech outcomes at 5 years of age based on the Swedish quality registry for patients born with cleft palate with or without cleft lip Kristina Klintö1* , Marie Eriksson2, Avni Abdiu3, Karin Brunnegård4, Jenny Cajander5, Emilie Hagberg6, Malin Hakelius7, Christina Havstam8, Hans Mark9, Åsa Okhiria10, Petra Peterson11, Kristina Svensson12 and Magnus Becker1 Abstract Background: The objective of the Swedish cleft lip and palate registry (CLP registry) is to promote quality control, research and improvement of treatment, by the comparison of long-term results. The aim was to compare data from the CLP registry among the six treatment centres, regarding data on surgery and speech outcomes at 5 years of age. Methods: The participants were 430 children born in Sweden from 2009 to 2014, with cleft palate with or without cleft lip and without known syndromes and/or additional malformations. The number of primary and secondary palatal surgeries up to 5 years of age, timing of the last primary palatal surgery, percentage consonants correct, percentage non-oral speech errors and perceived velopharyngeal competence at 5 years were assessed. Multivariable binary logistic regression adjusted for sex and cleft type was used to compare results between the six centres. Results: At one centre (centre 4), the palate was closed in one to three stages, and at the remaining centres in one or two stages. At centre 4, more children underwent a higher number of palatal surgeries, and the last primary palatal surgery was performed at a higher age. Children in centre 4 were also less likely to achieve ≥86% correct consonants (OR = 0.169, P = < 0.001), have no non-oral speech errors (OR = 0.347, P = < 0.001), or have competent or marginally incompetent velopharyngeal competence (OR = 0.244, P = < 0.001), compared to the average results of the other centres. No clear association between patient volume and speech outcome was observed. Conclusions: The results indicated the risk of a negative speech result if the last primary palatal surgery was performed after 25 months of age. Whether the cleft in the palate was closed in one or two stages did not affect speech outcome. The Swedish CLP registry can be used for open comparisons of treatment results to provide the basis for improvements of treatment methods. If deviating negative results are seen consistently at one centre, this information should be acted upon by further investigation and analysis, making changes to the treatment protocol as needed. Keywords: Cleft lip and palate, Registry, Surgery, Speech *Correspondence: 1 Department of Clinical Sciences in Malmö, Lund University, Malmö, Sweden Full list of author information is available at the end of the article Background In Sweden, about one in 500 children are born with cleft lip with or without cleft palate (CL/P), which on average gives 175 births annually. These children receive © The Author(s) 2022. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativeco mmons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. Klintö et al. BMC Pediatrics (2022) 22:303 treatment by a multidisciplinary team at one of six regional cleft lip and palate (CLP) centres, all connected to the Swedish quality registry for patients born with CL/P (CLP registry). The CLP registry was initiated to enable the continuous evaluation of treatment results at the Swedish CLP centres, with the objective to promote quality control, research and improvements in treatment, via the comparison of long-term results [1]. There are various procedures for primary cleft palate surgery [2]. The procedures differ regarding timing, staging and technique. Speech development benefits from early closure of the hard palate [3, 4], whereas maxillary growth may benefit from delayed closure of the palate [4, 5]. Today, two-stage palatal closure with delayed hard palate closure is used at four of six Swedish CLP centres, with the objective of promoting maxillary growth [5]. At the other two Swedish CLP centres, the palate is closed in one stage. Surgical protocols for primary palatal surgery have a high degree of diversity and poor evidence base [6]. An aggravating circumstance for research in the area is that the population with cleft palate with or without cleft lip (CP ± L) is small and heterogeneous. Therefore, it may take a long time to collect data of larger groups of children. Timing, techniques for surgery and methods and materials for data collection may then change over time and violate the standardised evaluation of treatment outcome [7]. Multi-centre studies, such as the Scandcleft randomised trials [6] and the TOPS trial [8], allow for the recruitment of larger data sets during a period of time where these variables are kept constant. However, in randomised controlled trials requiring the participating surgeons to master a new surgical technique, a learning curve could be expected, which may influence the results [9]. This raises ethical issues. Other challenges may include the recruitment of patients at CLP centres where the annual case load is low, and rules of research governance, which may increase the associated costs [9]. Four hundred and forty-eight children born with non-syndromic unilateral cleft lip and palate (UCLP) participated in the Scandcleft trials. In all trials, lip and soft palate closure at 3–4 months and hard palate closure at 12 months was the common method. In trial 1, this method was compared with lip and soft palate closure at 3–4 months and hard palate closure at 36 months, in trial 2 with lip closure at 3–4 months and hard and soft palate closure at 12 months and in trial 3 with lip and hard palate closure at 3–4 months and soft palate closure at 12 months. Speech and dentofacial development served as the primary outcomes. The only statistically verified finding in the Scandcleft trials was that delaying hard p (...truncated)