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Objective: Identification of patient factors influencing velopharyngeal function for speech following initial cleft palate repair., Design: A literature search of relevant databases from inception until 2018 was performed using medical subject headings and keywords related to cleft palate, palatoplasty and speech assessment. Following three stage screening data extraction was performed., Setting: Systematic review and meta-analysis of relevant literature., Patients/participants: Three hundred and eighty-three studies met the inclusion criteria, comprising data on 47 658 participants., Interventions: Individuals undergoing initial palatoplasty., Main Outcome Measures: Studies including participants undergoing initial cleft palate repair where the frequency of secondary speech surgery and/or velopharyngeal function for speech was recorded., Results: Patient factors reported included cleft phenotype (95% studies), biological sex (64%), syndrome diagnosis (44%), hearing loss (28%), developmental delay (16%), Robin Sequence (16%) and 22q11.2 microdeletion syndrome (11%). Meta-analysis provided strong evidence that rates of secondary surgery and velopharyngeal dysfunction varied according to cleft phenotype (Veau I best outcomes, Veau IV worst outcomes), Robin Sequence and syndrome diagnosis. There was no evidence that biological sex was associated with worse outcomes. Many studies were poor quality with minimal follow-up., Conclusions: Meta-analysis demonstrated the association of certain patient factors with speech outcome, however the quality of the evidence was low. Uniform, prospective, multi-centre documentation of preoperative characteristics and speech outcomes is required to characterise risk factors for post-palatoplasty velopharyngeal insufficiency for speech., Systematic Review Registration: Registered with PROSPERO CRD42017051624., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

To investigate the relationship between patient-related factors (sex, cleft type, cleft extent, and Robin Sequence [RS]) and speech outcome at 5 years of age for children born with a cleft palate ±  lip (CP ± L)., 3157 Children (1426 female:1731 male) with a nonsyndromic CP ± L, born between 2006 and 2014 in England, Wales, and Northern Ireland., Perceptual speech analysis utilized the Cleft Audit Protocol for Speech-Augmented (CAPS-A) rating and UK National Speech Outcome Standards: Speech Standard 1 (SS1)-speech within the normal range, SS2a-no structurally related speech difficulties or history of speech surgery, and SS3-speech without significant cleft-related articulation difficulties., Odds of achieving SS1 were lower among boys (aOR 0.771 [CI 0.660-0.901]), those with clefts involving the lip and palate (vs palate only) (UCLP-aOR 0.719 [CI 0.591-0.875]; BCLP-aOR 0.360 [CI 0.279-0.463]), and clefts involving the hard palate (incomplete-aOR 0.701 [CI 0.540-0.909]; complete-aOR 0.393 [CI 0.308-0.501]). Similar relationships with these patient factors were observed for SS3. SS2 was affected by the extent of hard palate involvement (complete; aOR 0.449 [CI 0.348-0.580]). Although those with CP and RS were less likely to meet all 3 standards than those without RS, odds ratios were not significant when adjusting for sex and cleft extent., Sex, cleft type, and extent of hard palate involvement have a significant impact on speech outcome at 5 years of age. Incorporating these factors into risk-adjustment models for service-level outcome reporting is recommended., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Objectives: (1) To explore differences in educational attainment between children born with isolated clefts and the general population at ages 5, 7 and 11 years; (2) to describe longitudinal changes in attainment among children with cleft through primary education., Design: Analysis of Cleft Registry and Audit Network data linked to national educational outcomes., Setting: English state schools., Patients: 832 children born with isolated cleft, aged 5 years in 2006-2008., Main Outcome Measures: Difference in teacher-assessed attainment between children with a cleft and general population at each age, for all children and by cleft type. Percentage of children with low attainment at age 5 years who had low attainment at age 11 years, for all children and by cleft type., Results: Children with a cleft had lower attainment than the general population in all subject areas (Z-score range: -0.29 (95% CI -0.36 to -0.22) to -0.22 (95% CI -0.29 to -0.14)). This difference remained consistent in size at all ages, and was larger among children with a cleft affecting the palate (cleft palate/cleft lip and palate (CP/CLP)) than those with a cleft lip (CL). Of 216 children with low attainment in any subject at age 5 years, 54.2% had low attainment in at least one subject at age 11 years. Compared with children with CL, those with CP/CLP were more likely to have persistent low attainment., Conclusions: An educational attainment gap for children born with isolated clefts is evident throughout primary education. Almost half of children with low attainment at age 5 years achieve normal attainment at age 11 years., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2023. No commercial re-use. See rights and permissions. Published by BMJ.)

Objective: To quantify differences in number and timing of first primary cleft lip and palate (CLP) repair procedures during the first year of the COVID-19 pandemic (1 April 2020 to 31 March 2021; 2020/2021) compared with the preceding year (1 April 2019 to 31 March 2020; 2019/2021)., Design: National observational study of administrative hospital data., Setting: National Health Service hospitals in England., Study Population: Children <5 years undergoing primary repair for an orofacial cleft Population Consensus and Surveys Classification of Interventions and Procedures-fourth revisions (OPCS-4) codes F031, F291)., Main Exposure: Procedure date (2020/2021 vs 2019/2020)., Main Outcomes: Numbers and timing (age in months) of first primary CLP procedures., Results: 1716 CLP primary repair procedures were included in the analysis. In 2020/2021, 774 CLP procedures were carried out compared with 942 in 2019/2020, a reduction of 17.8% (95% CI 9.5% to 25.4%). The reduction varied over time in 2020/2021, with no surgeries at all during the first 2 months (April and May 2020). Compared with 2019/2020, first primary lip repair procedures performed in 2020/2021 were delayed by 1.6 months on average (95% CI 0.9 to 2.2 months). Delays in primary palate repairs were smaller on average but varied across the nine geographical regions., Conclusion: There were significant reductions in the number and delays in timing of first primary CLP repair procedures in England during the first year of the pandemic, which may affect long-term outcomes., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2023. Re-use permitted under CC BY. Published by BMJ.)

Background: Although the United Kingdom (UK) cleft surgeons follow a similar training pathway, and cleft centers adhere to similar protocols regarding the timing of palate surgery and surgical technique, speech outcomes still vary between centers., Objective: To explore the training experiences of consultant cleft lip and palate (CL/P) surgeons, performing a Sommerlad radical intravelar veloplasty (IVVP) and their approach to teaching others., Design: An exploratory, qualitative approach was adopted to understand the views of UK cleft surgeons performing a Sommerlad radical IVVP and discuss what was important during training and upon qualifying as a consultant., Method: A semi-structured interview schedule was designed, interviews were conducted in-person or via videoconferencing, depending on preference and availability, with interested surgeons. The interviews were recorded, transcribed, and checked for accuracy. Analysis involved inductive thematic analysis., Results: Fourteen cleft consultants from the UK participated (3F:11M). Seven of the consultants were trained in plastic surgery and four in maxillofacial surgery. Seven themes were identified from the thematic analysis. Three themes, namely Learning to perform palate repair, Teaching others to perform palate repair, and Ongoing learning as a consultant are discussed., Conclusions: Cleft palate repair is clearly a technically challenging procedure to learn and teach with the potential to cause harm if performed incorrectly. Positive changes have been made to improve exposure to palate surgery, encourage practice away from the patient, and increase supervised practical experience. The role of colleagues in providing mentorship and support appears invaluable. We provide some simple recommendations that may improve the training experience and ensure parity for all trainees.

Background: There may be many reasons for delays to primary cleft surgery. Our aim was to investigate the age of children undergoing primary cleft lip or primary cleft palate repair in 5 cleft centers within the United Kingdom. Identify the reasons for delayed primary cleft lip repair (beyond 6 months) and delayed primary palate repair (beyond 13 months). Identify children who had a cleft lip and/or palate (CL±P) that was intentionally unrepaired and the reasons for this., Methods: A retrospective, multicenter review of patients born with a CL±P between December 1, 2012, and December 31, 2016. Three regional cleft centers, comprising of 5 cleft administrative units in the United Kingdom participated., Results: In all, 1826 patients with CL±P were identified. Of them, 120 patients had delayed lip repair, outside the expected standard of 183 days. And, 178 patients in total had delayed palate repair, outside the expected standard of 396 days. Twenty (1%) patients had an unrepaired cleft palate., Conclusions: This large retrospective review highlights variations between centers regarding the timing of lip and palate surgery and details the reasons stated for delayed primary surgery. A small number of patients with an unrepaired cleft palate were identified. All had complex medical problems or comorbidities listed as a reason for the decision not to operate and 50% had a syndromic diagnosis. The number of patients receiving delayed surgery due to comorbidities, being underweight or prematurity, highlights the importance of the cleft specialist nurse and pediatrician within the cleft multidisciplinary team.

Background: Although cleft surgeons in the United Kingdom follow a similar training pathway, and cleft centers adhere to similar protocols regarding timing of palate surgery and surgical technique, speech outcomes still vary significantly between centers., Objective: To explore if differences in technique exist between individual surgeons, performing a Sommerlad radical intravelar veloplasty (IVVP)., Design: An exploratory, qualitative approach was adopted to understand the views of UK cleft surgeons performing a Sommerlad radical IVVP for primary cleft palate repair and to discuss what was important in the adoption, adaptation, and evolution of this technique within their own practice., Method: A semistructured interview schedule was designed. Interviews were conducted in person or via videoconferencing, with interested surgeons. The interviews were recorded, transcribed, and checked for accuracy. Analysis involved inductive thematic analysis., Results: Fourteen cleft consultants from the United Kingdom participated (3 females and 11 males). Seven of the consultants were trained in plastic surgery and 4 in oral and maxillofacial surgery. Eight themes were identified from the thematic analysis. One theme-Surgical Variation-is discussed., Conclusions: The findings provide insight into areas of variation seen within one surgical technique of cleft palate repair. These variations may have arisen to accommodate heterogeneity in the patient population or may have evolved in relation to different experiences of training or influences of colleagues. Further work is needed to explore the reasons for these differences in technique and to identify if any of these subtle differences contributed to variability in outcomes.

Objective: Identification of patient factors influencing velopharyngeal function for speech following initial cleft palate repair. Design: A literature search of relevant databases from inception until 2018 was performed using medical subject headings and keywords related to cleft palate, palatoplasty and speech assessment. Following three stage screening data extraction was performed. Setting: Systematic review and meta-analysis of relevant literature. Patients/Participants: Three hundred and eighty-three studies met the inclusion criteria, comprising data on 47 658 participants. Interventions: Individuals undergoing initial palatoplasty. Main Outcome Measures: Studies including participants undergoing initial cleft palate repair where the frequency of secondary speech surgery and/or velopharyngeal function for speech was recorded. Results: Patient factors reported included cleft phenotype (95% studies), biological sex (64%), syndrome diagnosis (44%), hearing loss (28%), developmental delay (16%), Robin Sequence (16%) and 22q11.2 microdeletion syndrome (11%). Meta-analysis provided strong evidence that rates of secondary surgery and velopharyngeal dysfunction varied according to cleft phenotype (Veau I best outcomes, Veau IV worst outcomes), Robin Sequence and syndrome diagnosis. There was no evidence that biological sex was associated with worse outcomes. Many studies were poor quality with minimal follow-up. Conclusions: Meta-analysis demonstrated the association of certain patient factors with speech outcome, however the quality of the evidence was low. Uniform, prospective, multi-centre documentation of preoperative characteristics and speech outcomes is required to characterise risk factors for post-palatoplasty velopharyngeal insufficiency for speech. Systematic Review Registration: Registered with PROSPERO CRD42017051624. [ABSTRACT FROM AUTHOR]

Objective : To investigate the relationship between patient-related factors (sex, cleft type, cleft extent, and Robin Sequence [RS]) and speech outcome at 5 years of age for children born with a cleft palate ± lip (CP ± L). Participants : 3157 Children (1426 female:1731 male) with a nonsyndromic CP ± L, born between 2006 and 2014 in England, Wales, and Northern Ireland. Outcome Measure : Perceptual speech analysis utilized the Cleft Audit Protocol for Speech–Augmented (CAPS-A) rating and UK National Speech Outcome Standards: Speech Standard 1 (SS1)—speech within the normal range, SS2a—no structurally related speech difficulties or history of speech surgery, and SS3—speech without significant cleft-related articulation difficulties. Results : Odds of achieving SS1 were lower among boys (aOR 0.771 [CI 0.660-0.901]), those with clefts involving the lip and palate (vs palate only) (UCLP—aOR 0.719 [CI 0.591-0.875]; BCLP—aOR 0.360 [CI 0.279-0.463]), and clefts involving the hard palate (incomplete—aOR 0.701 [CI 0.540-0.909]; complete—aOR 0.393 [CI 0.308-0.501]). Similar relationships with these patient factors were observed for SS3. SS2 was affected by the extent of hard palate involvement (complete; aOR 0.449 [CI 0.348-0.580]). Although those with CP and RS were less likely to meet all 3 standards than those without RS, odds ratios were not significant when adjusting for sex and cleft extent. Conclusion : Sex, cleft type, and extent of hard palate involvement have a significant impact on speech outcome at 5 years of age. Incorporating these factors into risk-adjustment models for service-level outcome reporting is recommended. [ABSTRACT FROM AUTHOR]

Background: Although the United Kingdom (UK) cleft surgeons follow a similar training pathway, and cleft centers adhere to similar protocols regarding the timing of palate surgery and surgical technique, speech outcomes still vary between centers. Objective: To explore the training experiences of consultant cleft lip and palate (CL/P) surgeons, performing a Sommerlad radical intravelar veloplasty (IVVP) and their approach to teaching others. Design: An exploratory, qualitative approach was adopted to understand the views of UK cleft surgeons performing a Sommerlad radical IVVP and discuss what was important during training and upon qualifying as a consultant. Method: A semi-structured interview schedule was designed, interviews were conducted in-person or via videoconferencing, depending on preference and availability, with interested surgeons. The interviews were recorded, transcribed, and checked for accuracy. Analysis involved inductive thematic analysis. Results: Fourteen cleft consultants from the UK participated (3F:11M). Seven of the consultants were trained in plastic surgery and four in maxillofacial surgery. Seven themes were identified from the thematic analysis. Three themes, namely Learning to perform palate repair, Teaching others to perform palate repair, and Ongoing learning as a consultant are discussed. Conclusions: Cleft palate repair is clearly a technically challenging procedure to learn and teach with the potential to cause harm if performed incorrectly. Positive changes have been made to improve exposure to palate surgery, encourage practice away from the patient, and increase supervised practical experience. The role of colleagues in providing mentorship and support appears invaluable. We provide some simple recommendations that may improve the training experience and ensure parity for all trainees. [ABSTRACT FROM AUTHOR]

Background: There may be many reasons for delays to primary cleft surgery. Our aim was to investigate the age of children undergoing primary cleft lip or primary cleft palate repair in 5 cleft centers within the United Kingdom. Identify the reasons for delayed primary cleft lip repair (beyond 6 months) and delayed primary palate repair (beyond 13 months). Identify children who had a cleft lip and/or palate (CL±P) that was intentionally unrepaired and the reasons for this. Methods: A retrospective, multicenter review of patients born with a CL±P between December 1, 2012, and December 31, 2016. Three regional cleft centers, comprising of 5 cleft administrative units in the United Kingdom participated. Results: In all, 1826 patients with CL±P were identified. Of them, 120 patients had delayed lip repair, outside the expected standard of 183 days. And, 178 patients in total had delayed palate repair, outside the expected standard of 396 days. Twenty (1%) patients had an unrepaired cleft palate. Conclusions: This large retrospective review highlights variations between centers regarding the timing of lip and palate surgery and details the reasons stated for delayed primary surgery. A small number of patients with an unrepaired cleft palate were identified. All had complex medical problems or comorbidities listed as a reason for the decision not to operate and 50% had a syndromic diagnosis. The number of patients receiving delayed surgery due to comorbidities, being underweight or prematurity, highlights the importance of the cleft specialist nurse and pediatrician within the cleft multidisciplinary team. [ABSTRACT FROM AUTHOR]

Background: Although cleft surgeons in the United Kingdom follow a similar training pathway, and cleft centers adhere to similar protocols regarding timing of palate surgery and surgical technique, speech outcomes still vary significantly between centers. Objective: To explore if differences in technique exist between individual surgeons, performing a Sommerlad radical intravelar veloplasty (IVVP). Design: An exploratory, qualitative approach was adopted to understand the views of UK cleft surgeons performing a Sommerlad radical IVVP for primary cleft palate repair and to discuss what was important in the adoption, adaptation, and evolution of this technique within their own practice. Method: A semistructured interview schedule was designed. Interviews were conducted in person or via videoconferencing, with interested surgeons. The interviews were recorded, transcribed, and checked for accuracy. Analysis involved inductive thematic analysis. Results: Fourteen cleft consultants from the United Kingdom participated (3 females and 11 males). Seven of the consultants were trained in plastic surgery and 4 in oral and maxillofacial surgery. Eight themes were identified from the thematic analysis. One theme--Surgical Variation--is discussed. Conclusions: The findings provide insight into areas of variation seen within one surgical technique of cleft palate repair. These variations may have arisen to accommodate heterogeneity in the patient population or may have evolved in relation to different experiences of training or influences of colleagues. Further work is needed to explore the reasons for these differences in technique and to identify if any of these subtle differences contributed to variability in outcomes. [ABSTRACT FROM AUTHOR]

Objective: To assess the number of publications in peer-reviewed journals that are generated from the verbal presentations at the annual conference of the Craniofacial Society of Great Britain and Ireland. Design: A list of the verbal presentations (2000 to 2009) was obtained from the Craniofacial Society of Great Britain and Ireland website. Using a web-based PubMed search engine, a search was made using title, key words, and main authors. Main Outcome Measure: The primary outcome measure was the presentation's publication in a peer-reviewed journal. Secondary measures included specialty of the first author, the journal in which the article was published and its impact factor, and topic of the article. Results: Of 318 verbal presentations, 67 (21.07%) went on to be published in a peer-reviewed journal. By topic, 50.7% were surgical and 12% concerned speech. The first author was in the plastic surgical specialty in 29.9% and in either speech-language therapy or orthodontics in 17.9% each of papers. In addition, 50.7% of papers were published in the The Craniofacial-Cleft Palate Journal. The overall 2-year impact factor was 0.941. Mean lead time to publication was 29.02 months (range, 2 to 110 months). Conclusions: The publication rate is low in comparison with the rate of 44.5% given for all specialties in a Cochrane review in 2007. This may be related to the specialist nature of the subject matter or to the type of research presented at the conference and the difficulty in carrying out high-quality research on cleft lip and palate due to limited numbers and a long lead time to outcomes. [ABSTRACT FROM AUTHOR]

Individuals with orofacial clefts (OFCs) may be at an increased risk of developing autism spectrum disorder (ASD) and attention deficit hyperactivity disorder (ADHD). This systematic review provides a summary of the most recent data regarding the prevalence of ASD and ADHD in the OFC population and compares this to the general paediatric population. Multiple databases were searched including PubMed/Medline and Embase in July 2024, following the Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines and was registered in PROSPERO (CRD42024565219). 1025 papers were identified for screening, of which nine were included in the report. Percentage prevalence was calculated and compared to global prevalence or control populations where available. Overall, ASD prevalence among children with a cleft ranged from 0 to 50% (Mean = 2.87%; SD = 5.40) compared to ~ 1% globally (WHO) and ADHD prevalence ranged from 2.34 to 31.71% (Mean = 3.63%; SD = 4.30) compared to ~ 5% globally (NICE). Matched control populations showed larger differences. Isolated cleft palate was associated with higher rates than isolated cleft lip or combined cleft lip and palate. Prevalence in individuals with syndromic clefts appeared greater still (Mean = 14.80%; SD = 16.58) although populations were small. Children with OFCs demonstrate increased prevalence of ASD compared to the average paediatric population. Evidence for increased prevalence of ADHD is less clear, with varying rates across studies. Children with isolated cleft palate or cleft associated with genetic syndromes appear most at risk, although further research is needed. [ABSTRACT FROM AUTHOR]