Martin Persson

Academic achievement in individuals with cleft: a population-based register study.

Objective The focus of this study was to determine whether there were any significant differences in academic achievement between students with a cleft and the general population of Swedish students at the typical time of graduation from compulsory school (usually 16 years of age). Design A retrospective population-based study. Data were obtained from the Swedish Medical Birth Register for the years 1973 through 1986 and were linked to the Swedish School–Grade Register. Participants A total of 511 individuals with cleft palate (CP), 651 individuals with cleft lip (CL), and 830 individuals with cleft lip and palate (CLP) were compared with a control group consisting of 1,249,404 individuals. Main Outcome Measures (1) Not receiving school leaving certificate; (2) odds of receiving lowest grade and reduced odds of receiving a high grade in the following subjects: (a) Mathematics, (b) English, (c) Swedish, (d) Physical Education, and (e) grade point average (GPA). Results The group with cleft had higher odds of not receiving leaving certificates in comparison with the general population. They also had higher odds of receiving the lowest grade and/or reduced odds of receiving a high grade in the subjects analyzed together, with strong evidence of lower GPA in comparison with the general population. Individuals with CP were affected the most, followed by individuals with CLP; least affected were individuals with CL. Conclusion This study clearly indicates that adolescents with cleft lip and/or palate in Sweden experience significant deficits in their educational achievements in compulsory school.

Speech and Language Therapy Interventions for Children With Cleft Palate: A Systematic Review

Objective (1) To examine the evidence for the effectiveness of differences in timing and type of speech and language therapy for children with cleft palate with or without a cleft lip and (2) to identify types of interventions assessed. Design Nine databases, including MEDLINE and EMBASE, were searched between inception and March 2011 to identify published articles relating to speech and language therapy for children with cleft palate with or without cleft lip. Studies that included at least 10 participants and reported outcome measures for speech and/or language measures were included. Studies where the experimental group had less than 90% of children with cleft palate with or without cleft lip were excluded. Two reviewers independently completed inclusion assessment, data extraction, and risk of bias assessment for all studies identified. Results A total of 17 papers were evaluated: six randomized control trials and 11 observational studies. Studies varied widely on risk of bias, intervention used, and outcome measures reported. None of the studies had a low risk of bias. In terms of intervention approaches, seven studies evaluated linguistic approaches and 10 evaluated motor approaches. Outcomes measures did not support either approach over the other, and based on data reported it was difficult to ascertain which approach is more effective for children with cleft palate with or without cleft lip. Conclusions The review found little evidence to support any specific intervention. Key uncertainties need to be identified and adequately powered, methodologically rigorous studies conducted to provide a secure evidence base for speech-language therapy practice in children with cleft palate with or without cleft lip.

Centralization of Services for Children Born With Orofacial Clefts in the United Kingdom: A Cross-Sectional Survey

Objective To examine current provision of cleft lip and/or palate services in the U.K. and compliance with recommendations made by the Clinical Standards Advisory Group (CSAG) in 1998. Design Cross-sectional questionnaire survey. Setting All 11 services within the U.K. providing care for children born with a cleft lip and palate. Participants Members from each healthcare specialty in each U.K. cleft team. Interventions Self-administered postal questionnaires enquired about the provision of cleft services. Data were collected about the overall cleft service, team coordination, hearing, orthodontics, pediatric dentistry, primary cleft surgery, psychology, restorative dentistry, secondary surgery, specialist cleft nursing, and speech and language therapy. Results Questionnaires were returned from members of 130/150 cleft teams (87%) and these showed that U.K. cleft services have been restructured to 11 centralized services with 17 primary operative sites and 61 peripheral sites. All services provide care through a multidisciplinary (MDT) model, but the composition of each team varies. Primary cleft surgery and orthodontics were the only specialties that were represented in all cleft teams. Specialties may be represented in a team but their attendance at MDT clinics is variable. Only one team met all of the CSAG recommendations. Conclusions Our survey shows that cleft services have centralized over the last 10 years, and an MDT model of care has been adopted. Further research is needed to show how this has influenced outcomes and to see whether some models of centralized care are associated with better outcomes.

A cross-sectional survey of 5-year-old children with non-syndromic unilateral cleft lip and palate: the Cleft Care UK study. Part 1: background and methodology.

Structured Abstract Objectives We describe the methodology for a major study investigating the impact of reconfigured cleft care in the United Kingdom (UK) 15 years after an initial survey, detailed in the Clinical Standards Advisory Group (CSAG) report in 1998, had informed government recommendations on centralization. Setting and Sample Population This is a UK multicentre cross-sectional study of 5-year-olds born with non-syndromic unilateral cleft lip and palate. Children born between 1 April 2005 and 31 March 2007 were seen in cleft centre audit clinics. Materials and Methods Consent was obtained for the collection of routine clinical measures (speech recordings, hearing, photographs, models, oral health, psychosocial factors) and anthropometric measures (height, weight, head circumference). The methodology for each clinical measure followed those of the earlier survey as closely as possible. Results We identified 359 eligible children and recruited 268 (74.7%) to the study. Eleven separate records for each child were collected at the audit clinics. In total, 2666 (90.4%) were collected from a potential 2948 records. The response rates for the self-reported questionnaires, completed at home, were 52.6% for the Health and Lifestyle Questionnaire and 52.2% for the Satisfaction with Service Questionnaire. Conclusions Response rates and measures were similar to those achieved in the previous survey. There are practical, administrative and methodological challenges in repeating cross-sectional surveys 15 years apart and producing comparable data.

Cleft Care UK study. Part 5: Child psychosocial outcomes and satisfaction with cleft services

Structured Abstract Objectives To describe the impact of cleft service centralization on parental perceptions of child outcomes and satisfaction with care from the Cleft Care UK (CCUK) study and compare them to the Clinical Standards Advisory Group (CSAG) study that took place 15 years earlier. Setting and Sample Population A subgroup of respondents from a UK multicentre cross-sectional study (CCUK) of 5-year-olds born with non-syndromic unilateral cleft lip and palate. Materials and Methods Data on parents’ perceptions of child self-confidence and their satisfaction with treatment outcomes and service provision were collected via self-report questionnaires. Data were compared with findings from the 1998 CSAG study. Results Fewer parents in the CCUK study perceived their children as having poor self-confidence than in the 1998 CSAG study (8 and 19%, respectively). At least 81% of parents report satisfaction with the child’s facial features after surgery and 98% report being satisfied with the care received. These results are similar to those reported in 1998. There is no evidence of an adverse impact on families’ ability to attend appointments at the cleft clinic following centralization. Levels of reported problems (around 30%) with attendance were similar to those reported by CSAG. Conclusion Centralization of cleft services appears to have improved parental perceptions of some child outcomes but has made little difference to already high levels of parental satisfaction with cleft care services. Centralization is not associated with an increase in the proportion of families who find it difficult to attend appointments.

The Effectiveness of Psychosocial Intervention for Individuals with Cleft Lip and/or Palate:

Objective The aim of this review was to assess the effectiveness of different psychological interventions for children and adults with cleft lip and/or palate and their parents. Design We searched six databases including MEDLINE and EMBASE to June 2013 and checked bibliographies. We included research that evaluated any psychological intervention in studies in which at least 90% of the participants had cleft lip and/or palate or were parents of those with cleft lip and/or palate. Studies containing less than 90% were excluded unless they reported results separately for those with cleft lip and/or palate, or raw data were available upon request from the authors. Inclusion assessment, data extraction, and risk of bias assessment were carried out independently by two reviewers. Results Seven studies were identified as inclusions, with only two studies being included in the full data analysis (one of which failed to meet the full inclusion criteria). The five remaining studies were included only in a narrative synthesis because data were available for people or parents of those with cleft lip and/or palate only. This highlights a distinct dearth of research into psychological intervention within the field of cleft lip and/or palate. Conclusions The review found no evidence to support any specific intervention. Key uncertainties need to be identified and addressed. Adequately powered, methodologically rigorous randomized controlled trials are needed to provide a secure evidence base for psychological intervention techniques in participants with cleft lip and/or palate and their parents.

Using service rationalisation to build a research network: lessons from the centralisation of UK services for children with cleft lip and palate

In the UK around a thousand children are born annually with a cleft lip and/or palate that requires treatment. In the last decade services have been centralised in the UK reducing the 57 centres operating on these children in 1998, down to 11 centres or managed clinical networks in 2011. While the rationale for centralisation was to improve the standard of care (and in so doing the outcome) for children born with cleft lip and/or palate, research was central to this process. We illustrate how research informed and shaped this service rationalisation and how it facilitated the emergence of a research culture within the newly configured teams. We also describe how these changes in service provision were linked to the development of a national research strategy and to the identification of the resources necessary to support this strategy.

Why are multi-centre clinical observational studies still so difficult to run?

Despite the introduction of IRAS, obtaining R&D approvals for multicentre studies is a major rate limiting step. Our experience with cleft research demonstrates that this approvals process must be drastically and rapidly improved if we wish to continue attract clinical research to the UK.

An Evaluation of Current Services Available for People Diagnosed with Head and Neck Cancer in the UK (2009–2010)

AIMS To evaluate current care and service provision for people with head and neck cancer in the UK. MATERIALS AND METHODS Self-report questionnaires for cancer networks, clinical leads of oncology units and leads for multidisciplinary teams (MDTs) were designed. These questionnaires were based on a previous survey. Questionnaires were sent out between 2009 and 2010. RESULTS Questionnaires were received from all networks (n = 37), most oncology units (48 of 53) and most MDTs (51 of 63). Care for people with head and neck cancer is increasingly being provided by a centralised MDT. The membership of these teams varies; facilities available for team meetings are fit for purpose in most cases. MDTs are meeting frequently (weekly meetings in 96%) and discussing on average 18 cases at each meeting (95% confidence interval 15-21 cases). Most oncologists have access to all common anti-cancer drugs and most have access to all forms of radiotherapy. Intensity-modulated radiotherapy is not yet available in some oncology units (28%). A small number of units have only one oncologist (13%). Despite audit and research being part of the rationale for MDT working, regular discussion of morbidity and mortality is unusual (40%) and use of a database to record decisions is not universal. Only seven centres record decisions into the Data for Head and Neck Oncology database. Reported recruitment to studies is generally low (<2% of cases enrolled in studies in 62%). CONCLUSIONS Head and neck cancer care is increasingly provided through a centralised MDT. Increased resources and further changes in practice are required to implement current National Health Service cancer policy. Teams need to improve recording of their decision-making, discuss morbidity and mortality and support recruitment to clinical studies.

Educational achievements in Pierre Robin Sequence

Abstract The aim of this retrospective population-based study was to determine if there is any significant difference in academic achievement between students with a Pierre Robin Sequence (PRS) compared with the general population of Swedish students at the typical time of graduation from compulsory school (usually at 16 years of age). The data were obtained from the Swedish Medical Birth Register for the years 1973–1986 and linked to the Swedish School-Grade Register, which generated a data set of 68 individuals with PRS that were compared with a control group consisting of 1,249,404 individuals. The following outcomes were measured: not receiving a school leaving certificate; their odds of receiving lowest grade; and reduced odds of receiving high grade in the following subjects: mathematics; English; Swedish; and physical education, and their grade point average. For the control group 2.74% did not receive their leaving certificate, while for the individuals with PRS 9.68% (OR = 4.00; 95% CI = 1.81–8.86) did not receive their leaving certificate. There were no differences between the groups in mathematics, English, and Swedish, while, in physical education, individuals with PRS had significantly reduced odds of receiving a high grade in the relative grading system. Individuals with PRS had a significantly lower grade point average: 3.00 ± 0.09 in comparison to the control group: 3.23 ± 0.002, p = 0.002. This study indicates that individuals with PRS experience significant difficulties in their educational achievements in compulsory school in Sweden.