Analysis and Reporting of Randomized Trials in Cleft Palate Surgery: Learning from the Timing of Primary Surgery (TOPS) Trial.

The Timing of Primary Surgery (TOPS) trial was published August 2023 in the New England Journal of Medicine and is a milestone achievement for a study focused on cleft palate. Due to the complexity of outcome reporting in cleft and the rarity of such comparative trials, TOPS presents a useful opportunity to critically review the design, analysis and reporting strategies utilised. This perspective article focused on the inclusion of participants, the choice of the primary outcome measure and the analysis of ordinal data within the trial. Considerations for future comparative studies in cleft care are discussed.

Identifying associations between dental arch relationship scores, relative deprivation and other cleft audit outcomes. Part 1.

OBJECTIVES: To assess whether dental arch relationship scores are associated with speech, audiological and psychology outcomes in patients with complete unilateral cleft lip and palate (UCLP) and bilateral cleft lip and palate (BCLP). SETTING AND SAMPLE POPULATION: One hundred 5-year-old children with non-syndromic complete UCLP and forty-two 5-year-olds with non-syndromic complete BCLP from the United Kingdom. MATERIAL AND METHODS: Data were collected from five-year audit clinics for dental arch relationships, speech, hearing and psychology outcomes. Associations were assessed between ordinal prognostic covariates and the dental arch relationship outcome using a Cochrane-Armitage test for trend and comparisons with binary prognostic covariates using chi-square/Fisher's exact test. This was extended to assess associations between five-year audit outcomes and a measure of deprivation. RESULTS: There was no evidence of associations between dental arch relationships and outcomes for audiology and speech. Individual domain analysis of the psychology outcomes within the UCLP cohort reported a poor outcome for Pro-social score to be associated with a poor 5-year-old index score (P = .018). For the BCLP sample, a good outcome for the BCLP Deciduous Dentition Yardstick was associated with a good conduct score (P = .01). CONCLUSION: There was no association with the 5-year-old index/BCLP Deciduous Dentition Yardstick score and outcomes reported for audiology and speech. For the UCLP and BCLP cohorts, a poor dental arch relationship outcome may be associated with issues with some aspects of psychological well-being, associated with peer interactions and conduct. The deprivation index of samples had no association with the outcomes assessed.

Identifying associations between dental arch relationship scores, relative deprivation and other cleft audit outcomes. Part 2.

Identify associations between dental arch relationship scores, oral health status and deprivation index in patients with complete unilateral cleft lip and palate (UCLP) and bilateral cleft lip and palate (BCLP). Ninety-two 5-year-old children with non-syndromic complete UCLP and thirty-nine 5-year-old children with non-syndromic complete BCLP from the United Kingdom. Data were collected from the 5-year audit outcomes submitted to the Cleft Registry and Audit Network (CRANE). The index of multiple deprivation (IMD) and Welsh index of deprivation were used to assess a relative measure of deprivation. Comparisons of 5-year-old index/BCLP Deciduous Dentition Yardstick outcome against IMD and dmft are performed using multivariable linear regression models. Both UCLP and BCLP had a high percentage of children with dmft >0 (47% and 49%, respectively). The mean dmft for the UCLP cohort was 2.8 and 2.6 for the BCLP cohort. In the UCLP group, a poorer 5-year-old index was associated with an increased dmft score (P = .023) and higher level of deprivation (P = .010). In the BCLP group, there was no significant associations between BCLP Deciduous Dentition Yardstick, dmft and IMD. A poorer dental arch relationship outcome may be associated with higher level of area deprivation and oral health status, in children with UCLP. Those with a poor outcome for the 5-year-old Index are more likely to have increased caries experience.

A Closer Look at Delayed Primary Cleft Surgery and Unrepaired Cleft Lip and/or Palate in 5 UK Cleft Centers.

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.

A Standard Set of Outcome Measures for the Comprehensive Appraisal of Cleft Care.

Care of the patient with cleft lip and/or palate remains complex. Prior attempts at aggregating data to study the effectiveness of specific interventions or overall treatment protocols have been hindered by a lack of data standards. There exists a critical need to better define the outcomes-particularly those that matter most to patients and their families-and to standardize the methods by which these outcomes will be measured. This report summarizes the recommendations of an international, multidisciplinary working group with regard to which outcomes a typical cleft team could track, how those outcomes could be measured and recorded, and what strategies may be employed to sustainably implement a system for prospective data collection. It is only by agreeing on a common, standard set of outcome measures for the comprehensive appraisal of cleft care that intercenter comparisons can become possible. This is important for quality-improvement endeavors, comparative effectiveness research, and value-based health-care reform.

Are NHS cleft services in England ready for delegation to integrated care systems?

Cleft care services in the UK have been nationally funded since centralisation 25 years ago and during this time have been able to demonstrate improved clinical outcomes. Integrated care systems have been introduced into legislature as part of the Health Care Act of 2022 and will be responsible for the paradigm shift of allocating funds on a regional basis for cleft care services in England from 2024. The proposed population-based funding formulas present an opportunity to improve current inequities in cleft care, including access to speech therapy and adult services. However, the regional footprint of integrated care systems does not align with that of the centralised cleft service system and represents a threat to the standardised patient-centred care that has taken two decades to build. Awareness needs to be raised so that cleft care providers can proactively adapt to this mandatory change to service funding to ensure that clinical standards are maintained and continue to improve.

The role of primary surgery in the management of orofacial clefting.

Primary cleft surgery refers to the planned surgical procedure(s) necessary to reconstruct an orofacial defect according to a specified protocol; in the context of a unilateral cleft lip and palate, this would include lip repair (with vomer flap closure of the hard palate), subsequent repair of the residual soft palate cleft and finally, the alveolar bone graft. This paper will provide an overview of the goals of primary surgical reconstruction, including the utility of pre-surgical orthopaedic techniques, including nasoalveolar moulding. The surgical methods of primary lip repair in both unilateral and bilateral clefts will be outlined and the concept of a primary rhinoplasty will be introduced. The principal techniques of cleft palate reconstruction will be delineated for a variety of cleft phenotypes. The final element of the primary reconstructive sequence is the alveolar bone graft using cancellous bone harvested from the iliac crest at approximately 8-10 years of age. The role of optimising oral hygiene prior to bone grafting cannot be over-emphasised and the utility of pre-surgical orthodontic arch expansion is also discussed. In the UK, bone graft outcomes are audited using the Kindelan score, which is evaluated from the six-month postoperative upper standard occlusal radiograph.

Secondary surgery techniques to optimise functional and aesthetic outcomes in orofacial clefting.

Although the goal of primary surgery is to avoid the need for secondary surgery, in a proportion of patients this is not always possible. Secondary, or revisional, surgery is frequently undertaken in patients with orofacial clefts and may present a complex and challenging problem for the multidisciplinary team. There are a broad range of functional and aesthetic issues that secondary surgery might aim to address. These include palatal fistulae (that may be symptomatic for air, fluid or food), velopharyngeal insufficiency (typically manifesting with decreased intelligibility or nasal regurgitation), suboptimal cleft lip scars (that can significantly impact upon the psychosocial wellbeing of the patient), and nasal asymmetry (that is frequently accompanied by nasal airway concerns). Unilateral and bilateral clefts are each associated with a characteristic nasal deformity that requires a tailored surgical approach. Suboptimal maxillary growth in repaired orofacial clefts may impact negatively on both appearance and function; surgical correction through orthognathic surgery can be transformational for the patient. The general dental practitioner, cleft orthodontist and restorative dentist all play a critical role in this process.

Non-surgical management of paediatric and adolescent mandibular condyles: A retrospective review of 49 consecutive cases treated at a tertiary referral centre.

INTRODUCTION: Mandibular fractures account for over 50% of all facial fractures in children, with the majority of these occurring at the condyle. There is currently no consensus on the management of mandibular condyle fractures in the paediatric population. Treatment options range from conservative management, with or without maxillomandibular fixation (MMF) and physiotherapy, to open reduction and internal fixation (ORIF). METHODS: This was a retrospective review of all patients who attended Alder Hey Children's Hospital with fractured condyle(s) between the years 2000 and 2015. All patients were managed non-surgically and included those managed conservatively and/or with MMF. The following variables were recorded: age, sex, mechanism of injury, concomitant mandibular fractures, imaging, SORG classification, complications and follow-up intervals. The following exclusion criteria applied: patients who underwent open reduction internal fixation of their condylar fracture and patients aged 16 years or over. RESULTS: Forty-nine patients (38 male, 11 female) underwent non-surgical management of condylar fractures during the 15-year study period. The mean age at time of injury was 12 years (range 2-15 years). The etiology of fractures comprised mechanical falls (n = 22), assault (n = 14), sport (n = 5), road traffic accident (n = 3), epileptic fit (n = 1), and unknown mechanism (n = 4). The mean length of hospital stay was 3 days, ranging from 0 to 14 days. Thirty-seven patients had a concomitant mandibular fracture and 12 had an isolated unilateral condylar fracture. Follow-up intervals ranged from 1 to 133 weeks, with a median length of 12 weeks. Fifteen patients underwent MMF, while 34 had soft diet and physiotherapy only. Ninety-two per cent of patients had no complications. No patient had failure of treatment requiring a return to theatre and/or ORIF. Occlusion, interincisal distance, asymmetry and TMJ symptoms were examined in 43/49 patients who attended their postoperative appointments. Four patients (8%) had complications in the postoperative period, namely: lateral open bite (n = 2), deviation to the affected side on wide opening (n = 1), and non-specific TMJ dysfunction (n = 1). None of these patients presented with any subjective symptoms and none developed asymmetry or needed any further surgical intervention. CONCLUSION: Paediatric and adolescent mandibular condyle fractures can be safely and predictably managed using conservative methods. In our, albeit small, study cohort all patients were managed conservatively, and all had a satisfactory outcome with no requirement for further operative intervention.

Cleft lip and palate: diagnosis and management.

Cleft lip and palate is the most common congenital facial anomaly in children, which can affect appearance, speech, hearing, growth, psychosocial wellbeing and social integration. This article provides an overview of the condition for the benefit of all health-care professionals.