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.

"It's On Your Shoulders Now" Transitioning from Child-to-Adult UK Cleft Lip/Palate Services: An Exploration of Young Adults' Narratives.

Objectives: Treatment for cleft lip and/or palate (CL/P) in the United Kingdom is administered on a standardised pathway from diagnosis to early adulthood, with options to be re-referred in later life. At age 16, patients become responsible for their treatment decisions. Evidence from the wider health literature indicates this transition can be challenging and that this population may require additional support.

Enhancing the vermilion in adult secondary cleft lip repair with a continuous V plasty without closure of the donor defect-a case series.

The aim of this paper was to describe a modification to an old method to enhance the vermillion in adult cleft patients. We present ten consecutive patients who requested enhancement of the upper lip vermillion. The technique involves a continuous V plasty within the non-visible mucosa to elevate the vermillion.

An essential overview of orofacial clefting.

An appreciation of the embryologic development of the face is essential to understanding the anatomic variation observed in this phenotypically broad condition. Embryologically, the nose, lip and palate are separated into the primary and secondary palate, which are divided anatomically by the incisive foramen. The epidemiology of orofacial clefting is reviewed together with the contemporaneous cleft classification systems that enable comparisons to be made between international centres for audit and research purposes.

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.

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 Development, Deployment, and Evaluation of the CLEFT-Q Computerized Adaptive Test: A Multimethods Approach Contributing to Personalized, Person-Centered Health Assessments in Plastic Surgery.

Background: Routine use of patient-reported outcome measures (PROMs) and computerized adaptive tests (CATs) may improve care in a range of surgical conditions. However, most available CATs are neither condition-specific nor coproduced with patients and lack clinically relevant score interpretation. Recently, a PROM called the CLEFT-Q has been developed for use in the treatment of cleft lip or palate (CL/P), but the assessment burden may be limiting its uptake into clinical practice.

Reflective Foreign Bodies Mimicking Fistulae of the Hard Palate in the Paediatric Population.

Palatal foreign bodies remain relatively rare, consequently, delays in diagnosis and misdiagnosis can occur leading to unnecessary anxiety and invasive investigations. We report three children with a reflective disc from inside a confetti balloon masquerading as a fistula of the hard palate. Awareness of this foreign body phenomenon enabled timely diagnosis in subsequent patients; hence the need to highlight such cases to the global cleft community.

Transitioning from child to adult cleft lip and palate services in the United Kingdom: Are the NICE Guidelines reflected in young adults' experiences?

Cleft lip and/or palate (CL/P) is one of the most common congenital conditions worldwide. Individuals born with CL/P will embark on a long-term treatment pathway throughout childhood and often into adulthood. As they grow older, young people become more involved in medical decisions.

The Impact of the Covid-19 Pandemic on Cleft Lip and Palate Service Delivery for New Families in the United Kingdom: Medical and Community Service Provider Perspectives.

Objectives: Professionals in the United Kingdom providing care to new families affected by cleft lip and/or palate (CL/P) had to adapt to ensure families' needs were met during a time of uncertainty due to Covid-19. The aims of this study were to explore the impacts of the pandemic on CL/P care provision for new families from the perspectives of professionals working in medical and community settings along with any personal impact on professionals and their reflections on the future of CL/P care.

Design: Semistructured interviews (n  =  27) were completed about experiences from March 2020 to October 2020 with consultant cleft surgeons (n  =  15), lead clinical nurse specialists (n  =  8), and staff working at the Cleft Lip and Palate Association (n  =  4).

Psychometric Validation of the CLEFT-Q Patient Reported Outcome Measure: A Prospective Study to Examine Cross-Sectional Construct Validity.

Objective: CLEFT-Q is a condition-specific patient-reported outcome measure (PROM) for patients with cleft lip and/or palate (CL/P). The aim of this study was to examine the cross-sectional construct validity of the CLEFT-Q scales.

Design: Construct validity was assessed through a prospective study that tested hypotheses regarding correlations of scores with other PROMs that measure related constructs.

Isolation, Uncertainty and Treatment Delays: Parents' Experiences of Having a Baby with Cleft Lip/Palate During the Covid-19 Pandemic.

Objectives: Previous literature finds that having a child with a cleft lip and/or palate (CL/P) may pose social and emotional challenges for parents. For parents of children born during the Covid-19 pandemic, such challenges may be heightened. Further, novel demands brought about by the pandemic could have caused additional hardships.

Deeper Understanding of Appearance in Orofacial Clefts: A Structural Equation Model of the CLEFT-Q Appearance Scales.

Background: The CLEFT-Q is a patient-reported outcome measure with seven scales measuring elements of facial appearance in cleft lip and/or palate. We built on the validated CLEFT-Q structural model to describe conceptual relationships between these scales, and tested our hypothesis through structural equation modeling (SEM). In our hypothesized model, the appearance of the nose, nostrils, teeth, jaw, lips, and cleft lip scar all contribute to overall facial appearance.

Recursive Partitioning vs Computerized Adaptive Testing to Reduce the Burden of Health Assessments in Cleft Lip and/or Palate: Comparative Simulation Study.

Background: Computerized adaptive testing (CAT) has been shown to deliver short, accurate, and personalized versions of the CLEFT-Q patient-reported outcome measure for children and young adults born with a cleft lip and/or palate. Decision trees may integrate clinician-reported data (eg, age, gender, cleft type, and planned treatments) to make these assessments even shorter and more accurate.

Objective: We aimed to create decision tree models incorporating clinician-reported data into adaptive CLEFT-Q assessments and compare their accuracy to traditional CAT models.

Hearing, Speech, Language, and Communicative Participation in Patients With Apert Syndrome: Analysis of Correlation With Fibroblast Growth Factor Receptor 2 Mutation.

Unlabelled: Apert syndrome (AS) is caused by the heterozygous presence of 1 of 2 specific missense mutations of the fibroblast growth factor receptor 2 (FGFR2) gene. The 2 adjacent substitutions, designated p.Ser252Trp (S252W) and p.

FACE-Q Craniofacial Module: Part 1 validation of CLEFT-Q scales for use in children and young adults with facial conditions.

Background: The CLEFT-Q includes 12 independently functioning scales that measure appearance (face, nose, nostrils, teeth, lips, jaws), health-related quality of life (psychological, social, school, speech distress), and speech function, and an eating/drinking checklist. Previous qualitative research revealed that the CLEFT-Q has content validity in noncleft craniofacial conditions. This study aimed to examine the psychometric performance of the CLEFT-Q in an international sample of patients with a broad range of facial conditions.

The Throat Pack Debate: A Review of Current Practice in UK and Ireland Cleft Centers.

Introduction: The use of throat packs during oropharyngeal surgery has long been a topic of debate among cleft surgeons. The advantage of inserting an absorbent tulle within the pharynx must be weighed against the risk of unintended retention postoperatively. Despite safety check mechanisms in place, retention may occur with potentially life-threatening consequences.

Velopharyngeal Incompetence in Children With 22q11.2 Deletion Syndrome: Velar and Pharyngeal Dimensions.

The surgical management of velopharyngeal incompetence (VPI) in children with 22q11.2 deletion syndrome (22q11.2 DS) is challenging.

Optimising the computerised adaptive test to reliably reduce the burden of administering the CLEFT-Q: A Monte Carlo simulation study.

Background: Computerised adaptive testing (CAT) has the potential to transform plastic surgery outcome measurement by making patient-reported outcome measures (PROMs) shorter, individualised and more accurate than pen-and-paper questionnaires.

Objectives: This paper reports the results of two optimisation studies for the CLEFT-Q CAT, a CAT intended for use in the field of cleft lip and/or palate. Specifically, we aimed to identify the optimal score estimation and item selection methods for using this CAT in clinical practice.

Response to Achieving Consensus in the Measurement of Psychological Adjustment to Cleft Lip and/or Palate at Age 8+ Years.

In this letter, we discuss the recently published paper by Stock et al, entitled Achieving Consensus in the Measurement of Psychological Adjustment to Cleft Lip and/or Palate at Age 8+ Years.

Further construct validation of the CLEFT-Q: Ability to detect differences in outcome for four cleft-specific surgeries.

Background: The CLEFT-Q is a patient-reported outcome measure developed for use in patients with cleft lip and/or palate. A significant indicator of the CLEFT-Q's validity relates to its ability to detect differences between the impact of specific aspects of clefting before and after surgery. This study compares relevant sub-scale scores of the CLEFT-Q for patients requiring four specific surgical treatments against those who either have had surgery or never needed surgery.

Computerised adaptive testing accurately predicts CLEFT-Q scores by selecting fewer, more patient-focused questions.

Background: The International Consortium for Health Outcome Measurement (ICHOM) has recently agreed upon a core outcome set for the comprehensive appraisal of cleft care, which puts a greater emphasis on patient-reported outcome measures (PROMs) and, in particular, the CLEFT-Q. The CLEFT-Q comprises 12 scales with a total of 110 items, aimed to be answered by children as young as 8 years old.

Objective: In this study, we aimed to use computerised adaptive testing (CAT) to reduce the number of items needed to predict results for each CLEFT-Q scale.

CLEFT-Q: Detecting Differences in Outcomes among 2434 Patients with Varying Cleft Types.

Background: Measuring the patient perspective is important in evaluating outcomes of cleft care. Understanding how treatment outcomes vary depending on cleft type may allow for better planning of treatments, setting of expectations, and more accurate benchmarking efforts. The CLEFT-Q is a patient-reported outcome measure for patients with cleft lip and/or palate.

Use of the Sphenoid Flap in Repair of the Wide Cleft Palate.

Objective: In wide palatal defects, closure of the nasal layer can prove a considerable challenge. Mobilizing nasal flaps posteriorly usually facilitates soft palate closure. However, the defect is often too wide within the hard palate; hence, bilateral vomerine flaps are frequently required.