Decision Considerations and Strategies for Lip Surgery in Patients with Cleft lip/Palate: A Qualitative Study.

Objective: To qualitatively assess surgeons decision making for lip surgery in patients with cleft lip/palate (CL/P). Design: Prospective, non-randomized, clinical trial. Setting: Clinical data institutional laboratory setting. Patients Participants: The study included both patient and surgeon participants recruited from four craniofacial centers. The patient participants were babies with a CL/P requiring primary lip repair surgery (n=16) and adolescents with repaired CL/P who may require secondary lip revision surgery (n=32). The surgeon participants (n=8) were experienced in cleft care. Facial imaging data that included 2D images, 3D images, videos, and objective 3D visual modelling of facial movements were collected from each patient, and compiled as a collage termed the Standardized Assessment for Facial Surgery (SAFS) for systematic viewing by the surgeons. Interventions: The SAFS served as the intervention. Each surgeon viewed the SAFS for six distinct patients (two babies and four adolescents) and provided a list of surgical problems and goals. Then an in-depth-interview (IDI) was conducted with each surgeon to explore their decision-making processes. IDIs were conducted either in person or virtually, recorded, and then transcribed for qualitative statistical analyses using the Grounded Theory Method. Results: Rich narratives/themes emerged that included timing of the surgery; risks/limitations and benefits of surgery; patient/family goals; planning for muscle repair and scarring; multiplicity of surgeries and their impact; and availability of resources. For diagnoses/treatments, surgeons agreed, and level of surgical experience was not a factor. Conclusions: The themes provided important information to populate a checklist of considerations to serve as a guide for clinicians.

Decision Considerations and Strategies for Lip Surgery in Patients with Cleft lip/Palate: A Qualitative Study.

Objective: To qualitatively assess surgeons' decision making for lip surgery in patients with cleft lip/palate (CL/P). Design: Prospective, non-randomized, clinical trial. Setting: Clinical data institutional laboratory setting. Patients Participants: The study included both patient and surgeon participants recruited from four craniofacial centers. The patient participants were babies with a CL/P requiring primary lip repair surgery (n=16) and adolescents with repaired CL/P who may require secondary lip revision surgery (n=32). The surgeon participants (n=8) were experienced in cleft care. Facial imaging data that included 2D images, 3D images, videos, and objective 3D visual modelling of facial movements were collected from each patient, and compiled as a collage termed the 'Standardized Assessment for Facial Surgery (SAFS)' for systematic viewing by the surgeons. Interventions: The SAFS served as the intervention. Each surgeon viewed the SAFS for six distinct patients (two babies and four adolescents) and provided a list of surgical problems and goals. Then an in-depth-interview (IDI) was conducted with each surgeon to explore their decision-making processes. IDIs were conducted either 'in person' or virtually, recorded, and then transcribed for qualitative statistical analyses using the Grounded Theory Method. Results: Rich narratives/themes emerged that included timing of the surgery; risks/limitations and benefits of surgery; patient/family goals; planning for muscle repair and scarring; multiplicity of surgeries and their impact; and availability of resources. In general, there was surgeon agreement for the diagnoses/treatments. Conclusions: The themes provided important information to populate a checklist of considerations to serve as a guide for clinicians.

Orthodontic Preparation for Secondary Alveolar Bone Grafting in Patients with Complete Cleft Lip and Palate.

This article provides an overview of the orthodontic preparation prior to secondary alveolar bone grafting of alveolar defects in those with complete cleft lip and palate. Use of cone beam computed tomography in diagnosis and treatment planning for addressing alveolar clefts, the rationale for maxillary expansion prior to alveolar bone grafting, key steps in differential maxillary expansion, potential adverse effects, and outcomes associated with maxillary expansion are provided in this overview.

An Overview of Craniosynostosis Craniofacial Syndromes for Combined Orthodontic and Surgical Management.

This article provides an overview of epidemiology, genetics, and common orofacial features of those with craniosynostosis. Patients with craniosynostosis require several surgical procedures along with continuum of care. The earliest surgical interventions are done during the first few years of life to relieve the fused sutures. Midface advancement, limited phase of orthodontic treatment, and combined orthodontics/orthognathic surgery treatment are usually required during later years. This article presents several examples of cases with outcomes associated with these procedures.

Etiology of Cleft Lip Lower Lip Deformity: Use of an Objective Analysis to Measure Severity.

OBJECTIVE: Measure lower lip thickness and eversion in patients with cleft lip and palate (CLP) and maxillary hypoplasia. The specific aims were to (1) compare lower lip thickness/eversion in patients with CLP to noncleft controls with maxillary hypoplasia, (2) determine differences between patients with unilateral CLP (UCLP) and bilateral CLP (BCLP), and (3) document changes in the lower lip that occur with Le Fort I advancement. DESIGN: Retrospective case-control study. SETTING: Tertiary care center. PATIENTS/PARTICIPANTS: Patients with available pre- and postoperative CT scans and 2D lateral photographs who had a Le Fort I advancement between 2009 and 2017. There were 32 patients with CLP (17 females; mean age 17.7 ± 1.9 years) and 33 noncleft controls (21 females; mean age 18.8 ± 2.6 years). MAIN OUTCOME MEASURES: Lower lip thickness and eversion. RESULTS: Patients with CLP and maxillary hypoplasia have a significantly thicker lower lip (P = .019) and outward rotation of the vermilion border (P = .003) compared to noncleft controls. The lower lip was significantly thicker in patients with BCLP than in those with UCLP (P = .035). Lower lip thickness and rotation did not change after maxillary advancement. CONCLUSIONS: Patients with CLP and maxillary hypoplasia have a thicker and more everted lower lip than noncleft controls. Patients with BCLP have a significantly thicker lower lip than those with UCLP. Strain of the lower lip musculature appears to be an important contributor to the development of the cleft lip lower lip deformity.

A predictive model for obstructive sleep apnea and Down syndrome.

Obstructive sleep apnea (OSA) occurs frequently in people with Down syndrome (DS) with reported prevalences ranging between 55% and 97%, compared to 1-4% in the neurotypical pediatric population. Sleep studies are often uncomfortable, costly, and poorly tolerated by individuals with DS. The objective of this study was to construct a tool to identify individuals with DS unlikely to have moderate or severe sleep OSA and in whom sleep studies might offer little benefit. An observational, prospective cohort study was performed in an outpatient clinic and overnight sleep study center with 130 DS patients, ages 3-24 years. Exclusion criteria included previous adenoid and/or tonsil removal, a sleep study within the past 6 months, or being treated for apnea with continuous positive airway pressure. This study involved a physical examination/medical history, lateral cephalogram, 3D photograph, validated sleep questionnaires, an overnight polysomnogram, and urine samples. The main outcome measure was the apnea-hypopnea index. Using a Logic Learning Machine, the best model had a cross-validated negative predictive value of 73% for mild obstructive sleep apnea and 90% for moderate or severe obstructive sleep apnea; positive predictive values were 55% and 25%, respectively. The model included variables from survey questions, medication history, anthropometric measurements, vital signs, patient's age, and physical examination findings. With simple procedures that can be collected at minimal cost, the proposed model could predict which patients with DS were unlikely to have moderate to severe obstructive sleep apnea and thus may not need a diagnostic sleep study.

Clinician's Primer to ICD-10-CM Coding for Cleft Lip/Palate Care.

On October 1, 2015, the United States required use of the Clinical Modification of the International Classification of Diseases, 10th Revision (ICD-10-CM) for diagnostic coding. This primer was written to assist the cleft care community with understanding and use of ICD-10-CM for diagnostic coding related to cleft lip and/or palate (CL/P).

Effect of infant surgical orthopedic treatment on facial growth in preadolescent children with unilateral and bilateral complete cleft lip and palate.

OBJECTIVE: To examine the impact of dentofacial infant orthopedic treatment (DFIO) on facial growth in preadolescent children with unilateral complete cleft lip and palate (UCCLP) and bilateral complete cleft lip and palate (BCCLP). METHODS: This is a retrospective study of patients with UCCLP and BCCLP treated at a single center. The treatment group had DFIO, and the control group did not have DFIO. Regression models were used to compare outcomes between the study and control groups. RESULTS: The study sample comprised 81 patients (54 had DFIO and 27 did not have DFIO). Among those with UCCLP, those who had DFIO had a shorter maxillary length (-2.12 mm; P = .04) and shorter lower anterior facial height (-2.77 mm; P = .04) compared with controls. Among those with BCCLP, there were no significant differences between the treatment and control groups. CONCLUSIONS: DFIO treatment could result in shorter maxillary length and lower anterior facial height in those with UCCLP.

Operative and Immediate Postoperative Outcomes of Using a Latham-Type Dentomaxillary Appliance in Patients With Unilateral Complete Cleft Lip and Palate.

OBJECTIVE: The objective of this study is to examine the operative and immediate postoperative effects of the use of a dentomaxillary appliance (DMA). DESIGN: Retrospective chart review of 40 treated patients with unilateral complete cleft lip and palate who had a comprehensive set of pre-, peri-, and postoperative records. SETTING: Boston Children's Hospital. PATIENTS: Forty treated patients with unilateral complete cleft lip and palate. INTERVENTIONS: Use of DMA. MAIN OUTCOME MEASURES: Outcome variables of interest included cleft lip width reduction following use of DMA and odds of having primary gingivoperiostetoplasty (GPP). RESULTS: The study sample included 40 subjects (31 boys and 9 girls). The average age at the time of DMA insertion was 11 weeks. The average width of the alveolar cleft prior to DMA insertion was 10.77 mm. The mean cleft width reduction was 8.66 mm. Each 1-mm increase in pre-DMA cleft width was associated with a 0.631-mm reduction in cleft width (P < .001). Thirty-three patients (82.5%) had a GPP procedure. Each 1-mm increase in post-DMA width was associated with a lower odds of having a GPP (odds ratio = 0.32, 95% confidence interval = 0.14-0.77, P = .01). CONCLUSIONS: Post-DMA width was the significant factor associated with the performance of GPP. The use of DMA is associated with a significant reduction in the width of the cleft, and outcomes are predictable without any major adverse events or complications.

Change in Mandibular Position in Patients With Syndromic Craniosynostosis After Midfacial Advancement With Distraction Osteogenesis.

OBJECTIVE: Characterize mandibular morphology in patients with syndromic craniosynostosis and document changes in mandibular position following midfacial advancement using distraction osteogenesis (DO). DESIGN: Retrospective chart review and analysis of cephalometric radiographs. SETTING: Tertiary care center. PATIENTS: Patients with syndromic craniosynostosis who had midfacial advancement with DO at Boston Children's Hospital between 2000 and 2012. Mandibular morphology was characterized in 26 patients (15 boys and 11 girls) with a mean age of 11 years, 9 months. Pre- and postoperative analyses were performed for 17 (10 boys and 7 girls) of the 26 patients with a mean age of 11 years, 9 months. MAIN OUTCOME MEASURES: Mandibular morphology and mandibular position. Data were compared to standard data from the Michigan Growth Study. RESULTS: Comparison of preoperative mandibular measurements to standard data showed that patients with syndromic craniosynostosis have a shorter mandibular body and length and an obtuse gonial angle. Comparison of pre- and postoperative cephalograms showed that, following midfacial advancement with DO, the maxilla moved forward and the mandible moved backward and downward. CONCLUSIONS: Patients with syndromic craniosynostosis have a smaller mandible length and obtuse gonial angle. Correction of midfacial hypoplasia with DO results in inferior and posterior mandibular movement. Clinicians can use this information to counsel patients regarding anticipated changes in facial profile and the need for adjunct procedures.

FaceReview: supporting interactive exploration of linked heterogeneous datasets for unilateral cleft lip and palate.

Children with unilateral cleft lip and palate (UCLP) suffer from negative public perceptions. A better treatment strategy should be established to help them live an ordinary life with improved perceptions. To do that, it is important to understand the relationship between physical facial features and perceptual judgment. In this paper, we present FaceReview, a new visualization system to support interactive exploration of a heterogeneous multidimensional dataset with facial measurement data and subjective judgment data. To seamlessly link the two data, we design FaceReview based on information visualization techniques that are proven to be useful and therefore commonly used, such as brushing and linking, small multiples, and dynamic query. Our design decisions successfully support exploratory tasks of our collaborators. We present a case study to show the efficacy of FaceReview.

Combined push-pull distraction for correction of syndromic midfacial hypoplasia.

PURPOSE: Gradual midfacial advancement, applying the principle of distraction osteogenesis, reduces the restriction of the soft tissues and results in bony consolidation without need for grafting. The midface can be distracted by either pushing it forward, using semiburied devices, or pulling it forward by a rigid external device. For each method there are inherent technical problems, such as controlling the vector of movement, symmetry of advancement, and differential movement of the upper/lower face. We have used a combination of the 2 methods, called "push-pull," in an effort to control the distraction process. The purpose of this paper is to describe our push-pull distraction technique and summarize our early experience. MATERIALS AND METHODS: Ten patients (5 males and 5 females) with a mean age of 11 years 2 months underwent midfacial advancement using push-pull distraction. Two orthodontists, blinded for landmark identification, traced preoperative and postoperative cephalograms and determined linear and angular measurements of midfacial position. A Student t test was used to assess differences between the cephalometric measures on the 2 radiographs. Interexaminer reliability was calculated by an intraclass correlation coefficient. RESULTS: Postdistraction cephalograms were taken a mean of 10 months (range, 3 to 20 months) after removal of the devices. Patients exhibited improvement at all levels of the midface after distraction. There was a statistically significant sagittal advancement from the infraorbital rim to dentoalveolus. The central midface was sufficiently advanced as shown by an improved convexity, nasolabial angle, and upper labial protrusion. There were no significant differences between examiners for any of the measurements in this study. CONCLUSIONS: Push-pull distraction permits 1) equal movement at both the upper and lower facial levels, 2) advancement of the central midface, and 3) symmetric movement of the zygomaticomaxillary complexes. This method also provides a backup, in case one device malfunctions. In combination, the advantages of each device are additive; whereas the weaknesses are not. The push-pull technique is a practical method for midfacial distraction until a better single device is fabricated.