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Objective : The aim of this study was to examine internal responsiveness and estimate minimally important differences (MIDs) for CLEFT-Q scales. Design : In this prospective cohort study, participants completed the CLEFT-Q appearance and health-related quality of life (HRQL) scales before and six months after cleft-related surgery. Setting : Seven cleft centres in Canada, USA and UK participated. Patients/Participants : Patients were ages 8–29 years with CL/P. Interventions : Patients underwent rhinoplasty, orthognathic or cleft lip scar revision surgery. Main Outcome Measure(s) : Internal responsiveness was examined using Cohen's d effect sizes (ESs) based on the following interpretation: 0.20–0.49 small, 0.50–0.79 moderate and ≥ 0.80 large. MIDs were estimated using two distribution-based approaches. Results : Participants had a rhinoplasty (n = 31), orthognathic (n = 21) or cleft lip scar revision (n = 18) surgery. Most participants were males (56%) and aged 8–11 years (41%). Following rhinoplasty, ESs were larger for the nose (0.92, p = 0.001) and nostrils (0.94, p < 0.001) scales than for the face scale (0.51, p = 0.003). MIDs ranged between 6.2–10.4. For orthognathic surgery, larger ES was observed for the jaws scale (1.80, p < 0.001) compared with the teeth (1.16, p < 0.001), face (1.15, p = 0.001) and lips (0.94, p < 0.001) scales. MIDs ranged between 5.9–14.4. In the cleft lip scar revision sample, the largest ES was observed for the nose scale (0.76, p = 0.03), followed by lips (0.58, p = 0.009) and cleft lip scar (0.50, p = 0.043) scales. MIDs ranged between 6.4–12.3. Conclusions : CLEFT-Q detected change in key outcomes for three cleft-specific surgeries, providing evidence of its responsiveness. Estimated MIDs will aid in interpreting this PROM. [ABSTRACT FROM AUTHOR]

Analyze contributing factors toward satisfaction after cleft lip and palate treatments in Senyum Bali Foundation. Qualitative case study, an interview with a semi-structured question from February to April 2023 by purposive sampling informants including parents, foundation staff, and plastic surgeon. Unsatisfying nose and teeth appearance due imperfect effects after surgery and requirement of more procedures. Mini model theory study that includes the appearance of the face and lips; speech and hearing; the functions of feeding, masticatory, breathing; psychosocial, quality of life, treatment cost-effectiveness, funding, health services, physician, implementing cultural contexts, surgical outcomes, and parents' feedback. [ABSTRACT FROM AUTHOR]

Introduction: Treacher Collins syndrome is a rare congenital disease characterized by the multiple craniofacial malformations. Although the deformities affecting patients with Treacher Collins syndrome have been well characterized, the effects of these malformations to clinical severity of the syndrome are not well understood. Objective: To determine the association of specific Treacher Collins mandibular malformations with clinical severity. Design: A retrospective radiographic observational study. Setting: Study conducted at a single institution, a quaternary craniofacial care center Patients: 54 patients with Treacher Collins syndrome. Interventions: Computed tomography (CT), clinical photographs and medical history were included in this analysis. Mandibles were isolated from CT data and reconstructed in three dimensions using Mimics software. Cephalometric measurements were performed on CT data. Clinical severity was determined by Teber and Vincent scores. Association of craniofacial dysmorphology to clinical severity was determined by Spearman rank coefficient. Main outcome measures: The main results obtained were the measurements of the mandibles and the quantification of the malformations of the evaluated patients Results: Among the most frequent findings in the sample are hypoplasia of the zygomatic complex, descending palpebral cleft and mandibular hypoplasia. Patients with a lower ramus/corpus ratio had a higher (more severe) Teber and Vincent classification. Conclusion: Patients with the most compromised mandible are also the patients with the highest number of malformations, thus, the most severe patients. [ABSTRACT FROM AUTHOR]

Objective: To develop an appropriate Chinese version of the CLEFT-Q through translation and cultural adaptation and to evaluate its reliability and validity. Design: The English CLEFT-Q was translated into Chinese following the International Society for Pharmacoeconomics and Outcomes Research guidelines, including cognitive debriefing interviews, and its reliability and validity were assessed. Participants: Patients (N = 246) were mostly in active orthodontic treatment, had a mean age of 14.7 ± 4.4 years, 29% were female, and were born with isolated cleft lip ± alveolus (12%), cleft palate (1%), or cleft lip and palate (87%). Main Outcome Measures: The Chinese CLEFT-Q, including 13 subscales covering Appearance, Health-Related Quality of Life (HRQOL), and Facial Function. Criterion validity instruments included the Negative Physical Self, Satisfaction with Life Scale, and Scale of Positive and Negative Experience. Results: The wording of 67 items was adapted in the final translation. The internal consistency of the Chinese version of the CLEFT-Q was high based on Cronbach's alphas of 0.85 to 0.98 and split-half reliability of 0.85 to 0.92. Exploratory and confirmatory factor analyses yielded three factors, which demonstrated construct validity by broadly matching the structure of the original CLEFT-Q. The Appearance and HRQOL dimensions had weak to moderate correlations (r = −0.35 to 0.67) with the corresponding instruments for criterion validity. Conclusions: The Chinese version of the CLEFT-Q is a patient-reported outcome measure that can reflect the quality of life of Chinese patients with cleft lip and/or palate with good reliability and validity. [ABSTRACT FROM AUTHOR]

Objective: To determine associations of demographic, morphologic, and treatment protocol parameters with quality of life (QoL), appearance/speech satisfaction, and psychological adjustment. Design: Observational study utilizing retrospective report of protocol variables and current outcome variables. Setting: Six North American cleft treatment clinics. Participants: Children, ages 8.0–10.99 years, with Cleft Lip ± Alveolus, Cleft Palate, Cleft Lip and Palate, and parents (N = 284). Outcome Measures: Pediatric QoL Inventory (PedsQL): Parent, Child, Family Impact Module (FIM); Patient Reported Outcome Measurement Information System (PROMIS); Child Behavior Checklist (CBCL); CLEFT-Q. Results: Outcome scores were average with few differences by cleft type. Multiple regression analyses yielded significant associations (Ps <.05) between socioeconomic status, race, and age at assessment and parent- and self-reported measures. Females had higher PROMIS Depression (β=.20) but lower CBCL Affective (β = −.16) and PROMIS Stigma scores (β= −.24). Incomplete cleft lip was associated with lower PROMIS Depression, and more positive ratings of CLEFT-Q: Nose, Nostril, Lip Scar; CBCL Competence scores, (βs = −.17 to.17). Younger Age at Lip Closure was associated with higher CBCL School Competence (β= −.18). Younger Age at Palate Closure was associated with higher Child PedsQL Total, Physical, Psychosocial QoL, and better CLEFT-Q Speech Function (βs = −.18 to −.15). Furlow Palatoplasty was associated with more CBCL Externalizing Problems (β =.17) higher CBCL Activities (β =.16). For all diagnoses, fewer Total Cleft-Related Surgeries was associated with lower PROMIS Stigma and higher CBCL Total Competence and Activities (βs = −.16 to.15). Conclusions: Demographic characteristics, lip morphology, and treatment variables are related to later psychological functioning. [ABSTRACT FROM AUTHOR]

Objective: Given prior craniofacial research reporting higher risk for negative social interactions, this study aimed to explore experiences of stigma described by children with cleft lip and/or palate (CL/P). Design: Qualitative interviews were conducted with children in three sessions (totaling 90–180 min) using creative activities in their homes and/or and walking interviews in their neighborhood. Interview transcriptions were interpreted primarily with thematic analysis following theoretical frameworks of the stigmatization process and self-stigma concept. Setting: Participants were recruited through Operation Smile Colombia. Interviews took place at children's homes and neighborhoods within three Colombian regions (Boyacá, Bogotá and Cundinamarca). Participants: Children (N = 12) with CL/P aged between 6–12 years were interviewed. Results: Themes fit within the stigmatization process, starting with labeling and stereotyping, such as a range of mockery, and group separation by peers highlighting their not belonging and being socially 'other'. Status loss themes included negative appraisals of cleft-related differences and being perceived as 'ill' and 'imperfect'. Social exclusion themes reflected limited social interactions and loneliness. Self-stigma themes included shame about speaking with peers and anticipation of negative social interactions. Conclusions: The study results suggest that the process of stigmatization and self-stigma adversely affect social interactions for children with a cleft in multiple ways. Healthcare practitioners and policy makers can help address the potential consequences of stigma by implementing interventions at micro, meso and macro levels. [ABSTRACT FROM AUTHOR]

Objective: The birth of a child with a craniofacial anomaly (CFA) can have a profound psychological impact on the family and the parental relationship. The purpose of this study was to qualitatively investigate how a child's CFA condition affected parents' couple relationship. Setting: All patients with a CFA are followed-up by the National Unit for Craniofacial Surgery, a specialized and multidisciplinary team. Hence, participants were recruited within a centralized treatment setting. Design: We used a qualitative approach to explore the relationship experiences of parents of children with CFAs. The interviews were analysed using a hermeneutic-phenomenological approach. Participants: The study included 13 parents, nine mothers and four fathers of children with a range of different CFAs. At the time of the interview, 10 participants were married, one was cohabiting, and two were divorced. Results: Most participants perceived their partners as committed and engaged in caring for their affected child and involved in the family's everyday life, and described a strengthened relationship to their partner after the child with a CFA was born. However, some participants struggled in their relationships with their partners, and did not receive the comfort and support they needed during this critical time, leading to feelings of distance and loneliness. Conclusions: Craniofacial teams should be mindful of the importance of the environment surrounding the child, such as parental relationship and family function. Therefore, a comprehensive approach should be included in team-based care, and couples and families in need of extra support should be referred to relevant specialists. [ABSTRACT FROM AUTHOR]

Objective: To evaluate a method of measuring the change in palatal length and shape following maxillary advancement using synchronous lateral videofluoroscopy and voice recording in order to understand how movement of the maxilla may affect VPI risk in patients with cleft lip and/or palate (CL/P). Design: Retrospective cohort study of children with cleft lip and/or palate. Setting: Single center, tertiary children's hospital. Participants: Patients with cleft lip and/or palate who underwent maxillary advancement between 2016–21 inclusive. Interventions: Maxillary advancement surgery, including those who underwent concurrent mandibular procedures. Main outcome measures: The length of the soft palate and the genu angle were measured throughout palatal dynamic range. Pre- and post-operative measurements were compared using a one sided T-test, with subgroup analysis for patients with clinical VPI. Results: Ten patients were examined. The mean distance of maxillary advancement was 10.5 mm. The average increase in pre-genu soft palate length was 2.8 mm in the resting position and 2.9 mm in the closed position. The genu angle decreased in the closed position by 16.3 degrees. Conclusions: The soft palate showed limited ability to lengthen following maxillary advancement and this may explain the risk of VPI. There was partial compensation by the muscle sling of the palate as demonstrated by a more acute post-operative genu angle and this suggests one reason for the variability of VPI reported. Future research is required to investigate how length and shape changes measured using this method can predict VPI risk. [ABSTRACT FROM AUTHOR]

Objective: This study used three-dimensional measurements to provide a nasolabial analysis of patients with unilateral cleft lip and palate (UCLP), bilateral cleft lip and palate (BCLP), and controls across different races and ethnicities. Design: A retrospective comparative study. Setting: Tertiary care pediatric institution. Patients, Participants: The study included 90 patients with UCLP, 43 patients with BCLP, and 90 matched controls. Patients are separated as self-identified Caucasian, Hispanic, or African American. Main Outcome Measure(s): Nasal length, nasal protrusion, columellar height, columellar width, tip width, alar width, alar base width, nasolabial angle, upper lip length, philtrum length, nostril height, and nostril width. Results: All UCLP groups had significantly greater columella and tip widths and decreased nasolabial angles than controls. All BCLP groups had significantly greater columella width, tip width, nasolabial angle, and nostril widths. Upper lip length, philtrum length, and nostril height were significantly decreased in BCLP compared to controls. Across UCLP groups, African Americans had significantly decreased nasal protrusion and columella height and a significantly increased columella width compared to Caucasians and Hispanics. Alar and alar base widths were significantly different between all groups. Across BCLP groups, the Caucasian nostril width was significantly less than the African Americans. Conclusions: These findings suggest that when correcting nasolabial characteristics in patients with cleft lip, it is important to consider racial and ethnic differences to achieve a normal appearance. Specifically, goals for alar width, alar base width, nasal tip, and projection should be tailored to the patient's race and ethnicity. [ABSTRACT FROM AUTHOR]

Objective: To investigate condylar bone density (BD) in children with craniofacial microsomia (CFM) and identify factors that contribute to early stage condylar resorption (CR) after mandibular distraction osteogenesis (MDO). Design: Retrospective study. Setting: Craniofacial department of a plastic surgery hospital. Patients: Fifty-one children with CFM classified as Pruzansky IIa based on complete pre-(T0) and post-MDO (T1) computed tomography (CT) data. Intervention and Main outcome measurements: Mimic 21.0 (Materialise Inc., Belgium) was used to measure bilateral BD and condylar height (CH) and volume (CV) of affected side. Children were split into groups based on either affected side BD or the distraction length (DL,25 mm as cutoff).Bilateral BD was compared using a paired t-test in each group. The CH and CV of affected side at T0 and T1 were compared. The relative values of the CH and CV (CH ratio) and the volume (CV ratio) of the affected side were compared across the groups. Results: The BD was lower on affected side than on unaffected side. Regarding BD, CH and CV decreased after MDO in group I, while the CH ratio and CV ratio of group I was lower than that of groups II and III. Regarding DL, the CV ratio was lower in Group L than Group S. Conclusions: The condylar bone quality on affected side is compromised in type IIa CFM. A low BD in combination with a larger distraction distance may increase the risk of CR; therefore, MDO in patients with such characteristics should be postponed. [ABSTRACT FROM AUTHOR]

Objective: Cleft lip and/or palate (CL/P) can have long-lasting effects on the appearance, function, and psychosocial wellbeing of patients. The CLEFT-Q questionnaire is a patient-reported outcomes instrument specifically designed to assess the health-related quality of life of patients with CL/P. The aim of this study was to produce and linguistically validate a Finnish version of the CLEFT-Q questionnaire. Design: The CLEFT-Q questionnaire was translated into Finnish following guidelines of the International Society for Pharmacoeconomics and Outcomes Research. Pilot testing with cognitive debriefing interviews was conducted on patients of the target age range of the questionnaire, 8–29, and with various cleft types. Results: The CLEFT-Q questionnaire translated readily into Finnish. A review of the backward translation led to two words being changed. Thirteen patients – ten females and three males – with a median age of 14 years, participated in the cognitive debriefing interviews. The interviews led to further nine word changes. The pilot study data suggested that the performance of the Finnish version of the instrument is in line with the original CLEFT-Q questionnaire. Conclusions: The Finnish version of CLEFT-Q produced here is linguistically valid and ready for use in the evaluation of the health-related quality of life of patients with CL/P. However, future work is needed to further assess the validity and the reliability of the CLEFT-Q in the Finnish patient population. [ABSTRACT FROM AUTHOR]

Objective: To translate and validate CLEFT-Q©, patient-reported outcome measure for patients with cleft lip and/or palate (CL and/or P), into Indonesian. CLEFT-Q© covers the domains of appearance, facial function, health-related quality of life and consists of scales describing outcomes after cleft surgery. Design: The CLEFT-Q© instrument was translated according to the International Society of Pharmacoeconomics and Outcomes Research guidelines, including translation, cognitive debriefing, and field-testing. Setting: Dr. Cipto Mangunkusumo Hospital, Indonesia; independent CL and/or P support groups. Patients: Patients ages 8-29 with a history of repaired CL and/or P were grouped based on age. Those unable to complete the questionnaire independently were excluded. Interventions: The primary objective was reliable translation of the CLEFT-Q® instrument. Each scale was assessed for its internal consistency (Cronbach's alpha) and validity (inter-item correlation), and sub-group analyses were performed based on age group. Results: Forward and back translation revealed 25(13.3%) and 12(6.3%) of items were difficult to translate. Cognitive debriefing revealed 10(5.3%) items were difficult to understand, with the lowest reliability on the facial appearance scale (α=0.27). Other scales demonstrated acceptable to excellent reliability (α=0.53–0.68). Field testing revealed acceptable reliability and validity of the translation (α = 0.74-0.92; 69% ideal range of inter-item correlation). Sub-group analyses revealed patients in the <11y.o. and >18y.o. groups had the lowest scores on the "cleft lip scar" scale while those 11-18y.o. had the lowest scores on the "nostrils" scale. Conclusion: Iterative translation and cultural adaptation of CLEFT-Q© into Indonesian demonstrated reliability and validity of the tool, supported by acceptable to excellent internal consistency and ideal inter-item correlation. [ABSTRACT FROM AUTHOR]

Objective: To assess the ability of current 3D printing technology to generate a craniofacial bony and soft tissue anatomical model for use in simulating the performance of a fronto-orbital advancement (FOA) osteotomy and then to further assess the value of the model as an educational tool. Design: Anatomic models were designed with a process of serial anatomic segmentation/design, 3D printing, dissection, and device refinement. A validation study was conducted with 5 junior and 5 senior plastic surgery residents. The validation study incorporated a multiple-choice Knowledge Assessment test (KA), an Objective Structured Assessment of Technical skills (OSATs), a Global Rating Scale (GRS) and a Michigan Standard Simulation Experience Scale (MiSSES). We compared the scores of both the junior and senior residents and compared junior resident scores, before and after viewing a lecture/demonstration. Results: MiSSES showed high face validity with a score of 85.1/90, signifying high satisfaction with the simulator learning experience. Simulation and the lecture/demonstration improved the junior resident average KA score from 5.6/10 to 9.6/10 (P =.02), OSATs score from 32.4/66 to 64.4/66 (P <.001) and GRS score from 13.9/35 to 27.5/35 (P <.001). The senior residents OSATs score of 56.3/66 was higher than the pre-lecture juniors (32.4/66) (P <.001), but lower than the post-lecture juniors (64.4/66) (P <.001). Conclusion: We have successfully fabricated a 3D printed craniofacial simulator capable of being used as an educational tool alongside traditional surgical training. Next steps would be improving soft tissue realism, inclusion of patient and disease specific anatomy and creation of models for other surgical specialties. [ABSTRACT FROM AUTHOR]

Objective: The objective of this study was to use data from Smile Train's global partner hospital network to identify patient characteristics that increase odds of fistula and postoperative speech outcomes. Design: Multi-institution, retrospective review of Smile Train Express database. Setting: 1110 Smile Train partner hospitals. Patients/Participants: 2560 patients. Interventions: N/A. Main Outcome Measure(s): Fistula occurrence, nasal emission, audible nasal emission with amplification (through a straw or tube) only, nasal rustle/turbulence, consistent nasal emission, consistent nasal emission due to velopharyngeal dysfunction, rating of resonance, rating of intelligibility, recommendation for further velopharyngeal dysfunction assessment, and follow-up velopharyngeal dysfunction surgery Results: The patients were 46.6% female and 27.5% underweight by WHO standards. Average age at palatoplasty was 24.7 ± 0.5 months and at speech assessment was 6.8 ± 0.1 years. Underweight patients had higher incidence of hypernasality and decreased speech intelligibility. Palatoplasty when under 6 months or over 18 months of age had higher rates of affected nasality, intelligibility, and fistula formation. The same findings were seen in Central/South American and African patients, in addition to increased velopharyngeal dysfunction and fistula surgery compared to Asian patients. Palatoplasty technique primarily involved one-stage midline repair. Conclusions: Age and nutrition status were significant predictors of speech outcomes and fistula occurrence following palatoplasty. Outcomes were also significantly impacted by location, demonstrating the need to cultivate longitudinal initiatives to reduce regional disparities. These results underscore the importance of Smile Train's continual expansion of accessible surgical intervention, nutritional support, and speech-language care. [ABSTRACT FROM AUTHOR]

Objective: To identify specific areas for improvement in cleft lip repair teaching. Design: Secondary analysis of prospectively-collected, blinded data. Setting: Three residency programs rotating at a single academic children's hospital. Participants: Plastic surgery residents, and craniofacial/pediatric plastic surgery fellows. Main Outcome Measures: Mean scores for each skill in an 18-item Unilateral Cleft Lip Repair competency assessment tool (UCLR) (1–3 scale for each item) were rank ordered. Correlation between level of training (PGY) and performance on steps of the procedure was examined using Pearson R. Results: Simulation participants (n = 26) scored highest on skills in the "Marking" subscale (2.38–2.63 mean score). Procedural steps that scored lowest were: closing the nasal floor (2.00), repairing oral mucosa (2.15) and avoiding over/under-dissection (2.19). Interestingly, none of these skills correlated with PGY, suggesting they do not improve with training. Conclusions: These results suggest that marking cleft lip repair is taught well in our current teaching environment, while steps like closing the nasal floor and repairing the oral mucosa are taught less well. Improved teaching of these steps could be achieved with deliberate instruction, video, digital simulation, and high fidelity simulation. [ABSTRACT FROM AUTHOR]

Objective : Using both endoscope and exoscope in cleft soft palate surgery is not widespread, despite the potential advantages related to view magnification, ergonomic posture of the surgeon, and involvement of the surgical team. Design : The aim of the current study is to compare endoscopic (Olympus Visera©) and exoscopic (Karl-Storz Vitom©) assistance in cleft soft palate surgery in a preclinical cadaver setting. Setting : A formalin fixed specimen was dissected to mimic the anatomical conditions of a cleft soft palate. Participants : Ten young surgeons with limited experience in transoral surgery were involved in the exercitation on the specimen. Interventions : The exercitation consisted of 4 tasks: (1) device setting; (2) identification of muscle plane; (3) muscle suturing; (4) oral mucosa suturing. Main outcomes : Participants were timed while performing each task both with exoscope and endoscope and asked to fill in 2 questionnaires related to the visual systems used (NASA Task Load System TLS and VAS 1-10). Results : All surgeons completed the 4 tasks with both the endoscope and exoscope. The execution times were similar except for faster setting of the exoscope. Participants felt that completing surgical exercises using the exoscope required less physical, intellectual, and temporal efforts compared to the endoscope. The exoscope was also more appreciated for its handling, 3D visualization, and limited encumbrance. Conclusions : Exoscope scored better both at NASA TLS and VAS 1-10 and required a faster setting than endoscope. Further clinical in-vivo studies are required to explore the advantages of these devices in cleft palate repair. [ABSTRACT FROM AUTHOR]

Background: During periods of civil strife, the need for trauma care and lack of sufficient cleft surgeons causes an increase in children left untreated with cleft lip and palate deformities. During the Syrian war, some cleft care was provided through visiting charities, with surgeries performed both in Syria and neighboring countries. There is a need to increase the number of adequately trained cleft surgeons available in such regions so that care can be achieved beyond mission trips. Methodology: Cleft lip and palate repair workshops were delivered to 50 doctors in Syria. Pre-workshop, trainees received supplementary learning material. During the workshop, attendees received didactic teaching followed by 2 simulation sessions. Pre- and post-workshop, attendees completed questionnaires regarding their confidence and ability to perform cleft lip and palate repair. Results: Pre-workshop, 96% of workshop attendees had never independently performed cleft lip repair while 100% of attendees had not previously performed cleft palate repair. The mean pre-workshop confidence score was 2.452. Post-workshop, the mean confidence score was 3.503. Confidence rating scores significantly improved (P <.001). Conclusion: The workshop delivered in Syria, together with this cleft lip and palate simulator provides an effective training tool that may support surgical training globally, particularly those in low-income countries. Further support is needed by charity organizations to ensure the continued delivery of such training. [ABSTRACT FROM AUTHOR]

Objective : To translate and culturally adapt the original English version of the CLEFT-Q into Thai (Thai CLEFT-Q). Design : A pilot study. Setting : A single tertiary referral hospital in Thailand. Participants : Patients with cleft lip or palate (CL/P) aged between 8 and 29 years. Interventions : The CLEFT-Q was translated and culturally adapted from English into Thai using the good practice guidelines developed by the International Society for Pharmacoeconomics and Outcomes Research (ISPOR). Two forward translations and reconciliation, 1 back translation, and 2 sets of cognitive debriefing interviews with patients were performed to develop the Thai CLEFT-Q. Results : The English version of the CLEFT-Q consists of 13 scales (119 items) assessing appearance, health-related quality of life (HRQOL), and facial function. The forward translations revealed 33.6% inconsistencies. They were related to narrow meaning (2.5%) and improper (16.8%) or incorrect (14.3%) wording or phrasing of the items. After reconciliation, the back translation showed 5 inconsistencies (4.2%) with the original version. However, no changes were needed because the reconciliation version was consistent with the source version. Sixteen participants underwent cognitive debriefing interviews, which revealed a comprehensive interpretation of the Thai CLEFT-Q. Interim reports revealed fair facial appearance scores, albeit with HRQOL and facial function outcomes. Conclusions : Translation and cultural adaptation of the Thai CLEFT-Q provided evidence of its transferability and equivalence with the original English version. Feasible comprehension of the Thai version was also achieved. [ABSTRACT FROM AUTHOR]

Objectives: To determine the UK prevalence of behavioral problems in 5-year-old children born with isolated or syndromic cleft lip and/or palate (CL/P) compared to the general population and identify potentially associated factors. Design: Observational study using questionnaire data from the Cleft Collective 5-Year-Old Cohort study and three general population samples. Main Outcome Measure: The Strengths and Difficulties Questionnaire (SDQ). Participants: Mothers of children (age: 4.9-6.8 years) born with CL/P (n = 325). UK general population cohorts for SDQ scores were: Millennium Cohort Study (MCS) (n = 12 511), Office of National Statistics (ONS) normative school-age SDQ data (n = 5855), and Avon Longitudinal Study of Parents and Children (ALSPAC) (n = 9386). Results: By maternal report, 14.2% of children born with CL/P were above clinical cut-off for behavioral problems, which was more likely than in general population samples: 7.5% of MCS (OR = 2.05 [1.49-2.82], P < 0.001), 9.8% of ONS (OR = 1.52 [1.10-2.09], P = 0.008), and 6.6% of ALSPAC (OR = 2.34 [1.70-3.24], P < 0.001). Children in the Cleft Collective had higher odds for hyperactivity, emotional and peer problems, and less prosocial behaviors. Maternal stress, lower maternal health-related quality of life and family functioning, receiving government income support, and maternal smoking showed evidence of association (OR range: 4.41-10.13) with behavioral problems, along with maternal relationship status, younger age, and lower education (OR range: 2.34-3.73). Conclusions: Findings suggest elevated levels of behavioral problems in children born with CL/P compared to the general population with several associated maternal factors similar to the general population. [ABSTRACT FROM AUTHOR]

Objective : This study evaluated the effectiveness of academic screening measures in relation to parent-reported diagnoses. Design : Multicenter, retrospective cohort study including structured interviews, questionnaires, and chart reviews. Setting : Six North American cleft centers. Participants : Children (n = 391) with cleft lip and/or palate, ages 8 to 10 years of age (192 male) and their guardians were recruited during regular clinic visits. Main Outcome Measures : Parent and child ratings on the Pediatric Quality of Life Inventory (PedsQL) School Scale, child report on CleftQ School Scale, parent report on the Adaptive Behavior Assessment System-Third Edition Functional Academics (ABAS-FA) Scale and Child Behavior Checklist (CBCL) School Competency Scale, parent interview, and medical chart review. Results : Risk for concerns ranged from 12% to 41%, with higher risk reflected on the CBCL-SC compared to other measures. Males with cleft palate were consistently at the highest risk. Only 9% of the sample had a parent-reported diagnosis of a learning or language disability. Ratings from the ABAS-FA and CBCL-SC had the highest utility in identifying those with language and/or learning concerns. Conclusions : As cleft teams work to develop standardized batteries for screening and monitoring of patients, it is important to evaluate the effectiveness of measures in identifying those at highest risk. When screening for language and learning disorders, questions related to potential academic struggles, such as increased school effort or increased school distress, are most useful. Referrals for follow-up evaluation are recommended for those identified at high risk. [ABSTRACT FROM AUTHOR]

Objective : Midface hypoplasia (MFH) is a long-term sequela of cleft lip and palate repair, and is poorly understood. No study has examined the aggregate data on sagittal growth restriction of the midface following repair of the lip, but not palate, in these patients. Methods : A systematic review of 3780 articles was performed. Twenty-four studies met inclusion criteria and 11 reported cephalometric measurements amenable to meta-analysis. Patients with Veau class I-III palatal clefts were included so long as they had undergone only lip repair. Groups were compared against both noncleft and unrepaired controls. Results : Cephalometrics were reported for 326 patients (31.3% female). Noncleft controls had an average SNA angle of 81.25° ± 3.12°. The only patients demonstrating hypoplastic SNA angles were those with unilateral CLP with isolated lip repair (77.4° ± 4.22°). Patients with repaired CL had SNA angles similar to noncleft controls (81.4° ± 4.02°). Patients with unrepaired CLP and CL tended toward more protruding maxillae, with SNA angles of 83.3° ± 4.04° and 87.9° ± 3.11°, respectively. Notably, when comparing SNA angles between groups, patients with CLP with isolated lip repair had significantly more hypoplastic angles compared to those with repaired CL (P <.0001). Patients with CLP with isolated lip repair were also more hypoplastic than noncleft controls (P <.0001). In contrast, there was no significant difference between the SNA of patients with repaired CL and controls (P =.648). Conclusion : We found that cleft lip repair only appeared to contribute to MFH in the setting of concurrent cleft palate pathology, suggesting that scarring from lip repair itself is unlikely to be the predominant driver of MFH development. However, studies generally suffered from inadequate reporting of timing, technique, follow-up time, and cleft severity. [ABSTRACT FROM AUTHOR]

Objective: To investigate the relationship between perception of craniofacial deformity, geometric head features, and 3D head shape analyzed by statistical shape modeling (SSM). Patients: A total of 18 unoperated patients with scaphocephaly (age = 5.2 ± 1.1m)—6 were followed-up after spring-assisted cranioplasty (SAC) (age = 9.6 ± 1.5m)—and 6 controls (age = 6.7 ± 2.5m). Main Outcome Measures: 3D head shapes were retrieved from 3D scans or computed tomography (CTs). Various geometrical features were measured: anterior and posterior prominence, take-off angle, average anterior and posterior lateral and horizontal curvatures, cranial index (CI) (cranial width over length), and turricephaly index (TI) (cranial height over length). SSM and principal component analysis (PCA) described shape variability. All models were 3D printed; the perception of deformity was blindly scored by 9 surgeons and 1 radiologist in terms of frontal bossing (FB), occipital bulleting (OB), biparietal narrowing (BN), low posterior vertex (LPV), and overall head shape (OHS). Results: A moderate correlation was found between FB and anterior prominence (r = 0.56, P <.01) and take-off angle (r = − 0.57, P <.01). OB correlated with average posterior lateral curvature (r = 0.43, P < 0.01) similarly to BPN (r = 0.55, P <.01) and LPV (r = 0.43, P <.01). OHS showed strong correlation with CI (r = − 0.68, P <.01) and TI (r = 0.63, P <.01). SSM Mode 1 correlated with OHS (r = 0.66, p <.01) while Mode 3 correlated with FB (r = − 0.58, P <.01). Conclusions: Esthetic cranial appearance in craniofacial patients is correlated to specific geometric parameters and could be estimated using automated methods such as SSM. [ABSTRACT FROM AUTHOR]

Introduction: Alveolar bone grafting aims to restore bony continuity of the alveolus and provide optimal periodontal support for teeth adjacent to the cleft. We created a survey of cleft surgeons to assess the current standard of care regarding this procedure. Methods: A multiple choice survey was implemented using Qualtrics software and emailed to a list of 708 surgeons from the ACPA membership directory. Correlation between various provider factors and treatment practices was assessed with Fisher's exact test and likelihood ratio tests. Results: The response rate was 17.5%. Eighty-seven percent of providers preferred to perform grafts prior to secondary canine eruption while 10% favored before central incisor eruption. Eighty-one percent favored palatal expansion prior to bone grafting. Wide variability existed regarding the time to initiate postoperative orthodontics; 43% waited 4 to 6 months. Sixty-four percent of surgeons now utilize cone beam CT to assess graft take. The majority of respondents utilized cancellous bone autograft (92%) from the anterior iliac crest (97%) as graft material. Seventy percent used three or more modalities for post-operative pain control management. Early career surgeons (0–5 years) appeared more likely to use non-autologous materials (p <.01) for grafting. Conclusion: Alveolar bone grafting prior to secondary canine eruption remains the most common strategy but other protocols are employed. Surgeons utilize multiple modalities for radiographic evaluation and most often use autologous cancellous bone as the primary grafting material. There is no true consensus on the perioperative timing and sequencing of orthodontic manipulation while principles of multimodal perioperative pain control appear widely accepted. [ABSTRACT FROM AUTHOR]

Introduction: The squamosal suture (SQS) joins the temporal to the parietal bones bilaterally and is a poorly described site of craniosynostosis. SQS fusion is thought to occur as late as the fourth decade of life and beyond; however, we have incidentally noted its presence among our pediatric patients and hypothesize that it may occur earlier in life and more frequently than previously believed. Methods: A retrospective review of imaging performed on pediatric patients was completed to identify patients with SQS synostosis. This included a review of clinical notes as well as computed tomography (CT) images obtained by our craniofacial clinic. Relevant patient data and imaging were reviewed. Results: Forty-seven patients were identified with SQS synostosis, 21 were female (45%). Age at the time of radiographic diagnosis was 10.1 ± 8.4 years (range 17 days to 27 years). A majority of patients had bilateral SQS synostosis (57%), with a relatively even distribution of unilateral right (23%) versus left (19%). SQS was an isolated finding (no other suture involvement) in 15 patients (32%), all of whom were normocephalic and did not require surgical intervention. Thirty-two patients (68%) had concomitant craniosynostosis of other sutures, most commonly sagittal and coronal. Nine patients (19%) underwent surgery to correct cranial malformations—all these patients had multi-suture synostosis (P = 0.012). Twenty-seven patients (57%) had SQS synostosis diagnosed incidentally compared to 20 (43%) who were imaged with suspicion for synostosis. In those who were symptomatic, common findings included developmental delay, elevated intracranial pressure, hydrocephalus, seizures, and visual/hearing impairments. Ten patients (21%) were syndromic, the most frequent of which was Crouzon syndrome. No single pattern of calvarial malformation could be definitively described for SQS synostosis. Conclusion: Given that most isolated SQS synostosis cases were normocephalic, asymptomatic, and discovered incidentally, it is likely that there are many cases of unidentified SQS synostosis. The significance of SQS synostosis is currently unclear, and warrants further investigation into this phenomenon, its natural course, and its potential presence in the spectrum of normal development. [ABSTRACT FROM AUTHOR]

Objectives : To (1) appraise current international classification and clinical management strategies for craniofacial microsomia (CFM) and microtia, and (2) to assess agreement with the European Reference Network "European Guideline Craniofacial Microsomia" recommendations on screening and monitoring. Design : This was a cross-sectional online survey study. The survey consisted of 44 questions on demographics, diagnostics and classification, obstructive sleep apnea, feeding difficulties, speech and language development, hearing, ocular abnormalities, visual development, orthodontic screening, genetic counselling, psychological wellbeing, and extracraniofacial anomalies. Participants : Respondents were participants of 3 international cleft and craniofacial conferences, members of the American Cleft Palate and Craniofacial Association and members of the International Society for Auricular Reconstruction. Respondents were requested to complete 1 questionnaire per multidisciplinary team. Results : Fifty-seven responses were received from 30 countries (response rate ∼3%).The International Consortium for Health Outcomes Measurement diagnostic criteria were used by 86% of respondents, though 65% considered isolated microtia a mild form of CFM. The Orbit, Mandible, Ear, Facial Nerve and Soft Tissue classification system was used by 74% of respondents. Agreement with standardized screening and monitoring recommendations was between 61% and 97%. A majority of respondents agreed with screening for extracraniofacial anomalies (63%-68%) and with genetic counselling (81%). Conclusions : This survey did not reveal consistent agreement on the diagnostic criteria for CFM. Respondents mostly supported management recommendations, but frequently disagreed with the standardization of care. Future studies could focus on working towards international consensus on diagnostic criteria, and exploring internationally feasible management strategies. [ABSTRACT FROM AUTHOR]

Objective: This paper describes 20 years of microtia and craniofacial microsomia (CFM) psychosocial and healthcare studies and suggests directions for clinical care and research. Design: A narrative review of papers January 2000 to July 2021 related to psychosocial and healthcare experiences of individuals with microtia and CFM and their families. Results: Studies (N = 64) were mainly cross-sectional (69%), included a range of standardized measures (64%), and were with European (31%), American (27%), or multinational (23%) samples. Data were generally collected from both patients and caregivers (38%) or patient self-report (35%). Sample sizes were 11 to 25 (21%), 26 to 50 (19%), 51 to 100 (22%), or over 100 (38%). Studies addressed 5 primary topics: (1) Healthcare Experiences, including Medical Care, Hearing Loss/Amplification, Diagnostic Experiences, and Information Preferences; (2) Psychosocial Experiences, including Teasing, Behavioral Adjustment, Psychosocial Support, and Public Perception; (3) Neurocognitive Functioning and Academic Assistance; (4) Pre- and Post-Operative Psychosocial Outcomes of Ear Reconstruction/Canaloplasty; and (5) Quality of Life and Patient Satisfaction. Conclusions: Care involved multiple specialties and was often experienced as stressful starting at diagnosis. Psychosocial and neurocognitive functioning were generally in the average range, with possible risk for social and language concerns. Coping and resiliency were described into adulthood. Satisfaction and positive benefit of ear reconstruction/canaloplasty were high. Care recommendations include increasing: hearing amplification use, microtia and CFM knowledge among providers, efficient treatment coordination, psychosocial support, academic assistance, and advances to minimize surgical scarring. This broad literature overview informs clinical practice and research to improve psychosocial outcomes. [ABSTRACT FROM AUTHOR]

Objective: This multicenter study aimed to compare demographic, operative, and short-term outcomes data between open and minimally invasive surgical approaches for craniosynostosis repair utilizing the American College of Surgeon's National Surgical Quality Improvement Program Pediatric (NSQIP-P) database and highlight surgical disparities among races and ethnicities. Design: Retrospective review of large multicenter database. Setting: Freestanding general acute care children's hospitals, children's hospitals within a larger hospital, specialty children's hospitals, or general acute care hospitals with a pediatric wing. Patients and Participants: A total of 4931 pediatric patients underwent craniosynostosis correction within the NSQIP-P database from 2013 to 2019. Interventions: None. Main Outcome Measure(s): Demographic information included age at surgery, sex, race, and ethnicity. Operative and outcomes measures included operative time, anesthesia time, days until discharge, postoperative complications, blood transfusions, 30-day readmission, and 30-day unplanned return to operating room. Results: Patients who underwent minimally invasive surgery had significantly shorter operative and anesthesia times (p <.001; p <.001), fewer days until discharge (p <.001), fewer postoperative complications (p <.05), and less blood transfusions (p <.001). The proportion of White patients was significantly higher in the minimally invasive surgery group (p <.01), whereas Black and Hispanic patients had a significantly higher proportion in the open surgery group (p <.001; p <.001). Additionally, the percentage of patients undergoing minimally invasive surgery increased from 3.8% in 2014 to over 13% in 2019. Conclusions: This study adds to a growing consensus that minimally invasive surgery has significantly decreased operative time, anesthesia time, transfusion rates, length of hospital stay, and postoperative complications compared to open surgery. Racial and ethnic surgical disparities showed larger proportions of Black and Hispanic populations undergoing open procedures. [ABSTRACT FROM AUTHOR]

Objective: Children and adolescents with orofacial clefts may experience ongoing psychosocial impacts due to the continuous nature of cleft treatments, facial and dental differences, and speech and hearing difficulties. The aim of this qualitative systematic review was to better understand the experiences of children and adolescents with orofacial clefts. Design: A systematic search strategy using PubMed, Embase, Emcare, Scopus, and Web of Science databases was performed to identify relevant qualitative studies evaluating the lived experience of children and adolescents with orofacial clefts from inception through to June 2021. Eligible studies were critically appraised using the Joanna Briggs methodology and a meta-aggregative approach. Results: The search identified 2466 studies, with 13 found to meet the inclusion criteria. Extraction of 155 findings resulted in 27 categories, which were meta-aggregated into 7 overarching synthesized findings. These 7 core findings included aspects of child experience and findings that enhanced or impeded child experience at the individual, family, and community levels. Conclusions: Factors that impeded child experience at the individual, family, and community levels were more pronounced than factors that enhanced their experience among children and adolescents with orofacial clefts. Further initiatives are needed to provide support to individuals, families, and school communities to enhance children's experience of orofacial cleft during the formative childhood and adolescent years. [ABSTRACT FROM AUTHOR]

Objectives : Rapid palatal expanders (RPEs) are commonly used in patients with cleft lip and palate (CLP) prior to secondary alveolar bone grafting (SABG). Their position and size can impede tongue movement and affect speech. This study assessed changes in perception and production of speech over the course of RPE treatment. Design : Prospective longitudinal. Setting : Tertiary university-affiliated hospital. Participants : Twenty-five patients with unilateral CLP treated with Fan-type RPEs, and their parents. Interventions : Patient and parent speech questionnaires and patient speech recordings were collected at baseline before RPE insertion (T1), directly after RPE insertion (T2), during RPE expansion (T3), during RPE retention (T4), directly after RPE removal but before SABG (T5), and at short-term follow-up after RPE removal and SABG (T6). Main Outcome Measures : Ratings for patient and parent questionnaires, first (F1) and second (F2) formants for vowels /a/, /i/, and /u/, and nasalance scores for non-nasal and nasal sentences, were obtained and analyzed using mixed model analyses of variance. Results : Ratings worsened at T2. For the vowel /a/, F1 and F2 were unchanged at T2. For the vowel /i/, F1 increased and F2 decreased at T2. For the vowel /u/, F1 was unchanged and F2 decreased at T2. Nasalance was unchanged at T2. All outcome measures returned to T1 levels by T4. Conclusions : RPE insertion resulted in initial adverse effects on speech perception and production, which decreased to baseline prior to removal. Information regarding transient speech dysfunction and distress may help prepare patients for treatment. [ABSTRACT FROM AUTHOR]

We recently published the 3-month follow-up of 2 neonates with Robin sequence whose mandibular hypoplasia and restricted airway were successfully treated with an orthodontic airway plate (OAP) without surgical intervention. Both infants were successfully weaned off the OAP after several months of continuous use. We present the course of OAP treatment in these patients with a focus on breathing, feeding, and facial growth during their first year of life. Both infants demonstrated stable mandibular projection, resolution of obstructive sleep apnea, and normal development. [ABSTRACT FROM AUTHOR]

Objective : Measuring the impact of orthognathic surgery on quality of life is of significant importance in patients with cleft deformities. Standardized tools such as patient-reported outcome measures (PROMs) are needed to fully comprehend patients' needs and perceptions. Therefore, the availability of reliable, valid, and comprehensive questionnaires for patients is essential. The aim of this study is to identify PROMs measuring the impact of orthognathic surgery on quality of life in patients with cleft deformities and to evaluate the identified PROMs. Methods : A systematic search of the literature was performed according to the preferred reporting items for systematic reviews and meta-analysis guidelines. All validated PROMs, regarding the impact of orthognathic surgery on quality of life in patients with cleft deformities, were identified and assessed according to the quality criteria proposed for measurement properties of health status questionnaires. Results : An electronic search yielded 577 articles. After a full-text review of 87 articles, 4 articles met the inclusion criteria, comprising 58 PROMs. Of these 58 PROMs, 1 PROM (the CLEFT-Q) has been validated to measure the impact of orthognathic surgery on patients with a facial cleft. Evaluation of methodological quality of the included articles and assessment of the measurement properties of the CLEFT-Q show that the CLEFT-Q scores relatively good for all available measurement properties, making it suitable for immediate use. Conclusion: The CLEFT-Q was found to be the only valid instrument so far to measure the impact of orthognathic surgery on the quality of life in patients with cleft deformities. [ABSTRACT FROM AUTHOR]

Objective: There have been few studies on the anatomy of palatine aponeurosis (PA). Herein, we elucidated the relationship between the PA and soft palate muscles and pharyngeal muscles. Design: Two cadaveric specimens were dissected to observe the gross anatomy of the PA. Six cadaveric specimens were processed and scanned by micro-computed tomography to determine the elaborate anatomy. Images were exported to Mimics software to reconstruct a three-dimensional model. Results: The PA covered the anterior (32.1%-38.8%) of the soft palate, extending from the tensor veli palatini (TVP) and connecting to 3 muscles: palatopharyngeus (PP), uvula muscle, and superior pharyngeal constrictor (SC). The SC and PP are attached to the PA on the medial side of the pterygoid hamulus. SC muscle fibers were attached to the hamulus, forming a distinct gap between the hamulus. Some muscle fibers of the PP and uvula originated from the PA. The PA extended from the TVP to the midline and the posterior edge of the hard palate. The PA was not uniformly distributed, which was complementary to the attached muscles in thickness. Conclusions: PA, as a flexible fibrous membrane, maintains the shape of the soft palate. It extends from the TVP and covers anteriorly about one-third of the soft palate. The PA provides a platform for the soft palate muscles and pharyngeal muscles, connecting to the PP, uvula muscle, and SC. These muscles are important for palatopharyngeal closure and middle-ear function. It is necessary to minimize the damage to the PA during surgical interventions. [ABSTRACT FROM AUTHOR]

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. Setting: Seven cleft centres in Canada, the USA, and UK were involved. Patients/Participants: Patients were aged eight to 29 years with CL/P. Interventions: Before undergoing rhinoplasty, orthognathic, cleft lip scar revision, and alveolar bone graft, participants were asked to complete the following PROMs: CLEFT-Q (9 scales), Child Oral Health Impact Profile (socio-emotional subscale) and Cleft Hearing Appearance and Speech Questionnaire (features 1 subscale). Main Outcome Measure(s): The correlation coefficients examining the relationship between the scales were the main outcome measures. Correlations (Spearman) were calculated and interpreted as follows: <0.3 weak, 0.30 to 0.50 moderate, ≥0.50 strong. Results: Participants (n = 177) were mostly male (61%) and aged between eight and 11 years (42%). Overall, 38 of 52 (73%) hypotheses tested were supported. More specifically, 20 of 26 (77%) hypotheses about correlations between the appearance scales were supported, two of three (67%) hypotheses about correlations between the health-related quality of life scales were supported, and 16 of 23 (70%) hypotheses about correlations between the appearance and health-related quality of life scales were supported. Conclusions: Cross-sectional construct validity of the CLEFT-Q scales adds further evidence of the psychometric properties of this instrument. [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]

Objective: To investigate the effect of laser bio-modulation irradiation therapy on the scar after surgical correction of unilateral cleft lip. Design: a comparative, open-label study. Setting: we conducted the study in a university based tertiary hospital that recruited early wound healers of unilateral cleft lip correction. Patients: Eighty patients were divided into two groups: In study's group, patients undergo laser bio-modulation irradiation (n = 60); in the control group, patients were followed-up without intervention (n = 20). Intervention: In the study's group, patients underwent low-power diode Laser with wavelength of 806 nm and power of 100 mw. Main outcome: The change in the scar of cleft lip patients, which was assessed by clinical examination and ultrasound. Results: The median pigmentation score was significantly lower in the laser group (median = 1; IQR = 1–2) than the control group (median 2; IQR 1–3), with p-value of <0.001. Likewise, the median height score was significantly lower in the laser group (median = 1; IQR = 1–1) than the control group (median 1.5; IQR 1.5–2), with p-value of 0.001. The median pliability score was significantly lower in the laser group (median = 1; IQR = 1–1) than the control group (median 2.5; IQR 1–3), with p-value of <0.001. Finally, the median vascularity score was significantly lower in the laser group (median = 1; IQR = 1–1) than the control group (median 1.5; IQR 1–2), with p-value of <0.001. Conclusion: laser bio-modulation irradiation therapy demonstrates a potential efficacy in managing the hypertrophic scars after surgical repair of unilateral cleft lip. [ABSTRACT FROM AUTHOR]

Objective : This study aimed to describe the social determinants of health (SDoH) for patients receiving multidisciplinary team care in a Cleft Palate-Craniofacial program, develop responsive and consistent processes to include trauma-informed psychosocial histories, promote discussions about additional "non-medical" factors influencing health and surgical outcomes, and demonstrate that these activities are feasible in the context of multidisciplinary patient-provider interactions. Design : Single-site, cross-sectional study using a questionnaire. Setting : Participants were recruited from a provincial quaternary care Cleft Palate-Craniofacial program at British Columbia Children's Hospital in Vancouver, BC, Canada. Participants : 290 families completed the questionnaire. Results : 34% of families experience significant barriers to accessing primary health care, 51% struggle financially, and 11% scored four or more on the Adverse Childhood Experiences scale. Furthermore, 47% reported not having adequate social support in their lives, and 5% reported not feeling resilient at the time of the survey. Conclusion : Patients with cleft and craniofacial anomalies have complex needs that extend beyond the surgical and medical care they receive. It is critical that all Cleft and Craniofacial teams incorporate social histories into their clinic workflow and be responsive to these additional needs. Discussions surrounding SDoH and adversity are welcomed by families; being involved in the care and decision-making plans is highly valued. Healthcare providers can and should ask about SDoH and advocate for universal access to responsive, site-based, social work support for their patients. [ABSTRACT FROM AUTHOR]

Background: Higher rates of postoperative complication following cleft lip or palate repair have been documented in low resource settings, but their causes remain unclear. This study sought to delineate patient, surgeon, and care environment factors in cleft complications in a low-income country. Design: Prospective outcomes study. Setting: Comprehensive Cleft Care Center. Patients: Candidate patients presenting for cleft lip or palate repair or revision. Interventions: Patient anthropometric, nutritional, environmental and peri- and post-operative care factors were collected. Post-operative evaluation occurred at standard 1-week and 2-month postoperative intervals. Main Outcome Measures: Complication was defined as fistula, dehiscence and/or infection. Results: Among 408 patients enrolled, 380 (93%) underwent surgery, of which 208 (55%) underwent lip repair (124) or revision (84), and 178 (47%) underwent palate repair (96) or revision (82). 322 (85%) were evaluated 1 week and 166 (44%) 2 months postoperatively. 50(16%) complications were identified, including: 25(8%) fistulas, 24(7%) dehiscences, 17(5%) infections. Mid-upper arm circumference (MUAC) ≤12.5 cm was associated with dehiscence after primary lip repair (OR = 28, p = 0.02). Leukocytosis ≥11,500 on pre-operative evaluation was associated with dehiscence (OR = 2.51, p = 0.04) or palate revision fistula (OR = 64, p < 0.001). Surgeons who performed fewer previous-year palate repairs had higher likelihood of palate complications, (OR = 3.03, p = 0.01) although there was no difference in complication rate with years of surgeon experience or duration of surgery. Conclusions: Multiple patient, surgeon, and perioperative factors are associated with higher rates of complication in a low-resource setting, and are potentially modifiable to reduce complications following cleft surgery. [ABSTRACT FROM AUTHOR]

Objective: Oculoauriculovertebral spectrum (OAVS) is a genetically and clinically heterogeneous disorder that occurs due to a developmental field defect of the first and second pharyngeal arches. Even though recent whole exome sequencing studies (WES) have led to identification of several genes associated with this spectrum in a subset of individuals, complete pathogenesis of OAVS remains unsolved. In this study, molecular genetic etiology of OAVS was systematically investigated. Design/Setting/Patients: A cohort of 23 Turkish patients with OAVS, referred to Hacettepe University Hospital, Department of Pediatric Genetics from 2008 to 2018, was included in this study. Minimal diagnostic criteria for OAVS were considered as unilateral microtia or hemifacial microsomia with preauricular skin tag. The cohort was clinically reevaluated for craniofacial and extracranial findings. Molecular etiology was investigated using candidate gene sequencing following copy number variant (CNV) analysis. WES was also performed for 2 of the selected patients. Results: Patients in the study cohort presented similar demographic and phenotypic characteristics to previously described patients in the literature except for a higher frequency of bilaterality, cardiac findings, and intellectual disability/developmental delay. CNV analysis revealed a possible genetic etiology for 3 patients (13%). Additional WES in 1 of the 2 patients uncovered a novel heterozygous nonsense variant in Elongation factor Tu GTP-binding domain-containing 2 (EFTUD2) causing mandibulofacial dysostosis with microcephaly (MFDM), which clinically overlaps with OAVS. Conclusion: Detailed clinical evaluation for any patient with OAVS is recommended due to a high rate of accompanying systemic findings. We further expand the existing genetic heterogeneity of OAVS by identifying several CNVs and a phenotypically overlapping disorder, MFDM. [ABSTRACT FROM AUTHOR]

Objective: The aim of this study was to confirm the pathogenic variants, explore the genotype–phenotype correlation and characteristics of Chinese patients with Treacher Collins syndrome (TCS). Design: Clinical details of 3 TCS family cases and 2 sporadic cases were collected and analyzed. Whole-exome sequencing and Sanger sequencing were conducted to detect causative variants. Setting: Tertiary clinical care. Patients: This study included 8 patients clinically diagnosed with TCS who were from 3 familial cases and 2 sporadic cases. Main Outcome Measures: When filtering the database, variants were saved as rare variants if their frequency were less than 0.005 in the 1000 Genomes Project Database, the Exome Aggregation Consortium (ExAC) browser, and the Novogene database, or they would be removed as common ones. The pathogenic variants identified were verified by polymerase chain reaction. The sequencing results were analyzed by Chromas 2.1 software. Results: Two novel pathogenic variants (NM_000356.3: c.537del and NM_000356.3: c.1965_1966dupGG) and 2 known pathogenic variants (NM_000356.3: c.1535del, NM_000356.3: c.4131_4135del) were identified within TCOF1 which are predicted to lead to premature termination codons resulting in a truncated protein. There was a known missense SNP (NM_015972.3: c.139G>A) within POLR1D. No phenotype–genotype correlation was observed. Instead, these 8 patients demonstrated the high genotypic and phenotypic heterogeneity of TCS. Conclusions: This study expands on the pathogenic gene pool of Chinese patients with TCS. Besides the great variation among patients which is similar to international reports, Chinese patients have their own characteristics in clinical phenotype and pathogenesis mutations. [ABSTRACT FROM AUTHOR]

Objective: To date, the recording of outcomes of interventions for velopharyngeal dysfunction (VPD) has not been standardized. This makes a comparison of results between studies challenging. The aim of this study was to develop a core outcome set (COS) for reporting outcomes in studies examining the management of VPD. Design: A two-round Delphi consensus process was used to develop the COS. Patients, Participants: The expert Delphi panel comprised patients and caregivers of patients with VPD, surgeons and speech and language therapists specializing in cleft palate, and researchers with expertise in VPD. Interventions: A long list of outcomes was derived from the published literature. In each round of a Delphi survey, participants were asked to score outcomes using the Grading of Recommendations, Assessment, Development, and Evaluations scale of 1 to 9, with 1 to 3 labeled "not important," 4 to 6 labeled "important but not critical," and 7 to 9 labeled "critical." Main outcome measure: Consensus criteria were specified a priori. Outcomes with a rating of 75% or more of the panel rating 7 to 9 and 25% or fewer rating 1 to 3 were included in the COS. Results: A total of 31 core outcomes were identified from the Delphi process. This list was condensed to combine topic areas to produce a final COS of 10 outcomes, including both processes of care and patient-reported outcomes that should be considered for reporting in future studies of VPD. Conclusions: Implementation of the COS-VPD will facilitate consistency of outcomes data collection and comparison of results across studies. [ABSTRACT FROM AUTHOR]

Objective: To determine the current applications of 3-dimensional (3D) printing in the care of patients with cleft lip and palate. We also reviewed 3D printing limitations, financial analysis, and future implications. Design: Retrospective systematic review. Methods: Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines were used by 3 independent reviewers. Articles were identified from Cochrane library, Ovid Medline, and Embase. Search terms included 3D printing, 3 dimensional printing, additive manufacturing, rapid prototyping, cleft lip, and cleft palate. Exclusion criteria included articles not in English, animal studies, reviews without original data, oral presentations, abstracts, opinion pieces, and articles without relevance to 3D printing or cleft lip and palate. Main Outcome Measures: Primary outcome measure was the purpose of 3D printing in the care of patients with cleft lip and palate. Secondary outcome measures were cost analysis and clinical outcomes. Results: Eight-four articles were identified, and 39 met inclusion/exclusion criteria. Eleven studies used 3D printing models for nasoalveolar molding. Patient-specific implants were developed via 3D printing in 6 articles. Surgical planning was conducted via 3D printing in 8 studies. Eight articles utilized 3D printing for anatomic models/educational purposes. 3-Dimensional printed models were used for surgical simulation/training in 6 articles. Bioprinting was utilized in 4 studies. Secondary outcome of cost was addressed in 8 articles. Conclusion: 3-Dimensional printing for the care of patients with cleft lip and palate has several applications. Potential advantages of utilizing this technology are demonstrated; however, literature is largely descriptive in nature with few clinical outcome measures. Future direction should be aimed at standardized reporting to include clinical outcomes, cost, material, printing method, and results. [ABSTRACT FROM AUTHOR]

Objective: To report speech outcomes following Orticochea pharyngoplasty in 43 patients with cleft palate and noncleft velopharyngeal dysfunction. Design: A retrospective surgical audit of patients undergoing Orticochea pharyngoplasty between 2004 and 2012, with speech as a primary outcome measure. Setting: Patients known to a regional UK cleft center. Methods: Forty-three patients underwent Orticochea pharyngoplasty by a single surgeon in a UK regional cleft center. Twenty-one patients had undergone a prior procedure for velopharyngeal dysfunction. Pre- and postoperative speech samples were assessed blindly using the Cleft Audit Protocol for Speech-Augmented by a specialist cleft speech and language therapist, external to the team. Speech samples were rated on the following parameters: hypernasality, hyponasality, audible nasal emission, nasal, turbulence, and passive cleft speech characteristics. Statistical differences in pre- and postoperative speech scores were tested using the Wilcoxon matched-pairs signed-ranks test. Inter- and intrareliability scores were calculated using weighted Cohen κ. Results: Whole group: A statistically significant difference in pre- and postoperative scores for hypernasality (P <.001), hyponasality (P <.05), nasal emission (P <.01), and passive cleft speech characteristics (P <.01) were reported. Patients with cleft diagnoses: A statistically significant difference in scores for hypernasality (P <.001), nasal emission (P <.01), and passive cleft speech characteristics (P <.01) were reported for this group of patients. Patients with noncleft diagnoses: The only parameter to demonstrate a statistically significant difference was hypernasality (P <.01) in this group. Conclusions: Orticochea pharyngoplasty is a successful surgical procedure in treating velopharyngeal dysfunction in both the cleft and noncleft populations. [ABSTRACT FROM AUTHOR]

Aim: To assess occlusal outcomes of orthodontic treatment for patients with complete cleft lip and palate. Design: Retrospective assessment using the Peer Assessment Rating (PAR) index. Setting: Consecutive patients treated by one consultant orthodontist at a tertiary care cleft center. Participants: One hundred twenty-seven patients with either complete unilateral cleft lip and palate (UCLP) or bilateral cleft lip and palate (BCLP) consecutively treated with fixed appliances. Intervention: Fixed orthodontic appliance treatment and orthognathic surgery when required. Outcomes: The PAR index assessment was carried out by a calibrated-independent assessor. Treatment duration, the number of patient visits, and data on dental anomalies were drawn from patient records and radiographs. Results: One hundred two patients' study models were assessed after exclusions. Mean start PAR score for UCLP (n = 71) was 43.9 (95% CI, 41.2-46.6, SD 11.5), with a mean score reduction of 84.3% (95% CI, 81.9-86.7, SD 10.1). The UCLP mean treatment time was 23.7 months with 20.1 appointments. Mean start PAR score for BCLP (n = 31) was 43.4 (95% CI, 39.2-47.6, SD 11.4), with a mean score reduction of 80.9% (95% CI, 76.3-85.5, SD 12.5). The BCLP mean treatment time was 27.8 months with 20.5 appointments. Conclusion: These results compare well with other outcome reports, including those for patients without a cleft, and reflect the standard of care provided by an experienced cleft orthodontist. As with high-volume surgeons, orthodontic treatment for this high need group is favorable when provided by a high-volume orthodontist. These findings may be used for comparative audit with similar units providing cleft care. [ABSTRACT FROM AUTHOR]

Objective: To assess whether children with torticollis have quantifiably greater facial asymmetry than their age-matched controls using 3-dimensional (3D) photogrammetry. Design: We retrospectively analyzed patients diagnosed with torticollis and age-matched volunteers who underwent 3D photogrammetry of their faces. We calculated the root mean square deviation (RMSD) between native and reflected facial images, as a measure of asymmetry. Two observers independently measured RMSD values for all study participants. The Spearman correlation coefficient evaluated interobserver reliability. The Wilcoxon rank-sum test with Bonferroni adjusted P values for multiple comparisons. Setting: Institutional. Participants: Twenty patients diagnosed with torticollis and 12 age-matched volunteers. Patients were analyzed on a computer database and volunteers were selected and consented in the hospital. We excluded patients with a history of facial trauma, facial operations, or other craniofacial diagnoses. Interventions: Facial surface scans were obtained using the Canfield Vectra stereophotogrammetry system. The technology captures surface anatomy without radiation. Main Outcome Measures: RMSD comparisons between patients with torticollis and age-matched controls. Results: Compared to controls, patients with torticollis had statistically significant greater full face, upper third, and middle third facial asymmetry. There was a trend toward greater asymmetry of the lower facial third. Conclusions: We used 3D photogrammetry to quantitate facial asymmetry from torticollis. We found greater asymmetry in patients with torticollis than in their unaffected peers. All areas of the face appeared to be affected, though the asymmetry in the lower facial third just failed to reach significance. [ABSTRACT FROM AUTHOR]

Objective: This study evaluates skeletal and dental outcomes of LeFort I advancement surgery in patients with cleft lip and palate (CLP) with varying degrees of maxillary skeletal hypoplasia. Design: Retrospective study. Method: Lateral cephalograms were digitized at preoperative (T1), immediately postoperative (T2), and 1-year follow-up (T3) and compared to untreated unaffected controls. Based on the severity of cleft maxillary hypoplasia, the sample was divided into 3 groups using Wits analysis: mild : ≤0 to ≥−5 mm; moderate : <−5 to >−10 mm; and severe : ≤−10 mm. Participants: Fifty-one patients with nonsyndromic CLP with hypoplastic maxilla who met inclusion criteria. Intervention: LeFort I advancement. Main Outcome Measure: Skeletal and dental stability post-LeFort I surgery at a 1-year follow-up. Results: At T2, LeFort I surgery produced an average correction of maxillary hypoplasia by 6.4 ± 0.6, 8.1 ± 0.4, and 10.7 ± 0.8 mm in the mild, moderate, and severe groups, respectively. There was a mean relapse of 1 to 1.5 mm observed in all groups. At T3, no statistically significant differences were observed between the surgical groups and controls at angle Sella, Nasion, A point (SNA), A point, Nasion, B point (ANB), and overjet outcome measures. Conclusions: LeFort I advancement produces a stable correction in mild, moderate, and severe skeletal maxillary hypoplasia. Overcorrection is recommended in all patients with CLP to compensate for the expected postsurgical skeletal relapse. [ABSTRACT FROM AUTHOR]

Objective: The aim of this study is to compare the short- and long-term reproducibility of verbal and nonverbal facial expressions of normal people using dynamic 3-dimensional (3-D) imaging. Design: Prospective, cross-sectional, controlled study. Setting: Peking University School and Hospital of Stomatology, Beijing, China. Patients and Participants: Twenty-seven participants, 12 males and 15 females, were recruited for this study. Methods: A 3-D dynamic system was applied to capture the process of 4 nonverbal facial expressions (smile lips closed, smile lips open, lip purse, cheek puff) and 2 verbal facial expressions (/i:/, /u:/) at an initial time point, 15 minutes later, and 1 week later. Key frames were selected from each expression recording sequence. Main Outcome Measures: The root mean square (RMS) between each key frame and its corresponding frame at rest was calculated. ΔRMS reflected the difference of the same key frames between the different sessions of the same expression of the same participant. The reproducibility of different facial expressions at different time intervals were analyzed. Results: There was no significant difference in verbal and nonverbal expression repeatability during a 15-minute interval, except for cheek puff motion. Following a 1-week interval, verbal expression repeatability was superior to that of nonverbal expressions (P <.01). Compared with nonverbal expressions, the repeatability of verbal expressions did not obviously decrease with the increase in recording interval. Conclusions: Dynamic 3-D imaging is a useful technique for facial expression analysis. Verbal expressions showed greater reproducibility than nonverbal expressions. [ABSTRACT FROM AUTHOR]

Objective: Patients with Richieri-Costa-Pereira syndrome (RCPS) present severe craniofacial alterations and frequently require orthodontic and surgical procedures. Thus, this study aims to describe the craniofacial relationships in patients with RCPS. Design: Panoramic radiographs and lateral cephalometric teleradiographs of 7 patients with RCPS and 7 age- and sex-matched nonsyndromic patients were analyzed. Cephalometric measurements were used to determine the size of apical bases, the relationship between them, the pattern of craniofacial growth, and the facial heights of the patients. Interobservers' concordance was verified by intraclass coefficient. For comparison between the groups, paired t test was employed. P values <.05 indicated statistical significance. Results: Average age of patients with RCPS was 18.5 years. Six patients were female. All patients with RCPS had Pierre-Robin sequence while 2 also presented cleft mandible. Most patients with RCPS had missing lower central incisors (100%), lower lateral incisors (85.7%), lower second premolars (85.7%), and/or upper lateral incisors (57.1%). Concordance between observers was excellent for all cephalometric measurements (0.87-0.99). Patients with RCPS presented severe craniofacial alterations when compared to control group: sella–nasion–B point (SNB) angle (73.8o ± 4.86o vs 78.85o ± 4.53o, P =.029), maxillary length (7.89 cm ± 0.58 cm vs 16.36 cm ± 0.75 cm, P =.001), mandibular length (9.90 cm ± 0.46 cm vs 20.61 cm ± 0.45 cm, P =.001), upper anterior face height (5.41 cm ± 0.50 cm vs 9.40 cm ± 0.47 cm, P =.001), lower anterior face height (5.48 cm ± 0.75 cm vs 11.66 cm ± 0.55 cm, P =.001), and posterior face height (6.70 cm ± 0.33 cm vs 13.65 cm ± 1.06 cm, P =.001). There was no difference in SNB, A point–nasion–B point, pogonion–nasion–B point, and mandibular place angles between the groups (P >.05). Conclusion: Patients with RCPS present deficient development of maxilla and mandible when compared with nonsyndromic patients. [ABSTRACT FROM AUTHOR]

Objective: To gain more insight into the assessment of "atypical" nasal and lip appearance outcomes compared to "typical" appearance outcomes after unilateral cleft lip and palate (UCLP) repair, when judged by professionals, patients with repaired UCLP, and laypeople. Design: An online survey containing 3 series of photographs with various degrees of "typical" and "atypical" nasal and lip appearance outcomes after UCLP repair was sent to 30 professionals, 30 patients with repaired UCLP, and 50 laypeople in 2 countries. Participants were instructed to rank the photographs from excellent to poor based on overall appearance. Mean rank positions of photographs were analyzed and differences in mean rank score between "typical" and "atypical" results were assessed using a T-test. Agreement of ranking between the 3 groups was assessed with an analysis of variance analysis. Setting: Amsterdam UMC, location VUmc, Netherlands and Boston Children's Hospital, Boston, USA. Patients: Photographs of 6- to 18-year-old patients with repaired UCLP. Results: "Atypical" appearance outcomes were ranked significantly less favorably (small nostril: P = 0.00; low vermillion border: P = 0.02; whistling deformity: P = 0.00) compared to "typical" outcomes. Difference between professionals, patients and laypeople in rank positioning the photographs was not statistically significant (P = 0.89). Conclusions: Noses with a smaller nostril and lips containing a whistling deformity were perceived as poorer outcome compared to the "typical" results. Professionals, patients, and laypeople are in agreement when assessing these outcomes. [ABSTRACT FROM AUTHOR]

Introduction: Oronasal fistulae following palatoplasty may affect patients' quality of life by impacting their ability to eat, speak, and maintain oral hygiene. We aimed to quantify the impact of previous oronasal fistula repair on patients' quality of life using patient-reported outcome psychometric tools. Methods: A cross-sectional study of 8- to 9-year-old patients with cleft palate and/or lip was completed. Patients who had a cleft team clinic between September 2018 and August 2019 were recruited. Participants were divided into 2 groups (no fistula, prior fistula repair). Differences in the individual CLEFT-Q and Child Oral Health Impact Profile-Short Form 19 (COHIP-SF 19) Oral Health scores between the 2 groups were evaluated using a multivariate analysis controlling for Veau classification and syndromic diagnosis. Results: Sixty patients with a history of cleft palate were included. Forty-two (70%) patients had an associated cleft lip. Thirty-two (53.3%) patients had no history of fistula and 28 (46.7%) patients had undergone a fistula repair. CLEFT-Q Dental, Jaw, and Speech Function were all higher in patients without a history of a fistula repair; however, none of these differences were statistically significant. The COHIP-SF 19 Oral Health score demonstrated a significantly lower score in the fistula group, indicating poorer oral health (P =.05). Conclusions: One would expect that successful repair of a fistula would result in improved function and patient satisfaction, but the consistent trend toward lower CLEFT-Q scores and significantly increased COHIP-SF 19 Oral Health scores in our study group suggests that residual effects linger and that the morbidity of a fistula may not be completely treated with a secondary correction. [ABSTRACT FROM AUTHOR]

Objective: To evaluate evolving practice patterns in secondary cleft rhinoplasty. Design: Retrospective review of data submitted during Maintenance of Certification (MOC). Setting: Evaluation of MOC data from the American Board of Plastic Surgery. Participants: Tracer data for secondary cleft rhinoplasty were reviewed from August 2006 through March 2020, and the data subdivided from 20062012 and 20132020 to evaluate changes in practice patterns. Interventions: Practice patterns in tracer data were compared to those from evidence-based medicine (EBM) literature over this time period. Main Outcome Measures: Practice patterns were compared to EBM trends during the study period. Results: A total of 90 cases of secondary cleft rhinoplasty were identified. The average age at operation was 13 years (range 4-77). Cumulative data demonstrated 61% to present with nasal airway obstruction and 21% to have undergone primary nasal correction at the time of cleft lip repair; 72% of patients experienced no complications, with the most common complications being asymmetry (10%) and vertical asymmetry of alar dome position (6%). Cartilage graft was used in 68% of cases, with 32% employing septal cartilage. Change in practice patterns between 2006 to 2012 and 2013 to 2020 demonstrated increase in dorsal nasal surgery (26% vs 43%, P =.034), use of osteotomies (14% vs 38%, P =.010), septal resection and/or straightening (26% vs 48%, P =.034), and turbinate reduction (8% vs 30%, P =.007). Conclusions: These tracer data provide long-term data by which to evaluate evolving practice patterns for secondary cleft rhinoplasty. When evaluated relative to EBM literature, future research to further improve outcomes can be better directed. [ABSTRACT FROM AUTHOR]