Cleft Palate-Craniofacial Journal最新文献

Objective: To describe factors affecting anxiety, depression, stress, and self-esteem in parents of children with microtia.

Design: Cross-sectional correlational study.

Participants: Parents (N = 117) of children with microtia were recruited nationally through a Turkish craniofacial non-governmental organization's social media.

Main outcome measures: Demographics, parental report of microtia experiences and concerns, the Depression Anxiety Stress Scale, and the Rosenberg Self-Esteem Scale.

Results: The mean parental age was 35.61 ± 5.96 years and 74.4% were female. The mean age of their children was 6.08 ± 4.22 years, 69.2% were male, 93.8% had unilateral microtia, and 14.5% had ear reconstruction. Some parents had low self-esteem (24.8%) and were in the severe range for depression (30.7%), anxiety (25.6%), and stress (21.4%). Depression (M = 7.43, SD = 5.35) was associated with worry about child's future (β = 0.25, P = .013), microtia information given around birth (β = -0.20, P = .024), and child no ear surgery (β = -0.23, P = .008), while stress (M = 8.21, SD = 5.37) was associated with worry about the child's future (β = 0.28, P = .008). Parental self-esteem (M = 29.59, SD = 5.10) was related to child having ear surgery (β = 0.19, P = .047) and information about microtia provided around birth (β = 0.22, P = .018). There were no significant associations with Anxiety (M = 6.04, SD = 4.82) identified by multiple regression.

Conclusions: While most parents were in the average to moderate range for clinical concerns, a fifth to a third of participants were in the severe range for depression, anxiety, and stress and a quarter of participants for low self-esteem. Informing families about the microtia treatment process, having the child have surgery, and interventions for reducing their future concerns may provide psychological relief for them.

Objective: Apert syndrome (AS) is a rare congenital craniofacial disorder that requires a multidisciplinary approach to treatment and multiple surgeries. Given that cleft palate (CP) is presented in some of these cases, this poses an additional risk of aggravating obstructed airways after closure. The timing and outcome of CP repair in these patients remains disputed and requires additional attention.

Design: This retrospective analysis included patients diagnosed with CP and AS, born between 1950 and 2020, and treated at our institution. Data were collected from medical records and evaluated using descriptive statistics.

Setting: Data analyses were conducted at Sahlgrenska University Hospital in Gothenburg, Sweden.

Patients/participants: A registry of 83 patients with AS resulted in a cohort of 26 patients also presenting with CP.

Main outcome measures: Postoperative complications, requirement for intensive care, and reoperations following CP repair.

Results: CP incidence among all registered patients was 31%. Patients undergoing CP repair at low age (mean: 22.5 months) tended to experience more frequent postoperative complications and requirements for intensive care. Among the evaluated cohort with medical records describing CP repair (n = 14), 29% experienced postoperative complications, all of which involved aggravation of obstructed airways.

Conclusions: This study highlights the importance of airway assessment before and after CP repair in AS. The findings suggest that surgical outcomes might benefit from postponing CP repair, avoiding combined surgeries, and operating in two stages when indicated. However, additional and larger studies are required.

Objective: Children with cleft lip and/or palate (CL/P) are at increased risk for Sleep Disordered Breathing (SDB), particularly Obstructive Sleep Apnea (OSA). At our institution, routine screening for SDB is performed using the Chevrin Pediatric Sleep Questionnaire (PSQ). This analysis is a practice audit looking at the outcomes of screening children with CL/P.

Design/setting/patients/participants: A single-center, retrospective analysis was done of all non-syndromic patients with CL/P over the age of 36 months over a 4-year period. Children with known OSA were eliminated from analysis.

Main outcome measures: Univariate logistic regression was used to assess predictors for SDB (PSQ score > 8) amongst various patient, disease, and treatment characteristics. Outcomes of those screened were tracked.

Results: Of the 239 patients in the study cohort, 43 (18%) had positive PSQs. These subjects were more likely to have class III dental occlusion with maxillary retrusion (OR = 2.65, 95% CI: 1.2-5.8, p = 0.02). There were no differences amongst age, type of cleft, Veau classification, BMI, or history of pharyngeal surgery. One third of the group did not complete recommended testing. Twenty-five subjects with positive sleep screening underwent subsequent polysomnography and 21 (84%) had OSA.

Conclusion: Routine screening reveals a significant proportion of patients with CL/P with symptoms suggestive of OSA. While several patients did not complete confirmatory testing, those who completed a PSG had a high rate of identification of OSA. After excluding children with known OSA, patients with SDB are also likely to have class III dental occlusion and maxillary retrusion.

Objective: To present a rare case of an isolated paramedian cleft of the lower lip. We describe the second case of isolated paramedian cleft of the lower lip. The pregnancy was realized in the fifth month prior to which the mother gives history of consumption of non-steroidal anti-inflammatory drugs (N.S.A.I.D.s) and a computed tomography (C.T.) scan. The cleft was repaired with a z-plasty for the mucosa and a z-plasty for the skin. We achieved good vermilion bulk, white roll continuity and functionality. We attributed this cleft to maternal exposure to NSAIDs and radiation during early pregnancy.

Objective: To introduce a highly innovative imaging method to study the complex velopharyngeal (VP) system and introduce the potential future clinical applications of a VP atlas in cleft care.

Design: Four healthy adults participated in a 20-min dynamic magnetic resonance imaging scan that included a high-resolution T2-weighted turbo-spin-echo 3D structural scan and five custom dynamic speech imaging scans. Subjects repeated a variety of phrases when in the scanner as real-time audio was captured.

Setting: Multisite institution and clinical setting.

Participants: Four adult subjects with normal anatomy were recruited for this study.

Main outcome: Establishment of 4-D atlas constructed from dynamic VP MRI data.

Results: Three-dimensional dynamic magnetic resonance imaging was successfully used to obtain high quality dynamic speech scans in an adult population. Scans were able to be re-sliced in various imaging planes. Subject-specific MR data were then reconstructed and time-aligned to create a velopharyngeal atlas representing the averaged physiological movements across the four subjects.

Conclusions: The current preliminary study examined the feasibility of developing a VP atlas for potential clinical applications in cleft care. Our results indicate excellent potential for the development and use of a VP atlas for assessing VP physiology during speech.

To assess the effect of secondary alveolar bone grafting (SABG) on maxillary growth in patients with unilateral cleft lip or palate (UCL/P).

Systematic review and Meta-analysis.

Pubmed, Cochrane Library, Embase, Scopus, Web of Science and manual search was performed to assess the maxillary growth following SABG in UCL/P patients.

Non-syndromic patients with UCL/P.

Comparison of maxillary growth between patients with UCL/P who underwent SABG and UCL/P patients who had not undergone SABG or non-cleft control.

39 of the identified 233 articles were assessed for inclusion and exclusion criteria after duplicate removal and title and abstract reading. 7 articles (1 prospective, and 6 retrospective studies) were included in the qualitative analysis and 2 articles were subjected to quantitative analysis. Four studies had a low risk of bias and three studies had a moderate risk of bias. Meta-analysis revealed a significant reduction of SNA and no significant difference in ANB in the SABG group compared to the non-cleft control group(I²= 0%). There was no significant difference in ANB between SABG and non-cleft control; however, results showed high heterogeneity(I²= 83%). Meta-analysis of SNA and ANB showed no significant difference between SABG and the cleft control group; however, there was high heterogeneity.

The studies showed a low to moderate risk of bias. SABG causes inhibition of maxillary growth in patients with cleft lip or palate when compared to patients with non-cleft control. Due to high heterogeneity, comparison to cleft control showed insufficient evidence.

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.

Assess agreement of hypernasality and audible nasal emission (ANE) ratings between audio-recordings and a clinic setting.

Cross-sectional study using retrospective clinical recordings.

Audio-recording ratings by two trained speech language pathologists.

Percent agreement and intra- and inter-rater reliability of perceptual ratings.

Intra-rater reliability (AC2) of 167 audio-recorded speech samples for the primary and secondary raters, respectively, was 0.82 and 0.79 for hypernasality; for ANE, it was 0.57 and 0.75. Inter-rater reliability was 0.77 for hypernasality and 0.63 for ANE. When comparing ratings made from audio-recording versus the original clinical ratings, intra-rater reliability was 0.85 and 0.61 (primary and secondary rater, respectively) for hypernasality and 0.21 and 0.34 for ANE.

Ratings for hypernasality made from audio recordings were consistent with clinical evaluation, while ratings of ANE were not. ANE ratings made from audio recordings may not be a valid measure of velopharyngeal insufficiency speech characteristics.

Posterior cranial vault distraction osteogenesis (PCVDO) is a relatively new paradigm in the treatment of syndromic craniosynostosis, having first been introduced in 2009. PCVDO directly addresses the underdeveloped cranial vault and appears to allow for a larger increase in intracranial volume when compared to traditional techniques. Although reported as safe in the literature, critical appraisal is still required as PCVDO is a relatively uncommon procedure that may require greater numbers to detect true complication rates. The overall reported incidence of serious complications in PCVDO to date is low. This presentation highlights a rare case of sagittal sinus obstruction following posterior cranial vault distraction and raises questions as to the safest technical considerations when planning the operation.

Objective: Investigate associations between socioeconomic indicators of healthcare access with family compliance with cleft-related otologic and audiologic care within an interdisciplinary model.

Design: Retrospective case series.

Subjects and setting: Children born 2005-2015 who presented to the Cleft-Craniofacial Clinic (CCC) at a quaternary care children's hospital.

Interventions: Associations between main outcome measures and Area Deprivation Index (ADI), median household income for zip code, distance from hospital, and insurance status were evaluated.

Main outcome measures: Cleft types, ages at presentation to outpatient clinic (cleft, otolaryngology, and audiology), and ages at procedures (first tympanostomy tube insertion (TTI), lip repair, and palatoplasty) were measured.

Results: Most patients were male (147/230, 64%) with cleft lip and palate (157/230, 68%). Median age at first cleft, otolaryngology, and audiology visits were 7 days, 86 days, and 5.9 months, respectively. Private insurance predicted lower no-show rates (p = .04). Age at first CCC visit was younger for patients with private insurance (p = .04) and older for those who lived further from the hospital (p = .002). Age at lip repair was positively correlated with national ADI (p = .03). However, no socioeconomic status (SES) proxy or proximity to hospital was associated with delays in first otolaryngology or audiology examination or TTI.

Conclusion: Once children become established within an interdisciplinary CCC, SES appears to bear little influence on cleft-related otologic and audiologic care. Future efforts should aim to elucidate which aspects of the interdisciplinary model maximize multisystem cleft care coordination and increase access for higher risk populations.