Cleft Palate-Craniofacial Journal最新文献

Objective: International outreach for cleft lip and/or palate care has traditionally been characterized by foreign groups and individuals holding surgical outreach trips in low- and middle-income countries. However, this "magic bullet" approach has often been criticized for prioritizing short-term results that may disrupt local workflow. The presence and impact of local organizations that support cleft care and take on capacity building initiatives has not been well explored.

Design: Eight countries previously studied as having the highest Google search demand for CL/P were chosen for the scope of the study. Local NGOs in regions were identified through a web search, and information was collected regarding the location, objectives, partnerships, and work conducted thus far.

Results: Countries with a strong combination of local and international organizations included Ghana, Philippines, Nepal, Kenya, Pakistan, India, and Nigeria. The country with minimal to no local NGO presence included Zimbabwe. Local NGOs often supported education and research, training of providers and staff, spreading community awareness, offering interdisciplinary care, and opening cleft clinics and hospitals. Unique initiatives included starting the first school for children with CL/P, enrolling patients in the national healthcare to cover CL/P care, and monitoring the referral system to improve efficiencies in the healthcare system.

Conclusions: Moving towards a mindset of capacity building not only involves bilateral partnerships between international host sites and visiting organizations, but also collaboration with local NGOs that have a deep understanding of local communities. Successful partnerships may help address the complex challenges regarding CL/P care faced by LMICs.

Objective: To discuss advantages, disadvantages and strategical application of anatomical subunit approximation technique in complete unilateral cleft lip repair.

Design: Analysis of consecutive 28 cases.

Setting: A single surgeon experience at a university hospital.

Patients: Among 77 patients who underwent cleft lip repair between May 5, 2019 and June 30, 2021, 28 patients with complete unilateral cleft lip who received cheiloplasty by author's technique.

Main outcome measures: Surgical outcomes were assessed by frontal view medical photographs obtained postoperatively at follow-up visits.

Results: The average length difference between cleft and noncleft sides was <10% for transverse lip length and philtral height. Nostril sill height demonstrated about twenty percent of difference with much variable results. Although the one sample t-test revealed that differences exist between the cleft and noncleft side, they were within an acceptable range.

Conclusions: Understanding the strengths and limitations of anatomical subunit approximation technique enabled strategic application in complete unilateral cleft lip repair. Satisfactory outcomes were achieved not only regarding the scar placement but also regarding the symmetry of the lips and augmentation of nasal sill.

Objectives: Few evidence-based psychosocial programs exist within craniofacial care. This study (a) assessed feasibility and acceptability of the Promoting Resilience in Stress Management-Parent (PRISM-P) intervention among caregivers of children with craniofacial conditions and (b) described barriers and facilitators of caregiver resilience to inform program adaptation.

Design: In this single-arm cohort study, participants completed a baseline demographic questionnaire, the PRISM-P program, and an exit interview.

Participants: Eligible individuals were English-speaking legal guardians of a child <12-years-old with a craniofacial condition.

Intervention: PRISM-P included 4 modules (stress-management, goal-setting, cognitive-restructuring, meaning-making) delivered in 2 one-on-one phone or videoconference sessions 1-2 weeks apart.

Main outcome measures: Feasibility was defined as >70% program completion among enrolled participants; acceptability was defined as >70% willingness to recommend PRISM-P. Intervention feedback and caregiver-perceived barriers and facilitators of resilience were summarized qualitatively.

Results: Twenty caregivers were approached and 12 (60%) enrolled. The majority were mothers (67%) of a child <1-year-old diagnosed with a cleft lip and/or palate (83%) or craniofacial microsomia (17%). Of these, 8 (67%) completed PRISM-P and 7 (58%) completed interviews; 4 (33%) were lost-to-follow-up before PRISM-P and 1 (8%) before the interview. Feedback was highly positive, with 100% willing to recommend PRISM-P. Perceived barriers to resilience included uncertainty about their child's health; facilitators included social support, parental identity, knowledge, and control.

Conclusions: PRISM-P was acceptable among caregivers of children with craniofacial conditions but not feasible based on program completion rates. Barriers and facilitators of resilience support the appropriateness of PRISM-P for this population and inform adaptation.

To determine the sensitivity and specificity of velar notching seen on nasopharyngoscopy for levator veli palatini (LVP) muscle discontinuity and anterior positioning.

Nasopharyngoscopy and MRI of the velopharynx were performed on patients with VPI as part of their routine clinical care. Two speech-language pathologists independently evaluated nasopharyngoscopy studies for the presence or absence of velar notching. MRI was used to evaluate LVP muscle cohesiveness and position relative to the posterior hard palate. To determine the accuracy of velar notching for detecting LVP muscle discontinuity, sensitivity, specificity, and positive predictive value (PPV) were calculated.

A craniofacial clinic at a large metropolitan hospital.

Participants: Thirty-seven patients who presented with hypernasality and/or audible nasal emission on speech evaluation and completed nasopharyngoscopy and velopharyngeal MRI study as part of their preoperative clinical evaluation.

Among patients with partial or total LVP dehiscence on MRI, presence of a notch accurately identified discontinuity in the LVP 43% (95% CI 22-66%) of the time. In contrast, the absence of a notch accurately indicated LVP continuity 81% (95% CI 54-96%) of the time. The PPV for the presence of notching to identify a discontinuous LVP was 78% (95% CI 49-91%). The distance from the posterior edge of the hard palate to the LVP, known as effective velar length, was similar in patients with and without notching (median 9.8 mm vs 10.5 mm, P = 1.00).

The observation of a velar notch on nasopharyngoscopy is not an accurate predictor of LVP muscle dehiscence or anterior positioning.

The current study examined stop consonant production in children with cleft lip and/or palate (CP ± L) 2-6 months following palatal surgery.

Prospective comparative study.

Multisite institutional.

Participants included 113 children with repaired CP ± L (mean age = 16 months) who were participating in the multicenter CORNET study.

Parents of participants were asked to record approximately two hours of their child's vocalizations/words at home using a Language ENvironmental Analysis (LENA^TM) recorder. Four ten-minute audio-recorded samples of vocalizations were extracted from the original recording for each participant and analyzed for presence of oral stop consonants. A minimum of 100 vocalizations were required for analysis.

Preliminary findings indicate that at least one oral stop was evident in the consonant inventory for 95 of the 113 children (84%) at the time of their post-surgery 16-month recording, and 80 of these children (71%) were producing two or more different stops. Approximately 50% of the children (57/113) produced the three voiced stops, and eight of the children (7%) were producing all six stop consonants.

The findings of this study suggest that the majority of children with repaired CP ± L from English-speaking homes are producing oral stops within six months following palatal surgery. Similar to same-age children without CL ± P, voiced stops were more frequently evident in the children's inventories than voiceless stops. In contrast to findings of previous reports suggesting place of articulation differences, a somewhat comparable percentage of children in this study produced voiced bilabial, alveolar, and velar stops.

Superior Semicircular Canal Dehiscence (SSCD) is a recently-defined developmental defect may be associated with several craniofacial anomalies such as Cleft Lip/Palate (CLP). The present study aimed to compare subjects with unilateral and bilateral CLP and normal controls in terms of Superior Semicircular Canal (SSC) bone thickness and pattern.

A total of 238 Cone Beam Computed Tomography (CBCT) images were collected from 52 unilateral Cleft Lip and Palate (UCLP) subjects (104 temporal bones) and 38 Bilateral Cleft Lip and Palate (BCLP) (76 temporal bones) subjects and 148 healthy controls (296 temporal bones). The SSC bone thickness was measured twice and validated by a maxillofacial radiologist. The samples were then classified into five categories based on bone thickness: papyraceous or thin, normal, thick, pneumatized, and dehiscence. After all, the UCLP, BCLP, and normal control groups were compared concerning the SSC pattern and thickness.

The results revealed no significant difference among the three groups regarding the SSC pattern and thickness based on gender. The SSC patterns (P value = .001) and SSC thickness (0.01) were strongly correlated to the cleft type. The thinnest bone thickness and the highest incidence of SSCD were observed among the subjects with BCLP.

The results showed a significant association between the SSC patterns and SSC thickness and the study groups.

Objective: Families increasingly use online resources to acquire medical information about their child's condition with little understanding of the legitimacy of the source of information or of the information itself. We evaluate the quality and readability of online information related to positional head shape conditions and identify unmet needs for healthcare providers to improve online patient education.

Design: The search terms "flat head baby," "brachycephaly," and "plagiocephaly" were queried on the Google search engine and the first 20 websites for each were reviewed. Included websites were evaluated for quality using the DISCERN Instrument and readability using the Flesch-Kincaid Reading Grade Level (FKGL) and Flesch Reading Ease Score (FRES). Websites were categorized by upload source and results were compared using one-way ANOVA.

Results: 38 websites met inclusion criteria. There was no significant correlation between DISCERN score and Google search rank between the three search terms. Professional organizations provided websites with the highest mean DISCERN score (56.3) and commercial websites with the lowest score (36.6, P = .003), indicating "good" and "poor" quality content, respectively. Readability assessments showed an overall average FKGL of 9.9 and FRES of 54.4, suggesting "fairly difficult". Hospitals provided the most website results and tended to publish lower quality information, yet are the most readable.

Conclusions: High quality websites written at an appropriate reading level for the general public are lacking. A review of online resources for positional head shape conditions can be used to derive recommendations to improve the content of online patient education for pediatric healthcare.

Objective: This study investigates the effectiveness of demineralized bone matrix (DBX) to close alveolar clefts in patients previously treated with bone morphogenic protein-2 (BMP-2) who remained with bone nonunion.

Design: This is an IRB-approved retrospective, single-center study.

Setting: This study was conducted at a tertiary academic center.

Patients/participants: We searched for all surgical encounters with the Current Procedural Terminology (CPT) code 42210 from the years 2013-2019. Included patients were diagnosed with cleft alveolus, previous BMP-2 exposure and required revision bone grafting during mixed dentition for persistent alveolar defects.

Interventions: 17 patients underwent revision alveolar bone grafting (ABG) with either DBX (n = 10) or autograft (n = 7) to repair persistent bony cleft.

Main outcome measure(s): The primary study outcome measured was alveolar bone graft revision failure described as continued alveolar nonunion.

Results: The median age at revision ABG was 13.1 ± 3.3 years, with a mean follow-up time of 4.9 years (1.1-9.2 years). Patients were 53% male, 47% had a unilateral cleft lip and alveolus. 58.8% of patients were treated with DBX in the cleft, 41.2% treated with autograft from iliac crest. Overall, 11.8% (n = 2) of all revisions failed, requiring a second revision. The average time to reoperation was 2.06 years, and both were re-grafted with autograft. There was no statistically significant difference between the type of bone graft source used and the failure rate obtained (P = .1544).

Conclusions: DBX and autologous iliac crest bone grafts achieve similar alveolar union rates during revision ABG in patients treated with previous BMP-2 to the alveolar cleft.

Objective: This study aimed to determine whether increased raising of the back of the tongue is evident in children with repaired cleft palate with or without cleft lip (CP+/-CL). We hypothesized that children with CP+/-CL would show increased raising of the tongue dorsum, a compensatory pattern.

Method: Secondary data analysis of mid-sagittal ultrasound tongue imaging data from 31 children with CP+/-CL and 29 typically developing children were used. We annotated the consonants /ʃ, t, s, k/ at the point of maximum constriction in an /aCa/ environment. Children with CP+/-CL said the tokens 10 times, typically developing children said them once. We automatically fitted splines to the tongue contour and extracted the Dorsum Excursion Index (DEI) for each consonant. This metric measures the relative use of the tongue dorsum, with more posterior consonants having higher values. We compared DEI values across groups and consonants using a linear mixed effects model. DEI was predicted by the interaction of consonant (baseline: /ʃ/) and speaker type (baseline: TD), including by-speaker random slopes for consonant and random intercepts for speaker.

Results: Overall DEI was not higher in children with CP+/-CL compared to typically developing children. Between groups the only significant difference was the position of /k/ relative to /ʃ/, where the difference between these two consonants was smaller in the children with CP+/-CL.

Conclusions: There was no support for the hypothesis that increased raising of the tongue dorsum is a common characteristic in children with repaired CP+/-CL. However, individual children may present with this pattern.

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.