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PURPOSE: Craniofacial microsomia (CFM) is a congenital condition that can be associated with feeding challenges in infants. As part of the larger 'Craniofacial microsomia: Accelerating Research and Education (CARE)' program, this study described caregivers' early feeding experiences. DESIGN AND MATERIALS: US-based caregivers of 34 children with CFM participated in remote narrative interviews. Two authors completed inductive thematic analysis in an iterative process until consensus was reached. RESULTS: Caregivers' narratives outlined the inherent challenges of feeding an infant with special healthcare needs. The first theme 'Navigating Challenges and Managing Expectations' describes the distress participants experienced when they were unable to breastfeed and the negative emotional effect of switching to formula. The second theme 'Making Adaptations' outlines the methods participants tried, including breast pumps and feeding tubes. The third theme 'Accessing Support' describes participants' interactions with healthcare providers and challenges accessing feeding support. The final theme 'Growing from Adversity' recounts participants' relief once their child established a feeding pattern and the personal growth gained from their experiences. CONCLUSIONS: Caregivers reported several feeding related challenges associated with CFM, many of which negatively affected their wellbeing. Negative consequences were particularly pronounced in cases where caregivers' feeding experiences differed from their expectations. Participants identified challenges in accessing reliable feeding information and support. Despite difficult experiences, caregivers cited some positive outcomes, including increased confidence and resilience. PRACTICE IMPLICATIONS: Holistic feeding information and support for families affected by CFM should be inclusive of several feeding methods to improve care delivery, child health, and the caregiver experience.
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Cuidadores , Humanos , Femenino , Masculino , Cuidadores/psicología , Lactante , Adulto , Síndrome de Goldenhar/enfermería , Lactancia Materna/psicología , Investigación Cualitativa , Estados Unidos , Preescolar , Adaptación Psicológica , Recién NacidoRESUMEN
OBJECTIVE: Children with craniofacial microsomia (CFM) have complex healthcare needs, resulting in evaluations and interventions from infancy onward. Yet, little is understood about families' treatment experiences or the impact of CFM on caregivers' well-being. To address this gap, the NIH-funded 'Craniofacial microsomia: Accelerating Research and Education (CARE)' program sought to develop a conceptual thematic framework of caregiver adjustment to CFM. DESIGN: Caregivers reported on their child's medical and surgical history. Narrative interviews were conducted with US caregivers (n = 62) of children aged 3-17 years with CFM. Transcripts were inductively coded and final themes and subthemes were identified. RESULTS: Components of the framework included: 1) Diagnostic Experiences, including pregnancy and birth, initial emotional responses, communication about the diagnosis by healthcare providers, and information-seeking behaviors; 2) Child Health and Healthcare Experiences, including feeding, the child's physical health, burden of care, medical decision-making, surgical experiences, and the perceived quality of care; 3) Child Development, including cognition and behavior, educational provision, social experiences, and emotional well-being; and 4) Family Functioning, including parental well-being, relationships, coping strategies, and personal growth. Participants also identified a series of "high" and "low" points throughout their journey and shared their priorities for future research. CONCLUSIONS: Narrative interviews provided rich insight into caregivers' experiences of having a child with CFM and enabled the development of a conceptual thematic framework to guide clinical care and future research. Information gathered from this study demonstrates the need to incorporate evidence-based psychological support for families into the CFM pathway from birth onward.
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OBJECTIVE: Craniofacial microsomia (CFM) is a broad clinical term used to describe a congenital condition most commonly involving the underdevelopment of the external ear, mandible, soft tissues, and facial nerve. Despite medical advances, understanding of the psychological health and healthcare experiences of individuals with CFM and their caregivers remains limited. This article describes a research program designed to address these knowledge gaps, and identify opportunities for psychosocial intervention and improved healthcare provision. DESIGN: The Craniofacial microsomia: Accelerating Research and Education (CARE) research program aims to: 1) Conduct up to 160 narrative interviews with individuals and caregivers to validate a conceptual framework; 2) Administer an online international survey of up to 800 individuals with CFM and caregivers to identify predictors of psychological distress; 3) Perform up to 60 semi-structured interviews with healthcare providers and advocacy leaders to examine the extent to which current healthcare provisions address identified patient needs; and 4) Establish a participant registry to build a longitudinal database and develop an international community. RESULTS: Teams in the USA and UK have been established, alongside an international, interdisciplinary Advisory Committee. Data analysis for Aim 1 is ongoing and informing the delivery of Aims 2-3. Aim 4 is also in development. A dedicated website serves as a recruitment tool, educational resource, and mechanism for engaging with the CFM community. CONCLUSIONS: The CARE program provides a comprehensive approach to understanding the experiences of individuals with CFM and their caregivers. Challenges encountered and lessons learned are shared for the benefit of the community.
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ABSTRACT: Children with craniofacial microsomia (CFM) are at increased risk for educational and social concerns. This study describes intervention services and frequency of teasing in a multinational population of children with CFM. Caregivers of children with CFM ages 3 to 18âyears in the US and South America were administered a questionnaire. Additional information was gathered from medical charts and photographs. Participants (Nâ=â169) had an average age of 10.1â±â6.2âyears, were primarily male (60%), and from the US (46%) or Colombia (32%). Most participants had microtia and mandibular hypoplasia (70%). They often had unilateral (71%) or bilateral (19%) hearing loss and 53% used a hearing aid. In the US, special education services were provided for 48% of participants enrolled in school; however, similar services were rare (4%) in South America and reflect differences in education systems. Access to any intervention service was higher in the US (80%) than in South America (48%). Caregivers reported children showed diagnosis awareness by an average age of 4.4â±â1.9âyears. Current or past teasing was reported in 41% of the children, starting at a mean age of 6.0â±â2.4âyears, and most often took place at school (86%). As half of the US participants received developmental and academic interventions, providers should screen for needs and facilitate access to services. Given diagnosis awareness at age 4 and teasing at age 6, providers are encouraged to assess for psychosocial concerns and link to resources early in treatment.
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Microtia Congénita , Síndrome de Goldenhar , Adolescente , Cuidadores , Niño , Preescolar , Síndrome de Goldenhar/epidemiología , Humanos , Masculino , Padres , PrevalenciaRESUMEN
OBJECTIVE: The study aim was to assess behavioral adjustment in preschool children with and without craniofacial microsomia (CFM). DESIGN: Multisite cohort study of preschoolers with CFM ("cases") or without CFM ("controls"). PARTICIPANTS: Mothers (89%), fathers (9%), and other caregivers (2%) of 161 preschoolers. OUTCOME MEASURE: Child Behavior Check List (CBCL 1.5-5); linear regressions with standardized effect sizes (ES) adjusted for sociodemographic confounds. RESULTS: Child Behavior Check Lists for 89 cases and 72 controls (average age 38.3 ± 1.9 months). Children were male (54%), white (69%), and of Latino ethnicity (47%). Cases had microtia with mandibular hypoplasia (52%), microtia only (30%), or other CFM-associated features (18%). Nearly 20% of cases had extracranial anomalies. Composite CBCL scores were in the average range compared to test norms and similar for cases and controls. On the subscales, cases' parents reported higher Anxious/Depressed scores (ES = 0.35, P = .04), Stress Problems (ES = 0.40, P = .04), Anxiety Problems (ES = 0.34, P = .04), and Autism Spectrum Problems (ES = 0.41, P = .02); however, the autism subscale primarily reflected speech concerns. Among cases, more problems were reported for children with extracranial anomalies and certain phenotypic categories with small ES. CONCLUSIONS: Behavioral adjustment of preschoolers with CFM was comparable to peers. However, parental reports reflected greater concern for internalizing behaviors; thus, anxiety screening and interventions may benefit children with CFM. Among cases, more problems were reported for those with more complex presentations of CFM. Craniofacial microsomia-related speech problems should be distinguished from associated psychosocial symptoms during developmental evaluations.
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Microtia Congénita , Síndrome de Goldenhar , Adulto , Cuidadores , Preescolar , Estudios de Cohortes , Femenino , Humanos , Masculino , MadresRESUMEN
OBJECTIVE: To examine neurodevelopment in preschool-aged children with craniofacial microsomia (CFM) relative to unaffected peers. DESIGN: Multisite, longitudinal cohort study. SETTING: Tertiary care centers in the United States. PARTICIPANTS: We included 92 children with CFM ("cases") through craniofacial centers and clinics. Seventy-six children without CFM (controls) were included from pediatric practices and community advertisements. This study reports on outcomes assessed when participants were an average age of 38.4 months (SD = 1.9). MAIN OUTCOME MEASURES: We assessed cognitive and motor skills using the Bayley Scales of Infant and Toddler Development, third edition (Bayley-III), and language function using subtests from the Clinical Evaluation of Language Fundamentals-Preschool, second edition (CELF-P2). RESULTS: Case-control differences were negligible for Bayley-III cognitive (effect sizes [ES] = -0.06, P = .72) and motor outcomes (ES = -0.19, P = .25). Cases scored lower than controls on most scales of the CELF-P2 (ES = -0.58 to -0.20, P = .01 to .26). Frequency counts for "developmental delay" (ie, one or more scores > 1 SD below the normative mean) were higher for cases (39%) than controls (15%); however, the adjusted odds ratio = 1.73 (P = 0.21) was not significant. Case-control differences were most evident in children with microtia or other combinations of CFM-related facial features. CONCLUSIONS: Cognitive and motor scores were similar for preschool-aged children with and without CFM. However, children with CFM scored lower than controls on language measures. We recommend early monitoring of language to identify preschoolers with CFM who could benefit from intervention.
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Síndrome de Goldenhar , Niño , Desarrollo Infantil , Preescolar , Cognición , Discapacidades del Desarrollo , Humanos , Lactante , Desarrollo del Lenguaje , Estudios Longitudinales , Estados UnidosRESUMEN
OBJECTIVE: To characterize operative care for cleft lip and/or palate (CL/P) based on location (ie, from American Cleft Palate Craniofacial Association [ACPA]-approved multidisciplinary teams or from community providers). DESIGN: Cross-sectional analysis of Healthcare Cost and Utilization Project State Inpatient Database and State Ambulatory Surgery & Services Database databases for North Carolina from 2012 to 2015. SETTING/PATIENTS AND MAIN OUTCOME MEASURES: Clinical encounters for children with CL/P undergoing operative procedures were identified, classified by location as "Team" versus "Community," and characterized by demographic, geographic, clinical, and procedural factors. A secondary evaluation reviewed concordance of team and community practices with an ACPA guideline related to coordination of care. RESULTS: Three teams and 39 community providers performed a total of 3010 cleft-related procedures across 2070 encounters. Teams performed 69.7% of total volume and performed the majority of cleft procedures, including cleft lip repair, palate repair, alveolar bone grafting, and correction of velopharyngeal insufficiency. Community locations principally offered myringotomy and rhinoplasty. Team care was associated with higher guideline concordance. CONCLUSIONS: American Cleft Palate Craniofacial Association -approved team-based care accounts for the majority of cleft-related care in North Carolina; however, a substantial volume of cleft-related procedures was provided by community providers, with 3 providers accounting for the vast majority of community cases.
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Labio Leporino , Fisura del Paladar , Niño , Labio Leporino/cirugía , Fisura del Paladar/cirugía , Estudios Transversales , Humanos , North CarolinaRESUMEN
OBJECTIVE: The Craniofacial microsomia: Longitudinal Outcomes in Children pre-Kindergarten (CLOCK) study is a longitudinal cohort study of neurobehavioral outcomes in infants and toddlers with craniofacial microsomia (CFM). In this article, we review the data collection and methods used to characterize this complex condition and describe the demographic and clinical characteristics of the cohort. SETTING: Craniofacial and otolaryngology clinics at 5 study sites. PARTICIPANTS: Infants with CFM and unaffected infants (controls) ages 12 to 24 months were recruited from the same geographical regions and followed to age 36 to 48 months. METHODS: Phenotypic, neurodevelopmental, and facial expression assessments were completed during the first and third waves of data collection (time 1 and time 3, respectively). Medical history data were taken at both of these time points and during an intermediate parent phone interview (time 2). RESULTS: Our cohort includes 108 cases and 84 controls. Most cases and controls identified as white and 55% of cases and 37% of controls identified as Hispanic. Nearly all cases had microtia (95%) and 59% had mandibular hypoplasia. Cases received extensive clinical care in infancy, with 59% receiving care in a craniofacial clinic and 28% experiencing at least one surgery. Study visits were completed at a study site (92%) or at the participant's home (8%). CONCLUSIONS: The CLOCK study represents an effort to overcome the challenges of characterizing the phenotypic and neurodevelopmental outcomes of CFM in a large, demographically and geographically diverse cohort.
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Microtia Congénita , Síndrome de Goldenhar , Preescolar , Estudios de Cohortes , Femenino , Síndrome de Goldenhar/cirugía , Humanos , Lactante , Estudios Longitudinales , Masculino , Resultado del Tratamiento , Estados UnidosRESUMEN
BACKGROUND: Limited quality of life data exist for pediatric chronic rhinosinusitis (CRS) patients undergoing endoscopic sinus surgery (ESS). Further exploration of the following areas will enhance understanding and support clinical decision-making: baseline and post-ESS general and disease-specific quality of life, parent vs. child report, and correlation of nasal endoscopy to sinus CT scores. METHODOLOGY: A prospective cohort study evaluated CRS patients age 5-18 undergoing ESS. Surveys were completed at two timepoints: (1) pre-ESS and (2) 30-90 days post-ESS, with parents completing general (PedsQLTM) and CRS-specific (SNOT-16 and SN-5) quality of life surveys and children completing PedsQLTM and SNOT-16 surveys. Preoperative Lund-Kennedy nasal endoscopy and Lund-Mackay sinus CT scores were calculated. Where appropriate, outcomes were stratified by cystic fibrosis status. RESULTS: Impaired preoperative general quality of life was evidenced by parent proxy-report of PedsQLTM scores in 10 cystic fibrosis and 11 non-CF patients. ESS was associated with decreased sinus symptoms at 1-3 months postoperatively with SN-5 change scores of -1.85 and -2.2, in CF and non-CF patients, respectively. Parents reported worse CRS symptoms via higher preoperative SNOT-16 scores than their children did. Nasal endoscopy and sinus CT scores correlated with a Spearman correlation coefficient of 0.51. Scores not reaching statistical significance included CF-related CRS SNOT-16 change scores and PedsQLTM general quality of life change scores. CONCLUSION: In pediatric patients with CRS electing ESS, general quality of life is impaired preoperatively and sinus symptoms improve significantly 1-3 months after sinus surgery. Parents report statistically worse CRS symptom scores than their children do. Nasal endoscopy scores in this cohort correlated with sinus CT scores.
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Fibrosis Quística/patología , Senos Paranasales/cirugía , Rinitis/cirugía , Humanos , Evaluación de Procesos y Resultados en Atención de Salud , Periodo Posoperatorio , Estudios Prospectivos , Calidad de VidaRESUMEN
Oral causes of dysphagia in infancy may involve the lips, the tongue, or the palate. Whereas ankyloglossia is commonly diagnosed in infants with dysphagia, assessment of the need for surgical intervention may be less straightforward. Tongue size (macroglossia) may be associated with dysphagia as it may cause limitation of movement of the food or milk bolus by the lips or cheeks. Congenital conditions such as cleft lip and palate, micrognathia, or craniofacial microsomia may also be associated with dysphagia. Diagnosis and treatment of these conditions can be improved with the engagement of lactation and feeding experts as well as multidisciplinary craniofacial teams.
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Trastornos de Deglución , Lengua , Humanos , Trastornos de Deglución/etiología , Trastornos de Deglución/diagnóstico , Trastornos de Deglución/terapia , Lactante , Lengua/fisiopatología , Niño , Anquiloglosia , Fisura del Paladar/complicaciones , Fisura del Paladar/cirugía , Labio Leporino/complicaciones , Labio Leporino/cirugía , Labio/fisiopatología , Anomalías de la Boca/cirugía , Anomalías de la Boca/complicaciones , Micrognatismo/complicacionesRESUMEN
The upper aerodigestive system is closely intertwined from an embryologic and functional perspective. Laryngotracheal anatomic abnormalities, such as laryngomalacia, stenosis, vocal cord paralysis, and laryngeal clefts, affect not only the respiratory function but also the swallow function. Laryngotracheal pathology can interfere with the suck-swallow-breathe mechanism in infants. It can also exacerbate gastroesophageal reflux. Chronic aspiration secondary to laryngotracheal anomalies can result in respiratory and pulmonary complications. Surgical treatment of laryngotracheal anomalies can also cause transient or long-term swallow dysfunction. Multidisciplinary approaches and clinical assessment of swallowing are important in patients with laryngotracheal pathology.
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Trastornos de Deglución , Laringe , Humanos , Trastornos de Deglución/etiología , Trastornos de Deglución/diagnóstico , Laringe/anomalías , Laringe/cirugía , Niño , Lactante , Laringomalacia/complicaciones , Laringomalacia/cirugía , Laringomalacia/diagnóstico , Reflujo Gastroesofágico/complicaciones , Reflujo Gastroesofágico/diagnóstico , Enfermedades de la Laringe/cirugía , Enfermedades de la Laringe/complicaciones , Parálisis de los Pliegues Vocales/etiología , Deglución/fisiología , Anomalías CongénitasRESUMEN
Objective: In the USA, the American Cleft Palate Association-Craniofacial Association (ACPA-CPF) specifies guidelines to ensure craniofacial teams function in a multidisciplinary fashion to provide care in a sequence that mirrors the patient's needs. Among these guidelines is the expectation that teams hold regular multidisciplinary team meetings to discuss patient care. The purpose of this study was to identify factors that contribute to optimal team functioning during these meetings. The impact of the virtual format on meetings was also evaluated. Design: One-hour semi-structured interviews of craniofacial team members, queried on current and ideal team meetings, were conducted from December 2020 - January 2021. Interviews were recorded, transcribed, and coded to identify common themes until thematic saturation was achieved. Setting/Participants: Sixteen team members from three major American craniofacial centers were interviewed virtually. Results: Three major categories were identified as integral to successful meetings: (1) meeting content, (2) meeting processes, and (3) team dynamics. Within these categories are several factors that participants reported as important to optimize their multidisciplinary meetings: key content for discussions, integration of patient perspective, recognition of collaboration in determining proper sequence of treatments, an attentive discussion leader promoting equitable discussions, managing absences, and team diversity. Participants also identified benefits and pitfalls of hosting meetings in the virtual setting, including lack of engagement and awkward communication. Conclusion: Diverse groups of disciplines are expected to come together during craniofacial multidisciplinary meetings to formulate intricate care plans for complex patients. In this study, craniofacial team members have identified several key factors to optimize these meetings. The research group used these factors to propose a self-evaluation instrument for meeting quality improvement, provided in printable format. Further studies should evaluate how implementation of these factors affects meetings and ultimately guides patient care.
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OBJECTIVE: To describe the early health care experiences of parents of children with craniofacial microsomia (CFM), a congenital diagnosis often identified at birth. DESIGN: Qualitative descriptive. SETTING: Homes of participants. PARTICIPANTS: Parents of 28 children with CFM from across the United States. METHODS: We interviewed participants (27 mothers individually and one mother and father together) via telephone or teleconference and used reflexive thematic analysis to derive themes that represented early health care experiences of parents of children with CFM. RESULTS: Participants' narratives included detailed recounting of their birth and early care experiences. We identified two overarching themes. The first overarching theme, Stressors, included four subthemes that represented difficulties related to emotional reactions and negative experiences with health care providers. The second overarching theme, Finding Strength, included four subthemes that represented participants' positive adjustment to stressors through independent information seeking about CFM, adaptive coping, positive experiences with health care providers, and drawing on external supports. CONCLUSION: Participants often described early experiences as challenging. Findings have implications for improving early care, including increasing open and supportive communication by health care professionals, expanding access to CFM information, screening for mental health concerns among parents, strengthening coping among parents, and linking families to resources such as reliable online CFM information and early intervention programs.
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Adaptación Psicológica , Padres , Investigación Cualitativa , Humanos , Femenino , Masculino , Padres/psicología , Adulto , Estados Unidos , Síndrome de Goldenhar/psicología , Síndrome de Goldenhar/diagnóstico , Estrés Psicológico/psicología , Niño , Recién Nacido , Preescolar , LactanteRESUMEN
An otolaryngology evaluation is an important aspect of care for any patient with craniofacial conditions. Otolaryngologists are commonly involved in the care of these patients to treat chronic otitis media, which frequently requires placement of pressure equalization tubes; however, there are many other otorhinolaryngologic issues that are addressed. Patients with craniofacial conditions may struggle with nasal obstruction, airway compromise, obstructive sleep apnea, dysphonia, swallowing, chronic sinonasal disease, and otologic disease.
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Apnea Obstructiva del Sueño , HumanosRESUMEN
Despite efforts at many institutions, nationally, women still lag behind their male counterparts in leadership, promotion, and seniority. In this paper, we describe the efforts to improve the environment for women faculty at one large academic medical center through the creation of an Association of Professional Women in Medical Sciences. Over the years, the group has helped influence policies that directly affect women faculty, provided high-yield programming on topics related to women's success, and created interprofessional networking opportunities for women faculty. We describe the challenges and successes of this group to serve as a model and inspiration for other institutions.
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As healthcare systems become more complex, medical education needs to adapt in many ways. There is a growing need for more formal leadership learning for healthcare providers, including greater attention to health disparities. An important challenge in addressing health disparities is ensuring inclusive excellence in the leadership of healthcare systems and medical education. Women and those who are underrepresented in medicine (URMs) have historically had fewer opportunities for leadership development and are less likely to hold leadership roles and receive promotions. One successful initiative for improved learning of medical leadership-presented as a case example here-is the Academic Career Leadership Academy in Medicine (ACCLAIM) at the University of North Carolina at Chapel Hill School of Medicine. ACCLAIM is uniquely designed for faculty identified as having emerging leadership potential, with an emphasis on women and URMs. Using a leadership learning system approach, annual cohorts of participants (Scholars) interactively participate in a multi-faceted nine-month long learning experience, including group (e.g., guest-speaker weekly presentations and exercises) and individual learning components (e.g., an individual leadership project). Since its initiation in 2012 and through 2021, 111 Scholars have participated in ACCLAIM; included were 57% women and 27% URMs. Two important outcomes described are: short-term impact as illustrated by consistent improvements in quantitively measured leadership knowledge and capabilities; and long-term leadership growth, whereby half of the ACCLAIM graduates have received academic rank promotions and almost two-thirds have achieved new leadership opportunities, with even higher percentages observed for women and URMs; for example, 87% of URMs were either promoted or achieved new leadership positions. Also consistently noted, through qualitative assessments, are broader healthcare system knowledge and shared tactics for addressing common challenges among Scholars. This case example shows that the promotion of leadership equity may jointly enhance professional development while creating opportunities for systems change within academic medical centers. Such an approach can be a potential model for academic medical institutions and other healthcare schools seeking to promote leadership equity and inclusion.
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Educación Médica , Docentes Médicos , Humanos , Femenino , Masculino , Liderazgo , Centros Médicos Académicos , AprendizajeRESUMEN
OBJECTIVES: Facial dysmorphic disorder (FDD), a variant of body dysmorphic disorder, occurs when individuals are preoccupied with perceived defects in their facial appearance. Cleft lip and/or palate (CL/P) requires many clinical interventions and has significant psychological impacts on a patient's perception of appearance. This study identified psychological burdens related to living as an adult with CL/P and characterizes the degree of FDD symptoms in an adult craniofacial population. METHODS: This was a prospective, single-center, cross-sectional case-control study using semi-structured interviews and symptom assessments at a university-based craniofacial center. Patients without CL/P undergoing non-cosmetic facial surgery were recruited as controls (n = 20). Patients with an orofacial cleft (n = 30) were recruited from medical and dental providers at the University of North Carolina. Body Dysmorphic Disorder-Yale Brown Obsessive Compulsive Scale (BBD-YBOCS) scores were collected from a control population and patients with CL/P to assess FDD severity. RESULTS: Demographic factors such age, biological sex, and ethnicity had no significant impact on FDD symptom scores. Patient with CL/P were more likely to have significant FDD symptoms (BDD-YBOCS greater than 16) than patients without CL/P (OR 10.5, CI95 2.7-41.1), and had a mean difference in FDD symptoms scores of 10.04 (p < 0.0001; CI95 5.5-14.6). Patients with CL/P seen by a mental health provider in the past 3 months had 3-fold lower overall FDD symptom scores (OR 0.081; CI95 0.0085-0.77). CONCLUSIONS: Adults with CL/P would benefit from treatment for cleft-specific needs and psychological support as they face unique stressors related to their appearance, including an increase in FDD-associated symptoms. This study emphasizes the importance of recognizing psychological symptoms and providing ongoing multidisciplinary care to adults with CL/P. LEVEL OF EVIDENCE: 3; Individual case-control study Laryngoscope, 133:818-821, 2023.
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Trastorno Dismórfico Corporal , Labio Leporino , Fisura del Paladar , Humanos , Adulto , Labio Leporino/complicaciones , Labio Leporino/cirugía , Fisura del Paladar/complicaciones , Fisura del Paladar/cirugía , Estudios Transversales , Estudios de Casos y Controles , Estudios ProspectivosRESUMEN
Objective Individuals with cleft lip and palate (CLP) are at a high risk of developing obstructive sleep apnea (OSA). Hypothetically, the severity of OSA might be associated with the morphology of the upper airway (UAW) and the characteristics of the airflow. Thus, the present study aimed to assess and compare, in adults with CLP and skeletal class-III discrepancy, with or without OSA, simulations of airflow resistance and pressure according to the geometrical characteristics of the UAW and cephalometric parameters. Materials and Methods According to the results of type-I polysomnography tests, the sample ( n = 21) was allocated in 2 groups: 1) without OSA (N-OSA; n = 6); and 2) with OSA (OSA; n = 15). Cephalometric measurements were performed on the cone-beam computed tomography (CBCT) scans of the groups. After three-dimensional (3D) reconstructions, the volume (V) and minimal cross-sectional area (mCSA) of the UAW were generated. Computational fluid dynamics (CFD) simulations were used to assess key airflow characteristics. The results were presented at a significance level of 5%. Results The UAW pressure values and airway resistance did not differ between the groups, but there was a tendency for more negative pressures (26%) and greater resistance (19%) in the OSA group. Volume and mCSA showed a moderate negative correlation with resistance and pressure. The more inferior the hyoid bone, the more negative the pressures generated on the pharyngeal walls. Conclusion The position of the hyoid bone and the geometry of the UAW (V and mCSA) exerted effects on the airway-airflow resistance and pressure. However, key airflow characteristics did not differ among subjects with CLP, were they affected or not by OSA.