The Development, Deployment, and Evaluation of the CLEFT-Q Computerized Adaptive Test: A Multimethods Approach Contributing to Personalized, Person-Centered Health Assessments in Plastic Surgery.

BACKGROUND: Routine use of patient-reported outcome measures (PROMs) and computerized adaptive tests (CATs) may improve care in a range of surgical conditions. However, most available CATs are neither condition-specific nor coproduced with patients and lack clinically relevant score interpretation. Recently, a PROM called the CLEFT-Q has been developed for use in the treatment of cleft lip or palate (CL/P), but the assessment burden may be limiting its uptake into clinical practice. OBJECTIVE: We aimed to develop a CAT for the CLEFT-Q, which could facilitate the uptake of the CLEFT-Q PROM internationally. We aimed to conduct this work with a novel patient-centered approach and make source code available as an open-source framework for CAT development in other surgical conditions. METHODS: CATs were developed with the Rasch measurement theory, using full-length CLEFT-Q responses collected during the CLEFT-Q field test (this included 2434 patients across 12 countries). These algorithms were validated in Monte Carlo simulations involving full-length CLEFT-Q responses collected from 536 patients. In these simulations, the CAT algorithms approximated full-length CLEFT-Q scores iteratively, using progressively fewer items from the full-length PROM. Agreement between full-length CLEFT-Q score and CAT score at different assessment lengths was measured using the Pearson correlation coefficient, root-mean-square error (RMSE), and 95% limits of agreement. CAT settings, including the number of items to be included in the final assessments, were determined in a multistakeholder workshop that included patients and health care professionals. A user interface was developed for the platform, and it was prospectively piloted in the United Kingdom and the Netherlands. Interviews were conducted with 6 patients and 4 clinicians to explore end-user experience. RESULTS: The length of all 8 CLEFT-Q scales in the International Consortium for Health Outcomes Measurement (ICHOM) Standard Set combined was reduced from 76 to 59 items, and at this length, CAT assessments reproduced full-length CLEFT-Q scores accurately (with correlations between full-length CLEFT-Q score and CAT score exceeding 0.97, and the RMSE ranging from 2 to 5 out of 100). Workshop stakeholders considered this the optimal balance between accuracy and assessment burden. The platform was perceived to improve clinical communication and facilitate shared decision-making. CONCLUSIONS: Our platform is likely to facilitate routine CLEFT-Q uptake, and this may have a positive impact on clinical care. Our free source code enables other researchers to rapidly and economically reproduce this work for other PROMs.

Measuring the Unilateral Cleft Lip Nasal Deformity: Lateral Deviation of Subnasale Is a Clinical and Morphologic Index of Unrepaired Severity.

OBJECTIVE: Objective measurement of pre-operative severity is important to optimize evidence-based practices given that the wide spectrum of presentation likely influences outcomes. The purpose of this study was to determine the correlation of objective measures of form with a subjective standard of cleft severity. DESIGN: 3D images were ranked according to severity of nasal deformity by 7 cleft surgeons so that the mean rank could be used as the severity standard. PATIENTS: 45 patients with unilateral cleft lip and 5 normal control subjects. INTERVENTIONS: Each image was assessed using traditional anthropometric analysis, 3D landmark displacements, and shape-based analysis to produce 81 indices for each subject. MAIN OUTCOME: The correlation of objective measurements with the clinical severity standard. RESULTS: Lateral deviation of subnasale from midline was the best predictor of severity (0.86). Other strongly-correlated anthropometric measurements included columellar angle, nostril width ratio, and lateral lip height ratio (0.72, 0.80, 0.79). Almost all shape-based measurements had tight correlation with the severity standard, however, dorsum deviation and point difference nasolabial symmetry were the most predictive (0.84, 0.82). CONCLUSIONS: Quantitative measures of severity transcend cleft type and can be used to grade clinical severity. Lateral deviation of subnasale was the best measure of severity and may be used as a surrogate of uncoupled premaxillary growth; it should be recorded as an index of pre-operative severity with every cleft lip repair. The correlation of other measures evaluated clarify treatment priorities and could potentially be used to grade outcomes.

Using "Real-World Data" to Study Cleft Lip/Palate Care: An Exploration of Speech Outcomes from a Multi-Center US Learning Health Network.

OBJECTIVE: To assess the ability of a cleft-specific multi-site learning health network registry to describe variations in cleft outcomes by cleft phenotypes, ages, and treatment centers. Observed variations were assessed for coherence with prior study findings. DESIGN: Cross-sectional analysis of prospectively collected data from 2019-2022. SETTING: Six cleft treatment centers collected data systematically during routine clinic appointments according to a standardized protocol. PARTICIPANTS: 714 English-speaking children and adolescents with non-syndromic cleft lip/palate. INTERVENTION: Routine multidisciplinary care and systematic outcomes measurement by cleft teams. OUTCOME MEASURES: Speech outcomes included articulatory accuracy measured by Percent Consonants Correct (PCC), velopharyngeal function measured by Velopharyngeal Competence (VPC) Rating Scale (VPC-R), intelligibility measured by caregiver-reported Intelligibility in Context Scale (ICS), and two CLEFT-Q™ surveys, in which patients rate their own speech function and level of speech distress. RESULTS: 12year-olds exhibited high median PCC scores (91-100%), high frequency of velopharyngeal competency (62.50-100%), and high median Speech Function (80-91) relative to younger peers parsed by phenotype. Patients with bilateral cleft lip, alveolus, and palate reported low PCC scores (51-91%) relative to peers at some ages and low frequency of velopharyngeal competency (26.67%) at 5 years. ICS scores ranged from 3.93-5.0 for all ages and phenotypes. Speech Function and Speech Distress were similar across phenotypes. CONCLUSIONS: This exploration of speech outcomes demonstrates the current ability of the cleft-specific registry to support cleft research efforts as a source of "real-world" data. Further work is focused on developing robust methodology for hypothesis-driven research and causal inference.

Systematic Review and Critical Appraisal of the Evidence Base for Nasoalveolar Molding (NAM).

OBJECTIVE: To critically appraise the body of scientific literature supporting the risks and efficacy of nasoalveolar molding (NAM), specifically in contrast to alternative methods of presurgical infant orthopedics (PSIO) or to treatment without PSIO. MAIN OUTCOME MEASURES: Five outcome domains were considered: nasolabial aesthetics; dentoalveolar relationship; midfacial growth; cost and burden of care; and number of anesthetic events. DESIGN: MEDLINE, Embase, and Scopus were queried for articles from the first description of the Grayson-Santiago NAM technique (1993) through December 13, 2021. After the application of inclusionary and exclusionary criteria, selected articles were critically appraised using a systematic framework that included risk of bias assessment using the Cochrane RoB 2.0 and ROBINS-I tools. RESULTS: A total of 88 studies were included. Level-I and -II evidence showed on par or better approximation and alveolar alignment achieved by NAM compared to other PSIO. Level-II and -III evidence showed improved nasolabial aesthetics compared to other PSIOs. Level-II and -III evidence supported no harm to maxillofacial skeletal growth through age 12. Sparse level-III evidence supported a reduced number of labial or nasal revisions following NAM. Level-II and -III evidence showed NAM requiring upfront cost and frequent appointments but reducing caregiver psychosocial burden and reducing long-term costs compared to select alternatives. Many studies carried a high risk of bias. CONCLUSIONS: Current evidence supports the overall efficacy of NAM regarding short/mid-term outcomes, with a low risk of negative effects on midfacial growth or dental development. The high risk of bias discovered in many papers underscores the need for robust study design in future research.

Barriers and Facilitators to the International Implementation of Standardized Outcome Measures in Clinical Cleft Practice.

OBJECTIVE: To identify barriers and facilitators to international implementation of a prospective system for standardized outcomes measurement in cleft care. DESIGN: Cleft teams that have implemented the International Consortium for Health Outcomes Measurement Standard Set for cleft care were invited to participate in this 2-part qualitative study: (1) an exploratory survey among clinicians, health information technology professionals, and project coordinators, and (2) semistructured interviews of project leads. Thematic content analysis was performed, with organization of themes according to the dimensions of the reach, effectiveness, adoption, implementation and maintenance (RE-AIM) framework: reach, effectiveness, adoption, implementation, and maintenance. RESULTS: Four cleft teams in Europe and North America participated in this study. Thirteen participants completed exploratory questionnaires and 5 interviewees participated in follow-up interviews. Survey responses and thematic content analysis revealed common facilitators and barriers to implementation at all sites. Teams reach patients either via email or during the clinic visit to capture patient-reported outcomes. Adopting routine data collection is enhanced by aligning priorities at the organizational and cleft team level. Streamlining workflows and developing an efficient data collection platform are necessary early on, followed by pilot testing or stepwise implementation. Regular meetings and financial resources are crucial for implementing, sustaining, analyzing collected data, and providing feedback to health care professionals and patients. Fostering patient-centered care was articulated as a positive outcome, whereas time presented challenges across all RE-AIM dimensions. CONCLUSIONS: Identified themes can inform ongoing implementation efforts. Intentionally investing time to lay a sound foundation early on will benefit every phase of implementation and help overcome barriers such as lack of support or motivation.

Rasch Analysis of Patient- and Parent-Reported Outcome Measures in the International Consortium for Health Outcomes Measurement Standard Set for Cleft Lip and Palate.

OBJECTIVES: The aim of this study was to evaluate the psychometric performance of the patient- and parent-reported measures in the International Consortium for Health Outcomes Measurement (ICHOM) Standard Set for Cleft Care, and to identify ways of improving concept coverage. METHODS: Data from 714 patients with cleft lip and/or palate, aged 8 to 9, 10 to 12.5, and 22 years were collected between November 2015 and April 2019 at Erasmus University Medical Center, Boston Children's Hospital, Duke Children's Hospital, and from participating sites in the CLEFT-Q Phase 3 study. The Standard Set includes 9 CLEFT-Q scales, the Nasal Obstruction Symptom Evaluation (NOSE) questionnaire, the Child Oral Health Impact Profile-Oral Symptoms Scale (COHIP-OSS), and the Intelligibility in Context Scale (ICS). Targeting, item-fit statistics, thresholds for item responses, and measurement precision (PSI) were analyzed using Rasch measurement theory. RESULTS: The proportion of the sample to score within each instruments range of measurement varied from 69% (ICS) to 92% (CLEFT-Q teeth and COHIP-OSS). Specific problems with individual items within the NOSE and COHIP-OSS questionnaires were noted, such as poor item fit to the Rasch model and disordered thresholds (6 of 10). Reliability measured with PSI was above 0.82 for the ICS and all but one CLEFT-Q scale (speech distress). PSIs were lowest for the COHIP-OSS (0.43) and NOSE questionnaire (0.35). CONCLUSION: The patient- and parent-reported components within the facial appearance, psychosocial function, and speech domains are valid measures; however, the facial function and oral health domains are not sufficiently covered by the CLEFT-Q eating and drinking, NOSE, and COHIP-OSS, and these questionnaires may not be accurate enough to stratify cleft-related outcomes.

A Toolbox of Surgical Techniques for Palatal Fistula Repair.

OBJECTIVE: To provide an inventory of oronasal fistula repair techniques alongside expert commentary on which techniques are appropriate for each fistula type. DESIGN: A 4-stage approach was used to develop a consensus on surgical techniques available for fistula repair: (1) in-person discussion of oronasal fistula cases among cleft surgeons, (2) development of a schema for fistula management using transcripts of the in-person case discussion, (3) evaluation of the preliminary schema via a web-based survey of additional cleft surgeons, and (4) revision of the management schema using survey responses. PARTICIPANTS: Six cleft surgeons participated in the in-person case discussion. Eleven additional surgeons participated in the web-based survey. Participants had diverse training experiences, having completed residency and fellowship at 20 different hospitals. RESULTS: A schema for fistula management was developed, organized by fistula location. The schema catalogues all viable approaches for each location. For fistulae involving the soft palate, the schema stresses the importance of evaluating for velopharyngeal insufficiency (VPI) and incorporating VPI management into fistula repair. For fistulae involving the hard palate, the schema separately enumerates the techniques available for nasal lining repair and for oral lining repair in each region. The schema also catalogues the diversity of approaches to lingual- and labioalveolar fistula, including variation in timing, orthodontic preparation, and simultaneous alveolar bone grafting. CONCLUSIONS: This study employed consensus methods to create a comprehensive inventory of available fistula repair techniques and to identify preferential techniques among a diverse group of surgeons.

Provision and Utilization of Team- and Community-Based Operative Care for Patients With Cleft Lip/Palate in North Carolina.

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.

Power-Assisted Particulate Bone Grafting Effectively Prevents Osseous Defects After Cranial Vault Reconstruction.

BACKGROUND: Cranial vault reconstruction (CVR) is the gold standard in the operative treatment of craniosynostosis. Full thickness osseous defects (FTOD) of the calvaria have been observed in 5% to 15% patients after CVR, with higher rates cited in the fronto-orbital advancement (FOA) subset. Particulate bone graft (PBG) harvested manually has been shown to decrease FTOD after FOA from 24% to 5.5%. The authors used a modified technique using a powered craniotome, with the hypothesis that the technique would also improve outcomes. METHODS: A retrospective review was performed of patients who underwent CVR for craniosynostosis between 2004 and 2014. Patient demographics, diagnosis, age, operative details, and postoperative care were reviewed in detail. Categorical, nonparametric variables were compared by Fisher exact tests. RESULTS: A total of 135 patients met inclusion criteria. The most common diagnoses were metopic (n = 41), sagittal (n = 33), and unilateral coronal craniosynostosis (n = 31); 65% (n = 88) underwent FOA, 29% (n = 39) underwent single-stage total vault reconstruction, and 6% (n = 8) had a posterior vault reconstruction. CVR was performed without PBG in 95 patients and with PBG in 40 patients. Without PBG, FTOD were discovered on clinical examination in 18% of patients (n=17): 11 presented with subcentimeter defects, while 6 had larger defects requiring revision cranioplasty (6% operative revision rate). Among those receiving PBG, 1 patient presented a subcentimeter FTOD (2.5% FTOD incidence and 0% operative revision rate). CONCLUSION: Particulate bone graft harvested with a powered device decreases the rate of FTOD and reoperation rate after CVR for craniosynostosis.

Socioeconomic Factors, Perioperative Complications, and 30-Day Readmission Rates Associated With Delayed Cranial Vault Reconstruction for Craniosynostosis.

BACKGROUND: Premature fusion of the cranial sutures can lead to significant neurocognitive, developmental, and esthetic consequences, especially if not corrected within the first year of life. This study aimed to identify the drivers of delayed cranial vault reconstruction (CVR) and its impact on complication and 30-day readmission rates among craniosynostosis patients. METHODS: The medical records of all children who underwent CVR for craniosynostosis between 2005 and 2017 at an academic institution were retrospectively reviewed. A delay in operation was defined by surgery performed >12 months of age. Patient demographics, comorbidities, perioperative complication rates, and 30-day readmission rates were collected. RESULTS: A total of 96 patients underwent primary CVR, with 79 (82.3%) patients undergoing nondelayed surgery and 17 (17.7%) patients undergoing surgery >12 months of age. Children undergoing delayed surgery were significantly more likely to be non-White (P < 0.0001), have Medicaid insurance (P = 0.023), and have a non-English primary language (P < 0.005). There was increased incidence of developmental disability identified at first consult (no-delay: 3.9% vs delay: 41.2%, P < 0.0001) and increased intracranial pressure (no-delay: 6.3% vs delay: 29.4%, P < 0.005) among children undergoing delayed surgery. The delayed cohort had a significantly higher unplanned 30-day readmission rate (no-delay: 0.0% vs delay: 5.9%, P = 0.03). CONCLUSION: Our study suggests that craniosynostosis patients who are non-White, have a non-English primary language, and have Medicaid insurance are at risk for delayed primary surgery, which may lead to increased 30-day readmission. Interventions are necessary to reduce craniosynostosis patients' barriers to care to minimize the sequelae associated with delayed surgery.

Implementation of a Standardized Data-Collection System for Comprehensive Appraisal of Cleft Care.

OBJECTIVE: Our objective is to describe the process of adapting a conceptual framework into a practical toolkit for one cleft team. DESIGN: This is a single-arm implementation study in a single institution. SETTING: Implementation took place at a mid-sized multidisciplinary clinic for patients with cleft lip and/or palate (CL/P) from urban/suburban and rural areas across North Carolina and neighboring states. PARTICIPANTS: Eligible participants were patients with CL/P from English-speaking families. Sixty patients entered and finished the study. INTERVENTIONS: The implementation of a prospective data collection system based on the International Consortium for Health Outcomes Measurement (ICHOM) standard set of outcome measures for CL/P was accomplished in multiple stages. Patient- and clinician-reported forms and protocols for gathering data were created. Team members were trained and the system was tested; finally, the system was deployed. MAIN OUTCOME MEASURES: Success was appraised using the RE-AIM framework to assess reach, effectiveness, adoption, implementation, and maintenance. RESULTS: Ninety-eight percent of patients and all team members agreed to participate. Ninety-four percent of required data were captured. Adaptations to friction points were made; specifically, visible reminders were affixed to charts, primary clinicians were required to assume data entry responsibility, and e-mail reminders were instituted. Development cost was US$7707; average time cost per clinician was 21 min/wk. CONCLUSIONS: Conceptual frameworks for outcomes studies must be tailored to their environments; otherwise, they cannot be practically implemented and sustained. We present this process for a cleft team using the ICHOM standard set. The process may help other teams implement the standard set or other conceptual frameworks.

Reliability of Oronasal Fistula Classification.

OBJECTIVE: Oronasal fistula is an important complication of cleft palate repair that is frequently used to evaluate surgical quality, yet reliability of fistula classification has never been examined. The objective of this study was to determine the reliability of oronasal fistula classification both within individual surgeons and between multiple surgeons. DESIGN: Using intraoral photographs of children with repaired cleft palate, surgeons rated the location of palatal fistulae using the Pittsburgh Fistula Classification System. Intrarater and interrater reliability scores were calculated for each region of the palate. PARTICIPANTS: Eight cleft surgeons rated photographs obtained from 29 children. RESULTS: Within individual surgeons reliability for each region of the Pittsburgh classification ranged from moderate to almost perfect (κ = .60-.96). By contrast, reliability between surgeons was lower, ranging from fair to substantial (κ = .23-.70). Between-surgeon reliability was lowest for the junction of the soft and hard palates (κ = .23). Within-surgeon and between-surgeon reliability were almost perfect for the more general classification of fistula in the secondary palate (κ = .95 and κ = .83, respectively). CONCLUSIONS: This is the first reliability study of fistula classification. We show that the Pittsburgh Fistula Classification System is reliable when used by an individual surgeon, but less reliable when used among multiple surgeons. Comparisons of fistula occurrence among surgeons may be subject to less bias if they use the more general classification of "presence or absence of fistula of the secondary palate" rather than the Pittsburgh Fistula Classification System.

Academic Achievement Among Children With Nonsyndromic Orofacial Clefts : A Population-Based Study.

OBJECTIVE: Children with orofacial clefts (OFCs) may experience poor reading proficiency, learning disabilities, and academic underachievement. We examined the association between nonsyndromic (NS) OFCs and end-of-grade (EOG) performance in reading and math from third through eighth grade in a sample subgroup. PARTICIPANTS: We identified a cohort of 559 children with NS-OFCs and 6822 children without birth defects, classifying cleft type by cleft lip alone, with or without cleft alveolar ridge (CL); cleft lip with cleft palate (CL+P); and cleft palate only (CP). MAIN OUTCOME MEASURES: Using logistic regression, we estimated the odds of not meeting grade-level standards among children with NS-OFCs compared to unaffected peers. Using longitudinal analyses, we estimated the odds of not meeting grade-level standards and average change in test scores through eighth grade. RESULTS: Children with NS-OFCs were 1.22 (95% CI: 0.96, 1.83) times as likely not to meet grade-level standards in reading compared to unaffected peers. The effect was similar for math (OR: 1.17; 95% CI: 0.92, 1.48). Children with CL+P were 1.33 (95% CI: 0.86, 1.83) and 1.74 (95% CI: 1.19, 2.56) times as likely not to meet grade-level standard in reading and in both subjects, respectively, compared to unaffected peers. The average rate of change in both scores was similar for children with and without OFCs. CONCLUSIONS: Poor academic performance appears greatest for children with CL+P, a finding compatible with previous observations and hypothesized mechanisms associating orofacial clefts with subtle abnormalities in brain development. Academic performance monitoring and referral for academic assistance is warranted.

Risk Factors for Delayed Referral to a Craniofacial Specialist for Treatment of Craniosynostosis.

OBJECTIVE: To assess the impact of age at referral on treatment options in craniosynostosis and to identify risk factors for referral delays in this population. STUDY DESIGN: A retrospective cohort study was performed on patients with an abnormal head shape diagnosis treated at a single academic medical center between January 1, 2004 and January 1, 2014. Newly diagnosed patients with craniosynostosis were identified and referral patterns were examined. A multivariate logistic regression model was used to identify risk factors associated with the range of ages at initial referral. RESULTS: A total of 477 patients were evaluated at our institution, 197 of whom were subsequently diagnosed with craniosynostosis. The median age at initial appointment was 5.6 months (mean 8.2 months). Only 28% of children were referred within 3 months of birth. Patients referred within 3 months of birth were less likely to have had preappointment imaging than those patients referred slightly later (OR 2.53, CI 1.07-5.98, P = .035). Several variables were associated with referral after 12 months of age including multiple suture involvement (OR 4.21, CI 1.06-16.68, P = .041), minority race (OR 4.96, CI 1.91-12.9, P ≤ .0001), and referral by a nonpediatrician (OR 6.9, CI 1.73-27.49, P = .006). CONCLUSIONS: Obtaining imaging before referral to a specialist for abnormal head shape was associated with a delay in evaluation and potentially increases radiation exposure and limits treatment options in patients with craniosynostosis. In addition, children from minority groups, children referred from someone other than a pediatrician's office, and those with multiple suture craniosynostosis are at increased risk of delayed referral. Further studies into the cause of these delays are warranted.

Epidemiologic Analysis of Elective Operative Procedures in Infants Less Than 6 Months of Age in the United States.

BACKGROUND: This study uses publicly available data to analyze the total number of elective, potentially deferrable operative procedures involving infants <6 months of age in the United States. We investigated the factors associated with the performance of these procedures in this population. METHODS: The State Ambulatory Surgery Database was used to identify patients in California, North Carolina, New York, and Utah during the years of 2007-2010 who were younger than 6 months of age at the time that they underwent outpatient (ambulatory) surgery. Operations that could reasonably be postponed until 6 months of age were classified as potentially deferrable procedures. Hernia repairs were analyzed separately from other deferrable procedures. Primary outcomes included the total number of elective procedures and the number and rates of potentially deferrable procedures per state per year in this population. RESULTS: Over the study period, a total of 27,540 procedures were identified as meeting inclusion criteria; of those, 7832 (28%) were classified as potentially deferrable, 4315 of which were hernia repairs. The average rates of potentially deferrable nonhernia procedures in California, North Carolina, New York, and Utah were 8.3, 43.8, 30.0, and 11.7 per 10,000 person-years, respectively. In multivariable analysis, private insurance (odds ratio [OR] = 1.36), self-pay status (OR = 1.50), and treatment in a different state (OR = 0.48-3.16) were independent predictors of a potentially deferrable procedure being performed on an infant younger than 6 months. CONCLUSIONS: Potentially deferrable procedures are still performed in infants <6 months of age. There appears to be significant variation in timing of these procedures among states. Insurance status and geography may be independent predictors of a procedure being potentially deferrable.

Post-Surgical Relapse in Metopic Synostosis and Limitations of the Interfrontal Angle as an Outcome Measure.

BACKGROUND: The interfrontal angle (IFA) has been utilized as a surrogate for morphologic assessment of the anterior cranial region in metopic synostosis with success in characterizing presenting severity. Its utility as an outcome measure has not been assessed similarly. In this study, we aimed to determine whether meaningful information relative to shape, and relapse in particular, could be assessed using the IFA. METHODS: Patients with operatively treated isolated metopic synostosis were identified and included based on the availability of "previously obtained" low-dose computerized tomography (CT) scans at (1) preoperative and 2 postoperative (2) postoperative day-3 and (3) 1 year time points. Relapse was calculated as the percentage of the operatively induced change in IFA that was retained after the first postoperative year. RESULTS: After exclusionary criteria, 19 patients were identified. The mean IFA values at the 3 time points were 119.3° (±9.8), 139.6° (±6.3), and 135.3° (±7.1), chronologically. Of the 19 patients, 14 (73.7%) experienced relapse, with this subgroup averaging 62.4% retention of their surgical change after 1 year. Increased preoperative severity and delayed age at surgical intervention were concurrently associated with an increased occurrence of relapse (P = 0.011). Another model utilizes preoperative IFA to predict the change in IFA that would accompany surgery on a case-by-case basis (P < 0.0001). CONCLUSIONS: (1) Fronto-orbital advancement offers immediate correction in the frontal bandeau. (2) Surgical impact, using fronto-orbital advancement, can be predicted using preoperative severity. (3) A component of postoperative relapse occurs within the frontal bandeau and is detectable at 1 year via the IFA. Exclusively, the IFA cannot characterize holistic postoperative relapse due to its inherent anatomical constraints; this supports the cessation of routine postoperative computerized tomography scans for determining outcomes.

Morbidity and healthcare costs of vascular anomalies: a national study.

PURPOSE: This study aimed to define morbidities and costs related to modern-day medical care for children with vascular anomalies. METHODS: We reviewed the 2003-2009 Kids' Inpatient Database for pediatric patients (age < 21 years) hospitalized with hemangioma, arteriovenous malformation (AVM), or lymphatic malformation (LM). Patient characteristics, hospital complications, and hospital charges were compared by vascular anomaly type. Multivariable linear regression modeling was used to determine predictors of increasing hospital costs for patients with AVMs. RESULTS: In total, 7485 pediatric inpatients with vascular anomalies were identified. Frequently associated complications included chronic anemia (4.0%), sepsis (4.6%), and hypertension (2.4%). Children with AVM had the highest rate of in-hospital mortality, compared to those with hemangiomas or LM (1.0% vs. 0.1% vs. 0.3%, p < 0.001). AVMs were also associated with the highest median hospital charge, more than twice the cost for hemangiomas or LM ($45,875 vs. $18,909 vs. $18,919; p < 0.001). CONCLUSIONS: There is a significant rate of morbidity in children with vascular anomalies, most often from blood loss and infection. The greater cost of AVM care may be related to the higher mortality rate, associated complications, and complexity of procedures required treating them. Cost-effective management of vascular anomalies should target prevention and the early recognition of both chronic comorbidities and acute complications.

Classification of Cleft Lip/Palate: Then and Now.

Cleft lip and/or palate (CL/P) is phenotypically diverse, making classification difficult. This article explores the evolution of ideas regarding CL/P classification and includes the schemes described by Davis and Ritchie (1922) , Brophy (1923) , Veau (1931) , Fogh-Andersen (1943), Kernahan and Stark (1958) , Harkins et al. (1962) , Broadbent et al. (1968), Spina (1973) , and others. Based on these systems, a longhand structured form is proposed for describing CL/P in a way that is clear, comprehensive, and consistent. A complementary shorthand notation is also described to improve the utility and convenience of this structured form.

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

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

A Standard Set of Outcome Measures for the Comprehensive Appraisal of Cleft Care.

Care of the patient with cleft lip and/or palate remains complex. Prior attempts at aggregating data to study the effectiveness of specific interventions or overall treatment protocols have been hindered by a lack of data standards. There exists a critical need to better define the outcomes-particularly those that matter most to patients and their families-and to standardize the methods by which these outcomes will be measured. This report summarizes the recommendations of an international, multidisciplinary working group with regard to which outcomes a typical cleft team could track, how those outcomes could be measured and recorded, and what strategies may be employed to sustainably implement a system for prospective data collection. It is only by agreeing on a common, standard set of outcome measures for the comprehensive appraisal of cleft care that intercenter comparisons can become possible. This is important for quality-improvement endeavors, comparative effectiveness research, and value-based health-care reform.