The Caregiver Burden of Helmet Therapy following Endoscopic Strip Craniectomy: A Phenomenological Qualitative Study.

INTRODUCTION: This cohort study aimed to elucidate the caregiver burden of helmet therapy (HT), following endoscopic strip craniectomy (ESC) to treat craniosynostosis, in an effort to inform clinicians and future caregivers navigating this therapeutic option. METHODS: Fourteen caregivers of children with positional plagiocephaly (6) and craniosynostosis treated by ESC (8) undergoing HT at a single center were recruited via convenience sampling. Using a phenomenological qualitative approach, semi-structured interviews were conducted to understand the experience of HT for caregivers. Data collection and analysis were iterative and conducted until thematic saturation was reached. RESULTS: Emerging themes revealed five domains of caregiver burden: emotional, cognitive, physical, psychosocial, and financial. No caregiver felt the therapy was too burdensome to complete. Caregivers of both groups also expressed positive aspects of HT related to support from the team, the noninvasive nature of treatment, and the outcomes of therapy. Furthermore, caregivers report overall satisfaction with the process, stating willingness to repeat the treatment with subsequent children if required. CONCLUSION: HT is associated with five major domains of caregiver burden; however, none of the caregivers regret choosing this treatment option, nor was the burden high enough to encourage treatment cessation. This study will inform future prospective analyses that will quantify real-time caregiver burden throughout HT.

Evaluation of Research Diagnostic Criteria in Craniofacial Microsomia.

Characteristics of patients with craniofacial microsomia (CFM) vary in type and severity. The diagnosis is based on phenotypical assessment and no consensus on standardized clinical diagnostic criteria is available. The use of diagnostic criteria could improve research and communication among patients and healthcare professionals. Two sets of phenotypic criteria for research were independently developed and based on multidisciplinary consensus: the FACIAL and ICHOM criteria. This study aimed to assess the sensitivity of both criteria with an existing global multicenter database of patients with CFM and study the characteristics of patients that do not meet the criteria. A total of 730 patients with CFM from were included. Characteristics of the patients were extracted, and severity was graded using the O.M.E.N.S. and Pruzansky-Kaban classification. The sensitivity of the FACIAL and ICHOM was respectively 99.6% and 94.4%. The Cohen's kappa of 0.38 indicated a fair agreement between both criteria. Patients that did not fulfill the FACIAL criteria had facial asymmetry without additional features. It can be concluded that the FACIAL and ICHOM criteria are accurate criteria to describe patients with CFM. Both criteria could be useful for future studies on CFM to create comparable and reproducible outcomes.

A Systematic Review of the Cost-Effectiveness of Cleft Care in Low- and Middle-Income Countries: What is Needed?

OBJECTIVE: The objective of this paper is to conduct a systematic review that summarizes the cost-effectiveness of cleft lip and/or palate (CL/P) care in low- and middle-income countries (LMICs) based on existing literature. DESIGN: We searched eleven electronic databases for articles from January 1, 2000 to December 29, 2020. This study is registered in PROSPERO (CRD42020148402). Two reviewers independently conducted primary and secondary screening, and data extraction. SETTING: All CL/P cost-effectiveness analyses in LMIC settings. PATIENTS, PARTICIPANTS: In total, 2883 citations were screened. Eleven articles encompassing 1,001,675 patients from 86 LMICs were included. MAIN OUTCOME MEASURES: We used cost-effectiveness thresholds of 1% to 51% of a country's gross domestic product per capita (GDP/capita), a conservative threshold recommended for LMICs. Quality appraisal was conducted using the Joanna Briggs Institute (JBI) checklist. RESULTS: Primary CL/P repair was cost-effective at the threshold of 51% of a country's GDP/capita across all studies. However, only 1 study met at least 70% of the JBI criteria. There is a need for context-specific cost and health outcome data for primary CL/P repair, complications, and existing multidisciplinary management in LMICs. CONCLUSIONS: Existing economic evaluations suggest primary CL/P repair is cost-effective, however context-specific local data will make future cost-effectiveness analyses more relevant to local decision-makers and lead to better-informed resource allocation decisions in LMICs.

The First Hybrid International Educational Comprehensive Cleft Care Workshop.

OBJECTIVE: Describe the first hybrid global simulation-based comprehensive cleft care workshop, evaluate impact on participants, and compare experiences based on in-person versus virtual attendance. DESIGN: Cross-sectional survey-based evaluation. SETTING: International comprehensive cleft care workshop. PARTICIPANTS: Total of 489 participants. INTERVENTIONS: Three-day simulation-based hybrid comprehensive cleft care workshop. MAIN OUTCOME MEASURES: Participant demographic data, perceived barriers and interventions needed for global comprehensive cleft care delivery, participant workshop satisfaction, and perceived short-term impact on practice stratified by in-person versus virtual attendance. RESULTS: The workshop included 489 participants from 5 continents. The response rate was 39.9%. Participants perceived financial factors (30.3%) the most significant barrier and improvement in training (39.8%) as the most important intervention to overcome barriers facing cleft care delivery in low to middle-income countries. All participants reported a high level of satisfaction with the workshop and a strong positive perceived short-term impact on their practice. Importantly, while this was true for both in-person and virtual attendees, in-person attendees reported a significantly higher satisfaction with the workshop (28.63 ± 3.08 vs 27.63 ± 3.93; P = .04) and perceived impact on their clinical practice (22.37 ± 3.42 vs 21.02 ± 3.45 P = .01). CONCLUSION: Hybrid simulation-based educational comprehensive cleft care workshops are overall well received by participants and have a positive perceived impact on their clinical practices. In-person attendance is associated with significantly higher satisfaction and perceived impact on practice. Considering that financial and health constraints may limit live meeting attendance, future efforts will focus on making in-person and virtual attendance more comparable.

Endoscopic Versus Open Total Vault Reconstruction of Sagittal Craniosynostosis.

Sagittal craniosynostosis is the most common form of congenital cranial deformity. Surgical interventions are performed either open or endoscopic. Advancements in minimally invasive surgery have enabled the development of the endoscopic suturectomy technique. This is contrasted to the traditional open cranial vault reconstruction. There is a paucity of data comparing the head shape changes from both techniques. This study aims to compare the morphological outcome of endoscopic suturectomy versus total cranial vault reconstruction. METHODS: This is a retrospective comparative study involving 55 cases of sagittal craniosynostosis, 37 of which has open total cranial vault reconstruction and 18 had endoscopic suturectomy procedure. Preoperative and postoperative 3D photographs of both groups were analyzed and compared. The change in correction between preoperative and postoperative state was measured against a crowd-driven standard for acceptable head shape. RESULTS: Total cranial vault had higher percentage change between pre and postoperative cranial index than endoscopic suturectomy (14.7% versus 7.7%, P = 0.003). However, both techniques were able to achieve the minimum standard of 70% correction (TCV 107.5%, ES 100.4%, P = 0.02). CONCLUSION: Total cranial vault and endoscopic suturectomy are effective in correcting scaphocephaly among children with sagittal craniosynostosis. Additionally, both techniques are able to achieve a percentage correction that exceeds the 70% benchmark established by the lay public.

Concepts Important to Patients With Facial Differences: A Qualitative Study Informing a New Module of the FACE-Q for Children and Young Adults.

OBJECTIVE: The concepts important to children and young adults who undergo treatments for facial differences are not well-defined. Measurement of treatment outcomes from the patient's perspective is necessary to ensure goals of treatment are met. We aimed to identify concepts important to children and young adults with facial differences through a qualitative study. DESIGN: An interpretive description qualitative approach was followed. Semistructured interviews were conducted, transcribed verbatim, and coded using a line-by-line approach. Qualitative analysis led to the development of a conceptual framework of outcomes important to patients. SETTING: Interviews were conducted in Canada and the United Kingdom at home, by telephone, or in the hospital. PARTICIPANTS: Participants (N = 72) were recruited between May and June 2014 from craniofacial clinics at the Hospital for Sick Children (Toronto) and Great Ormond Street Hospital (London). Participants included anyone with a visible and/or functional facial difference aged 8 to 29 years and fluent in English, excluding patients with a cleft. The sample included 38 females and 34 males, with a mean age of 13.9 years, and included 28 facial conditions (11 facial paralysis, 18 ear anomalies, 26 skeletal conditions, and 17 soft tissue conditions). RESULTS: Analysis led to identification of important concepts within 4 overarching domains: facial appearance, facial function, adverse effects of treatment, and health-related quality of life (psychological, social, and school function). CONCLUSIONS: Our study provides an understanding of concepts important to children and young adults with facial differences.

Assessing Performance in Simulated Cleft Palate Repair Using a Novel Video Recording Setup.

OBJECTIVE: To test the feasibility of implementing a high-fidelity cleft palate simulator during a workshop in Santiago, Chile, using a novel video endoscope to assess technical performance. DESIGN: Sixteen cleft surgeons from South America participated in a 2-day cleft training workshop. All 16 participants performed a simulated repair, and 13 of them performed a second simulated repair. The repairs were recorded using a low-cost video camera and a newly designed camera mouth retractor attachment. Twenty-nine videos were assessed by 3 cleft surgeons using a previously developed cleft palate objective structured assessment of technical skill (CLOSATS with embedded overall score assessment) and global rating scale. The reliability of the ratings and technical performance in relation to minimum acceptable scores and previous experience was assessed. RESULTS: The video setup provided acceptable recording quality for the purpose of assessment. Average intraclass correlation coefficient for the CLOSATS, global, and overall performance score was 0.69, 0.75, and 0.82, respectively. None of the novice surgeons passed the CLOSATS and global score for both sessions. One participant in the intermediate group, and 2 participants in the advanced group passed the CLOSATS and global score for both sessions. There were highly experienced participants who failed to pass the CLOSATS and global score for both sessions. CONCLUSIONS: The cleft palate simulator can be practically implemented with video-recording capability to assess performance in cleft palate repair. This technology may be of assistance in assessing surgical competence in cleft palate repair.

Comparing Cephalic Index and Midsagittal Vector Analysis in Assessing Morphology in Sagittal Synostosis: A CT-Based Morphometric Analysis.

INTRODUCTION: Assessment of cranial dysmorphism in sagittal synostosis is often subjective but objective measures can be applied. These include cephalic index (CI) and midsagittal vector analysis (MSVA). OBJECTIVE: To assess discriminant validity, construct validity, and responsiveness of CI and MSVA measured from computed tomography (CT) in patients with sagittal synostosis. METHODS: Patients with nonsyndromic isolated sagittal synostosis with complete preoperative (n = 30) and postoperative (n = 13) CT data were included. Age-matched control group (n = 24) comprised of normocephalic patients who underwent CT for reasons related to trauma. OUTCOME MEASURES: Retrospective CT evaluation of CI and MSVA was conducted and correlated with a dysmorphism numeric rating scale (D-NRS) that measured surgeon-rated severity of sagittal synostosis. Responsiveness of CI and MSVA was evaluated using dysmorphism global rating of change (D-GRC). RESULTS: Thirty patients with sagittal synostosis were demographically similar to 24 normocephalic patients. The difference in CI and MSVA was statistically significant between normocephalic and scaphocephalic patients. Cephalic index had a good correlation with D-NRS (r = -0.665, ρ = -0.667), but not with MSVA (r = 0.250, ρ = 0.203). Change in CI (r = 0.738, ρ = 0.657) was well correlated with D-GRC, but not with MSVA (r = -0.409, ρ = -0.301). CONCLUSION: Cephalic index appears to quantify the severity of sagittal synostosis better than MSVA. Cephalic index also has better responsiveness than MSVA to measure a reduction in severity of disease; however, MSVA is a better descriptive craniometric measurement. Midsagittal vector analysis was able to quantify the shift in morphology in sagittal synostosis following surgical treatment.

Economy of Hand Motion During Cleft Palate Surgery Using a High-Fidelity Cleft Palate Simulator.

OBJECTIVE: The objectives of this study were to assess economy of hand motion of residents, fellows, and staff surgeons using a high-fidelity cleft palate simulator to (1) stratify performance for the purpose of simulator validation and (2) to estimate the learning curve. DESIGN: Two residents, 2 fellows, and 2 staff surgeons performed cleft palate surgery on a high-fidelity cleft palate simulator while their hand motion was tracked using an electromagnetic hand sensor. The time, number of hand movements, and path length of their hands were determined for 10 steps of the procedure. The magnitude of these metrics was compared among the 3 groups of participants and utilized to estimate the learning curve using curve-fitting analysis. RESULTS: The residents required the most time, number of hand movements, and path length to complete the procedure. Although the number of hand movements was closely matched between the fellows and staff, the overall total path length was shorter for the staff. Inverse curves were fit to the data to represent the learning curve and 25 and 113 simulation sessions are required to reach within 5% and 1% of the expert level, respectively. CONCLUSION: The simulator successfully stratified performance using economy of hand motion. Path length is better matched to previous level of experience compared to time or number of hand movements.

Psychometric findings and normative values for the CLEFT-Q based on 2434 children and young adult patients with cleft lip and/or palate from 12 countries.

BACKGROUND: Patients with cleft lip and/or palate can undergo numerous procedures to improve appearance, speech, dentition and hearing. We developed a cleft-specific patient-reported outcome instrument to facilitate rigorous international measurement and benchmarking. METHODS: Data were collected from patients aged 8-29 years with cleft lip and/or palate at 30 hospitals in 12 countries between October 2014 and November 2016. Rasch measurement theory analysis was used to refine the scales and to examine reliability and validity. Normative CLEFT-Q values were computed for age, sex and cleft type. RESULTS: Analysis led to the refinement of an eating and drinking checklist and 12 scales measuring appearance (of the face, nose, nostrils, teeth, lips, jaws and cleft lip scar), health-related quality of life (psychological, social, school, speech distress) and speech function. All scales met the requirements of the Rasch model. Analysis to explore differential item functioning by age, sex and country provided evidence to support the use of a common scoring algorithm for each scale for international use. Lower (worse) scores on CLEFT-Q scales were associated with having a speech problem, being unhappy with facial appearance, and needing future cleft-related treatments, providing evidence of construct validity. Normative values for age, sex and cleft type showed poorer outcomes associated with older age, female sex and having a visible cleft. INTERPRETATION: The CLEFT-Q represents a rigorously developed instrument that can be used internationally to collect and compare evidence-based outcomes data from patients aged 8-29 years of age with cleft lip and/or palate.

Content Validity of Patient-Reported Outcome Instruments used with Pediatric Patients with Facial Differences: A Systematic Review.

OBJECTIVE: The aim of this systematic review was to identify patient-reported outcome (PRO) instruments used in research with children/youth with conditions associated with facial differences to identify the health concepts measured. DESIGN: MEDLINE, EMBASE, CINAHL, and PsycINFO were searched from 2004 to 2016 to identify PRO instruments used in acne vulgaris, birthmarks, burns, ear anomalies, facial asymmetries, and facial paralysis patients. We performed a content analysis whereby the items were coded to identify concepts and categorized as positive or negative content or phrasing. RESULTS: A total of 7,835 articles were screened; 6 generic and 11 condition-specific PRO instruments were used in 96 publications. Condition-specific instruments were for acne (four), oral health (two), dermatology (one), facial asymmetries (two), microtia (one), and burns (one). The PRO instruments provided 554 items (295 generic; 259 condition specific) that were sorted into 4 domains, 11 subdomains, and 91 health concepts. The most common domain was psychological (n = 224 items). Of the identified items, 76% had negative content or phrasing (e.g., "Because of the way my face looks I wish I had never been born"). Given the small number of items measuring facial appearance (n = 19) and function (n = 22), the PRO instruments reviewed lacked content validity for patients whose condition impacted facial function and/or appearance. CONCLUSIONS: Treatments can change facial appearance and function. This review draws attention to a problem with content validity in existing PRO instruments. Our team is now developing a new PRO instrument called FACE-Q Kids to address this problem.

What Matters to Patients With Cleft Lip and/or Palate: An International Qualitative Study Informing the Development of the CLEFT-Q.

OBJECTIVE: The goal of treatment for individuals with cleft lip and/or palate (CL/P) is to improve physical, psychological, and social health. Outcomes of treatment are rarely measured from the patient's perspective. The aim of the study was to develop a conceptual framework for a patient-reported outcome (PRO) instrument for individuals with clefts (CLEFT-Q) by developing an in-depth understanding of issues that individuals consider to be important. DESIGN: The qualitative methodology of interpretive description was used. Setting, Participants, and Intervention: We performed 136 individual in-depth interviews with participants with clefts of any age, presenting for cleft care, across 6 countries. Parents were involved if the child was more comfortable. Interviews were audio-recorded, transcribed verbatim, and coded using constant comparison. The data were used to develop a refined conceptual framework. RESULTS: Participants described concepts of interest in 3 top-level domains, each of which included subdomains: appearance (face, nose, nostrils, teeth, lips, jaw, cleft lip scar), health-related quality of life (psychological, social, school, speech-related distress), and facial function (speech, eating/drinking). Participants were able to describe changes over time with regard to the 3 domains. CONCLUSIONS: A conceptual framework of concepts of interest to individuals with CL/P formed the basis of the scales in the CLEFT-Q. Each subdomain represents an independently functioning scale. Understanding what matters to patients is essential in guiding PRO measurement.

Bilateral Malar Reconstruction Using Patient-Specific Polyether Ether Ketone Implants in Treacher-Collins Syndrome Patients With Absent Zygomas.

The authors performed bilateral malar reconstruction using polyether ether ketone implants in 3 patients with Treacher-Collins syndrome with absent, as opposed to hypoplastic, zygomata. These patient-specific implants were fabricated using computed-aided design software reformatted from three-dimensional bony preoperative computed tomography images. The first time the authors performed this procedure the implant compressed the globe resulting in temporary anisocoria that was quickly recognized intraoperatively. The implant was immediately removed and the patient made a full-recovery with no ocular disturbance. The computer-aided design and manufacturing process was adjusted to include periorbital soft-tissue boundaries to aid in contouring the new implants. The same patient, and 2 further patients, subsequently underwent malar reconstruction using this soft tissue periorbital boundary fabrication process with an additional 2 mm relief removed from the implant's orbital surface. These subsequent procedures were performed without complication and with pleasing aesthetic results. The authors describe their experience and the salutary lessons learnt.

FGFR-associated craniosynostosis syndromes and gastrointestinal defects.

Craniosynostosis is a relatively common birth defect characterized by the premature fusion of one or more cranial sutures. Examples of craniosynostosis syndromes include Crouzon (CS), Pfeiffer (PS), and Apert (AS) syndrome, with clinical characteristics such as midface hypoplasia, hypertelorism, and in some cases, limb defects. Mutations in Fibroblast Growth Factor Receptor-2 comprise the majority of known mutations in syndromic forms of craniosynostosis. A number of clinical reports of FGFR-associated craniosynostosis patients and mouse mutants have been linked to gastrointestinal tract (GIT) disorders, leading to the hypothesis of a direct link between FGFR-associated craniosynostosis syndromes and GIT malformations. We conducted an investigation to determine GIT symptoms in a sample of FGFR-associated craniosynostosis syndrome patients and a mouse model of CS containing a mutation (W290R) in Fgfr2. We found that, compared to the general population, the incidence of intestinal/bowel malrotation (IM) was present at a higher level in our sample population of patients with FGFR-associated craniosynostosis syndromes. We also showed that the mouse model of CS had an increased incidence of cecal displacement, suggestive of IM. These findings suggest a direct relationship between FGFR-related craniosynostosis syndromes and GIT malformations. Our study may shed further light on the potential widespread impact FGFR mutations on different developmental systems. Based on reports of GIT malformations in children with craniosynostosis syndromes and substantiation with our animal model, GIT malformations should be considered in any child with an FGFR2-associated craniosynostosis syndrome. © 2016 Wiley Periodicals, Inc.

Skull base development and craniosynostosis.

Abnormal skull shape resulting in craniofacial deformity is a relatively common clinical finding, with deformity either positional (positional plagiocephaly) or related to premature ossification and fusion of the skull sutures (craniosynostosis). Growth restriction occurring at a stenosed suture is associated with exaggerated growth at the open sutures, resulting in fairly predictable craniofacial phenotypes in single-suture non-syndromic pathologies. Multi-suture syndromic subtypes are not so easy to understand without imaging. Imaging is performed to define the site and extent of craniosynostosis, to determine the presence or absence of underlying brain anomalies, and to evaluate both pre- and postoperative complications of craniosynostosis. Evidence for intracranial hypertension may be seen both pre- and postoperatively, associated with jugular foraminal stenosis, sinovenous occlusion, hydrocephalus and Chiari 1 malformations. Following clinical assessment, imaging evaluation may include radiographs, high-frequency US of the involved sutures, low-dose (20-30 mAs) CT with three-dimensional reformatted images, MRI and nuclear medicine brain imaging. Anomalous or vigorous collateral venous drainage may be mapped preoperatively with CT or MR venography or catheter angiography.

Optimization of Cranio-Orbital Remodeling: Application of a Mathematical Model.

Cranio-orbital remodeling aims to correct the dysmorphic skull associated with craniosynostosis. Traditionally, the skull is reconstructed into a shape that is subjectively normal according to the surgeon's perception. We present a novel technique using a mathematical algorithm to define the optimal location for bony osteotomies and to objectively reshape the fronto-orbital bar into an ideal normal skull contour. Using pre-operative computed tomography images, the abnormal skull contour at the frontal-orbital region was obtained for infants planned to undergo cranio-orbital remodeling. The ideal skull shape was derived from an age- and sex-matched normative skull library. For each patient, the mathematical technique of dynamic programming (DP) was applied to compare the abnormal and ideal skull shapes. The DP algorithm identifies the optimal location of osteotomy sites and calculates the objective difference in surface area remaining between the normative and dysmorphic skull shape for each solution applied. By selecting the optimal solution with minimal objective difference, the surgeon is guided to reproducibly recreate the normal skull contour with defined osteotomies. The DP algorithm was applied in 13 cases of cranio-orbital remodeling. Five female and 8 male infants with a mean age of 11 months were treated for craniosynostosis classified as metopic (n = 7), unicoronal (n = 4), or bicoronal (n = 2). The mean OR time was 190.2  min (SD 33.6), mean estimated blood loss 244  cc (SD 147.6), and 10 infants required blood transfusions. Compared with a historical crania-orbital remodeling group treated without application of the algorithm, there was no significant difference in OR time, estimated blood loss, or transfusion rate. This novel technique enables the craniofacial surgeon to objectively reshape the fronto-orbital bar and reproducibly reconstruct a skull shape resembling that of normal infants.

Is Linear Advancement Related to Relapse in Unilateral Cleft Lip and Palate Orthognathic Surgery?

OBJECTIVE: To investigate the stability of major versus minor Le Fort I maxillary advancements in unilateral cleft lip and palate (UCLP) patients. DESIGN: A retrospective longitudinal study was undertaken on 30 nonsyndromic UCLP patients treated with the same protocol at The Hospital for Sick Children, Toronto, Canada. Patients were grouped into major and minor movement groups based on planned surgical advancement. Standard lateral cephalometric radiographs were taken preoperatively (T1), immediately postoperatively (T2), and at least 1 year postoperatively (T3). Skeletal and dental variables were measured using cephalometric analysis. Stability was compared between groups using repeated-measures analysis of variance. Linear regression analysis was used to assess the relationship between advancement and relapse for the entire study population. RESULTS: A mean maxillary advancement of 9.8 mm and 4.9 mm was seen for the major (n = 10) and minor (n = 20) movement groups, respectively. The mean skeletal horizontal relapse was 1.8 mm (18%) for the major advancement group and 1.5 mm (31%) for the minor advancement group. There was no significant difference in skeletal horizontal relapse between the groups (P > .05). The correlation coefficient (r) between linear horizontal advancement and relapse was calculated to be .31 (P > .05). Dental horizontal relapse was not significant for either the major or minor groups, and no significant difference was found between the groups (P > .05). CONCLUSION: Skeletal and dental relapse was found to be unrelated to the amount of maxillary linear advancement using conventional Le Fort I osteotomies in UCLP.

Single versus segmental maxillary osteotomies and long-term stability in unilateral cleft lip and palate related malocclusion.

PURPOSE: To investigate the stability of single-piece versus segmental (2-piece) maxillary advancement in patients with unilateral cleft lip and palate (UCLP) treated using conventional Le Fort I orthognathic surgery. PATIENTS AND METHODS: A retrospective study was undertaken in 30 patients with nonsyndromic UCLP treated with the same surgical and orthodontic protocol from 2002 through 2011. Standard lateral cephalometric radiographs were taken preoperatively, immediately postoperatively, and at least 1 year postoperatively. Patients were divided into single-piece and segmental Le Fort I groups based on planned surgical movement. Postoperative movements were compared between groups using repeated measures analysis of variance. RESULTS: The mean skeletal horizontal advancement was 7.3 and 7.5 mm in the single-piece and segmental groups, respectively. The skeletal horizontal relapse was 1.3 mm (18%) for the single-piece group and 1.9 mm (25%) for the segmental group. The skeletal surgical extrusion was 2.7 mm for the 2 groups. The skeletal vertical relapse was 0.6 mm (22%) and 1.5 mm (56%) for the single-piece and segmental groups, respectively. The mean dental horizontal postoperative movement was an advancement of 0.4 mm for the single-piece group and a relapse of 0.2 mm (3%) for the segmental group. The mean dental vertical relapse was 0.1 mm (4%) for the single-piece group and 0.3 mm (11%) for the segmental group. There was no statistically significant difference in relapse between the single-piece and segmental groups for all movements (P>.05). CONCLUSION: Skeletal and dental relapse was similar between single-piece and segmental maxillary advancements using conventional Le Fort I orthognathic surgery in patients with UCLP.

Computed tomography-based morphometric analysis of extended strip craniectomy for sagittal synostosis.

BACKGROUND: The purpose of this study is 2-fold: (1) to identify computed tomography (CT)-based morphometric parameters that differentiate the normal skull from one with sagittal synostosis and (2) to evaluate correction of sagittal synostosis with extended strip craniectomy and postoperative helmeting based on morphometric parameters. METHODS: An institutional review board-approved, retrospective review was carried out at the Hospital for Sick Children for all patients who underwent an extended strip craniectomy and postoperative helmeting for sagittal synostosis from 1999 to 2005. Inclusion criteria consisted of patients who underwent a routine craniofacial CT preoperatively and 12 months postoperatively. Craniofacial CT scans of age-matched control subjects were used for preoperative and postoperative comparison. RESULTS: Thirty-nine patients with sagittal synostosis met inclusion criteria. Median age at preoperative CT was 3.0 months. Nine control subjects were identified, with a median age at CT scan of 5.0 months. Patients with sagittal synostosis preoperatively had a significantly longer maximum cranial length, smaller maximum cranial breadth, more acute frontal takeoff and occipital incline angles, lower cephalic index, and an anteriorly positioned vertex. Postoperative CT scans (median, 17.0 months) were compared with 10 control subjects (median, 19.0 months). Patients with sagittal synostosis postoperatively had equivalent maximum cranial breadth, frontal takeoff, and occipital incline angles as compared with controls. Sagittal synostosis patients remained with a significantly longer maximum cranial length, lower cephalic index, and anteriorly positioned vertex. CONCLUSIONS: Twelve months following extended strip craniectomy and helmeting for sagittal synostosis, CT-based morphometric analysis demonstrated correction of cranial breadth, frontal bossing, and occipital bulleting. Skull length and vertex position did not fully correct.

Cephalometric outcomes of orthognathic surgery in hemifacial microsomia.

Hemifacial microsomia is a hypoplastic disorder of the first and second branchial arches that significantly impacts on the development of the jaws, leading to malocclusion and facial asymmetry. There is little in the literature regarding the application of orthodontic/orthognathic approaches to the correction of these deformities and the stability of the surgical results. To address this, a retrospective chart review of 10 patients with complete orthodontic records and greater than 1 year of follow-up was performed. Posteroanterior cephalograms were assessed by modified Grummons analysis to determine mandibular offset (deviation of the chin point from the skeletal midline) and occlusal cant. These measurements were performed at 3 time points (T1: preoperative, T2: immediate postoperative, T3: follow-up) to elucidate the surgical movement (T2-T1), the postoperative relapse (T3-T2), and the net gain movement (T3-T1). Maxillary movements were quantified, and the occlusal cant was expressed as a ratio between vertical heights of the maxilla at the first molar on each side. One sample t test demonstrated statistically significant surgical movement and net gain. Relapse was statistically insignificant. Repeated-measures analysis of variance demonstrated similar results for chin point position relative to the putative midline. Our results suggest that a combined orthodontic/orthognathic approach at skeletal maturity delivers improved occlusal outcomes in the long term as assessed by chin point deviation and occlusal cant, but secondary surgery rates are higher than those for orthognathic surgery in other patient groups. We advocate limiting surgery to skeletal maturity whenever possible to achieve stable long-term results while limiting morbidity and number of procedures.