Spectrum of Disease in Hospitalized Newborns with Congenital Micrognathia: A Cohort of 3,236 Infants at North American Tertiary-Care Intensive Care Units.

OBJECTIVE: To describe the spectrum of disease and burden of care in infants with congenital micrognathia from a multicenter cohort hospitalized at tertiary care centers. STUDY DESIGN: The Children's Hospitals Neonatal Database was queried from 2010 through 2020 for infants diagnosed with micrognathia. Demographics, presence of genetic syndromes, and cleft status were summarized. Outcomes included death, length of hospitalization, neonatal surgery, and feeding and respiratory support at discharge. RESULTS: Analysis included 3,236 infants with congenital micrognathia. Cleft palate was identified in 1266 (39.1%). A genetic syndrome associated with micrognathia was diagnosed during the neonatal hospitalization in 256 (7.9%). Median (IQR) length of hospitalization was 35 (16, 63) days. Death during the hospitalization (n = 228, 6.8%) was associated with absence of cleft palate (4.4%, P < .001) and maternal Black race (11.6%, P < .001). During the neonatal hospitalization, 1289 (39.7%) underwent surgery to correct airway obstruction and 1059 (32.7%) underwent gastrostomy tube placement. At the time of discharge, 1035 (40.3%) were exclusively feeding orally. There was significant variability between centers related to length of stay and presence of a feeding tube at discharge (P < .001 for both). CONCLUSIONS: Infants hospitalized with congenital micrognathia have a significant burden of disease, commonly receive surgical intervention, and most often require tube feedings at hospital discharge. We identified disparities based on race and among centers. Development of evidence-based guidelines could improve neonatal care.

The Incidence of Thromboembolic Events During Hospitalizations Following Surgical Resection of Lower Extremity Sarcomas in Children: Utilizing the Pediatric Health Information System Database.

BACKGROUND: Venous thromboembolism (VTE) is a frequent occurrence during treatment for adults with sarcoma. The incidence and underlying risk factors of postsurgical VTE in children and adolescents undergoing resection of sarcoma are unknown. METHODS: Using International Classification of Disease revision-9 diagnostic and procedure codes, the Pediatric Health Information System database was queried for patients aged 18 years and younger, discharged from 2004 to 2015 with a diagnosis of lower extremity malignant neoplasm who had a tumor resection or amputation performed during the encounter. Malignant neoplasms of the pelvic bones and soft tissues were categorized as "pelvis tumors", whereas malignant neoplasms of bone and soft tissues of the lower limbs were categorized as "lower limb tumors". Hospitalizations were evaluated for the occurrence of VTE. Demographic characteristics (age at admission, sex, race, and race/ethnicity) and incidence of VTE were reported. RESULTS: There were 2400 patients identified. Of these, 19 experienced VTE (0.79%) during their surgical hospitalization encounter. By anatomic group, the rate of VTE was 1.4% (CI: 0.5%-3.2%) for tumors in the pelvis and 0.6% (CI: 0.3%-1.0%) in lower limb tumors. Categorizing by age, the incidence of VTE was 1.2% in patients aged zero to 5, 0.3% in patients 6 to 13, and 1.2% in patients 14 to 18 years old. (Table 1). The extremely low rate of VTE occurrence precluded further analysis of risk factors. CONCLUSIONS: In this analysis, postsurgical VTE during hospitalization after pelvic and lower extremity sarcoma resection was an uncommon event in children and adolescents. There seemed to be an increased incidence of postsurgical VTE in pelvic tumors when compared with lower limb tumors, however, the rarity of all events precluded formal statistical analysis. A more robust data set would be required to determine if there are subsets of children and adolescents with sarcoma at higher risk of VTE that could benefit from thromboprophylaxis in the postoperative setting. LEVEL OF EVIDENCE: Level II.

A Survey of Bone Grafting Practice Patterns in North American Cleft Surgeons.

INTRODUCTION: Alveolar bone grafting aims to restore bony continuity of the alveolus and provide optimal periodontal support for teeth adjacent to the cleft. We created a survey of cleft surgeons to assess the current standard of care regarding this procedure. METHODS: A multiple choice survey was implemented using Qualtrics software and emailed to a list of 708 surgeons from the ACPA membership directory. Correlation between various provider factors and treatment practices was assessed with Fisher's exact test and likelihood ratio tests. RESULTS: The response rate was 17.5%. Eighty-seven percent of providers preferred to perform grafts prior to secondary canine eruption while 10% favored before central incisor eruption. Eighty-one percent favored palatal expansion prior to bone grafting. Wide variability existed regarding the time to initiate postoperative orthodontics; 43% waited 4 to 6 months. Sixty-four percent of surgeons now utilize cone beam CT to assess graft take. The majority of respondents utilized cancellous bone autograft (92%) from the anterior iliac crest (97%) as graft material. Seventy percent used three or more modalities for post-operative pain control management. Early career surgeons (0-5 years) appeared more likely to use non-autologous materials (p < .01) for grafting. CONCLUSION: Alveolar bone grafting prior to secondary canine eruption remains the most common strategy but other protocols are employed. Surgeons utilize multiple modalities for radiographic evaluation and most often use autologous cancellous bone as the primary grafting material. There is no true consensus on the perioperative timing and sequencing of orthodontic manipulation while principles of multimodal perioperative pain control appear widely accepted.

An Algorithmic Approach for Deploying Buccal Fat Pad Flaps and Buccal Myomucosal Flaps Strategically in Primary and Secondary Palatoplasty.

OBJECTIVE: Recent publications have introduced the use of buccal myomucosal and fat pad flaps to augment palatal repairs with autologous tissue. We propose a workflow for intraoperative decision-making to introduce these adjuncts into standard palatoplasty procedures. DESIGN/PATIENTS: A retrospective chart review of a single-surgeon series of patients undergoing primary and secondary palatoplasties performed between October 2017 and November 2020 was completed after Institutional Review Board approval. MAIN OUTCOME MEASURES: Patient demographics, phenotype, operative details, and postoperative complications were recorded. RESULTS: Fifty-eight patients were included in a review. For those undergoing primary repair, 23.3% underwent a Furlow palatoplasty alone, 46.3% had a Furlow palatoplasty accompanied with acellular dermal matrix (ADM) and/or a buccal fat flap (BFF). A unilateral buccal myomucosal flap (BMMF) with or without augmentation with BFF or ADM was employed in 16.3% of the cases. Fourteen percent required a bilateral BMMF+/- ADM. Fistula occurrence was 2.3% (n = 1). For revisions, 27% underwent only a conversion Furlow palatoplasty, 26% had a conversion Furlow palatoplasty accompanied with ADM and/or a BFF, 33% had a unilateral BMMF or BMMF/ADM, and 14% required a bilateral BMMF+/- ADM. CONCLUSIONS: In severe phenotypes or complicated cases, buccal fat flaps and myomucosal flaps may be utilized. This approach has mostly replaced the use of ADM over time. An algorithmic approach to palatoplasty allows surgeons to tailor the extent of surgery to the needs of each patient.

Design and Validation of a 3D Printed Cranio-Facial Simulator: A Novel Tool for Surgical Education.

OBJECTIVE: To assess the ability of current 3D printing technology to generate a craniofacial bony and soft tissue anatomical model for use in simulating the performance of a fronto-orbital advancement (FOA) osteotomy and then to further assess the value of the model as an educational tool. DESIGN: Anatomic models were designed with a process of serial anatomic segmentation/design, 3D printing, dissection, and device refinement. A validation study was conducted with 5 junior and 5 senior plastic surgery residents. The validation study incorporated a multiple-choice Knowledge Assessment test (KA), an Objective Structured Assessment of Technical skills (OSATs), a Global Rating Scale (GRS) and a Michigan Standard Simulation Experience Scale (MiSSES). We compared the scores of both the junior and senior residents and compared junior resident scores, before and after viewing a lecture/demonstration. RESULTS: MiSSES showed high face validity with a score of 85.1/90, signifying high satisfaction with the simulator learning experience. Simulation and the lecture/demonstration improved the junior resident average KA score from 5.6/10 to 9.6/10 (P = .02), OSATs score from 32.4/66 to 64.4/66 (P < .001) and GRS score from 13.9/35 to 27.5/35 (P < .001). The senior residents OSATs score of 56.3/66 was higher than the pre-lecture juniors (32.4/66) (P < .001), but lower than the post-lecture juniors (64.4/66) (P < .001). CONCLUSION: We have successfully fabricated a 3D printed craniofacial simulator capable of being used as an educational tool alongside traditional surgical training. Next steps would be improving soft tissue realism, inclusion of patient and disease specific anatomy and creation of models for other surgical specialties.

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.

Determination of Ethnic Variation in Infant Nasolabial Anthropometry Using 3D Photographs: Implications for Bilateral Cleft Lip Nasal Correction.

OBJECTIVE: We aimed to assess significant ethnic variabilities in infants' nasolabial anthropometry to motivate variations in surgical correction of a synchronous bilateral cleft lip/nasal anomaly, specifically whether a long columella is a European feature, therefore accepting a short columella and/or delayed columellar lengthening suitable for reconstruction in ethnic patients. METHODS: Thirty-three infants without craniofacial pathology (10 African American [AA], 7 Hispanic [H], and 16 of European descent [C]), ages 3 to 8 months, presenting to the Johns Hopkins All Children's general pediatric clinic were recruited. Four separate 3D photographs (2 submental and frontal views each) were taken using the Vectra H1 handheld camera (Canfield Imaging). Eighteen linear facial distances were measured using Mirror 3D analysis (Canfield Imaging Systems). Difference between ethnicities was measured using analysis of variance with the Bonferroni/Dunn post hoc comparisons. Pearson correlation was employed for interrater reliability. All statistical analyses were carried out using SPSS version 21.0 (IBM Corp), with statistical significance set at P < .05. RESULTS: Nasal projection (sn-prn) and columella length (sn-c) did not differ significantly between groups (P = .9). Significant differences were seen between ethnic groups in nasal width (sbal-sbal [C-AA; P = .02]; ac-ac [C-AA; P = .00; H-AA; P = .04]; al-al [C-AA; P = .00; H-AA; P = .001]) and labial length (sn-ls [C-AA; P = .041]; sn-sto [C-AA; P = .005]; Cphs-Cphi L [C-AA; P = .013]; Cphs-Cphi R [C-AA; P = .015]). Interrater reliability was good to excellent and significantly correlated for all measures. CONCLUSIONS: African American infants exhibited wider noses and longer lips. No difference was noted in nasal projection or columella length, indicating that these structures should be corrected during the primary cleft lip and nasal repair for all patients and should not be deferred to secondary correction.

Degree of Asymmetry Between Patients With Complete and Incomplete Cleft Lips.

INTRODUCTION: Surgical outcomes for patients with complete cleft lips are not as ideal as those achieved for milder phenotypes. We hypothesized that in addition to the greater width of the cleft, patients with complete cleft lip and palate exhibit a greater degree of hypoplasia and asymmetry. METHODS: Stereophotographs of 14 infants with unrepaired unilateral complete and 14 with incomplete cleft lips were measured using Vectra imaging software (Canfield Imaging). Unpaired t tests were used to compare measured asymmetry between groups. Measurements included nasion to endocathion, sn-sbal, subnasale to alare (sn-al), subnasale to crista philtra, subalare to crista philtra (sbal-cphi), chelion to crista philtra, lateral lip element fullness, medial lip element fullness (mef), and non-cleft lip fullness. Duplicate measurements allowed Pearson correlation to be used to determine intra-rater reliability. Statistical significance was set at P < .05. RESULTS: Degree of asymmetry of the nasal base, sn-al, and sn-sbal was significantly greater for patients with complete clefts (P = .0001, P = .0001). Hypoplasia of the lateral lip element was seen when comparing lateral and mef (P = .04, P = .004) and lateral lip height (sbal-cphi''; P = .002). The degree of cupid's bow asymmetry did not differ between groups (P = .23). Intrarater reliability was high for all facial measures, ranging from 0.70 to 0.99. CONCLUSIONS: More severe, complete cleft lips demonstrate statistically significant greater asymmetry in surgically relevant dimensions. There was greater width of the nasal base. Vertical asymmetry of cupid's bow was unaffected by cleft severity, but that asymmetry was greater in patients with complete clefts due to hypoplasia of the lateral lip element. This may contribute to the less favorable results in these patients.

Patients With a History of Oronasal Fistula Repair Exhibit Lower Oral Health Measured With Patient-Centric Outcomes Measures.

INTRODUCTION: Oronasal fistulae following palatoplasty may affect patients' quality of life by impacting their ability to eat, speak, and maintain oral hygiene. We aimed to quantify the impact of previous oronasal fistula repair on patients' quality of life using patient-reported outcome psychometric tools. METHODS: A cross-sectional study of 8- to 9-year-old patients with cleft palate and/or lip was completed. Patients who had a cleft team clinic between September 2018 and August 2019 were recruited. Participants were divided into 2 groups (no fistula, prior fistula repair). Differences in the individual CLEFT-Q and Child Oral Health Impact Profile-Short Form 19 (COHIP-SF 19) Oral Health scores between the 2 groups were evaluated using a multivariate analysis controlling for Veau classification and syndromic diagnosis. RESULTS: Sixty patients with a history of cleft palate were included. Forty-two (70%) patients had an associated cleft lip. Thirty-two (53.3%) patients had no history of fistula and 28 (46.7%) patients had undergone a fistula repair. CLEFT-Q Dental, Jaw, and Speech Function were all higher in patients without a history of a fistula repair; however, none of these differences were statistically significant. The COHIP-SF 19 Oral Health score demonstrated a significantly lower score in the fistula group, indicating poorer oral health (P = .05). CONCLUSIONS: One would expect that successful repair of a fistula would result in improved function and patient satisfaction, but the consistent trend toward lower CLEFT-Q scores and significantly increased COHIP-SF 19 Oral Health scores in our study group suggests that residual effects linger and that the morbidity of a fistula may not be completely treated with a secondary correction.

Laryngoscopy Grade Improvement and Difficult Airway Resolution in Infants With Robin Sequence Undergoing Mandibular Distraction Osteogenesis: A Multi-Institutional Study.

OBJECTIVE: Mandibular distraction osteogenesis (MDO) aims to relieve tongue-based airway obstruction in Robin Sequence (RS). We investigated direct laryngoscopy grade (DLG) improvement and difficult airway (DA) resolution following MDO. DESIGN: Retrospective cohort analysis. SETTING: Three tertiary care institutions. PATIENTS: Sixty-four infants with RS who underwent a single MDO procedure in their first year of life were identified from January 2010 to January 2019. MAIN OUTCOME MEASURES: The primary outcome was DLG pre- and post-MDO. Secondary outcomes included DA designation, pre- and post-MDO polysomnographic assessment for obstructive sleep apnea (OSA), length of stay, need for gastrostomy, and major/minor adverse events. RESULTS: Median DLG improved from II pre-MDO to I at the time of distractor removal (n = 43, P < .001). No significant change was seen in patients with a third recorded time point (eg, palatoplasty; n = 78, P = .52). Twenty-six (47%) of 55 patients were designated as DA pre-MDO, and 10 (18%) of 55 patients retained the label post-MDO (P < .01). Five (50%) of these 10 patients appeared to be inappropriately retained. Median obstructive apnea-hypopnea index improved from 38.6 (range 31.2-62.8) pre-MDO to 2.9 (range 1-3.9) post-MDO (n = 12; P = .002). CONCLUSION: Mandibular distraction osteogenesis allowed for DLG improvement that was stably maintained as well as functional improvement in OSA, with minimal morbidity. Difficult airway designation persisted in the electronic record of some infants despite clinical resolution.

Le Fort 1 and Bimaxillary Osteotomies Increase the Length of Stay but not Postoperative Morbidity Compared to Mandibular Osteotomies and Single Jaw Procedures.

PURPOSE: The purpose of this study is to examine the association between type of facial osteotomies performed during orthognathic surgery and postoperative outcomes including complications, length of stay, and readmission. METHODS: A retrospective review of orthognathic surgery cases from the Pediatric Health Information System (PHIS) database from 2004 to 2014 was undertaken. Osteotomy procedures were classified as Le Fort 1 (LF 1), Mandibular osteotomy, Genioplasty or their combinations. Primary outcome variable was major complications. Secondary outcomes included postoperative LOS > 1 day and 90-day readmission. Random-intercept logistic regression models were utilized to assess the association between the type of osteotomy performed and the outcomes. Bonferroni approach was used to account for multiple comparisons. RESULTS: The sample included 5413 patients, with a mean age of 17.1 ±â€Š1.68 years and 60.65% were female. The most common procedures were LF1 (39.4%), followed by bimaxillary surgery (23%). Major complications occurred in 8.57% of patients, postoperative LOS > 1 day in 52.4% and 90-day all-cause readmission in 11.16%. In adjusted analyses comparing LF1 compared to mandibular osteotomies, there were no significant differences for major complications (OR = 0.78), 90-day readmission rate (OR = 0.98). However, LF1 was associated with an increased odds for LOS compared to mandibular osteotomies (OR = 1.42). Addition of osteotomies is associated with a significant increase in LOS (P < 0.001). CONCLUSIONS: Patients undergoing orthognathic surgery demonstrated increased length of stay for LF1 or bimaxillary osteotomies. Osteotomy type did not impact the odds of readmission or complications. The trends revealed should be helpful for patient counseling.

Maxillomandibular Advancement for the Treatment of Obstructive Sleep Apnea in Patients With Normal or Class I Malocclusion.

BACKGROUND: Maxillomandibular advancement is an effective surgical option for obstructive sleep apnea (OSA) that achieves enlargement of the upper airway by physically expanding the facial skeleton. The authors sought to determine whether an advancement of 10 mm predicts surgical success and if any correlation existed between the magnitude of mandibular/maxillary advancement and improvement in polysomnography metrics using aggregated individual patient data from multiple studies. METHODS: A search of the PubMed database was performed to identify relevant articles that included preoperative and postoperative polysomnography data and measurements of the advancement of both the maxillary and mandibular portions of the face in patients with normal or class I malocclusion. Each patient was stratified into "Success" or "Failure" groups based on criteria defining a "Success" as a 50% preoperative to post-operative decrease in AHI or RDI and a post-operative AHI or RDI <20. RESULTS: A review of the PubMed database yielded 162 articles. Review of these resulted in 9 manuscripts and a total of 109 patients who met the inclusion criteria. There was no statistically significant difference in the amount of anterior advancement of either the mandible (P = 0.96) or the maxilla (P = 0.23) between the "Success" or "Failure" groups. CONCLUSIONS: While there is a paucity of individual data available, the current data does not support an ideal amount of maxillary or mandibular advancement that is required to obtain a surgical success in the treatment of OSA. Until a multicenter, prospective, randomized trial is performed, surgical planning should be tailored to patient-specific anatomy to achieve the desired result.

Factors Influencing Timely Preparation of Alveolar Bone Grafting: A Survey of the ACPA.

OBJECTIVE: To identify factors that are commonly felt to impact a team's ability to ensure patients are orthodontically and dentally prepared for alveolar bone grafting at the appropriate time. DESIGN: Cross-sectional survey. SETTING: American Cleft Palate-Craniofacial Association (ACPA)-approved multidisciplinary cleft teams. PARTICIPANTS: Cleft team coordinators. INTERVENTIONS: Cleft team coordinators were asked to complete the survey. RESULTS: Fifty-three team coordinators from the 167 ACPA-certified cleft teams completed the survey (response rate = 32%). The majority (83.02%) of cleft teams feel having a case manager increases or would increase the rate of timely bone graft preparation. Orthodontic and dental coverage varied greatly between teams with university/hospital employed dentists and volunteer private practice orthodontists representing the most frequent models. The reported percentage of patients seeking regular dental and orthodontic care with providers affiliated with the team did not correlate with the likelihood of timely bone graft preparation (P = .17; P = .43). Great variability was noted in the manner and frequency of team communication with families during orthodontic bone graft preparation. The presence of a designated case manager on the team was not significantly associated with the percentage of patients who were reported to be orthodontically prepared for bone graft surgery (P = .25). CONCLUSION: Within our survey data, there is great variability noted in factors that may potentially impact orthodontic bone graft preparation. The majority of teams felt that a case manager did or would help with this process, however, our study does not support this fact. Significant recall bias concerning the success of bone graft preparation may affect these results.

Laser ablation of embryonic veins in children.

BACKGROUND: Klippel-Trenaunay syndrome (KTS) and related overgrowth syndromes are rare conditions that often present to academic pediatric units and multidisciplinary vascular anomaly clinics. These conditions are commonly associated with abnormalities of the superficial and deep venous structures and carry an increased risk of venous thromboembolism. METHODS: A retrospective chart review was completed of all patients treated at Johns Hopkins All Children's Hospital with endovenous laser ablation therapy (EVLT) for management of dilated embryonic veins in the setting of limb overgrowth. RESULTS: Three patients with limb overgrowth underwent EVLT between 2015 and 2017. All patients had successful occlusion of the targeted veins on post-procedure imaging. One patient developed a cellulitis successfully treated with oral antibiotics. CONCLUSIONS: Endovenous laser ablation therapy is a well-tolerated and safe procedure for prophylactic closure of abnormal superficial embryonic veins. This study supports the use of this technique in managing increased thromboembolic risk in this unique patient population and recognizes the need for a long-term study to determine its efficacy.

Alveolar soft-part sarcoma: Case demonstrating principles for uncommon vascular lesions.

BACKGROUND: Alveolar soft-part sarcoma (ASPS) is an uncommon malignancy that may present in a manner similar to benign vascular tumors. METHODS: A 6-year-old boy with autism spectrum disorder was referred to the Johns Hopkins All Children's Hospital vascular anomalies clinic for the evaluation of a tongue mass. RESULTS: Prior to the presentation, at 5 years of age, neck computed tomography (CT) was performed. This showed a well circumscribed, enhancing mass at the anterior aspect of the tongue. The radiologic impression was that this lesion was most likely a hemangioma. Two years later, the patient was evaluated in the vascular anomalies clinic. At that examination a 2-3 cm swelling was noted on the dorsal aspect of the tongue. The mass was fleshy and firm with discrete borders. Handheld Doppler examination indicated a high-flow lesion. The patient underwent an excisional biopsy. The lesion was identified as an alveolar soft-part sarcoma based on pathologic characteristics. CONCLUSION: Familiarity with common vascular tumors and malformations allows providers to diagnose the majority of these lesions on a combination of clinical history and physical examination. Atypical and combined lesions do benefit from imaging to help characterize and aid in the differential diagnosis. Biopsy enables definitive diagnosis but is necessary in the minority of cases. When in doubt, referral to a specialized, multidisciplinary vascular anomalies clinic will ensure that these patients receive management for this challenging collection of conditions.

Adding Depth to Cephalometric Analysis: Comparing Two- and Three-Dimensional Angular Cephalometric Measurements.

INTRODUCTION: Lateral cephalometric radiographs (LCR) have been the standard tool used for cephalometric analysis in craniofacial surgery. Over the past decade, a three-dimensional (3D) revolution in cephalometric analysis and surgical planning has been underway. To date, research has not validated whether cephalometric measurements taken from two-dimensional (2D) and 3D data sources are equivalent and interchangeable. The authors sought to compare angular cephalometric measurements taken with 2D and 3D modalities. METHODS: Sixty-two head CT scans (36 females, 26 males) with an average age of 63 ±â€Š20 years were studied. Twelve cephalometric angular measurements were taken from 3D reconstructed skulls using the software package Mimics 19.0 (Materialize; Leuven, Belgium). These same facial angles were measured from 2D lateral cephalograms reconstructed from the original CT scans using Dolphin 11.9. Measurements taken with both techniques were compared for agreement using a paired t test. Intra-class correlation coefficient assessment was used to determine inter-rater reliability. Statistical significance was set at P < 0.05. RESULTS: Five of the 12 angular measurements (SNA, SNB, MP-FH, U1-SN, and U1-L1) demonstrated statistically significant differences (P < 0.05) between the 2D and 3D analyses. All of these differences were less than the standard deviations for the respective measure. CONCLUSION: The differences between angular cephalometric values obtained from 2D LCRs and 3D CT reconstructions are small. This supports the practices of using 2D and 3D cephalometric data interchangeably in most applications. Clinicians must be selective in which measures they employ to maximize accuracy and care must be taken when measuring dental inclination with lateral cephalograms.

High-Fidelity Anthropometric Facial Measurements Can Be Obtained From a Single Stereophotograph From the Vectra H1 3-Dimensional Camera.

BACKGROUND: Anthropometry is a well-established means of measuring facial morphology. Although reliable, direct anthropometry can be time-consuming and not conducive to a busy clinical practice. The Vectra H1 handheld stereophotogrammetric system requires 3 stereophotographs taken from different perspectives to generate a three-dimensional (3D) surface. The time needed to take the 3 stereophotographs can increase the possibility of involuntary movements, precluding its use to assess young patients. To overcome this limitation, we evaluated if accurate linear facial measurements can be obtained from a single stereophotograph and compare these to the measurements taken by direct anthropometry. METHODS: Twenty pediatric patients, aged 0 to 10 years, who were undergoing minor surgical procedures at Johns Hopkins All Children's Hospital were recruited. Fourteen linear facial distances were obtained from each participant using direct anthropometry under general anesthesia. These same distances were measured using Mirror 3D analysis. Intraclass correlation was used to determine intrarater reliability on duplicate 3D images. RESULTS: Correlation coefficients between 3D imaging in frontal view and direct anthropometric measurements were excellent for 13 measures taken, ranging from 0.8 (subnasale to columella and subnasale to stomion superius) to 0.98 (nasion to subnasale and subnasale to labiale superius). Correlation coefficients between submental view and direct anthropometric measurements were excellent for 13 measures as well, ranging from 0.77 (subnasale to columella) to 0.98 (nasion to subnasale). CONCLUSIONS: Linear anthropometric measurements taken from 3D surfaces generated from a single stereophotograph correlate closely with direct anthropometric measures. This improves workflow and applicability of anthropometric studies to our youngest patients.

Comprehension of Online Educational Material Regarding Orofacial Clefts.

BACKGROUND: Cleft lip and palate are among the common birth defects in the United States. Management is complex and may be difficult for parents and patients to understand. The goal of this study was to evaluate the readability of online educational material provided by the American Society of Plastic Surgeons (ASPS) and the American Cleft Palate-Craniofacial Association (ACPA) websites regarding cleft lip and palate and to assess parents' comprehension. METHODS: Parents of children younger than 10 years of age presenting to the plastic and reconstructive surgery clinic at Johns Hopkins All Children's Hospital for conditions other than cleft lip and palate were recruited for participation. Parents were given the literature published on the ASPS and ACPA web sites to review and were asked to complete a 5-question test specifically targeted to the content of the passage they reviewed. We tested the readability of the 2 online passages using Readability Studio software (Oleander Software Solutions Ltd). RESULTS: Sixty parents completed the study. Across all groups, the mean score was 4.35 of a possible 5. The mean score was 4.4 for the ASPS web site and 4.29 for the ACPA web site. Fifty-three percent of participants answered all questions correctly. No significant difference in scores was noted between web sites (P = .48). CONCLUSIONS: Despite containing content of varying reading-level difficulty, the literature posted on the ACPA and ASPS web sites concerning cleft lip and palate conveyed information equally well to the target parent demographic with high levels of comprehension.

The Impact of Presurgical Infant Orthopedics on Oronasal Fistula Rates Following Cleft Repair: A Meta-Analysis.

BACKGROUND: Presurgical infant orthopedics (PSIO) techniques were introduced to improve the outcomes achieved when treating children with complete cleft lip and palate. The effect of PSIO on the incidence of postoperative fistulae has never been reliably demonstrated. We conducted a meta-analysis to assess the effectiveness of PSIO in reducing postoperative fistulas in patients with complete cleft lip and palate. METHODS: A search of the PubMed and Embase databases was performed to identify relevant articles that included primary palate repairs of patients with unilateral or bilateral complete clefts, reported the incidence of postoperative fistulae, and explicitly stated if PSIO was used. Details including author, number of subjects, use of PSIO, and fistula rate were cataloged. RESULTS: A review of the PubMed database yielded 1135 unique citations, and Embase yielded 507 articles. Review of these yielded 15 studies, comprising 1241 children, which met inclusion criteria. The overall rate of oronasal fistula development was 7.09%. The average fistula rate for studies using PSIO was 5.93% versus 9.71% in the non-PSIO group. This difference was not statistically significant ( P = .34). CONCLUSIONS: The use of PSIO prior to cleft lip and palate repair provides multiple benefits related to facial and nasal form and is supported by a body of literature. The effect of PSIO on the incidence of postoperative fistulae has received less attention in the literature. Our meta-analysis of the available literature does not provide evidence to support the premise that the use of PSIO affects the incidence of fistulae after cleft palate repair.

Clinical Outcomes Utilizing a "Modified Latham" Appliance for Presurgical Infant Orthopedics in Patients With Unilateral Complete Cleft Lip and Palate.

Presurgical infant orthopedic manipulation is utilized prior to cleft lip/nasal repair to facilitate a gingivoperiosteoplasty (GPP) and primary nasolabial repairs. The Latham dentomaxillary advancement appliance uses a screw that must be tightened daily to approximate the cleft segments in unilateral complete clefts. Our cleft center has been utilizing a "modified Latham" appliance since 1987, including an orthodontic elastic power chain to close the gap in a shorter amount of time. We performed a retrospective chart review of all patients undergoing treatment at Johns Hopkins All Children's Hospital (JHACH) with a unilateral complete cleft lip and palate between 1987 and 2017. Patients were identified by the International Classification of Diseases, Ninth Revision code (749.21). The majority of the patients represent the experience of the senior authors (E.R. and R.F.). Two hundred and eighty-one patients with unilateral complete cleft lip/palate were identified. Seventy-five patients were treated with a "modified Latham" appliance prior to their lip repair. The "modified Latham" appliance remained in place on average 20.6 days (range: 4-82), and average hospital stay after placement was 1.18 days. Nearly 96% of patients underwent a successful GPP at the time of nasolabial repair. Modification of the Latham appliance by utilizing an elastic power chain and eliminating the screw allows rapid closure of the alveolar cleft with limited need for adjustments and outpatient visits. Direct approximation of the palatal segments allows successful completion of a GPP in 95.9% of patients with limited dissection.