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1.
J Neurosurg Pediatr ; : 1-8, 2024 May 10.
Article En | MEDLINE | ID: mdl-38728754

OBJECTIVE: As many as 5% of normocephalic children may have a prematurely fused sagittal suture, yet the clinical significance and best course of management of this finding remain unclear. Providers in the Synostosis Research Group were surveyed to create a multicenter consensus on an optimal treatment and monitoring algorithm for this condition. METHODS: A four-round modified Delphi method was utilized. The first two rounds consisted of anonymous surveys distributed to 10 neurosurgeons and 9 plastic surgeons with expertise in craniosynostosis across 9 institutions, and presented 3 patients (aged 3 years, 2 years, and 2 months) with incidentally discovered fused sagittal sutures, normal cephalic indices, and no parietal dysmorphology. Surgeons were queried about their preferred term for this entity and how best to manage these patients. Results were synthesized to create a treatment algorithm. The third and fourth feedback rounds consisted of open discussion of the algorithm until no further concerns arose. RESULTS: Most surgeons preferred the term "premature fusion of the sagittal suture" (93%). At the conclusion of the final round, all surgeons agreed to not operate on the 3- and 2-year-old patients unless symptoms of intracranial hypertension or papilledema were present. In contrast, 50% preferred to operate on the 2-month-old. However, all agreed to utilize shared decision-making, taking into account any concerns about future head shape and neurodevelopment. Panelists agreed that patients over 18 months of age without signs or symptoms suggesting elevated intracranial pressure (ICP) should not undergo surgical treatment. CONCLUSIONS: Through the Delphi method, a consensus regarding management of premature fusion of the sagittal suture was obtained from a panel of North American craniofacial surgeons. Without signs or symptoms of ICP elevation, surgery is not recommended in patients over 18 months of age. However, for children younger than 18 months, surgery should be discussed with caregivers using a shared decision-making process.

2.
J Pediatr ; 265: 113799, 2024 Feb.
Article En | MEDLINE | ID: mdl-37879601

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.


Airway Obstruction , Cleft Palate , Micrognathism , Infant , Child , Humans , Infant, Newborn , Micrognathism/epidemiology , Micrognathism/surgery , Cleft Palate/epidemiology , Cleft Palate/surgery , Airway Obstruction/surgery , Intensive Care Units , North America , Retrospective Studies
3.
Cleft Palate Craniofac J ; : 10556656231207469, 2023 Oct 16.
Article En | MEDLINE | ID: mdl-37844605

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.

4.
Plast Reconstr Surg ; 2023 Sep 29.
Article En | MEDLINE | ID: mdl-37772883

BACKGROUND: For patients with cleft lip/palate, adolescence is a time of maxillofacial growth and complex psychosocial stressors. The personal significance of facial differences may change, making patient-reported outcomes measures (PROMs) invaluable. In this study, we use several scales from CLEFT-Q™ and FACE-Q™ to explore how aesthetic outcomes differ by age and by sex among patients with unilateral cleft lip/palate. MATERIALS AND METHODS: This was a multi-center, cross-sectional study that prospectively collected CLEFT-Q™ and FACE-Q™ data across six cleft treatment centers during clinical appointments from 2019-2022. Subjects were aged 8-22y with unilateral cleft lip, alveolus, and palate who had not undergone tertiary operative care (maxillary advancement or septorhinoplasty) at the time of survey response. Data cross-sections were prepared by age (8-10y, 11-13y, 14y+), by sex, and by age and sex together. RESULTS: Older age groups reported poorer aesthetic outcomes and worse appearance-related distress compared to younger groups. Although male and female subjects reported similar aesthetic outcomes, female subjects reported more appearance-related distress. When considered simultaneously, age and sex appear to have an intersectional impact on perceived aesthetic outcome and appearance-related distress during adolescence. CONCLUSIONS: This exploratory project suggests that patients with cleft lip/palate may perceive worsening of facial aesthetic throughout the course of adolescence, the exact pattern of which may be dependent on sex. Future work will evaluate this hypothesis using longitudinal cohorts. It will be important to investigate psychosocial factors that may impact these outcomes, and also to quantify the impact of tertiary operative care on these outcomes.

5.
Plast Reconstr Surg ; 2023 May 15.
Article En | MEDLINE | ID: mdl-37184511

BACKGROUND: Studies of infants with micrognathia, especially Robin Sequence (RS), are limited by its rarity and both phenotypic and diagnostic variability. Most knowledge of this condition is sourced from small, single-institution samples. METHODS: This is a cross-sectional study including infants with micrognathia admitted to 38 Children's Hospital Neonatal Consortium centers from 2010-2020. Predictor variables included demographic data, birth characteristics, cleft and syndrome status. Outcome variables included length of stay (LOS), death, feeding or respiratory support, and secondary airway operations. RESULTS: 1289 infants with micrognathia had a surgery to correct upper airway obstruction. Mean age and weight at operation were 34.8±1.8 weeks and 3515.4±42 grams, respectively. A syndromic diagnosis was made in 150 (11.6%) patients, with Stickler (5.4%) and Treacher Collins Syndromes (2.2%) most common. Operations included: mandibular distraction osteogenesis (MDO), 66.3%; tracheostomy, 25.4%; and tongue-lip adhesion (TLA), 8.3%. Tracheostomy patients had a lower birth weight, head circumference, gestational age, and APGAR scores. MDO patients were less likely to need a second airway operation compared to TLA patients (3.5%vs17.8%,p<0.001). The proportion of infants feeding exclusively orally at hospital discharge differed significantly, from most to least: MDO, TLA, tracheostomy. Hospital LOS was not statistically different for patients that had MDO and TLA, but was longer for those with primary tracheostomy. Mortality was low for all operations (0.5%). CONCLUSION: In this 1289 surgical patient cohort, MDO was associated with shorter hospital stay, improved oral feeding, and lower rates of secondary airway operations. Prospective multi-center studies are necessary to support these conclusions.

6.
Eplasty ; 23: e4, 2023.
Article En | MEDLINE | ID: mdl-36817363

Background: Dorsal augmentation is one of the more complication-prone procedures in rhinoplasty, demanding ample skill and experience to produce an aesthetic and functional result. Numerous autologous grafts, allografts, xenografts, and alloplastic materials have been attempted for augmentation with varying success. Each graft material has its own unique properties that require varying surgical intricacies. The relative risks and benefits of these strategies and materials are also variable and must be weighed. Alloplastic implants are commonly used in eastern countries, but, due to different patient populations and complication profiles, have found less acceptance among western surgeons. Methods: A PubMed search was done using the terms "dorsal augmentation rhinoplasty," "liquid rhinoplasty," and "grafts used for rhinoplasty." Publication dates ranging from 1957-2022 were included. Abstracts were screened for relevance, and references from each article were reviewed to identify further articles. Chapters on dorsal augmentation from two key rhinoplasty textbooks were also reviewed. Results: A total of 79 articles and 2 textbooks were formally screened; of these, 65 resources were included. Findings were grouped under the headings "facial analysis," "ethnic factors in graft selection," "graft characteristics," "septal cartilage," "auricular cartilage," "costal cartilage," "diced cartilage grafts," "bone grafts," "soft tissue grafts," "nonsurgical/liquid rhinoplasty," and "alloplastic implants." Conclusions: Current dorsal augmentation literature focuses on retrospective studies and expert experiences. There are limited quantitative and prospective studies present to delineate an ideal graft. With consistently advancing technology and knowledge of facial aesthetics, a surgeon must maintain a thorough understanding of the current graft and implant options to offer a specialized treatment plan for each deformity and patient.

7.
Eplasty ; 23: e7, 2023.
Article En | MEDLINE | ID: mdl-36817364

Background: The development of postoperative oronasal fistulae (ONF) is a complication that plagues all cleft surgeons to varying degrees. There is extensive literature discussing the incidence, functional impact, and treatment of ONF. The goal of this article is to provide an extensive review of the literature discussing the incidence, causative factors, functional impact, classification systems, and treatment of ONF. Methods: A literature review was performed using PubMed using the Medical Subject Heading terms "cleft palate" AND "fistula" OR "palatal fistula" OR "oronasal fistula". After review, a total of 356 articles were deemed relevant for this study. Results: Information regarding ONF care, prevention, and management in patients with cleft palate was collected from the articles included in this review. Treatment of ONF remains a challenging problem as there is not a consensus in the available literature on the best palatoplasty techniques for their prevention and treatment. A myriad of reconstructive options and adjunctive therapies exist, and their use is guided by the size and location of the fistula. Conclusions: Fistula treatment should be tailored to the specific needs of the patient, and consideration must be given to not only the ONF itself but also the patient's stage of growth and development. Large-scale, multicenter studies are needed in which ONF are described using standardized nomenclature, and improved outcomes reporting is necessary to better define an algorithm for a truly holistic approach to palate surgery and reduce the incidence of palatal fistula.

8.
Cleft Palate Craniofac J ; : 10556656221151096, 2023 Jan 12.
Article En | MEDLINE | ID: mdl-36635983

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.

9.
J Pediatr Orthop ; 43(4): 268-272, 2023 Apr 01.
Article En | MEDLINE | ID: mdl-36693390

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.


Health Information Systems , Sarcoma , Venous Thromboembolism , Adult , Adolescent , Humans , Child , Venous Thromboembolism/epidemiology , Venous Thromboembolism/etiology , Venous Thromboembolism/prevention & control , Anticoagulants/therapeutic use , Incidence , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Hospitalization , Sarcoma/epidemiology , Sarcoma/surgery , Sarcoma/complications , Risk Factors , Lower Extremity/surgery
10.
Cleft Palate Craniofac J ; 60(7): 865-874, 2023 Jul.
Article En | MEDLINE | ID: mdl-35262434

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.


Cleft Palate , Plastic Surgery Procedures , Humans , Retrospective Studies , Surgical Flaps , Cleft Palate/surgery , Adipose Tissue , Treatment Outcome
11.
Cleft Palate Craniofac J ; 60(11): 1366-1375, 2023 11.
Article En | MEDLINE | ID: mdl-36314735

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.


Alveolar Bone Grafting , Cleft Lip , Cleft Palate , Surgeons , Humans , Bone Transplantation/methods , Cleft Palate/surgery , Cleft Lip/surgery , Palatal Expansion Technique , Alveolar Bone Grafting/methods , North America , Retrospective Studies
12.
Cleft Palate Craniofac J ; 59(6): 693-700, 2022 06.
Article En | MEDLINE | ID: mdl-34132122

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.


Cleft Lip , Nose Diseases , Anthropometry , Child , Cleft Lip/diagnostic imaging , Cleft Lip/surgery , Ethnicity , Humans , Infant , Nasal Septum , Nose/abnormalities , Reproducibility of Results
13.
Cleft Palate Craniofac J ; 58(9): 1142-1149, 2021 09.
Article En | MEDLINE | ID: mdl-33353404

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.


Cleft Lip , Cleft Palate , Fistula , Child , Cleft Lip/surgery , Cleft Palate/surgery , Cross-Sectional Studies , Humans , Oral Fistula , Oral Health , Patient-Centered Care , Postoperative Complications , Quality of Life , Retrospective Studies , Treatment Outcome
14.
Cleft Palate Craniofac J ; 58(7): 805-814, 2021 07.
Article En | MEDLINE | ID: mdl-33030045

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.


Airway Obstruction , Osteogenesis, Distraction , Pierre Robin Syndrome , Airway Obstruction/surgery , Humans , Infant , Laryngoscopy , Mandible/surgery , Pierre Robin Syndrome/surgery , Retrospective Studies , Treatment Outcome
15.
Cleft Palate Craniofac J ; 58(5): 539-545, 2021 05.
Article En | MEDLINE | ID: mdl-32996328

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.


Cleft Lip , Cleft Palate , Cleft Lip/diagnostic imaging , Cleft Lip/surgery , Cleft Palate/diagnostic imaging , Cleft Palate/surgery , Humans , Infant , Lip/diagnostic imaging , Nose , Reproducibility of Results
16.
Cleft Palate Craniofac J ; 57(9): 1061-1068, 2020 09.
Article En | MEDLINE | ID: mdl-32552007

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.


Alveolar Bone Grafting , Cleft Lip , Cleft Palate , Bone Transplantation , Cleft Palate/surgery , Cross-Sectional Studies , Humans , Orthodontists , Surveys and Questionnaires
17.
J Craniofac Surg ; 31(6): 1734-1738, 2020 Sep.
Article En | MEDLINE | ID: mdl-32371693

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.


Jaw Diseases/surgery , Mandibular Osteotomy , Adolescent , Female , Genioplasty , Humans , Length of Stay , Male , Morbidity , Osteotomy, Le Fort , Retrospective Studies
18.
J Craniofac Surg ; 31(3): 716-719, 2020.
Article En | MEDLINE | ID: mdl-32049900

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.


Malocclusion, Angle Class I/surgery , Sleep Apnea, Obstructive/etiology , Humans , Malocclusion, Angle Class I/complications , Mandible/physiopathology , Mandibular Advancement , Maxilla/surgery , Polysomnography , Treatment Outcome
19.
J Craniofac Surg ; 30(5): 1568-1571, 2019 Jul.
Article En | MEDLINE | ID: mdl-31299770

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.


Cephalometry/methods , Adult , Aged , Aged, 80 and over , Face , Female , Humans , Imaging, Three-Dimensional/methods , Male , Middle Aged , Radiography, Dental , Reproducibility of Results , Skull/diagnostic imaging , Tomography, X-Ray Computed
20.
Plast Reconstr Surg ; 144(2): 424-431, 2019 08.
Article En | MEDLINE | ID: mdl-31348354

BACKGROUND: Previous attempts to study the effect of prophylactic antibiotics on the outcomes of cleft palate surgery have been hampered by the need for a very large sample size to provide adequate power to discern a potentially small therapeutic effect. This limitation can be overcome by querying large databases created by health care governing bodies. METHODS: Data from the Pediatric Health Information System database were used for this analysis. Patients, aged 6 to 18 months, who had undergone primary palatoplasty (International Classification of Diseases, Ninth Revision, code 27.62) between 2004 and 2009 were included. Subsequent repair of an oronasal fistula between 2004 and 2015 was identified by International Classification of Diseases, Ninth Revision, procedure code 21.82. Pharmacy billing records were used to determine antibiotic administration. Associations between antibiotic administration and fistula repair were assessed using random-intercept logistic regression adjusting for age, sex, race, and cleft type. RESULTS: Seven thousand one hundred sixty patients were available for analysis; of these, 460 (6.4 percent) had a subsequent repair of an oronasal fistula. Fistula rates were 5.9, 11.4, and 5.2 percent among patients given preoperative antibiotics, only postoperative antibiotics, and no antibiotics, respectively (p < 0.001). Multivariable analysis results showed that the odds of having an oronasal fistula among patients who were administered preoperative antibiotics did not differ significantly (statistically) from patients who did not receive antibiotics (OR, 0.88; 95 percent CI, 0.59 to 1.31). CONCLUSIONS: The treatment goal of primary palatoplasty is the successful repair of the cleft without an oronasal fistula. Administration of preoperative antibiotics did not significantly reduce the odds of subsequent fistula repair within the same Pediatric Health Information System institution following primary palatoplasty. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.


Antibiotic Prophylaxis/statistics & numerical data , Cleft Palate/surgery , Fistula/surgery , Nose Diseases/surgery , Oral Fistula/surgery , Cleft Lip/surgery , Female , Humans , Infant , Male , Postoperative Complications/surgery , Reoperation/statistics & numerical data , Surgical Wound Infection/prevention & control , United States
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