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Nasal Duplication: A Review of Literature and Case Report
Shen, Chen; Shetye, Pradip R; Flores, Roberto L
INTRODUCTION/UNASSIGNED:Nasal duplication is a rare congenital deformity with many subtypes including supernumerary nostril. The challenge of surgical correction is to achieve nasal symmetry and restore nasal airflow. However, there is no defined protocol for treatment, especially with regard to presurgical therapy. METHODS/UNASSIGNED:We performed a review of literature of studies reporting on patients with supernumerary nostril to complete this review. We then report on a patient with supernumerary nostril who was treated with nostril expansion therapy prior to surgical intervention. RESULTS/UNASSIGNED:We identified 59 cases of nostril duplication. Because of the rarity of the condition, treatment protocols varied greatly. For our patient, preoperative nasal appliance therapy was implemented for 3 months prior to surgical intervention. Patient was followed-up regularly for 1 year. DISCUSSION/UNASSIGNED:Although literature on nasal duplication is scarce, there is a general agreement that early intervention has psychological, anatomic, and functional benefits to the patient. In our case report, nostril expansion therapy was easy to implement and facilitated surgical reconstruction, resulting in aesthetic outcome and expanded airway 1 year postoperatively.
PMID: 33054357
ISSN: 1545-1569
CID: 4642772
The Effects of Nasoalveolar Molding on Nasal Proportions at the Time of Nasal Maturity
Maliha, Samantha G; Kantar, Rami S; Gonchar, Marina N; Eisemann, Bradley S; Staffenberg, David A; Shetye, Pradip R; Grayson, Barry H; Flores, Roberto L
BACKGROUND/UNASSIGNED:The aim of this study is to assess the effect of nasoalveolar molding (NAM) versus no-NAM on nasal morphology in patients with unilateral cleft lip and palate (UCLP) at the time of nasal maturity. METHODS/UNASSIGNED:A retrospective, single-institution review was conducted on all non-syndromic patients with UCLP. Inclusion criteria included age 14 years or above, unilateral cleft repair at the time of infancy, and adequate photography taken at nasal maturity and prior to rhinoplasty. Exclusion criteria included age less than 14 years, syndromic diagnosis, and rhinoplasty prior to nasal maturity. Ten parameters were measured twice from standardized clinical photographs using the Dolphin Imaging Software for establishment of intrarater reliability. Subjective analysis was achieved through completion of the Asher McDade grading scale by 3 expert cleft practitioners. RESULTS/UNASSIGNED:Nostril height, columellar angle, alar cant, vertical alar height, alar height angle, nasofacial angle, and nasolabial angle were found to be significantly less severe in patients who had undergone NAM in conjunction with surgical repair when compared with those who had undergone surgical repair alone. Asher McDade grading revealed significant improvement in nasal form, nasal symmetry/deviation, nasal profile, vermillion border, and overall score in patients who underwent NAM compared to no-NAM. CONCLUSION/UNASSIGNED:The use of presurgical NAM during infancy can improve nasal symmetry and nasal proportions at the time of nasal maturity.
PMID: 32851868
ISSN: 1545-1569
CID: 4575782
Simulation-Based Comprehensive Cleft Care Workshops: A Reproducible Model for Sustainable Education
Kantar, Rami S; Breugem, Corstiaan C; Keith, Kristen; Kassam, Serena; Vijayakumar, Charanya; Bow, Mikaela; Alfonso, Allyson R; Chahine, Elsa M; Ghotmi, Lilian H; Patel, Krishna G; Shetye, Pradip R; Santiago, Pedro E; Losee, Joseph E; Steinbacher, Derek M; Rossell-Perry, Percy; Garib, Daniela G; Alonso, Nivaldo; Mann, Robert J; Prada-Madrid, Jose Rolando; Esenlik, Elçin; Pamplona, María Del Carmen; Collares, Marcus VinÃcius Martins; Bennun, Ricardo D; Kummer, Ann; Giugliano, Carlos; Padwa, Bonnie L; Raposo-Amaral, Cassio Eduardo; Tse, Raymond; Sommerlad, Brian; Flores, Roberto L; Hamdan, Usama S
OBJECTIVE/UNASSIGNED:Evaluate simulation-based comprehensive cleft care workshops as a reproducible model for education with sustained impact. DESIGN/UNASSIGNED:Cross-sectional survey-based evaluation. SETTING/UNASSIGNED:Simulation-based comprehensive cleft care workshop. PARTICIPANTS/UNASSIGNED:Total of 180 participants. INTERVENTIONS/UNASSIGNED:Three-day simulation-based comprehensive cleft care workshop. MAIN OUTCOME MEASURES/UNASSIGNED:Number of workshop participants stratified by specialty, satisfaction with the workshop, satisfaction with simulation-based workshops as educational tools, impact on cleft surgery procedural confidence, short-term impact on clinical practice, medium-term impact on clinical practice. RESULTS/UNASSIGNED:< .001) surgery procedural confidence following the simulation sessions. Participants also reported a positive short-term and medium-term impact on their clinical practices. CONCLUSION/UNASSIGNED:Simulation-based comprehensive cleft care workshops are well received by participants, lead to improved cleft surgery procedural confidence, and have a sustained positive impact on participants' clinical practices. Future efforts should focus on evaluating and quantifying this perceived positive impact, as well reproducing these efforts in other areas of need.
PMID: 32729337
ISSN: 1545-1569
CID: 4540432
What Is the Burden of Care of Nasoalveolar Molding?
Alfonso, Allyson R; Ramly, Elie P; Kantar, Rami S; Wang, Maxime M; Eisemann, Bradley S; Staffenberg, David A; Shetye, Pradip R; Flores, Roberto L
OBJECTIVE/UNASSIGNED:This systematic review aims to evaluate nasoalveolar molding (NAM) in the context of burden of care defined as physical, psychosocial, or financial burden on caregivers. SEARCH METHODS/UNASSIGNED:Following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, 5 databases were searched from inception through December 24, 2019, for keywords and subject headings pertaining to cleft lip and/or palate and NAM. ELIGIBILITY CRITERIA/UNASSIGNED:Clinical studies on NAM with reference to physical (access to care, number of visits, distance traveled), psychosocial (caregiver perceptions, family interactions, breast milk feeding), and financial (direct and indirect costs) burden were included. DATA COLLECTION AND ANALYSIS/UNASSIGNED:Study selection was performed by 2 independent reviewers. RESULTS/UNASSIGNED:The search identified 1107 articles and 114 articles remained for qualitative synthesis. Burden of care domains were discussed but not measured in 43% of articles and only 25% assessed burden of care through a primary outcome. Of these, 20 articles reported on physical, 8 articles on psychosocial, and 12 articles on financial burden. Quality of evidence is limited by study design and risk of bias. CONCLUSION/UNASSIGNED:Nasoalveolar molding has been indiscriminately associated with burden of care in the literature. Although NAM may not be the ideal treatment option for all patients and families, the physical considerations are limited when accounting for the observed psychosocial advantages. Financial burden appears to be offset, but further research is required. Teams should directly assess the impact of this early intervention on the well-being of caregivers and advance strategies that improve access to care.
PMID: 32500737
ISSN: 1545-1569
CID: 4469462
Neonatal Mandibular Distraction Osteogenesis in Infants With Pierre Robin Sequence
Diep, Gustave K; Eisemann, Bradley S; Flores, Roberto L
Pierre Robin sequence is the constellation of micrognathia, glossoptosis, and tongue-based airway obstruction. When airway obstruction is severe, feeding, growth, and respiratory demise are at risk. Neonatal mandibular distraction osteogenesis is a technique which improves tongue-based airway obstruction and avoids tracheostomy in patients with severe expressions of Pierre Robin sequence. Its efficacy in relieving airway obstruction is well documented, and it has become the surgical intervention of choice at many craniofacial centers. However, this is an uncommon procedure which can be performed within the first weeks of life, offering little space for a learning curve. The success of neonatal distraction and avoidance of complications is highly dependent on proper surgical technique. This report provides a brief overview of the disease, details the technique of the senior surgeon with captioned videos, describes the protocol used at our institution and reports long-term outcomes with a case description.
PMID: 32209938
ISSN: 1536-3732
CID: 4358502
Temporomandibular Joint Ankylosis in Pediatric Patients With Craniofacial Differences: Causes, Recurrence and Clinical Outcomes
Ramly, Elie P; Yu, Jason W; Eisemann, Bradley S; Yue, Olivia; Alfonso, Allyson R; Kantar, Rami S; Staffenberg, David A; Shetye, Pradip R; Flores, Roberto L
BACKGROUND:The authors present an institutional experience treating congenital and acquired temporomandibular joint (TMJ) ankylosis, detailing outcomes and potential risk factors of recurrence. METHODS:Retrospective chart review identified patients with TMJ ankylosis (1976-2019). Clinical records, operative reports, and imaging studies were reviewed for demographics, surgical operations, and ankylosis including maximal interincisal opening (MIO) and re-ankylosis. RESULTS:Forty-four TMJs with bony ankylosis were identified in 28 patients (mean age at any initial mandibular surgery: 3.7; range:0-14 years). Follow-up was 13.7 ± 5.9 years. Sixteen (57.1%) patients had bilateral ankylosis; 27(96.4%) had syndromes. Nine patients had congenital ankylosis, 16 had iatrogenic ankylosis (4.5 ± 3.7 years from initial distraction osteogenesis or autologous mandibular reconstruction) referred from outside institutions in 6 instances, and 3 had post-infectious ankylosis. Patients having their first mandibular operation at a younger age had more frequent reoperations for recurrent TMJ ankylosis, although this did not reach statistical significance. Mean improvement in MIO was 21.4 ± 7.3 mm. Ankylosis recurred in 21 (75%) patients. Five patients with congenital TMJ ankylosis required gastrostomy and remained at least partially dependent. Five patients had tracheostomy at the time of TMJ ankylosis surgery: 2 were eventually decannulated and 3 required repeat tracheostomy after ankylosis recurrence and remained tracheostomy-dependent. CONCLUSION/CONCLUSIONS:The clinical course of TMJ ankylosis in children affected by craniofacial differences is complex and typically involves a high rate of recurrence and multiple reoperations despite initial improvement in postoperative MIO. Younger age at initial mandibular surgery and number of operations require further investigation as potential predictors of recurrent TMJ ankylosis as well as tracheostomy and gastrostomy dependence.
PMID: 32176014
ISSN: 1536-3732
CID: 4352402
Educational Resources in Craniofacial Surgery: The Case for User-Friendly Digital Simulators
Kantar, Rami S; Alfonso, Allyson R; Ramly, Elie P; Diaz-Siso, J Rodrigo; Flores, Roberto L
INTRODUCTION/BACKGROUND:Digital simulators are potential solutions to challenges facing surgical education. The authors sought to evaluate the reach and utilization of a freely-accessible craniofacial surgery digital educational simulator. More importantly, we compare usage patterns between web-based and mobile-based platforms. METHODS:A 3-way collaboration between academic, non-profit (myFace, New York, NY), and biotechnology (Biodigital, New York, NY) stakeholders in 2015 produced the Craniofacial Interactive Virtual Assistant Pro (CIVA-Pro). CIVA-Pro is a freely-accessible craniofacial surgery digital educational simulator. In addition to the web-based platform, a mobile-based platform was launched in 2017. Usage analytics were collected and analyzed. RESULTS:Since its launch, 751 registered users from 117 countries had accessed CIVA-Pro. The total number of sessions was 9531, including 7500 web and 2031 mobile sessions. The total screen time was 403.9 hours, 290.3 for the web and 113.6 for the mobile platform. Comparison of the mean monthly screen time and number of monthly sessions between platforms since 2017 demonstrated a significantly higher mean monthly screen time (60.1 ± 33.2 versus 29.4 ± 16.5 hours; P = 0.002) and number of sessions (110.2 ± 36.1 versus 58.1 ± 31.9; P < 0.0001) for the mobile-based platform. The mean screen time per session was comparable (P = 0.86). CONCLUSION/CONCLUSIONS:A freely available digital craniofacial surgery educational simulator designed for surgical trainees can achieve significant global reach. Significantly higher utilization of the mobile-based platform of the simulator as compared to the web-based platform reinforces the need to invest in user-friendly, easily accessible, and widely available digital educational resources by key stakeholders to ensure optimal plastic surgery trainee education.
PMID: 31985596
ISSN: 1536-3732
CID: 4293862
Sleep-Disordered Breathing and Airway Assessment Using Polysomnography in Pediatric Patients With Craniofacial Disorders
Bekisz, Jonathan M; Wang, Maxime M; Rickert, Scott M; Rodriguez, Alcibiades J; Flores, Roberto L
Children with cleft and craniofacial conditions commonly present with concurrent airway anomalies, which often manifest as sleep disordered breathing. Craniofacial surgeons and members of the multidisciplinary team involved in the care of these patients should appreciate and understand the scope of airway pathology as well as the proper means of airway assessment. This review article details the prevalence and assessment of sleep disordered breathing in patients with craniofacial anomalies, with emphasis on indications, limitations, and interpretation of polysomnography.
PMID: 32049904
ISSN: 1536-3732
CID: 4304462
Skeletal, soft tissue and globe position changes following le Fort i + III surgery in patients with mid-facial hypoplasia and proptosis [Meeting Abstract]
Liu, B; Grayson, B; McCarthy, J; Flores, R; Staffenberg, D; Rodriguez, E; Shetye, P
Background/Purpose: Our study quantifies changes in skeletal, soft tissue profile, and globe position in patients with syndromic craniosynostosis after Le Fort I + III (LF I + III) surgery. Methods/Description: Patients with syndromic craniosynostosis who underwent LF I + III at the time of facial maturity were followed for at least 1 year. Each lateral cephalometric radiograph was traced using Dolphin Imaging software and superimposed at the sella. Changes in positions of the different landmarks at T0 (preoperatively), T1 (immediate postoperatively), and T2 (1 year postoperatively) were measured by the software. Sixty-seven soft tissue and skeletal landmarks were digitized and measured. LF III skeletal changes were measured by changes in lateral orbit and orbitale. LF I skeletal changes were measured at the A point and U1. Corresponding soft tissue profile and globe position were studied. All data were measured along the x-axis.
Result(s): Twelve patients included in our study have the following syndromes: Crouzon (n = 6), Pfeiffer (n = 2), Apert (n = 1), Antley-Bixler (n = 1), cleidocranial dysplasia (n = 1) and frontonaso dysplasia (n = 1). Nine patients had previous history of LF III distraction. Standard descriptive statistics was used. Data were analyzed using paired T test. Lateral orbit advanced 5.49 mm (T0-T1) on average, with a P value of 1.3-5, and 5.94 mm (T0-T2) on average; 0.45 mm (T1-T2) change with a P value of .96 suggests the lateral orbit is stable. Similar advancement at orbitale is observed at 5.68 mm (T0-T1) and 6.42 mm (T0-T2). The globe moved anteriorly by 1.98 mm (T0-T1) with a P value of .025 and anteriorly by 0.944mm(T0-T2). The change between T2 and T1 is 1.04 mm (P value: .26), which suggests the globe moved backward after postsurgical swelling subsided. The ratios of movement (globe to lateral orbit) between T0-T1 and T0-T2 are 31% and 16%, respectively. The decrease in ratio can be attributed to the reduction in soft tissue swelling at T2. Restoring position of the globe relative to the lateral orbit decreases the risk of exposure keratitis, keratoconjunctivitis sicca, and corneal ulceration. Anterior nasal spine and point A were advanced by 9.38 and 10.08 mm, respectively, between T0 and T1, and 9.01 mm and 8.51 mm, respectively, between T1 and T2. At the occlusal level, U1 advanced 10 mm and L1 moved back 1.45 mm between T0 and T1. Menton moved back 1.25 mm (T0-T1) but advanced by 2.48 mm (T0-T2). This change in direction is due to splint use at T1 as it rotates mandible clockwise.
Conclusion(s): In our cohort, LFI + III surgery improved both midface deficiency and proptosis in those with syndromic craniosynostosis.Combined Le Fort I + III surgery allows surgeons to perform differential corrections of the midface at the orbital and the dentition level. This is ideal for proptosis correction and establishing optimal jaw relationship
EMBASE:631558314
ISSN: 1545-1569
CID: 4417632
Clinical course of temporomandibular joint ankylosis in pediatric patients with craniofacial anomalies [Meeting Abstract]
Ramly, E; Yu, J; Eisemann, B; Yue, O; Alfonso, A; Kantar, R; Staffenberg, D; Shetye, P; Flores, R
Background/Purpose: Temporomandibular joint (TMJ) ankylosis is an uncommon but debilitating condition which can affect feeding, speech, dental health, facial growth, and quality of life. We present an institutional experience treating congenital and acquired TMJ ankylosis, detailing outcomes and potential risk factors of recurrence. Methods/Description: Patients with ankylosis of the TMJ were identified through retrospective chart review (1976-2019). Clinical records, operative reports, and imaging studies were reviewed for demographics, surgical operations, and ankylosis including mean interincisal opening (MIO) and reankylosis.
Result(s): Forty-four TMJs with bony ankylosis were identified in 28 patients, 27(96.4%) of whom had syndromes. Mean age at any initial mandibular surgery was 3.7+/-3.6 (range: 0-14 years). Follow-up was 13.7 +/- 5.9 years. Sixteen (57.1%) patients had bilateral ankylosis. Nine cases of ankylosis were congenital, 16 were iatrogenic (4.5 +/- 3.7 years from initial distraction osteogenesis or autologous mandibular reconstruction) referred from outside institutions in 6 cases, and 3 were postinfectious. Patients having their first mandibular operation at a younger age had more frequent reoperations for recurrent TMJ ankylosis, although this did not reach statistical significance. Improvement in MIO was 21.4 +/- 7.3 mm. Ankylosis recurred in 21(75%) cases, 11 of which were iatrogenic, requiring an average of 2 reoperations (range: 1-8). Five patients with congenital TMJ ankylosis required gastrostomy and remained at least partially dependent. Five patients had tracheostomy at the time of TMJ ankylosis surgery: 2 were eventually decannulated and 3 required repeat tracheostomy after ankylosis recurrence and remained tracheostomy-dependent.
Conclusion(s): Craniofacial anomalies, younger age at mandibular surgery, and number of operations portend to increased risk of TMJ ankylosis as well as tracheostomy and gastrostomy dependence. Despite initial improvement in postoperative MIO, pediatric TMJ ankylosis is associated with high recurrence and multiple reoperations
EMBASE:631558218
ISSN: 1545-1569
CID: 4417672