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Prevalence of Dental Anomalies in Patients With Unilateral Cleft Lip and Alveolus Treated With Gingivoperiosteoplasty

Gibson, Travis L; Grayson, Barry H; Cutting, Court B; Shetye, Pradip R
OBJECTIVE/UNASSIGNED:To compare the prevalence of dental malformations and agenesis in patients who received or did not receive gingivoperiosteoplasty (GPP). DESIGN/UNASSIGNED:Retrospective cohort study. PATIENTS/UNASSIGNED:Review of patients born January 1, 2000, to December 31, 2007, with unilateral cleft lip and alveolus, with or without clefting of the secondary palate, who received GPP and/or secondary alveolar bone grafting (ABG). Patients were included if they had clinical images and dental radiographs available at ages 5 to 9 and 10 to 12 years. Ninety-four patients met the inclusion criteria; 46 treated with GPP, and 48 who did not receive GPP. OUTCOME MEASURES/UNASSIGNED:tests. RESULTS/UNASSIGNED:Cleft side lateral incisors were absent in 54% of GPP patients, compared to 50% in the no-GPP group. Two patients in the GPP group and 1 in the no-GPP group had supernumerary lateral incisors. Most lateral incisors were undersized or peg shaped in both the no-GPP (83.3%) and GPP (71.4%) groups. In the GPP group, 5 (10.9%) patients exhibited central incisor agenesis, and 3 had significant hypoplasia. In the no-GPP group, 4 (8.3%) patients exhibited central incisor agenesis, and 5 (10.5%) significant hypoplasia. These differences were not statistically significant. CONCLUSIONS/UNASSIGNED:Gingivoperiosteoplasty was not associated with increased prevalence of dental malformation or agenesis. When performed appropriately, GPP is a safe treatment technique that does not increase the risk of dental anomalies.
PMID: 34259074
ISSN: 1545-1569
CID: 4938562

Three-Dimensional Nasolabial Changes After Nasoalveolar Molding and Primary Lip/Nose Surgery in Infants With Bilateral Cleft Lip and Palate

Mancini, Laura; Avinoam, Shayna; Grayson, Barry H; Flores, Roberto L; Staffenberg, David A; Shetye, Pradip R
OBJECTIVE/UNASSIGNED:Utilize 3-dimensional (3D) photography to evaluate the nasolabial changes in infants with bilateral cleft lip and palate (BCLP) who underwent nasoalveolar molding (NAM) and primary reconstructive surgery. DESIGN/UNASSIGNED:coordinates to obtain the linear and angular measurements. Nasal form changes were measured and analyzed between T1 (0.5 months old), T2 (5 months old), and T3 (6 months old). Intraclass correlation coefficient was performed for intrarater reliability. Averaged data from the 3D images was statistically analyzed from T1 to T2 and T2 to T3 with Wilcoxon tests. Unaffected infant norms from the Farkas publication were used as a control sample. RESULTS/UNASSIGNED:After NAM therapy, statistically significant changes in the position of subnasale and labius superius improved nasolabial symmetry. Both retruded after NAM were displaced downward after NAM and surgical correction with respect to soft tissue nasion. The nasal tip's projection was maintained with NAM and surgical correction. The columella lengthened from 1.4 to 4.71 mm following NAM. CONCLUSIONS/UNASSIGNED:There was a significant improvement in the nasolabial anatomy after NAM, and this was further enhanced after primary reconstructive surgery.
PMID: 34032145
ISSN: 1545-1569
CID: 4887702

The Nasoalveolar Molding Cleft Protocol: Long-Term Treatment Outcomes from Birth to Facial Maturity

Yarholar, Lauren M; Shen, Chen; Wangsrimongkol, Buddhathida; Cutting, Court B; Grayson, Barry H; Staffenberg, David A; Shetye, Pradip R; Flores, Roberto L
BACKGROUND:The authors present outcomes analysis of the nasoalveolar molding treatment protocol in patients with a cleft followed from birth to facial maturity. METHODS:A single-institution retrospective review was conducted of cleft patients who underwent nasoalveolar molding between 1990 and 2000. Collected data included surgical and orthodontic outcomes and incidence of gingivoperiosteoplasty, alveolar bone grafting, surgery for velopharyngeal insufficiency, palatal fistula repair, orthognathic surgery, nose and/or lip revision, and facial growth. RESULTS:One hundred seven patients met inclusion criteria (69 with unilateral and 38 with bilateral cleft lip and palate). Eighty-five percent (91 of 107) underwent gingivoperiosteoplasty (unilateral: 78 percent, 54 of 69; bilateral: 97 percent, 37 of 38). Of those patients, 57 percent (52 of 91) did not require alveolar bone grafting (unilateral: 59 percent, 32 of 54; bilateral: 54 percent, 20 of 37). Twelve percent (13 of 107) of all study patients underwent revision surgery to the lip and/or nose before facial maturity (unilateral: 9 percent, six of 69; bilateral: 18 percent, seven of 38). Nineteen percent (20 of 107) did not require a revision surgery, alveolar bone grafting, or orthognathic surgery (unilateral: 20 percent, 14 of 69; bilateral: 16 percent, six of 38). Cephalometric analysis was performed on all patients with unilateral cleft lip and palate. No significant statistical difference was found in maxillary position or facial proportion. Average age at last follow-up was 20 years (range, 15 years 4 months to 26 years 10 months). CONCLUSIONS:Nasoalveolar molding demonstrates a low rate of soft-tissue revision and alveolar bone grafting, and a low number of total operations per patient from birth to facial maturity. Facial growth analysis at facial maturity in patients who underwent gingivoperiosteoplasty and nasoalveolar molding suggests that this proposal may not hinder midface growth. CLINICAL QUESTION/LEVEL OF EVIDENCE/METHODS:Therapeutic, IV.
PMID: 33890899
ISSN: 1529-4242
CID: 4847552

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

Sociodemographic Predictors of Treatment Success and Difficulty in Nasoalveolar Molding

Gibson, Travis L; Grayson, Barry H; Shetye, Pradip R
OBJECTIVE/UNASSIGNED:To assess social and demographic influences on caregiver success and difficulty with nasoalveolar molding (NAM). DESIGN/UNASSIGNED:Retrospective review identified patients who began NAM between April 22, 2013, and April 18, 2017, at the New York University Langone Medical Center. Records were reviewed, and the following sociodemographic data retrieved: parental marital status, parental ages, number of siblings, distance traveled to clinic, insurance coverage, concurrent medical conditions, and need for an interpreter. PATIENTS/UNASSIGNED:Patients were included if complete charting was available; 106 patients met the inclusion criteria; 79 patients with unilateral and 27 with bilateral clefts. OUTCOME MEASURES/UNASSIGNED:Chart entries indicating incorrect appliance usage, emergency visits, phone calls, and noncompliance were recorded. Alveolar cleft gap closure was measured on pre- and posttreatment models in unilateral cases. Multiple regression analyses were performed to assess the influence of social variables on these outcomes. RESULTS/UNASSIGNED:= .019). CONCLUSIONS/UNASSIGNED:Alveolar cleft gap closure was more successful for older fathers, younger mothers, and married couples. Married couples were also less likely to experience treatment difficulties such as incorrect appliance usage or inadequate duration of wear, as were those with private insurance coverage.
PMID: 32840124
ISSN: 1545-1569
CID: 4576232

The nasoalveolar molding cleft protocol: Long-term results from birth to facial maturity [Meeting Abstract]

Yarholar, L; Shen, C; Grayson, B; Cutting, C; Staffenberg, D; Shetye, P; Flores, R
Background/Purpose: We present the first long-term outcomes analysis of the nasoalveolar molding (NAM) treatment protocol on patients with a cleft followed from birth to facial maturity. Methods/Description: Single-institution retrospective review of all patients with a cleft who underwent NAM between the years 1990 and 2000. All study patients completed cleft care treatment at the same institution and were followed by the same team members. Our institution's treatment protocol offers NAM to patients with a significant cleft nasal deformity and/or widely displaced alveolar segments. All patients underwent primary cleft lip and nasal repair prior to the age of 6 months. Gingivoperiosteoplasty (GPP) is performed, when possible, at the time of lip repair. Cleft palate repair is performed by 1 year of age. Collected data include surgical and orthodontic outcomes of cleft care such as cleft lip and palate repair, GPP, alveolar bone grafting (ABG), speech surgery for velopharyngeal insufficiency (VPI), palatal fistula repairs, orthognathic surgery, and revision surgery to the nose and/or lip.
Result(s): A total of 135 patients met the inclusion criteria. Mean length of follow-up was 18.8 years. Eighty-nine patients presented with a unilateral cleft (UNI) and 46 with a bilateral cleft (BI); 84% (113/135) of patients underwent GPP (UNI: 78% [69/89]; BI: 96% [44/46]), 43% (58/135) of patients underwent ABG (UNI: 40% [36/89]; BI: 48% [22/46]), 18% (24/135) of patients underwent speech surgery for VPI (UNI: 14% [12/89]; BI: 26% [12/46]), 3% (4/135) of patients underwent palatal fistula repair (UNI: 0% [0/89]; BI: 9% [4/46]), 31% (42/135) underwent orthognathic surgery (UNI: 22% [20/89]; BI: 48% [22/46]), and 11% (15/135) underwent revision surgery to lip, nose, or both prior to facial maturity (UNI: 9% [8/89]; BI: 15% [7/46]]. Of the patients who underwent GPP, 61% (69/113) did not require ABG (UNI: 65% [45/69]; BI: 55% [24/44]) and 42% (48/113) required neither ABG nor orthognathic surgery (UNI: 51% [35/69]; BI: 30% [13/44]).
Conclusion(s): Clinical outcomes of the NAM treatment protocol from birth to facial maturity demonstrate a low rate of revision surgery to the lip and nose, as well as a low fistula and VPI rate. The frequency of orthognathic surgery reported in this study is consistent with published data. In addition, 42% of patients who underwent NAM with GPP required neither ABG nor orthognathic surgery
EMBASE:631558226
ISSN: 1545-1569
CID: 4417662

Developing a sustainable nasoalveolar molding program in outreach settings: An eight-year follow-up [Meeting Abstract]

Kassam, S; Toomey, N; Azurin, E; Ramly, E; Kantar, R; Johnson, A; Ahmed, M; Grayson, B; Hamdan, U
Background/Purpose: Global Smile Foundation (GSF) is a nonprofit foundation that provides comprehensive cleft care to underserved patients. GSF focuses on long-term follow-up and sustainability of local health-care teams, having engaged in 32 years of follow-up in Ecuador. In 2012, GSF added presurgical nasoalveolar molding (NAM) therapy for their patients in Guayaquil, Ecuador, as part of its sustainability and empowerment initiative. We present longitudinal data on 189 patients treated with NAM and discuss the challenges/barriers to its completion. Methods/Description: Data were collected from GSF surgical and dental records including patient diagnosis, completion/incompletion, and length of NAM therapy. Surgeon, patient age, peri, intra, and postoperative procedural data were collected for primary cleft lip/nose and palate repair, and any additional surgeries. Follow-up clinical and photographic data were retrieved to document long-term outcomes.
Result(s): A total of 207 patients were treated with presurgical therapy: 189 patients received NAM therapy, while 18 patients were treated with lip tape and/or nasal elevator. Of the 189 NAM patients who received NAM, long-term follow-up was available for 96 (50.8%) patients, while 84 (44.4%) were lost to follow-up or subsequently seen by another foundation, and 9 (4.8%) are currently undergoing NAM or awaiting primary surgery. Of the 96 patients with long-term followup, 70 (72.9%) had unilateral cleft lip and palate and 26 (27.1%) had bilateral cleft lip and palate; 64 (66.67%) were male and 32 (33.3%) were female. Of those 96 patients, 58 (60.4%) completed NAM therapy, 17 (17.7%) failed to complete it, and 21 (21.8%) had incomplete NAM documentation. The average age at NAM initiation was 36.36 +/-31.39 days (range: 0-157 days) and average length of NAM therapy was 118.98 +/- 82.68 days (range: 1-222 days). Patients underwent an average of 2.13 +/- 0.93 (range: 1-5) surgeries after NAM initiation, with an average of 0.17 +/- 0.43 (0-2) cleft lip/nose revisions, 0.06 +/- 0.28 (0-2) gingivoperioplasty, 0.06 +/- 0.28 (0-1) premaxillary setbacks, 0.07 +/- 0.30 (0-2) fistula repairs, and 0.03 +/- 0.17 (0-1) velopharyngeal insufficiency corrections. Over an 8-year period, 12 NAM providers were trained in Ecuador; 7 provided treatment in Ecuador, and 5 provided treatment internationally, making Ecuador a site for information exchange. Follow-up for NAM patients was an average of 2.00 +/- 1.77 (0.22-6.67) years after NAM initiation and 1.45 +/- 1.77 (0-6.52) years after their primary cleft lip/nose repair. This includes continued long-term follow-up and comprehensive cleft care in addition to NAM therapy.
Conclusion(s): With yearly patient follow-up and year-round partnership with local professionals, our model shows successful long-term delivery of NAM therapy as part of a sustainable comprehensive cleft care strategy in outreach settings
EMBASE:631558253
ISSN: 1545-1569
CID: 4417652

Aesthetic outcomes of patients with unilateral cleft lip and palate following nasoalveolar molding therapy in an outreach setting [Meeting Abstract]

Azurin, E; Toomey, N; Kassam, S; Johnson, A; Ramly, E; Kantar, R; Grayson, B; Hamdan, U
Background/Purpose: Global Smile Foundation (GSF) is a not for profit foundation whose founders and volunteers have been providing cleft care to underserved communities around the world for 32 years. In 2012, GSF incorporated nasoalveolar molding (NAM) into its treatment model in Guayaquil, Ecuador. We present an evaluation of nasolabial aesthetic outcomes and scarring in patients treated with NAM prior to primary cleft lip repair versus patients who were not, in similar outreach settings. Methods/Description: The Cleft Aesthetic Rating Scale (CARS) used frontal photographs taken at least 1-year post primary cleft lip repair to assess the nose: tip, nostrils (symmetry, size, flaring), and upper lip (vermillion symmetry and continuity and length of the philtrum), ranging from 1 (very good) to 5 (very poor). Photos were standardized to reveal only the nasolabial area and excluded any time points after any additional surgical revision to the nasolabial area. Each NAM time point was matched to a control time point based on their age at primary cleft lip repair (maximum of 3 months) and their time postoperative from primary cleft lift repair (maximum of 6 months). All included NAM and control patients had unilateral cleft lip and palate, and were from Ecuador. Patients with congenital syndromes other than cleft lip and palate (CLCP) affecting facial appearance were excluded. Twelve independent raters including 3 surgeons, 3 orthodontist, 3 pediatric dentists, and 3 medical students rated the photographs. As a modification to CARS, raters were also asked to assess scar quality utilizing a previously developed scar subtype scoring system.
Result(s): Of the 189 patients treated with NAM in Guayaquil since 2012, 96 patients had long-term follow-up, and 27 patients with 34 photographic time points qualified for inclusion, 15 (55.56%) male, 12 (44.44%) female. The average time post primary cleft lip repair was 2.19 +/- 1.65 months (range: 0.98-7.37 months). Matched to the patients who had received NAM were 31 control patients with 34 photographic time points, 26 (83.87%) male, 5 (16.13%) female with an average time post primary cleft lip repair of 2.19 +/- 1.63 months (range: 0.93-6.98 months) The average rating of Nasal aesthetics was significantly lower in patients who had received NAM compared to nasal controls (2.60 +/- 1.05 vs 2.82 +/- 1.12; P< .01).The average rating for lip aesthetics showed similar differences between patients who had received NAM and matched controls (2.23+/-0.96 vs 2.56+/- 1.07; P < .01). Similarly, the average rating of scar quality was significantly lower among patients who had received NAM (1.82+/-0.93 vs 2.03 +/- 0.89; P < .01).
Conclusion(s): Using the CARS and a modified scar rating scale, patients who had received NAM were found to have superior nose and lip aesthetic outcomes in comparison to non-NAM controls. This suggests that NAM is not only feasible in an outreach setting, but it is also associated with improved lip, nose, and scar outcomes
EMBASE:631558209
ISSN: 1545-1569
CID: 4417682

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

Maxillomandibular and occlusal relationships in preadolescent patients with syndromic craniosynostosis treated by LeFort III distraction osteogenesis: 10-year surgical and phenotypic stability

Gibson, Travis L; Grayson, Barry H; McCarthy, Joseph G; Shetye, Pradip R
INTRODUCTION/BACKGROUND:LeFort III distraction osteogenesis may be indicated in the treatment of syndromic craniosynostosis with severe midface retrusion. This study investigates long-term changes in patients undergoing distraction as children, and compares outcomes to an unaffected, untreated control. METHODS:Fifteen patients (9 males, 6 females) with syndromic craniosynostosis treated by LeFort III distraction at an average age of 4.9 ± 1.5 years were identified. Lateral cephalograms at predistraction, immediate, 1-, 5-, and 10-year postdistraction were superimposed using the best-fit of cranial base details. An untreated, unaffected matched control was obtained from the American Association of Orthodontists Foundation Legacy Collection. Differences in landmark location and cephalometric relationships were assessed between time points and between treatment and control groups. RESULTS:LeFort III distraction produced an average advancement of 14.86 ± 5.14 mm at A-point and 10.54 ± 3.78 mm at orbitale. This advancement produced overcorrection of anteroposterior occlusal relationships and phenotypic correction of midface position. Surgical stability over a 10-year follow-up was excellent. Posttreatment growth was characterized by absent anteroposterior maxillary growth, preservation of dentoalveolar development and maxillary remodeling, and delayed mandibular growth. Subsequent growth resulted in a long-term phenotypic relapse of pretreatment Class III maxillomandibular relationship and negative overjet. CONCLUSIONS:LeFort III distraction osteogenesis produces stable advancement of the midface. Overcorrection is required for long-term phenotypic stability because of deficient postdistraction sagittal midface growth. Late mandibular growth contributes to underestimation of the amount of distraction required to produce long-term phenotypic correction.
PMID: 31784011
ISSN: 1097-6752
CID: 4216292