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Comparative Analysis of Three-Dimensional Nasal Shape of Casts from Patients With Unilateral Cleft Lip and Palate Treated at Two Institutions Following Rotation Advancement Only (Iowa) or Nasoalveolar Molding and Rotation Advancement in Conjunction With Primary Rhinoplasty (New York)

Hosseinian, Banafsheh; Rubin, Marcie S; Clouston, Sean A P; Almaidhan, Asma; Shetye, Pradip R; Cutting, Court B; Grayson, Barry H
OBJECTIVES/OBJECTIVE:To compare 3-dimensional nasal symmetry in patients with UCLP who had either rotation advancement alone or nasoalveolar molding (NAM) followed by rotation advancement in conjunction with primary nasal repair. DESIGN/METHODS:Pilot retrospective cohort study. MATERIALS AND METHODS/METHODS:Nasal casts of 23 patients with UCLP from 2 institutions were analyzed; 12 in the rotation advancement only group (Iowa) and 11 in the NAM, rotation advancement with primary nasal repair group (New York). Casts from patients aged 6 to 18 years were scanned using the 3Shape scanner and 3-dimensional analysis of nasal symmetry performed using 3dMD Vultus software, Version 2507, 3dMD, Atlanta, GA. Cleft and noncleft side columellar height, nasal dome height, alar base width, and nasal projection were linearly measured. Inter- and intragroup analyses were performed using t tests and paired t tests as appropriate. RESULTS:; P = .02). Intergroup analysis performed on the most sensitive linear measure, alar base width, revealed significantly less asymmetry on average in group 2 than in group 1 ( P = .013). CONCLUSION/CONCLUSIONS:This study suggests the NAM followed by rotation advancement in conjunction with primary nasal repair approach may result in less nasal asymmetry compared to rotation advancement alone.
PMID: 29578802
ISSN: 1545-1569
CID: 3011262

Treacher Collins Syndrome and Tracheostomy: Decannulation Using Mandibular Distraction Osteogenesis

Ali-Khan, Safi; Runyan, Christopher; Nardini, Gil; Shetye, Pradip; Staffenberg, David; McCarthy, Joseph G; Flores, Roberto L
INTRODUCTION/BACKGROUND:Treacher Collins syndrome (TC) and Pierre Robin sequence (RS) are associated with hypoplastic mandible, glossoptosis, and consequent airway obstruction. Although TC and RS are often grouped together, airway outcomes of bilateral mandibular distraction osteogenesis (MDO) have not been specifically studied in TC. The purpose of this study is to report on the clinical outcomes of MDO in the TC patient population. MATERIALS AND METHODS/METHODS:A twenty-year single-institution retrospective review of all patients with TC who underwent bilateral MDO was performed. Twenty-four patients were identified after exclusion due to different diagnoses or insufficient medical records. Data on comorbidities, airway status, MDO operations, and complications were collected. Data were compared with published clinical outcomes in RS and data for 13 RS patients from our institution. RESULTS:Surgical success, defined as prevention of imminent tracheostomy or successful decannulation within 1 year after primary distraction, was observed in 21% of TC patients and 65% of RS patients (P = 0.01). Repeat distraction was necessary for 11 TC patients (46%) and 1 RS patient. Complications were divided into minor, moderate, and major based on need for invasive management. Overall, 67% of TC patients had complications, 20% of which were major. CONCLUSIONS:Clinical outcomes to airway function after MDO are significantly inferior in patients with TC compared with patients with RS. Repeat MDO and longer course to decannulation are more prevalent in patients affected by TC.
PMID: 29905603
ISSN: 1536-3708
CID: 3155322

Proptosis Correction in Pre-Adolescent Patients With Syndromic Craniosynostosis by Le Fort III Distraction Osteogenesis

Gibson, Travis L; Grayson, Barry H; McCarthy, Joseph G; Shetye, Pradip R
Le Fort III distraction osteogenesis may be indicated in the treatment of syndromic craniosynostosis with severe midface retrusion and proptosis. This study assesses the stability of proptosis correction over 10-years.A retrospective review identified 15 patients with syndromic craniosynostosis treated by Le Fort III distraction prior to age 10 (9 males, 6 females; age 4.9 ± 1.5 years). Untreated, non-craniosynostotic age- and gender-matched controls were obtained from historical growth records. Lateral cephalometric tracings at pre-surgery (T1), immediate (T2), 1 year (T3), 5 years (T4), and 10 years (T5) (n = 11) post-distraction were superimposed using the best-fit of cranial base. Proptosis severity was defined as the horizontal distance between the Ant. Globe cephalometric point and orbital rim landmarks Orbitale and Lat. Orbit.The orbital rim advanced 10.54 ± 3.78 mm (P < 0.001) at Orbitale and 9.73 ± 4.54 mm (P > 0.001) at Lat. Orbit from T1 to T2; Ant. Globe advanced 3.13 ± 3.02 mm (p 0.001). Proptosis decreased 7.41 ± 5.29 mm (P < .001) from Orbitale and 6.60 ± 6.50 mm (p 0.002) from Lat. Orbit. Comparison to controls demonstrated phenotypic correction. In craniosynostotic patients from T2 to T5, the bony orbital rim demonstrated non-significant remodeling posteriorly and inferiorly. Anterior Globe moved 3.79 ± 1.47 mm anteriorly (P < .001), which did not differ significantly from controls. Proptosis increased by 4.18 ± 2.94 mm in craniosynostotic patients from T2 to T5.Le Fort III distraction was stable, with no significant anteroposterior relapse of the maxilla or bony orbit. Phenotypic relapse of proptosis to pre-treatment levels occurred through deficient growth of the midface, surface resorption at the orbital rim, and preservation of normal forward movement of Ant. Globe.
PMID: 29771833
ISSN: 1536-3732
CID: 3121472

The Effect of Nasoalveolar Molding on Nasal Airway Anatomy: A 9-Year Follow-up of Patients With Unilateral Cleft Lip and Palate

Massie, Jonathan P; Bruckman, Karl; Rifkin, William J; Runyan, Christopher M; Shetye, Pradip R; Grayson, Barry; Flores, Roberto L
OBJECTIVE:To determine the effects of nasoalveolar molding (NAM) on nasal airway architecture. DESIGN/METHODS:Retrospective case-control study of patients with unilateral cleft lip treated with NAM vs without NAM. SETTING/METHODS:Tertiary referral center specializing in cleft and craniofacial care. Patients, Participants, and Interventions: Thirty-six patients with complete unilateral cleft lip and alveolus: 19 with NAM therapy and 17 without NAM therapy. MAIN OUTCOME MEASURES/METHODS:Cone beam computed tomography (CBCT) scans were compared in multiple coronal sections and were evaluated for linear and angular septal deviation, inferior turbinate hypertrophy, and linear and 2-dimensional airway area. RESULTS:There were no significant differences in linear or angular septal deviation, inferior turbinate area, linear stenosis, or airway area between NAM- and non-NAM-treated patients. CONCLUSIONS:NAM effectively molds the external nasal cartilage and structures but may have limited effects on internal nasal structures.
PMID: 29356619
ISSN: 1545-1569
CID: 2929392

Prevalence of dental anomalies in unilateral cleft lip and palate after gingivoperiosteoplasty [Meeting Abstract]

Gibson, T; Grayson, B; Shetye, P
Background/Purpose: Gingivoperiosteoplasty (GPP) performed with lip repair has been shown to eliminate the need for alveolar bone grafting in two-thirds of treated cleft sites. In patients who have received GPP and subsequently require alveolar bone grafting (ABG), bone fill may be more favorable than in patients treated by ABG alone. However, some reports have suggested that GPP increases the risk of dental anomalies. This study aimed to assess the prevalence of dental anomalies in patients who were treated by GPP compared to those treated by ABG without GPP. Methods/Description: A retrospective chart review was conducted to identify patients born January 1, 2000, to December 31, 2007, with unilateral complete cleft lip and alveolus, with or without cleft palate. Patients were included if they received GPP or ABG at our center, and had adequate panoramic radiographs and clinical images at ages 5 to 9 and 10 to 12 years. Clinical records were assessed for missing or malformed teeth by a blinded examiner. Cleft side lateral incisors were classified as absent, present, extracted, and supernumerary; cleft side lateral incisor morphology was classified as normal, undersized/ peg shaped, or severely malformed; cleft side central incisors were classified as absent, normal, or anomalous; and the number of cleft side premolars was recorded. Dental anomalies were compared between the GPP and no-GPP groups using the chi-square test.
Result(s): Ninety-four patients met inclusion criteria: 46 treated with GPP, and 48 patients who did not receive GPP. Among patients who received GPP, cleft-side lateral incisors were absent in 54% of patients, compared to 50% of patients who did not receive GPP. Two patients in the GPP group and 4 in the no-GPP group required lateral incisor extraction. Two patients in the GPP group and one in the no- GPP group had supernumerary lateral incisors. These differences were statistically nonsignificant (P = .919). The majority of lateral incisors were undersized or peg shaped in both the no-GPP (20, 83.3%) and GPP (15, 71.4%) groups. One patient in the GPP group had a severely malformed lateral incisor. These differences were not statistically significant (P = .442). Cleft side central incisors were present in the majority of patients. In the GPP group, 5 patients (10.9%) exhibited central incisor agenesis, and a further 3 had significant hypoplasia. In the no-GPP group, 4 patients (8.3%) exhibited central incisor agenesis, and 5 (10.5%) significant hypoplasia. There were no intergroup differences (P = .937). Eight patients in the GPP group and 14 in the no- GPP group were missing cleft side premolars; the difference was not statistically significant (P = .937).
Conclusion(s): In this sample, gingivoperiostoplasty was not associated with increased prevalence of agenesis or malformation of cleft side incisors or premolars. When performed appropriately, gingivoperiosteoplasty is a safe treatment technique that does not increase the risk of dental anomalies
EMBASE:629010836
ISSN: 1545-1569
CID: 4051412

Nasoalveolar molding in patients with bilateral clefts of the lip, alveolus, and palate [Meeting Abstract]

Shetye, P; Flores, R
Background/Purpose: Presurgical infant orthopedics has been employed since 1950 as an adjunctive neonatal therapy for the correction of cleft lip and palate. Most of these therapies did not address deformity of the nasal cartilage in unilateral and bilateral cleft lip and palate as well as the deficiency of the columella tissue in infants with a bilateral cleft. The nasoalveolar molding (NAM) technique is a unique approach to presurgical infant orthopedics to reduce the severity of the initial cleft of the alveolar and the nasal deformity, particularly in patients with bilateral cleft lip and palate. Methods/Description: In infants with bilateral cleft lip and palate, the premaxilla may be protrusive, mobile, and may show varying degree of asymmetrical displacement and rotation. In some instances, the premaxilla may be everted placed on top of the nasal tip with a very short columella length. Protruded premaxilla and the associated nasal deformity present a special challenge for the surgeon in achieving optimal repair during primary reconstructive surgery. This study session will demonstrate the NAM technique to treat patients with severe bilateral cleft lip and palate. The technique of correcting the protruded and asymmetrically displaced premaxilla, molding the alar cartilage and nonsurgical columella elongation will be discussed. Appliance design and weekly adjustment of the NAM appliance to accomplish the desired result will be presented. Special emphasis will be placed on leveling the premaxilla in asymmetric cases; retracting premaxilla in incomplete bilateral clefts and management of complications during the course of the NAM therapy will be discussed. For the successful outcome, the surgeon has to take the advantage of the NAM therapy during the primary repair. Surgical technique of 1-stage lip nose and alveolus surgery utilizing the presurgical preparation of infants with bilateral cleft lip and palate with NAM therapy will be discussed in detail. Long-term outcome of patients treated with NAM and primary reconstruction of nose lip and alveolus will be presented
EMBASE:629010833
ISSN: 1545-1569
CID: 4051422

Orthodontic management of patients with cleft lip and palate from infancy to skeletal maturity [Meeting Abstract]

Shetye, P; Figueroa, A
Background/Purpose:Management of patientswith cleft lip and palate is complex and requires a multidisciplinary team with several treatment interventions. Proper sequencing and timing of orthodontic and surgical treatment is important for successful long-term outcome and reducing the burden of care on the families. This presentation will focus on orthodontic management of patients born with cleft lip and palate from infancy to skeletal maturity. Methods/Description: The management of patients with cleft lip and cleft palate requires extended orthodontic treatment and an interdisciplinary approach in providing these patients with optimal aesthetics, function, and stability. Orthodontic or orthopedic management in infancy, primary, mixed, and permanent dentition and after the completion of facial growth will be discussed with a proper interdisciplinary approach to treatment planning and treatment sequencing during each phase of orthodontic and surgical treatment. This presentation will discuss the presurgical infant orthopedic, pre and post bone graft orthodontics, phase II comprehensive orthodontic treatment, and LeFort I distraction and orthognathic surgery at skeletal maturity. Long-term outcome of treatment will be presented of patients treated from birth to adulthood
EMBASE:629010809
ISSN: 1545-1569
CID: 4051432

Effects of alveolar cleft management on permanent canine position and eruption: comparing gingivoperiosteoplasty and secondary alveolar bone grafting [Meeting Abstract]

Gibson, T; Grayson, B; Flores, R; Shetye, P
Background/Purpose: Gingivoperiosteoplasty (GPP) performed concurrent with lip repair is an option for treating bony alveolar deficiency in patients with orofacial clefts. GPP has been demonstrated to produce bony continuity, eliminating the need for alveolar bone grafting (ABG) in two-thirds of treated cleft sites. The purpose of this study was to assess if early bone formation as produced by successful GPP influences maxillary canine eruption. Methods/Description: A retrospective chart review was conducted to identify patients born between January 1, 2000, and December 31, 2007, with unilateral complete cleft lip and alveolus, with or without cleft palate. Patients were included if they had successful GPP or ABG, and had panoramic or maxillary CBCT radiographs available at age 5 to 9 (T1) and 9 to 12 (T2) years, with a minimum of 6 months between radiographs. Panoramic images were excluded if a head positioning error produced an occlusal plane greater than 15degree from perpendicular to midline. Panoramic images were used to assess maxillary canine sector, angulation relative to midline and ipsilateral occlusal plane, and cusp tip height from ipsilateral occlusal plane. CBCT images were used to assess the horizontal distance between the canine cusp tip and the maxillary arch form. Clinical charts were reviewed to determine if canines erupted successfully or required intervention. Finally, canine mesial-distal and labio-lingual position after eruption was assessed using occlusal photographs. Outcomes in GPP and ABG groups were compared, and results were stratified by ipsilateral lateral incisor presence or absence.
Result(s): Seventy-nine patients met inclusion; 24 had successful bone fill after GPP, and 55 after ABG. In patients with cleft-side lateral incisors present, no significant differences were found between GPP and ABG groups in canine angulation, height, sector, eruptive outcome, or timing of eruption. When spontaneous canine eruption occurred, there was a statistically nonsignificant trend to more mesial eruptive position in patients who were treated with GPP. In patients with cleft-side lateral incisor agenesis, initial canine angulation did not differ. Patients who were treated with GPP demonstrated 10.8degree +/- 11.1degree spontaneous canine uprighting from T1 to T2, while canine angulation was maintained in the ABG group; this difference was statistically significant (P = .001). The GPP group demonstrated greater canine descent from T1 to T2, resulting in significantly less distance from the occlusal plane (5.8 +/- 4.8 mm) compared to the ABG group (9.4+/-4.2 mm). Horizontal distance to arch did not differ between the groups. In the GPP group, 75% of patients demonstrated successful spontaneous canine eruption, compared to 41% in the ABG group, though this did not reach statistical significance (P = .146).
Conclusion(s): Gingivoperiosteoplasty favorably influenced the angulation, height, and eruptive success of cleft-side canines in patients. These benefits were predominantly noted in patients with congenital absence of lateral incisors
EMBASE:629011173
ISSN: 1545-1569
CID: 4051482

Incidence of secondary midface advancement at the time of skeletal maturity in patients with a History of Early LeFort III Distraction Osteogenesis [Meeting Abstract]

Cho, G; Borab, Z; Gibson, T; Shetye, P; Grayson, B; Flores, R; McCarthy, J
Background/Purpose: LeFort III distraction osteogenesis is commonly recommended for children with syndromic craniosynostosis to reduce exorbitism, improve airway function, and decrease dysmorphism. This purpose of this study is to report on the long-term clinical outcomes of patients with syndromic craniosynostosis patients who have undergone early primary subcranial LeFort III distraction osteogenesis and who have been followed longitudinally through skeletal maturity. Methods/Description: Retrospective review of all patients who underwent LeFort III distraction osteogenesis between the ages of 3 and 11 years and were followed throughout development with longitudinal dental, medical, radiographic, and photographic evaluations conducted through skeletal maturity and beyond. Inclusion criteria entailed having preoperative medical photographs and cephalometric studies at 6 months and 1, 5, and 10 years postoperatively after the primary LeFort III distraction osteogenesis as well as cephalometric documentation 6 months and 1 year after the secondary midface advancement after skeletal maturity.
Result(s): Seventeen patients fulfilled inclusion criteria, with a mean age of 5.7 years at the time of initial LeFort III distraction. The mean advancement of point A was 14.9 mm anteriorly and 2.7 mm inferiorly along the x- and y-axis, respectively. Orbitale moved 10.5 mm anteriorly and 2.2 mm inferiorly along the x- and y-axis, respectively. At 10 years postoperatively, point A moved 3.4 mm anterior along the xaxis and 4.7 mm inferiorly along the y-axis, while orbitale moved 0.4 mm posteriorly and 3 mm inferiorly along the x- and y-axis, respectively. At the time of skeletal maturity, there was a return of occlusal disharmony from normal mandibular growth and a return of proptosis owing to remodeling of orbitale inferiorly, and the lateral orbital rim posteriorly, while the globe continued to grow in the anterior vector. All but 1 study patient underwent or is scheduled to undergo a secondary midface advancement at the LeFort III and LeFort I level after skeletal maturity was attained.
Conclusion(s): The data demonstrate that patients who undergo early LeFort III distraction osteogenesis before the age of mixed dentition will still most likely need a secondary midface advancement after skeletal maturity is reached given that there is a small degree of anterior growth at the level of the maxilla and no anterior growth at orbitale over time
EMBASE:629011081
ISSN: 1545-1569
CID: 4051502

Eruption of maxillary posterior permanent teeth following early conventional Lefort III advancement and LeFort III distraction surgeries [Meeting Abstract]

Gonchar, M; Grayson, B; Bekisz, J; McCarthy, J; Shetye, P
Background/Purpose: Early LeFort III (LFIII) surgery or LFIII distraction involve osteotomies and disjunction in the region of the maxillary tuberosity in proximity to the maxillary posterior tooth buds. The purpose of this study was to determine the effect of early LFIII advancement and/or distraction on survival of the maxillary posterior permanent dentition. Methods/Description: A retrospective review of patients with syndromic craniosynostosis treated by early LFIII surgery and distraction was conducted. Of 225 syndromic craniosynostosis patients enrolled between 1973 and 2006, a total of 50 patients satisfied the inclusion criteria: 1) surgical intervention prior to age 8 years; 2) two panoramic radiographs, one prior to surgery and one in adolescence; 3) no apparent abnormalities in the position of permanent tooth buds. Of the 50 patients, 25 underwent LFIII surgery and 25 underwent midface distraction (M = 21, F = 29, average age at time of surgery = 5 +/- 1.1 years with diagnoses of Crouzon (20), Apert (17) and Pfeiffer (13), syndromes). Panoramic radiographs presurgically (T1) and postsurgically (T2) were inspected by a trained observer. The tooth buds were classified as being present (P), displaced (D), impacted (I), ankylosed (ANK), extracted (E), or absent (A). SPSS software was used to carry out chi-squared analysis and Fisher exact test.
Result(s): In the LFIII surgery group, 94% of maxillary second molars (D = 16%, I = 8%, E = 6%, A = 64%) and 28% of maxillary first molars (D = 18%, I = 4%, ANK = 2%, E = 2%, A = 2%) experienced a disturbance in eruption. Of the displaced second molars, 75% were located in the maxillary sinus and 25% in the maxillary tuberosity. Of the displaced first molars, 78% were located in the maxillary sinus and 22% in the maxillary tuberosity. In the distraction group, 80% of maxillary second molars (D = 38%, ANK = 4%, E = 14%, A = 24%) and 18% of maxillary first molars (D = 10%, I = 2%, E = 2%, A = 4%) experienced a disturbance in eruption. Of the displaced second molars, 37% were located in the maxillary sinus and 63% in the maxillary tuberosity. Of the displaced first molars, 100% were located in the maxillary tuberosity. Traditional LFIII osteotomy was significantly more likely to result in an adverse event for maxillary second molars compared to distraction (chi2 = 4.33, P = .037).
Conclusion(s): The eruption of maxillary second molars had a high incidence of disruption following early LFIII intervention, with traditional LFIII surgery having greater negative consequences for the maxillary second molars compared to distraction. The maxillary first molars show significantly less disruption during early LFIII intervention with no significant differences noted between surgical procedures. Furthermore, a common disruption seen postsurgically is the displacement of the maxillary second molar tooth buds into the maxillary sinus, leading to the question if presurgical planning should include extraction/enucleation of the second molar tooth buds to avoid this sequela
EMBASE:629011060
ISSN: 1545-1569
CID: 4051532