Impact of protraction or orthognathic surgery for class III malocclusion on longitudinal quality of life in patients with cleft lip and palate.

OBJECTIVES: This study assessed overall quality of life (QoL) over time in youth with cleft lip and palate (CLP) undergoing maxillary protraction treatment or orthognathic surgery for class III malocclusion to identify any differences in QoL based on treatment group and outcome success. MATERIALS AND METHODS: A prospective longitudinal cohort study was conducted in two pediatric hospitals. The Short Form Health Survey (SF-12) measured physical and mental QoL prior to treatment, at maximal correction, at treatment completion, and at 1-year post treatment. Analyses included one-sample, two-sample, and paired t-tests and analyses of variance and covariance. RESULTS: Participants (N = 91) either completed protraction (n = 53) at age 11-14 or surgery (n = 38) at age 16-21. Participants were mostly Latinx (67%) males (55%) born with unilateral CLP (81%) and there were no demographic differences between the two groups other than age. The total sample's QoL was in the average range and significantly higher than national norms. No significant differences were found in QoL-based outcome success; however, the protraction group showed a gradual physical QoL improvement over time, while the surgery group experienced a temporary drop in physical QoL postoperatively. At treatment completion, higher physical QoL was associated with higher socioeconomic status. At a year post treatment, mental QoL was significantly higher for males. CONCLUSION: Both protraction and surgery appear to be acceptable treatment options in terms of overall QoL for youth with CLP. While treatment success did not impact QoL, there were some differences in physical QoL coinciding with the treatment phase as well as individual factors.

Comparison of Alveolar Bone Grafting Outcomes using CBCT in Individuals with UCLP Based on the Presurgical Orthodontic Treatment Methods.

OBJECTIVE: The purpose is to evaluate outcomes of alveolar bone grafting based on the pre-grafting orthodontic preparation methods. DESIGN: Retrospective analysis of individuals with unilateral cleft lip and palate. SUBJECTS AND SETTINGS: 28 individuals with non-syndromic UCLP from two craniofacial centers, 14 individuals each from XXXX and XXXX. INTERVENTIONS: The alignment group underwent maxillary expansion with incisors alignment while the non-alignment group underwent only maxillary expansion for presurgical orthodontic preparation. METHODS: Initial and post-surgical CBCT scans were compared to observe changes in angulation of the incisor adjacent to the cleft site, alveolar bony root coverage, and bone graft outcomes. RESULTS: In the alignment group, the buccolingual rotation decreased by 32.35 degrees (p = .0002), the anteroposterior inclination increased by 14.01 degrees (p = .0004), and the mesiodistal angulation decreased by 17.88 degrees (p = .0001). Alveolar bony coverage did not change after bone graft in both groups, and no difference was observed between the groups. Chelsea scale showed satisfactory bone graft outcome (category A, C) in 12 cases (85.71%) in the alignment group and 11 cases (78.51%) in the non-alignment group. The volumetric measurement showed the alignment group had better bone fill of 69.85% versus 51.45% in the non-alignment group (p = .0495). CONCLUSIONS: Alveolar bony coverage on the tooth adjacent to cleft sites did not change with alveolar bone grafting surgery in either of the alignment and non-alignment group. Presurgical orthodontic alignment does not induce root exposure nor poorer bone grafting outcome.

Dental Protraction Versus Surgery for Cleft Lip and Palate: A Budget Impact Analysis.

Class III malocclusion for individuals with cleft lip and palate has historically been managed with surgery. Orthodontic protraction is a noninvasive alternative that may be associated with lower costs. This analysis investigated the budget impact of protraction versus surgery from an institutional perspective. Using a decision tree, analysis was conducted using costs derived from Medicaid reimbursement codes and using actual institutional reimbursement. Probabilities of success, failure, and complications were based on a clinical trial comparing the 2 treatment modalities. One-way and probabilistic sensitivity analyses tested the robustness of results to model parameters. Based on Medicaid fee schedules and failure rates requiring additional surgery, the total cost of protraction was $79,506 versus $172,807 for surgery, resulting in $93,302 cost-savings per patient. The cost and probability of surgery success, as well as the cost of surgery failure and repeat surgery, had the largest impact on these cost-savings. Probabilistic sensitivity analysis showed cost-savings of nearly $92,000 or higher in >50% of simulations. This study showed that protraction is associated with lower costs than surgery and may present a cost-effective alternative to surgery in eligible, appropriate patients.

Is Le Fort I Distraction Enough to Treat Severe Midface Hypoplasia?

BACKGROUND: In severe cases of maxillary hypoplasia, Le Fort I distraction may be required for treatment. This study describes our experience with internal distraction devices and assesses our outcomes in patients with a negative overjet on average >15 mm. METHODS: A retrospective review of patients with a history of cleft lip and/or palate who underwent Le Fort I distraction at our institution from 11/2007-11/2017 was performed. Data regarding demographics, procedural details and outcomes were collected and analyzed. RESULTS: Twenty patients, 13 (65.0%) male and 7 (30.0%) female, were included. All (100%) patients had a history of cleft lip and/or palate and 2 (10.0%) of them had associated syndromes. All 20 patients underwent internal distraction. The average age at surgery was 17.8 years (range 15.2-20.7, SD 1.6 years). The average preoperative negative overjet was 19.27 mm (range 10-30, SD 5.63 mm). The mean total distraction length was 15.3 mm (range 0-30, SD 6.72 mm). There were no intraoperative complications, however, there were a total of 4 (20.0%) postoperative complications, 2 of which required reoperation due to device malfunction or displacement. 14 (70.0%) patients had repeat procedures to complete occlusal correction or correct relapse. CONCLUSIONS: Maxillary distraction alone was insufficient for correction of maxillary discrepancies averaging >15 mm. Instead, it's utility may be in positioning the maxilla for a more viable final advancement and fixation procedure, rather than being solely responsible for achieving normal occlusion during distraction.

Hospitalization Outcomes of Cleft Lip Repair in Neonates Across the United States.

OBJECTIVE: Cleft lip repair surgeries in neonates have shown to be effective and safe, resulting in less scarring and excellent aesthetic outcomes. However, existing studies are based on single-center experiences with limited numbers of patients and surgeons. Complication rates and hospital outcomes of neonatal lip repair have not yet been established at the national level. The objective of this study was to examine the association between age at cleft lip repair and hospital outcomes. DESIGN: Retrospective analysis of hospital discharge database. SETTING: Nationwide Inpatient Sample for years 2004 through 2010. PATIENTS: Patients under 12 months of age diagnosed with cleft lip with or without cleft palate. INTERVENTIONS: Surgical repair for cleft lip. MAIN OUTCOME MEASURES: Occurrence of complications. RESULTS: There were 10 132 cleft lip repair procedures in 2004-2010 in the United States. Mean age was 144 days with 2.1 days of hospital stay and $22 037 charges. Less than 2% were performed in neonates (0-28 days). The overall complication rate was 2.1%. Compared to 2-4 months, cleft lip procedures in neonates were associated with longer length of stay ( P = .001) and hospital charges ( P = .03). Cleft lip repair among neonates were 15 times more likely to develop complications ( P = .0004) even after adjusting for confounding factors. CONCLUSIONS: Cleft lip repair in neonates is associated with significantly higher complication rates as well as longer length of stay and more hospital charges. Purported benefits of neonatal cleft lip repair may not outweigh significant safety issues and hospitalization outcomes.

Three-Dimensional Cone Beam Computed Tomography Volumetric Outcomes of rhBMP-2/Demineralized Bone Matrix versus Iliac Crest Bone Graft for Alveolar Cleft Reconstruction.

BACKGROUND: Recent studies indicate that recombinant human bone morphogenetic protein-2 (rhBMP-2) in a demineralized bone matrix scaffold is a comparable alternative to iliac bone autograft in the setting of secondary alveolar cleft repair. Postreconstruction occlusal radiographs demonstrate improved bone stock when rhBMP-2/demineralized bone matrix (DBM) scaffold is used but lack the capacity to evaluate bone growth in three dimensions. This study uses cone beam computed tomography to provide the first clinical evaluation of volumetric and density comparisons between these two treatment modalities. METHODS: A prospective study was conducted with 31 patients and 36 repairs of the alveolar cleft over a 2-year period. Twenty-one repairs used rhBMP-2/DBM scaffold and 14 repairs used iliac bone grafting. Postoperatively, occlusal radiographs were obtained at 3 months to evaluate bone fill; cone beam computed tomographic images were obtained at 6 to 9 months to compare volumetric and density data. RESULTS: At 3 months, postoperative occlusal radiographs demonstrated that 67 percent of patients receiving rhBMP-2/DBM scaffold had complete bone fill of the alveolus, versus 56 percent of patients in the autologous group. In contrast, cone beam computed tomographic data showed 31.6 percent (95 percent CI, 24.2 to 38.5 percent) fill in the rhBMP-2 group compared with 32.5 percent (95 percent CI, 22.1 to 42.9 percent) in the autologous population. Density analysis demonstrated identical average values between the groups (1.38 g/cc). CONCLUSIONS: These data demonstrate comparable bone regrowth and density values following secondary alveolar cleft repair using rhBMP-2/DBM scaffold versus autologous iliac bone graft. Cone beam computed tomography provides a more nuanced understanding of true bone regeneration within the alveolar cleft that may contribute to the information provided by occlusal radiographs alone. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, II.

A Comparison between Osteotomy and Corticotomy-Assisted Tooth Movement.

Osteotomies and corticotomies used in combination with orthodontic tooth movement can activate different bone responses that may be exploited to accelerate tooth movement. Segmental osteotomies around dental roots can create a tooth-bearing transport disk that may be distracted and positioned with orthodontic appliances and archwires. In difficult craniofacial repairs, alveolar segments can be guided into position with archwires and orthodontic mechanics. The corticotomy extending into the marrow space can activate bone injury repair mechanisms that accelerate bone turnover as the alveolar bone surrounding the dental roots transitions from a demineralization phase to a fibrous replacement phase and, finally, a mineralization phase. The controlled demineralization and replacement of alveolar bone provides a window of opportunity for roots to move though less dense bone prior to remineralization. Although the corticotomies and osteotomies are minor surgeries compared to orthognathic surgery, the goal of future research is to produce similar bone responses by using smaller surgeries or by eliminating the surgeries altogether.

Missense mutation in the PTEN promoter of a patient with hemifacial hyperplasia.

The cellular mechanisms involved in the asymmetric facial overgrowth syndrome, hemifacial hyperplasia (HFH), are not well understood. This study was conducted to compare primary cell cultures from hyperplastic and normal HFH bone for cellular and molecular differences. Primary cultures developed from biopsies of a patient with isolated HFH showed a twofold difference in cell size and cell number between hyperplastic and normal bone. Microarray data suggested a 40% suppression of PTEN (phosphatase-tensin homolog) transcripts. Sequencing of the PTEN gene and promoter identified novel C/G missense mutation (position -1053) in the regulatory region of the PTEN promoter. Western blots of downstream pathway components showed an increase in PKBa/Akt1 phosphorylation and TOR (target of rapamcyin) signal. Sirolimus, an inhibitor of TOR, when added to overgrowth cells reversed the cell size, cell number and total protein differences between hyperplastic and normal cells. In cases of facial overgrowth, which involve PTEN/Akt/TOR dysregulation, sirolimus could be used for limiting cell overgrowth.

Apriori feasibility testing of randomized clinical trial design in patients with cleft deformities and Class III malocclusion.

OBJECTIVES: To assess the feasibility of randomizing treatment (surgical vs. non-surgical) for correction of a Class III malocclusion (underbite) resulting from an earlier repair of cleft lip and palate. MATERIALS AND METHODS: Surveys about willingness to accept randomized treatment during adolescence were mailed to the parents of cleft lip and palate patients under the care of Children's Hospital Los Angeles between 2005 and 2010. The inclusion criteria were patients with cleft lip and palate, Class III malocclusion due to maxillary deficiency, and absence of medical and cognitive contraindications to treatment. RESULTS: Out of 287 surveys, 82 (28%) were completed and returned; 47% of the subjects held a strong treatment preference (95% CI, 35-58%), while 30% were willing to accept randomization (95% CI, 20-41%). Seventy-eight percent would drop out of a randomized trial if dissatisfied with the assigned treatment (95% CI, 67-86%). The three most commonly cited reasons for being unwilling to accept random treatment assignment were 1) the desire for doctors to choose the best treatment, 2) the desire for parents to have input on treatment, and 3) the desire to correct the underbite as early as possible. CONCLUSION: Based on this study, parents and patients would be unwilling to accept a randomly assigned treatment and would not remain in an assigned group if treatment did not meet expectations. This highlight the limitations associated with randomization trials involving surgical modalities and provide justification for other research models (e.g., cohort studies) to compare two treatment options when randomization is not feasible.

Late maxillary protraction in patients with unilateral cleft lip and palate: a retrospective study.

OBJECTIVES: This retrospective study assessed the dentoskeletal effect of late maxillary protraction (LMP; reverse-pull headgear, Class III elastics, and maxillary sutural loosening) in unilateral cleft lip and palate (UCLP) patients versus a control group of untreated UCLP patients. MATERIALS AND METHODS: Cephalograms taken at age 13 to 14 years (T1) and 17 to 18 years (T2) were used for this study. The study group comprised 18 patients (10 male and 8 female, mean age at start of LMP therapy = 13.4 [0.45] years). A control groups of 17 patients (8 male and 9 female, mean age = 13.5 [0.44] years) was used for comparison. RESULTS: The repeated-measures analysis of variance showed statistically significant changes across time between groups for the following variables (mean difference [T2-T1] in the study group, 95% confidence interval): SNA (°) (1.95, 0.75 to 3.15), A ⊥ Na Perp (mm) (1.82, 0.86 to 2.77), CoA (mm) (2.92, 1.53 to 4.31), ANB (°) (3.13, 2.02 to 4.24), Wits (mm) (7.82, 5.01 to 10.54), Mx-Md Diff (mm) (0.62, -1.58 to 2.83), Occl P-SN (°) (-3.98, -5.99 to -1.98), overjet (mm) (8.82, 5.90 to 11.74), FMIA (°) (4.05, -0.05 to 8.15), and IMPA (°) (-5.77, -9.74 to -1.80). Late maxillary protraction created a slight open bite (0.66 mm). Trends for overeruption of mandibular incisors and an increase in lower face height (P = .07 for both) were noted in the study group. CONCLUSIONS: Late maxillary protraction produced a combination of skeletal changes (protraction of maxilla, improvement in the maxillo-mandibular skeletal relationship) and dental compensations (counterclockwise rotation of occlusal plane, retroclination of mandibular incisors) in patients with UCLP. Late maxillary protraction was also associated with some unwanted tooth movements (open bite tendency, mandibular incisors overeruption).

Clinicians and laypeople assessment of facial attractiveness in patients with cleft lip and palate treated with LeFort I surgery or late maxillary protraction.

OBJECTIVE: To assess the changes in the facial attractiveness (FA) in two groups of cleft lip and palate patients with Class III malocclusions treated using LeFort I surgery or late maxillary protraction. MATERIALS AND METHODS: Standardized pre- and post-treatment photographs were taken of 32 patients (17 corrected by orthognathic surgery and 17 by late maxillary protraction). The photographs were randomized and 42 clinicians and 121 laypeople rated them on a 10-point FA scale via a web-based survey. RESULTS: Clinicians' mean FA values increased from 4.45 to 5.16 [95% CI of mean difference (MD), 0.59-0.82, p<0.001] in surgical cases and 4.84 to 5.30 (95% CI of MD, 0.35-0.56, p<0.001) in protraction cases. The laypeople mean FA values increased from 5.07 to 5.54 (95% CI of MD, 0.40-0.53, p<0.001) in surgical cases and 5.51 to 5.68 (95% CI of MD, 0.11-0.23, p<0.001) in protraction cases. When patients combined, laypeople rated FA 0.64 points higher (95% CI, 0.54-0.74, p<0.001) in pre-treatment and 0.38 points higher (95% CI, 0.27-0.48, p<0.05) in post-treatment relative to clinicians. CONCLUSION: Both clinicians and laypeople perceived an improvement of FA after both treatments. Laypeople rated FA higher compared to clinicians.

A rare case of accessory maxilla and bilateral Tessier no. 7 clefts, a 10-year follow-up.

A Tessier no. 7 cleft is a lateral facial cleft which originates from the oral cavity and extends towards the tragus, involving both soft-tissue and skeletal components. A male patient presenting with both maxillary jaw duplication and bilateral Tessier no. 7 clefts, which has been reported only twice in the literature, is described. Bilateral facial clefts, macrostomia and chondro-cutaneous remnants were noted, which were repaired and resected. With further growth, facial asymmetry and asymmetric facial nerve dysfunction became apparent. Radiographic examination showed an accessory maxillary jaw and a flattened and hypoplastic right coronoid process. A maxillary alveolar cleft was also present between the left second bicuspid and the second permanent molar. This case may represent an under-recognized phenotype with an unusual combination of maxillary jaw duplication, macrostomia, Tessier no. 7 clefts, and chondro-cutaneous remnants. A long-term follow-up of these patients is recommended as they often develop craniofacial deformities later in life.

An unusual accessory mandible and a submucosal cleft palate-a case report and review of the literature.

An accessory mandible is a rare congenital anomaly that requires multidisciplinary management. This case report describes a female patient with an unusual accessory mandible, a dysplastic overgrowth of bone, containing teeth that extended from the midsymphyseal region. A submucosal cleft palate and cleft of the lower lip were also present. Her treatment plan took a staged approach with initial surgical resection of the accessory bone and teeth. The second stage, still in the planning phase, will correct the secondary deformity of an anterior open bite and will restore the missing lower anterior teeth. The original deformity and subsequent growth are discussed with the relevant literature.

Bilateral maxillary duplication: case report and literature review.

Accessory maxillary jaws are extremely rare occurrences. Currently, there is only 1 report of bilateral accessory maxillary jaws in the English-language literature. We present a case of a 7-year-old girl with bilateral bony exostoses extending from the maxillary tuberosities. The patient also had restricted protrusive and lateral excursive movements of the mandible. The histologic report revealed teeth in various developmental stages within the bony exostoses. We concluded that these structures were an isolated form of bilateral accessory maxillary jaws.

The nance obturator, a new fixed obturator for patients with cleft palate and fistula.

In patients with fistulas that impair function (e.g., feeding, resonance, intelligibility), obturators are used to improve feeding and reduce nasal air emission by occluding the abnormal opening between the oral and nasal cavities. This report describes a novel method for occluding an anterior palatal fistula in patients with cleft palates. The new design for a fixed obturator is based on the Nance appliance, which was originally used as a space maintainer, but has been redesigned for closing an anterior palatal fistula in a patient with cleft lip and palate. The Nance obturator may be used when the surgical closure of the fistula is not feasible and a removable device is not successful. As it is a fixed device, it does not require remaking with maxillary growth. The new design may also function as a fixed space maintainer to preserve molar anchorage and maxillary transverse width.

Protocols for Late Maxillary Protraction in Cleft Lip and Palate Patients at Childrens Hospital Los Angeles.

This paper describes the protocols used at Childrens Hospital Los Angeles (CHLA) to protract the maxilla during early adolescence. It is a modification of techniques introduced by Eric Liou with his Alternate Rapid Maxillary Expansion and Constriction (ALT-RAMEC) technique. The main differences between the CHLA protocol and previous maxillary protraction protocols are the age the protraction is attempted, the sutural loosening by alternating weekly expansion with constriction and the use of Class III elastics to support and redirect the protraction by nightly facemask wear. The CHLA protocol entirely depends on patient compliance and must be carefully taught and monitored. In a cooperative patient, the technique can correct a Class III malocclusion that previously would have been treated with LeFort 1 maxillary advancement surgery. Thus, it is not appropriate for patients requiring 2 jaw surgeries to correct mandibular prognathism, occlusal cants or facial asymmetry. The maxillary protraction appears to work by a combination of skeletal advancement, dental compensation and rotation of the occlusal planes. Microscrew/microimplant/temporary anchorage devices have been used with these maxillary protraction protocols to assist in expanding the maxilla, increasing skeletal anchorage during protraction, limiting dental compensations and reducing skeletal relapse.

Tisssue responses in corticotomy- and osteotomy-assisted tooth movements in rats: histology and immunostaining.

INTRODUCTION: The purpose of this histologic study was to examine underlying cellular responses to corticotomy- and osteotomy-assisted tooth movements. METHODS: Thirty-six rats were divided into 5 groups: corticotomy-assisted tooth movement (CO + TM), sham corticotomy without tooth movement (CO alone), osteotomy-assisted tooth movement (OS + TM), sham osteotomy without tooth movement (OS alone), and unassisted tooth movement (TM alone). Standard orthodontic springs were activated to produce mesial tooth movement. The rats were killed at 3, 21, and 60 days after activation for osteoclast and blood vessel counts, and immunostaining with proliferating cell nuclear antigen (PCNA), transforming growth factor beta 1 (TGF beta 1), vascular endothelial growth factor (VEGF), and osteocalcin were performed. RESULTS: The CO + TM group had significantly more osteoclasts at 3 days (P <0.005) compared with the OS + TM group. The alveolar bone surrounding the dental roots was replaced with multicellular tissue at 21 days in the CO + TM group but was intact in the OS + TM group with the exception of a distal distraction site. At day 21, immunostaining with PCNA, TGF beta 1, VEGF, and osteocalcin occurred at the mesial border of bone in the CO + TM group, whereas a diffuse pattern was observed in the distal distraction sites at 21 and 60 days in the OS + TM group. CONCLUSIONS: Corticotomy-assisted tooth movement produced transient bone resorption around the dental roots under tension; this was replaced by fibrous tissue after 21 days and by bone after 60 days. Osteotomy-assisted tooth movement resembled distraction osteogenesis and did not pass through a stage of regional bone resorption.

Clinical applications of orthodontic microimplant anchorage in craniofacial patients.

Microimplant anchors, also known as temporary anchorage devices, mini- and micro-screws, have been used to enhance orthodontic anchorage for difficult tooth movements. Here, the authors describe how microimplants can be used to help treat craniofacial patients by supporting distraction osteogenesis procedures, maxillary protraction procedures, cleft segment expansion and stabilization, and tooth movement into narrow alveolar cleft sites. While most craniofacial patients are treated without microimplants, it would be worthwhile to identify which cases could benefit from microimplant anchorage. As an adjunct to orthodontic treatment, the microimplant offers a potential method for solving troublesome orthodontic and surgical problems such as guiding distraction procedures with orthodontics when primary teeth are exfoliating, addressing residual maxillary cants after vertical distraction osteogenesis of a ramus, stabilizing an edentulous premaxilla, and moving teeth into atrophic alveolar ridges. These cases are presented to open a dialogue on their possible uses in craniofacial patients.

A novel model surgery technique for LeFort III advancement.

Current techniques for model surgery and occlusal splint fabrication lack the ability to mark, measure and plan the position of the orbital rim for LeFort III and Monobloc osteotomies. This report describes a model surgery technique for planning the three dimensional repositioning of the orbital rims. Dual orbital pointers were used to mark the infraorbital rim during the facebow transfer. These pointer positions were transferred onto the surgical models in order to follow splint-determined movements. Case reports are presented to illustrate how the model surgery technique was used to differentiate the repositioning of the orbital rim from the occlusal correction in single segment and combined LeFort III/LeFort I osteotomies.