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.

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.

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.

Premaxillary Setback in Bilateral Cleft Lip and Palate Repair.

OBJECTIVE: The aim of this study is to evaluate surgical outcomes and maxillofacial growth in patients undergoing primary lip repair with or without premaxillary setback. DESIGN: Retrospective review. SETTING: Children's Hospital of Los Angeles, California. PATIENTS AND PARTICIPANTS: Patients with bilateral cleft lip ± palate (BCLP) who underwent lip repair with or without premaxillary setback from January 1975 to September 2021. INTERVENTIONS: No intervention was performed. MAIN OUTCOME MEASURE(S): Patient demographics, comorbidities, and syndromic status were obtained. Indications for premaxillary setback, incidence of midface hypoplasia, orthodontic and/or orthognathic treatments, follow-up, complications, and revisions were recorded and analyzed. Comparisons among long-term outcomes, particularly the development of midface hypoplasia were made between groups. RESULTS: Thirty-one patients who underwent BCLP repair with premaxillary setback (BCLP + PS) and 31 matched control patients who underwent BCLP repair without premaxillary setback (BCLP - PS) were included. Among the 2 groups, multiple logistic regression demonstrated that when controlling for comorbidities, syndromic status, timing of lip repair, and timing of palate repair, premaxillary setback was neither significantly associated with the development of midface hypoplasia (P = .076) nor the timing of midface hypoplasia development (P = .940) in those that ultimately acquired this facial dysmorphology. CONCLUSIONS: While a high incidence of midface hypoplasia was seen in both BCLP ± PS and BCLP - PS, our findings demonstrate no difference in midface hypoplasia irrespective of premaxillary setback in the setting of BCLP. Future prospective studies investigating the downstream ramifications of our suggested selection criteria for premaxillary setback are warranted.

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.

Simultaneous LeFort III and LeFort I Osteotomies in Craniometaphyseal Dysplasia.

Craniometaphyseal dysplasia (CMD) is a rare genetic disease affecting bone metabolism with sclerosis of craniofacial bones. Orthognathic surgery has rarely been described in this patient population due to the bony thickness, making osteotomies challenging. We present a 19-year-old male with CMD with malocclusion, severe midface hypoplasia, and obstructive sleep apnea. With the aid virtual planning, we safely performed a combined LeFort III/I midface advancement to correct a negative overjet to improve occlusal balance, decrease scleral show, and diminish daytime sleepiness.

A novel nonsense substitution identified in the AMIGO2 gene in an Occulo-Auriculo-Vertebral spectrum patient.

OBJECTIVE: Craniofacial microsmia is the second most common congenital disorder with mostly unilateral defects of ear, temporomandibular joint, mandible, and muscles of facial expression and mastication. The objective of this study was to identify, if there were any, de novo germline or somatic variants in a patient with Occulo-Auriculo-Vertebral Spectrum (OAVS) using whole-exome sequencing. SETTINGS AND SAMPLE POPULATION: Trio/Family-based study of an OAVS proband. MATERIALS AND METHODS: Children's Mercy Hospital Institutional Review Board approved this study and a request-to-rely was procured from the University of Missouri Kansas City IRB. Informed assent/consent was obtained for all family members prior to any research activities. The peripheral blood/affected side tissues from corrective surgery of the proband and peripheral blood samples from unaffected parents were collected. The isolated genomic DNA were enriched for exomes and sequenced on an Illlumina HiSeq 2500 instrument yielding paired-end 125 nucleotide reads (84X coverage). Gapped alignment to reference sequences (GRCh37.p5) was performed with BWA and the GATK and analysis completed using custom-developed software. RESULTS: Analyses revealed that the proband carried a de novo germ line nonsense substitution (c.901C>T) in AMIGO2 gene, and missense substitutions in ZCCHC14 (c.1198C>T), and in SZT2 genes (c.2951C>T). CONCLUSIONS: The nonsense substitution in AMIGO2 gene introduces a premature stop codon possibly rendering the gene non-functional via nonsense-mediated pathway decay-therefore considered a stronger candidate. Further functional studies are required to confirm whether loss-of-function variants in AMIGO2 can cause OAVS.

A Programmable Expander for Patients With Cleft Lip and Palate.

This report describes a method to expand cleft segments of patients with cleft lip and palate prior to the secondary alveolar bone graft (ABG) using a reprogrammable nickel titanium expander. The expander can be reprogrammed with electric current to the desired width for each patient and delivers a gradual slow expansive spring force that expands and aligns the segments without further adjustments. Reprogramming the expander eliminates the need for multiple appliances and reduces chair time and patient appointments. The expander can be left in place during the ABG, because it does not interfere with a palatal flap. This alternative approach may be useful for patients who travel long distances to obtain cleft-related care.

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.

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).

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.

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.

Intercenter comparison of slow and rapid maxillary expansion in unilateral complete cleft lip and palate.

OBJECTIVE: The aim of this study was to compare the occlusal changes of rapid maxillary expansion (RME) and slow maxillary expansion (SME) in patients with unilateral complete cleft lip and palate (UCLP), by means of digital dental models. METHODS: Group RME was composed by 22 patients (13 males and 9 females), with mean age of 9.9 years, treated with rapid maxillary expansion with Hyrax appliance in Center 1. Group SME was composed by 29 patients (14 females and 15 males), with mean age of 10.7 years, treated with slow maxillary expansion with quad-helix appliance in Center 2. Digital dental models of the maxillary dental arch were obtained immediately pre-expansion (T1) and 6-month post-expansion (T2). Transversal distances, arch perimeter, arch length, palatal depth, palatal volume and posterior tooth inclination were digitally measured. Interphase and intergroup comparisons were performed with paired t-test and independent t-test, respectively. RESULTS: Intercanine expansion was 4 to 5mm in both groups, and increase in the intercanine distance was similar for both groups. RME group showed a greater increase in arch distances at the region of permanent premolar and molars, compared to SME group. Arch perimeter increase was greater for RME group, compared to SME. No differences were found for arch length, palatal depth, palatal volume and posterior tooth buccal tipping. CONCLUSION: RME and SME produced similar dentoalveolar outcomes, with greater amount of expansion on RME group.

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.

Cleft Lip and Palate: Le Fort I Distraction Using an Internal Device.

Maxillary hypoplasia is a commonly seen dentofacial anomaly in patients with cleft lip and palate after surgical repair of the cleft anomaly. To facilitate large horizontal movements of the maxilla, distraction osteogenesis is used to slowly stretch the soft tissue envelope with the skeletal advancement preventing tissue recoil and skeletal relapse. Internal distraction devices have the advantage of reducing the amount of physical and psychological stress placed on patients, families, and caregivers. The technique has been successful in producing stable results for large advancements of the facial skeleton when compared with conventional Le Fort I advancement and fixation of the maxilla.

Orthodontic and Surgical Principles for Distraction Osteogenesis in Children with Pierre-Robin Sequence.

Patients with Pierre-Robin sequence recalcitrant to nonsurgical intervention have historically required tracheostomy. Mandibular distraction provides a predictable alternative to tracheostomy. Orthodontic perioperative interventions should be considered, including overcorrection, placement of temporary anchorage devices, elastics, and molding the regenerate. Mandibular distraction can be technically difficult and may cause complications. Performed correctly, mandibular distraction provides patients with a better quality of life than tracheostomy.

Orthodontic Considerations for Cleft Orthognathic Surgery.

Preparation and planning for orthognathic surgery in late adolescence depends on the complexity of unresolved problems with which the patient presents. Different strategies are presented to address these unresolved problems in the adult patient with cleft lip and palate. Different surgical and orthodontic treatments are presented to correct the class III malocclusion in patients with cleft lip and palate in ranges that are analogous to the envelope of discrepancy. For complex cases, the principles of achievability, stability, and esthetics should guide the decision-making process for planning the preparation for orthognathic surgery.

Implant-supported distraction osteogenesis: a technique to advance the deficient maxilla.

We describe a technique in which osseointegrated implants are used as anchors to support a custom-fabricated distraction device to advance the deficient anterior maxilla. The absolute rigidity of osseointegrated implants in bone provides stable anchorage for successful distraction.

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.