Reverse Engineering Orthognathic Surgery and Orthodontics in Individuals with Cleft Lip and/or Palate: A Case Report.

This case report presents a virtual treatment simulation of the orthodontic treatment and surgery-first orthognathic surgery employed to treat a patient with a repaired unilateral cleft lip and alveolus with Class III malocclusion and lower third facial asymmetry. The patient exhibited a negative overjet of 9 mm, a missing lower right second premolar, and a 5 mm gap between the upper right central and lateral incisors with midline discrepancy. The three-dimensional virtual planning began with virtual pre-surgical orthodontics, followed by the positioning of the facial bones and teeth in their ideal aesthetic and functional positions. The sequence of steps needed to achieve this outcome was then reverse-engineered and recorded using multiplatform Nemostudio software (Nemotec, Madrid, Spain), which facilitated both surgical and orthodontic planning. The treatment included a two-piece segmental maxillary osteotomy for dental space closure, a LeFort I maxillary advancement, and a mandibular setback with bilateral sagittal split osteotomy to correct the skeletal underbite and asymmetry. A novel approach was employed by pre-treating the patient for orthognathic surgeries at age 11, seven years prior to the surgery. This early phase of orthodontic treatment aligned the patient's teeth and established the dental arch form. The positions of the teeth were maintained with retainers, eliminating the need for pre-surgical orthodontics later. This early phase of treatment significantly reduced the treatment time. The use of software to predict all the necessary steps for surgery and post-surgical orthodontic tooth movements made this approach possible. Multi-step virtual planning can be a powerful tool for analyzing complex craniofacial problems that require multidisciplinary care, such as cleft lip and/or palate.

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.

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.

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

ABSTRACT 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.

RESUMO Objetivo: O objetivo do presente estudo foi comparar, por meio de modelos digitais, as alterações oclusais após a expansão rápida da maxila (ERM) e após a expansão lenta da maxila (ELM) em pacientes com fissura labiopalatina completa unilateral (FLPU). Métodos: O grupo ERM foi composto por 22 pacientes (13 homens e 9 mulheres), com idade média de 9,9 anos, tratados com expansão rápida da maxila com aparelho Hyrax no Centro 1. O grupo ELM foi composto por 29 pacientes (14 mulheres e 15 homens), com idade média de 10,7 anos, tratados com expansão lenta da maxila com aparelho quadri-hélice no Centro 2. Modelos digitais da arcada dentária superior foram obtidos imediatamente pré-expansão (T1) e seis meses pós-expansão (T2). Distâncias transversais, perímetro da arcada, comprimento da arcada, profundidade do palato, volume do palato e inclinação dos dentes posteriores foram medidos digitalmente. As comparações interfases e intergrupos foram realizadas com teste t pareado e teste t independente, respectivamente. Resultados: A expansão da arcada na região dos caninos foi de 4 a 5 mm em ambos os grupos, e o aumento na distância intercaninos foi semelhante nos dois grupos. O grupo ERM apresentou maior aumento nas distâncias entre pré-molares e molares permanentes, em comparação ao grupo ELM. O aumento do perímetro da arcada foi maior para o grupo ERM, em relação ao ELM. Não foram encontradas diferenças para comprimento da arcada, profundidade do palato, volume do palato e inclinação vestibular dos dentes posteriores. Conclusão: ERM e ELM produziram resultados dentoalveolares semelhantes, com maior quantidade de expansão no grupo ERM.

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.

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.

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.

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.

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.

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.

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 Comparative Analysis of Recombinant Human Bone Morphogenetic Protein-2 with a Demineralized Bone Matrix versus Iliac Crest Bone Graft for Secondary Alveolar Bone Grafts in Patients with Cleft Lip and Palate: Review of 501 Cases.

BACKGROUND: Alveolar cleft reconstruction using iliac crest bone graft is considered standard of care for children with complete cleft lip and palate at the time of mixed dentition. Harvesting bone may result in donor-site morbidity and additional operating time and length of hospitalization. Recombinant human bone morphogenetic protein (rhBMP)-2 with a demineralized bone matrix is an alternative bone source for alveolar cleft reconstruction. The authors investigated the outcomes of rhBMP-2/demineralized bone matrix versus iliac crest bone graft for alveolar cleft reconstruction by reviewing postoperative surgical complications and cleft closure. METHODS: A retrospective chart review was conducted for 258 rhBMP-2/demineralized bone matrix procedures (mean follow-up, 2.9 years) and 243 iliac crest bone graft procedures (mean follow-up, 4.1 years) on 414 patients over a 12-year period. The authors compared complications, canine eruption, and alveolar cleft closure between the two groups. RESULTS: In the rhBMP-2/demineralized bone matrix group, one patient required prolonged intubation because of intraoperative airway swelling not thought to be caused by rhBMP-2, 36 reported facial swelling and one required outpatient steroids as treatment, and 12 had dehiscence; however, half of these complications resolved without intervention. Twenty-three of the 228 rhBMP-2/demineralized bone matrix patients and 28 of the 242 iliac crest bone graft patients required repeated surgery for alveolar cleft repair. Findings for canine tooth eruption into the cleft site through the graft were similar between the groups. CONCLUSIONS: The rhBMP-2/demineralized bone matrix appears to be an acceptable alternative for alveolar cleft repair. The authors found no increase in serious adverse events with the use of this material. Local complications, such as swelling and minor wound dehiscence, predominantly improved without intervention. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

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.