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Patients with bilateral complete cleft lip and palate (BCLP) may present a vertical excess of the premaxilla in childhood. This is a severe functional and aesthetic problem, where bone grafting is more challenging. The aim of this study was to describe a simple and reproducible non-surgical orthopaedic treatment for vertical excess of the premaxilla in the deciduous/early mixed dentition phase in BCLP patients. Six growing patients with complete BCLP with a severe vertical excess of the premaxilla were included. An intrusion device associated with a bonded rapid palatal expander was applied to intrude the premaxilla. Radiographic and photographic records obtained before and at the end of the orthodontic intrusion, at short- and long-term follow-up, were available. A flattening of the occlusal plane was achieved in all patients. Normalization of the position of the maxillary incisors and gingival display in relation to the upper lip was obtained, and an improvement in anterior nasal spine position was also observed in all cases. The novel technique described might be of assistance in treating BCLP children with vertical excess of the premaxilla during the deciduous/early mixed dentition phase. This simplified, easily reproducible method may allow the burden of care of this rare but complex problem affecting BCLP patients to be reduced significantly.

Aim This study compares long-term mandibular growth between a group of Beckwith-Wiedemann Syndrome (BWS) patients who underwent glossectomy at an early age and a group of patients not operated. Methods Cephalometric measurements were performed in BWS patients comparing the data obtained between a group of patients operated at an early age and a group of non-operated patients who declined surgery. Statistics included independent sample T-test. Results Twenty-four out of 78 BWS patients followed since birth completed longitudinal cephalometric x-rays at age 5, 10 and 15. Eighteen patients needed early surgery. Eleven families accepted glossectomy at 2.3 ± 1.3 years of age; seven declined surgery. No differences in mandibular growth were found between the two groups. Inclination of maxillary incisors results were statistically greater in the non-operated group (operated compared to the non-operated group: 103.58 ± 11.30 Vs 108.98 ± 12.47; p-value 0.0168 at 5; 107.06 ± 7.98 Vs 115.14 ± 7.05; p-value 0.0206 at 10; 109.80 ± 4.68 Vs 116.75 ± 5.28; p-value 0.0233 at 15). Conclusion Macroglossia has no role in the post-natal mandibular overgrowth in BWS and mandibular overgrowth is part of the syndrome. Therefore, early glossectomy does not change mandibular growth and does not prevent the development of class III skeletal malocclusion in these patients.

Objective: To evaluate patency of circummaxillary sutures in children with Apert, Crouzon, and Pfeiffer Syndromes and to compare it to a nonsyndromic matched control group. Design: Case–control study. Setting: Tertiary care public hospital. Materials and Methods: Thirty-eight computed tomography (CT) scans of patients affected by syndromic craniofacial synostosis (13 patients with Apert syndrome, 20 patients with Crouzon syndrome, and 5 patients with Pfeiffer syndrome), average age 5 ± 2.8 years, range 1.9 to 12 years, were compared to age- and sex-matched control CTs of 38 nonsyndromic children. Computed tomography scans of the study group had to be performed prior to any midfacial surgery. Main Outcome Measures: Midpalatal suture, zygomaticomaxillary sutures, and pterigomaxillary sutures were evaluated and scored. Results: The syndromic group showed a significant earlier ossification of all sutures compared to the nonsyndromic group. Significant differences were already present in early childhood and continued through adolescence. Conclusions: Based on the differences in terms of maxillary sutural ossification identified, midfacial hypoplasia does not seem to be only secondary to premature cranial base ossification, but also to primary synostosis of facial sutures, thus providing new insights into the pathogenesis of midface deficiency in children with craniofacial-synostosis. Care should be taken when planning any maxillary orthopedics, such as expansion or maxillary protraction, given the high frequency of early fusion of circummaxillary sutures.

Objective Describe the first hybrid global simulation-based comprehensive cleft care workshop, evaluate impact on participants, and compare experiences based on in-person versus virtual attendance. Design Cross-sectional survey-based evaluation. Setting International comprehensive cleft care workshop. Participants Total of 489 participants. Interventions Three-day simulation-based hybrid comprehensive cleft care workshop. Main Outcome Measures Participant demographic data, perceived barriers and interventions needed for global comprehensive cleft care delivery, participant workshop satisfaction, and perceived short-term impact on practice stratified by in-person versus virtual attendance. Results The workshop included 489 participants from 5 continents. The response rate was 39.9%. Participants perceived financial factors (30.3%) the most significant barrier and improvement in training (39.8%) as the most important intervention to overcome barriers facing cleft care delivery in low to middle-income countries. All participants reported a high level of satisfaction with the workshop and a strong positive perceived short-term impact on their practice. Importantly, while this was true for both in-person and virtual attendees, in-person attendees reported a significantly higher satisfaction with the workshop (28.63 ± 3.08 vs 27.63 ± 3.93; P = .04) and perceived impact on their clinical practice (22.37 ± 3.42 vs 21.02 ± 3.45 P = .01). Conclusion Hybrid simulation-based educational comprehensive cleft care workshops are overall well received by participants and have a positive perceived impact on their clinical practices. In-person attendance is associated with significantly higher satisfaction and perceived impact on practice. Considering that financial and health constraints may limit live meeting attendance, future efforts will focus on making in-person and virtual attendance more comparable.

A short columella and a flattened nasal tip are the characteristic stigmata of patients with complete bilateral cleft lip and palate (BCLP). Objective: The aim of this study was to assess the nasal shape of young adults with BCLP treated with primary surgical columella lengthening and nasoalveolar molding (NAM). Setting and Patients: A group of 28 young adult patients with BCLP (mean age: 19.1±1.4 years) was compared through normalized photogrammetry to a control of 28 age- and sex-matched noncleft young adults. Results: Nasal protrusion and length of the columella were not different from noncleft young adults. On the other hand, nasolabial angle, columellar width, interalar, and nasal tip width were significantly wider than the noncleft controls. Thus, 27% of the patients have requested at this time secondary correction of the excessive nasal width. Conclusions: Both NAM and primary rhinoplasty in patients with BCLP resulted in a near normal length of the columella and nasal projection until young adulthood. Nevertheless, width of all nasal features was significantly wider than the noncleft population and required secondary nasal correction in one-third of the sample.

Background: Closure of wide alveolar clefts with large soft tissue gaps and reconstruction of the dentoalveolar defect are challenging for the surgeon. Some authors successfully used interdental segmental distraction, which requires an additional surgical procedure. Objective: This study evaluates the effectiveness of tooth borne devices utilized to orthopedically advance the lesser segments, with a complete approximation of the soft tissue of the alveolar stumps, allowing traditional simultaneous soft tissue closure and bone grafting, and avoiding the need for supplementary surgery. Methods: Eight growing patients, 2 with unilateral complete cleft lip and palate (UCLP) and 6 with bilateral complete cleft lip and palate (BCLP), with large soft tissue and bony alveolar defects prior to bone grafting were prospectively selected. A banded rapid palatal expander (RPE) in BCLP and a modified RPE in UCLP combined with protraction face mask in younger patients or a modified Alt-Ramec in patients older than 12 years were applied. Radiographic and photographic records were available at T0, at the end of protraction (T1) and at least 1 year after bone grafting (T2). Results: Patients with large gaps showed a significant reduction in the bony cleft area and approximation of the soft tissues at T1. All patients received bone grafting with good healing and ossification at T2. Conclusion: In growing patients with UCLP and BCLP with large gaps, presurgical orthodontic protraction seems to be an efficient method to reduce the cleft defect, minimizing the risk of post grafting fistulas, reducing the need for supplementary surgical procedures.

Objective: The objective of this retrospective longitudinal study was to evaluate short- and long-term results of the application of the Liou Alt-RAMEC (alternate rapid maxillary expansion and constriction) technique, a late orthopedic maxillary protraction technique, with intraoral anchorage, in patients with cleft. Materials and Methods: Twenty-six patients with unilateral cleft lip and palate (UCLP) were consecutively treated with the Alt-RAMEC technique. The average age of the patients was 11.7 years (10.3-13.2 years) before protraction and 18.3 years (17.4-21.1 years) at long-term follow-up. A sample of nontreated patients with UCLP was used as a control group. It was matched for sex, skeletal class III, and age (11.3 years). The control sample had records at the end of growth (18.7 years). Results: The sagittal advancement of A-point, after the application of the technique, was 5.7 (2.17) mm. Some mandibular dentoalveolar and positional adaptation was noted. The position of the maxilla was stable in the long term. On the other hand, the UCLP control group showed hardly any growth at the maxillary level during the long-term follow-up period. Conclusion: Our results showed that the Alt-RAMEC technique, performed at the correct time, with a double-hinged expander, followed by class III spring or elastic traction, 24 hours per day, allows for satisfactory maxillary protraction, with, at this stage, apparently stable long-term results. Nevertheless, as only 50% of the patients had long-term follow-up data, we are still unable to predict the percentage of patients which will not eventually need orthognathic surgery.

Objective: Nasal stigma in patients with bilateral cleft lip and palate (BCLP) are a short columella and a flattened nasal tip. Design: The aim of this study was to evaluate the aesthetics of adolescents with BCLP, operated with a modified Cutting primary columella lengthening technique, associated to a modified Grayson orthopedic nasoalveolar molding (NAM). Setting and Patients: 72 BCLP patients were operated with this approach. Standardized photographic records were taken every 2 years. A group of 23 patients between 12 and 13 years of age was compared through normalized photogrammetry to a matched control of 23 noncleft adolescents. Results: Nasal protrusion and length of the columella were very close to normal. On the other hand, nasolabial angle and interalar width were still excessively wide compared to the noncleft sample. Conclusions: NAM and primary columella lengthening in BCLP has allowed to avoid traditional secondary columella lengthening at 5 to 6 years of age and given the patients a more pleasing, near-normal nasolabial appearance until adolescence. Some of the patients will require correction of the nasal width at a later stage.

Introduction: The objective of this prospective study was to compare the difference in pain perception between treatment with aligners (AL) and fixed appliances (FA) in patients affected by cleft and craniofacial anomalies (CFA). Methods: The sample consisted of 100 syndromic caucasian patients affected by various CFA from 2 different hospitals. Fifty patients treated with AL were matched for sex, age, and CFA with a control sample of 50 patients treated with FA. A modification of the Mc Gill Pain Questionnaire was adapted to our needs. Results: Statistical differences were found. Aligners induced more tightness and tension than FA, while FA induced more pain descriptors and patients reported a higher intake of painkillers. Conclusions: The results of this study documented a higher pain perception with FA than with AL in patients affected by CFA. The higher sensitivity to pain in cleft and craniofacial patients with fixed treatment could be related to their higher prior sensitization, given the past surgeries and orthodontic treatments. Thus, this study might suggest that Invisalign treatment might be a further interesting treatment option for patients with cleft in order to reduce their burden of orthodontic pain.

Introduction Cleft lip and palate (CLP) surgery interferes with maxillary growth and contributes to transversal and sagittal growth impairment. Our retrospective descriptive study aimed to evaluate maxillary bone shape in a homogenous unilateral CLP patient group using geometric morphometrics based on CT-scan data. Material and methods We included all patients with available CT-scans at the end of pubertal growth and operated on at Smile House of Milan, according to the standard protocol, involving two surgical steps: (1) primary closure of the lip and soft palate at 6 months of age and (2) early secondary gingivoalveoloplasty (GAP) associated with hard palate repair at 18–36 months. Shape differences between CLP and an age-matched control group were characterized using geometric morphometrics based on 15 3D landmarks. Results We included 16 unilateral CLP patients and 20 age-matched controls. Principal component and canonical variate analyses showed that the maxillary shape in CLP was significantly different from controls but that this difference was limited. Linear and angular measurements confirmed these differences. Conclusion Early secondary GAP results in satisfactory maxillary shape, with significant but limited differences relative to controls.

Objective The goal of this study was to evaluate the craniofacial morphology at 5 and 10 years of age and at the completion of growth, the need for final orthognathic surgery, and the orthodontic burden in a sample of patients with unilateral cleft lip and palate consecutively treated by the same surgeon with the same two-step protocol. Design A sample of 62 adult patients with unilateral cleft lip and palate was retrospectively collected (mean age, 17.5 years). Lateral cephalograms at three time points were traced. The need for orthognathic surgery was assessed, subdividing the sample into an orthognathic surgery group and nonorthognathic surgery group. Time and modality of orthodontic treatment were recorded. Results Cephalometric values related to maxillary growth (SNA, SNAns) and maxillomandibular relation (ANB, NAPg) were significantly different between the two groups already at 5 and 10 years of age. All patients presenting an ANB smaller than 2° at 5 years needed a Le Fort I osteotomy. Mandibular protrusion (SNB, SNPg) was not different at 5 and 10 years, but was different at the completion of growth. Patients with the same initial maxillomandibular relation did not show better growth when subjected to earlier or longer orthodontic treatment. Conclusion Patients needing final jaw surgery had a more severe skeletal discrepancy during early childhood. The ANB angle at 5 years allowed doctors to identify 45% of the need for orthognathic surgery. The final craniofacial pattern does not seem to change significantly with early or prolonged orthodontic treatment.

Introduction This article proposes an innovative and revolutionary diagnostic and therapeutic protocol for performing dentoalveolar osteotomies in office under local anaesthesia with piezoelectric surgery using a surgical acrylic guide produced through software-based planning. Methods The method was applied in the correction of crossbites, changing in the curve of Spee, incisal decompensations and dental ankylosis. Performing a preoperative CT with a special splint, optical scanning of the models and the subsequent planning with software has enabled us to produce a model with rapid prototyping with the design of the osteotomy on which the surgical guide was shaped, the use of the guide associated with piezoelectric surgery, allowed to perform surgery under local anaesthesia, with minimal invasiveness and high accuracy. Results Dentoalveolar immediate movements, with preservation of the roots of teeth involved, allow for rapid treatment of malocclusions which would be long and often difficult if not impossible to treat with orthodontics only. Dentoalveolar osteotomies associated to osteodistraction concepts, allow the orthodontist to achieve with accuracy the objectives required by the treatment plan. Conclusions GSOS is a new method, which, utilizing 3D optical scanning images of models, software and piezoelectric surgery, allows to perform dentoalveolar movements which may be dangerous to the roots or for the periodontal support, with orthodontics only. It dramatically reduces total surgical-orthodontic treatment time, with obvious great patient satisfaction.

Aim: Comparison of the long-term follow-up until the completion of growth of two homogeneous samples of children affected by hemifacial microsomia (HFM), one treated by mandibular distraction osteogenesis (DO) in the deciduous or early mixed dentition, the other not subjected to any treatment until adulthood. Material: Fourteen patients affected by vertically severe type I or II HFM were operated at an average age of 5.9 years with an average follow-up of 11.2 years. They were compared to a sample of eight patients who were never treated until the completion of growth. Methods: Mandibular vertical changes were measured on panoramic radiographs taken at different time points. Ratios between affected and non affected ramal heights were calculated and compared. Results: In the DO sample, after correction, mandibular vertical changes showed a gradual return of the asymmetry with growth in all patients. The ratio in the non treated sample was unchanged between the initial and the long term panoramic x-rays. Conclusion: The facial proportions of HFM patients are maintained, when not treated, throughout growth. The same proportions return to their original asymmetry after DO. Even though short term aesthetic and psychological advantages of distraction osteogenesis are well accepted, early surgery should only be applied after careful patient selection and honest clarification of the long term recurrence by genetically guided craniofacial growth pattern. © 2011 European Association for Cranio-Maxillo-Facial Surgery.

Long-term results after surgical treatment of the mandibular asymmetry in growing children with hemifacial microsomia (HFM), whether with osteotomies or distraction osteogenesis, have mostly shown a tendency towards the recurrence of the asymmetry. In contrast, in the literature we find sporadic case reports where the long-term post-surgical follow-up of patients diagnosed as HFM, are surprisingly stable. All these reports refer to patients who have substantially no soft tissue involvement, but only severe mandibular ramus and condyle deformities. The phenotypes of these cases are unexpectedly similar. The authors suggest, that it is possible that all of these cases might be isolated hemimandibular hypoplasias, misdiagnosed as HFM, which present a normal functional matrix and, therefore, tend to grow towards the original symmetry. Differential diagnosis between true HFM and this HFM-like isolated hemimandibular hypoplasia (pseudo-HFM) is of great importance given the very different prognosis and it is possible through the collaboration between not only surgeons and orthodontists, but also of geneticists and dysmorphologists.

Objective: To compare the short- and long-term craniofacial growth of patients operated with the Milan protocol to those operated with the Oslo protocol. Design: The Milan sample included 88 patients with unilateral cleft lip and palate (UCLP) at 5 years, 26 at 10 years, and 23 at the end of growth. The Oslo samples included 48 UCLP patients at 5 years, 29 at 10, and 23 at growth completion. Lateral cephalograms were used for comparison. An unpaired t test was run for the 5- and 10-year-old samples. The samples long term were matched for age and sex, and a paired t test was run. Results: There was no significant cephalometric difference in the maxillary prominence at 5 years, a mild but significant difference at 10 years, and again no difference at the end of growth. Nevertheless, at an older age, the need for orthognathic surgery was larger in the Milan sample (26%) than in the Oslo sample (13%). Conclusion: Although no statistically significant differences in the cephalometric measurements were found long term, the need for orthognathic surgery was clinically judged to be larger in the Milan sample. On the other hand, although the Milan protocol seemed to require more final jaw surgery, only the cases that needed an additional orthognathic procedure in the Milan group will undertake a third surgical step, while the Oslo protocol included three surgical steps for all the patients.

Objective:To evaluate the dental characteristics of patients subjected to a protocol that included early secondary gingivoalveoloplasty (ESGAP).Design:Panoramic radiographs of 87 patients with unilateral cleft lip and palate (UCLP) and 29 with bilateral cleft lip and palate (BCLP) were evaluated. Missing and supernumerary teeth were also quantified on the cleft and noncleft side and in the maxilla and mandible. Crown and root malformations and tooth rotations were quantified. A subsample in permanent dentition was extrapolated to analyze canine eruption patterns.Results:A total of 48.8% of the UCLP patients presented with missing permanent lateral incisors in the cleft area and 6.1% contralaterally. A total of 4.9% presented with missing second maxillary premolars on the cleft site and 1.2% contralaterally. A total of 7.3% presented with supernumerary lateral incisors, and 45% of the BCLP cleft sites presented with missing lateral incisors, while 25% of the cleft sites presented second maxillary premolars agenesis. Five percent of the cleft sites presented with supernumerary lateral incisors. Evaluation of the subsample in permanent dentition showed that 15.5% had a canine retention and 4.4% of the canines had to be surgically exposed. A significant association was observed between canine inclination and retention but not with absence of the lateral incisor.Conclusions:The frequency of dental anomalies in this sample was similar to other cleft populations. As surgical trauma has been suggested to damage forming teeth, the results of this study indicated that ESGAP has no detrimental influence on subsequent dental development.

The literature contains a number of case reports on successful orthopedic treatment of hemifacial microsomia (HFM), with surprising changes in the morphology of the condyles. All of these reports regard patients who have substantially no soft tissue involvement but only severe mandibular ramus and condyle deformities. A number of cases with unexpectedly similar phenotypes diagnosed as HFM are described. The authors suggest that it is possible that all of these cases might be misdiagnosed traumatic injuries of the condyle, which present a normal functional matrix and, therefore, with growth and with the help of functional stimulation, tend to grow toward the original symmetry.

Summary Aim After mandibular unilateral distraction osteogenesis (DO) a gradual reappearance of the vertical asymmetry during growth is observed. A pre- and post-surgical functional-orthodontic treatment was added to our distraction protocol in the attempt to increase long-term stability. In order to evaluate the actual efficacy of such a combined treatment, two samples of children affected by hemifacial microsomia were compared long-term. Material Ten children were treated by a combined orthodontic–distraction treatment, seven by distraction only. Method Only the vertical changes in the mandible and maxilla in the panoramic and postero-anterior cephalometric X-rays were measured. Results All of the patients showed a gradual return of the asymmetry with growth. Occlusal plane correction and, to a much lesser extent, mandibular vertical ramus height correction were better maintained over 5 years post-DO in the orthopaedic group. Conclusion Although orthopaedic treatment allows for a more stable occlusal plane and for a slower return of the mandibular vertical asymmetry, it has mainly a dento-alveolar effect. Therefore, the decision of applying an orthopaedic treatment associated with distraction, should be taken by surgeon and orthodontist together, considering both the advantages and the disadvantages of this treatment.

Summary Aim Prospective, longitudinal, clinical long-term follow-up study of a homogeneous sample of children affected by hemifacial microsomia and treated by mandibular distraction osteogenesis. Material Eight patients affected by types I and II hemifacial microsomia were operated on at an average age of 5.6 years with an average follow-up of 5.8 years. Methods Vertical changes were measured on postero-anterior cephalometric and panoramic radiographs taken sequentially. Results Angular changes of the infraorbital and nasal floor planes were not significant, showing that distraction osteogenesis starting after 5 years of age did not influence the maxillary skeletal base. Occlusal (plane) cant was reduced by 7° on average following distraction osteogenesis, showing good dentoalveolar plasticity. Mandibular vertical changes showed a gradual return of the asymmetry, with growth in all patients (the ratio between affected and non-affected rami returned by 77% of the correction obtained by means of distraction 5 years postoperatively). Conclusion Although aesthetic and psychological advantages of distraction osteogenesis are well accepted it should only be applied after careful patient selection and honest explanation of the long-term recurrence by genetically determined craniofacial growth patterns.

Objectives: We evaluated the correlation between the skeletal facial advancement through Le Fort III osteotomy and the modification of polysomnographic values in syndromic patients with sleep respiratory disorders affected by craniofacial synostosis. Methods: We selected a sample of 17 patients affected bymild to severe OSAS. All patients underwent a Le Fort III osteotomy. In five cases we used the “classical technique” and in 12 cases distraction osteogenesis. Sleep respiratory disorders were evaluated by pre-op and post-op polysomnography. All patients had pre-op and post-op cephalometric records. Results: The average midface advancement was 16mm (10–28mm)measured at A point. The average pre-op polysomnographic records were: ODI 28.6 (8–75), AHI 23.2 (8–50). The average post-op polysomnographic records were: ODI 3.88 (0.3–7), AHI 4.14 (0.2–3). In one tracheostomized patient we removed the tracheostomy and one patient with CPAP stopped the treatment. Conclusions: The respiratory disorders in syndromic patients with midfacial hypoplasia have different clinical manifestation: from snoring to acute respiratory distress requiring tracheostomy. Our study demonstrates an improvement of polysomnographic values in all patients, with a shift from severe to mild OSAS or even a complete resolution.