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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.

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.

The aim of this paper was to assess growth, speech, and aesthetic results at the completion of growth in patients with unilateral cleft lip and palate treated with the 2 stages Milan surgical protocol.Craniofacial growth was evaluated with cephalometric analysis and a theoretical need for orthognathic surgery.Nasolabial appearance was qualitatively assessed using the Asher McDade Aesthetic Index.Speech was assessed using the Gos.Sp.Ass '98 modified for Italian language scoring system.Burden of care was recorded in terms of number of secondary surgical procedures. All of the patients were treated and evaluated at San Paolo Hospital, Smile House, Milan.Fifty-two consecutive patients treated by the same surgeon were recalled, 12 patients did not come for assessment.The first surgical step (average age of 6 months) was cheilorhinoplasty (Millard modified Delaire technique) and soft palate rapair (Pigott). The second step (average age of 35 months) was hard palate and alveolar repair performed simultaneously with an early secondary gengivo alveolo plasty. Fifty-six percent of the patients did not need further surgery after the 2-stage surgery protocol.The 2-stage surgical protocol of Milano, Smile House, seems to be effective for treatment of unilateral cleft lip and palate, with good results in terms of speech, labial appearance, and alveolar cleft management. Nevertheless, maxillary growth was moderately impaired by the protocol.

Background: Ocular protrusion is usually measured as the deepestpart of the lateral orbital rim to the apex of the cornea. Severalstudies have been carried out to propose normative data for ocularprotrusion according to different age ranges. The protrusion of theeyeball seemed to increase during childhood and adolescence andbecome stable after the late teens during adulthood. Relationshipsof ocular protrusion with age are still not completely clarified. Thepurpose of the current retrospective study was to investigate ocularprotrusion on lateral x-rays at different growth stages.Methods: Ninety-five patients were retrospectively selected anddivided into 5 different groups according to ages. Subjects were ofboth sexes with an age range from 5 to 20 years (mean 12.3 years).Thecephalometricanalysisproposedinthecurrentstudyallowedtosuccessfully evaluate sagittal relationships of the orbital-globe andthe orbital-cavity ingrowing patients. Allother sagittal andverticalmeasurementsweresuccessfullycomputedbyonetrainedoperator.Results: Analysis of variance results with post hoc correctionsshowed for the orbital measurements indicating a significantincrease of the orbital protrusion from childhood to pubertal stage.Conclusions: Ocular protrusion measurements showed significantincrease from childhood (5–8 years) to teen age (13–15 years) andremainedstableafterthepubertalgrowthspurt(17–20years)intheexamined sample of patients.Key Words: Orbital growth, orbital-globe protrusion,development, aging(J Craniofac Surg 2015;26: 760–763)

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.

Background: The goal of this study was to evaluate with a three-dimensional method the long-term quality of alveolar ossification in unilateral cleft lip and palate patients who underwent early secondary gingivoalveoloplasty according to the Milan surgical protocol. Methods: The sample consisted of 63 computed tomographic scans of unilateral cleft lip and palate patients in permanent dentition. The average age at the time of assessment was 15.7 years. Alveolar thickness, nasoalveolar height, nasal floor ossification, and hard palate morphology were evaluated using dental, axial, and coronal cuts on computed tomographic scans and three-dimensional models. All measurements were normalized and ratios of the affected side versus the nonaffected side were provided. Volume measurements and ratios of each hemimaxilla were added. The presence or absence of the permanent lateral incisor on the cleft side was also recorded. Results: Alveolar thickness and height were ideal or good, respectively, in 89.5 and 91.4 percent of the sample. Insufficient ossification (

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.

The Milan surgical protocol includes the use of an early secondary gingivoalveoloplasty together with hard palate closure at 18 to 36 months, to avoid later bone grafting. The goal of this study was to evaluate the long-term quality of ossification in patients who have undergone early secondary gingivoalveoloplasty.The samples consisted of panoramic radiographs of 87 unilateral cleft lip-cleft palate and 29 bilateral cleft lip-cleft palate patients. The records available allowed for a longitudinal and a cross-sectional evaluation of the ossification in the cleft area. Alveolar bridging was assessed using a modified Bergland's scoring system. Nasal area ossification and canine inclination were each given three different qualitative scores.The alveolar bridging noted was type I (71.7 percent), type II (23.5 percent), and type III (4.8 percent) in the whole sample of unilateral and bilateral cleft lip-cleft palate patients. No type IV ossification was found. Longitudinal analysis showed that approximately one-fourth of the cleft sites improved after permanent tooth eruption, and very few worsened. An evaluation of permanent dentition in a group of 27 unilateral and nine bilateral cleft lip-cleft palate patients (mean age, 14.8 +/- 2.0 years) showed that 15.5 percent of the whole sample had canine retention and 4.4 percent of the whole sample had to be surgically exposed.Early secondary gingivoalveoloplasty seems to allow for adequate ossification in both the alveolar and the nasal regions. Permanent tooth eruption occurs at a normal rate. None of the patients has required a secondary alveolar bone graft.

Summary Background Maxillary distraction osteogenesis (DO) in cleft lip and palate patients has been described by several authors, but most studies have a relatively short follow-up and do not clearly separate growing patients from non-growing patients. Method The records of 22 consecutive patients affected by cleft lip and palate, who underwent Le Fort I osteotomy and maxillary distraction with a rigid external distractor (RED), were reviewed. The sample was subdivided into a growing and a non-growing group. All patients had pre-DO cephalometric records, immediately post DO, 12 months post DO and long-term records with a long-term follow-up of >5 years (range 5–13 years). As a control sample for the growing group, cleft children with a negative overjet not subjected to distraction or any protraction treatment during growth were followed up until the completion of growth. Results The average maxillary advancement in the growing group was 22.2 ± 5.5 mm (range: 15–32 mm); in the non-growing group, it was 17.7 ± 6.6 mm (range: 6–25 mm). Excellent post-surgical stability was recorded in the adult sample. On the other hand, growing children had an average 16% relapse in the first year post DO and an additional 26% relapse in the long-term follow-up. Conclusions This study seems to point out that early Le Fort I DO allows for the correction of very severe deformities. It is followed by a relatively high amount of true skeletal relapse in children with cleft lip and palate. Prognosis should be discussed in depth with the family and true aesthetic and psychological needs assessed.

The sagittal relationships between the orbital globe and the ocular orbit have great importance in aesthetic and functional eval- uations. These relationships are useful in diagnosis and treatment of some malformations in which the orbital volume is reduced such as craniostenosis. The aim of the present study was the cephalomet- ric evaluation of the ocular protrusion at different growth stages. Ninety-two patients with negative history of craniosynostosis, severe myopia, and skeletal class I were selected for the present study. Lateral teleradiography x-ray was performed for all the patients, and 5 groups were carried out based on the dentition phase. Cephalometric analyses were performed on the lateral teleradiography x-ray. Mean values of ocular protrusion (B-Or post) for females and males were found and digitized for the 5 groups tested. All other sagittal and ver- tical measurements were successfully computed by the trained opera- tor. B-Or post showed values between �14.93 mm and �16.90 mm among the groups. The cephalometric analysis proposed in the pres- ent study allowed to successfully evaluate sagittal and vertical rela- tionship of the orbital globe and the orbital cavity in growing patients. Moreover, the B-Or post maintained its value during growth in the present study.

Background: Midface distraction osteogenesis (DO) in craniofacial synostosis (CFS) patients has been described by several authors. However, very few cephalometric and computed tomography (CT) long-term follow-up studies are available. Method: A total of 40 consecutive patients affected by CFS subjected to Le Fort III and rigid external distraction (RED) were examined. All patients had pre-DO cephalometric records, immediately post-DO and 6-12 months post-DO. Twenty-seven patients had mid-term records (3 years post-DO) and 20 patients had long-term records (5-10 years post-DO). Fourteen patients had CT data within 1-year of DO, while 10 patients had long-term CT data (range 5-9 years). Results: Excellent post-surgical stability was recorded. Short- and long-term CT data demonstrated excellent ossification at the osteotomy sites post-DO. In the growing patients, surface resorption in the zygomatic-temporal and in the subspinal area (p < 0.05) was observed in the long-term follow-up, as well as a mild increment of the corrected exorbitism (p < 0.05), as only appositional and no sutural growth occurs post Le Fort III, whereby orbital volume does not increase after surgery. Conclusion: Significant advancement of the midface can be achieved and maintained through Le Fort III and RED. In the long term, in growing patients, in general a class III malocclusion does not re-occur, but physiological remodelling processes at the maxillary-zygomatic level, not coupled with sutural growth, tend to mildly re-express the original midfacial phenotype and the exorbitism. © 2011 British Association of Plastic, Reconstructive and Aesthetic Surgeons. Published by Elsevier Ltd. All rights reserved.

No consensus exists about the causes of restriction of maxillary growth in patients with cleft lip and palate (CLP). The aim of this study was to try to identify causes of this impairment other than the influence of surgical technique and skill. We analysed a sample of 129 consecutively treated 5-year-old children with unilateral cleft lip and palate (UCLP), who were operated on by the same surgeon with the same protocol. Multiple cephalometric measurements of the sample showed a wide distribution of values for maxillary growth. We selected SNA as a value describing maxillary position. Variables investigated were: initial cast measurements; timing of lip and of gingivoalveoloplasty (GAP)/palatal surgery; and presence of permanent lateral incisors. The significance of differences was investigated with Pearson's correlation and analysis of variance (ANOVA). The factor most significantly linked with maxillary protrusion was the presence or absence of the permanent lateral incisor, even when peg laterals and supernumerary laterals were considered. Initial width of the palate measured on infant casts correlated with maxillary growth, but the timing of GAP did not. Although surgical skill and technique may be the most important factors responsible for impairment of maxillary growth, inherent tissue hypoplasia, possibly the lack of lateral incisors, seems to be the most important non-iatrogenic factor.

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.

The Milan surgical protocol from 1988 has included repair of lip, nose, and soft palate at 6-9 months of age, and closure of the hard palate and alveolus with an early secondary gingivoalveoloplasty at 18-36 months. The goal of this study was to evaluate the long-term maxillary growth in patients with unilateral cleft lip and palate (UCLP) who had had the early secondary gingivoalveoloplasty, compared with the growth in a sample treated before 1988, by the same surgeon, with a surgical protocol that differed only by the method and the timing of alveolar closure. In the second group they were repaired by secondary bone grafting. The samples consisted of lateral cephalograms of the UCLP early secondary gingivoalveoloplasty sample (15 patients with a mean age of 18 (1.2) years) and of the UCLP bone graft sample (10 patients with a mean age of 19 (1.1) years). The early secondary gingivoalveoloplasty patients showed that maxillary growth was inhibited compared with the secondary bone graft group. Although the early secondary gingivoalveoloplasty allowed for early repair of the alveolus together with palatal repair, eliminating the need for secondary bone grafting, it seemed to have an inhibiting influence on maxillary growth that increased the need for Le Fort I osteotomies. Even with a Le Fort I osteotomy, the early secondary gingivoalveoloplasty allows the total number of operations to be kept down to three, as in most European protocols.

No true consensus exists regarding the causes of maxillary growth restriction in cleft lip and palate patients. The aim of this study was to evaluate a simplified method to identify the causes of this growth impairment. We analyzed a sample of 82 consecutively treated 5-year-old patients with unilateral complete cleft lip and palate, operated on by the same surgeon (R.B.). Multiple cephalometric measurements of the sample revealed a wide distribution of maxillary growth values. We selected Sella-Nasion A point angle (SNA) as a value describing maxillary position, and we arbitrarily isolated 2 "extreme" groups of patients: the 20 patients with the highest SNA were called "good growers" (group A), and the 20 patients with the lowest SNA were called "poor growers" (group B). Parameters investigated to search for different influencing factors within the groups were initial cast measurements, timing of lip and of gingivoalveoloplasty/palatal surgery, presence of lateral incisors, and family history of maxillary hypoplasia. Statistical differences were investigated with an independent-samples t-test. The 2 extreme groups had a significant difference (P < 0.01) in SNA and in lip protrusion relative to true vertical line. Cranial base angle was larger in group A. Timing of lip surgery in group B was earlier than in group A, but only close to significance. Timing of gingivoalveoloplasty did not differ. In group A, the permanent lateral incisor was missing in 20% of the patients; whereas in group B, it was missing in 82% of the patients (P < 0.01). Selecting cleft lip and palate population with different growth pattern might help in isolating the factors responsible for maxillary growth impairment. Congenitally missing laterals that could indicate inherent tissue hypoplasia seems to be the most important factor.

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.

This is a longitudinal cephalometric study of patients with craniofacial synostosis (Crouzon's and Apert's) syndromes who underwent Le Fort III advancement during early childhood. The objectives of the study were to quantify presurgical growth, surgical results, and surgical stability as well as long-term postsurgical growth. Of the 31 patients operated on during their growth period (less than 10 years of age), 17 had sufficient records to be included in the study: at infancy, before the Le Fort III osteotomy (average age=7.3 years, range: 4.8-10 years), and immediately after surgery. Ten of the patients were followed radiographically long term for an average of 6.1+/-2.7 years (range: 4.5-10.8 years). The presurgical abnormal midfacial vertical growth pattern was accurately described with a progressive class III discrepancy and an increasing exophthalmos. Remarkable postoperative stability of the maxillary segment was observed. After surgery, growth of the midface was measured, together with minimal, if any, anterior growth, similar to the presurgical growth pattern and rate. No actual detrimental or beneficial effect of surgery on subsequent growth was seen. This study quantitatively confirms that the standard Le Fort III osteotomy is a stable and effective but generally not definitive procedure in childhood. This study might also serve as a control sample to compare with groups of patients undergoing distraction osteogenesis to verify the actual advantages and shortcomings of this alternative technique.

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.