Maxillary distraction osteogenesis versus orthognathic surgery for cleft lip and palate patients.

BACKGROUND: Cleft lip and palate is one of the most common birth defects and can cause difficulties with feeding, speech and hearing, as well as psychosocial problems. Treatment of orofacial clefts is prolonged; it typically commences after birth and lasts until the child reaches adulthood or even into adulthood. Residual deformities, functional disturbances, or both, are frequently seen in adults with a repaired cleft. Conventional orthognathic surgery, such as Le Fort I osteotomy, is often performed for the correction of maxillary hypoplasia. An alternative intervention is distraction osteogenesis, which achieves bone lengthening by gradual mechanical distraction. This review is an update of the original version that was published in 2016. OBJECTIVES: To provide evidence regarding the effects and long-term results of maxillary distraction osteogenesis compared to orthognathic surgery for the treatment of hypoplastic maxilla in people with cleft lip and palate. SEARCH METHODS: Cochrane Oral Health's Information Specialist searched the following databases: Cochrane Oral Health's Trials Register (to 15 May 2018), the Cochrane Central Register of Controlled Trials (CENTRAL) (the Cochrane Library, 2018, Issue 4), MEDLINE Ovid (1946 to 15 May 2018), Embase Ovid (1980 to 15 May 2018), and LILACS BIREME Virtual Health Library (Latin American and Caribbean Health Science Information database; from 1982 to 15 May 2018). The US National Institutes of Health Trials Registry (ClinicalTrials.gov) and the World Health Organization International Clinical Trials Registry Platform were searched for ongoing trials. No restrictions were placed on the language or date of publication when searching the electronic databases. SELECTION CRITERIA: We included randomised controlled trials (RCTs) comparing maxillary distraction osteogenesis to conventional Le Fort I osteotomy for the correction of cleft lip and palate maxillary hypoplasia in non-syndromic cleft patients aged 15 years or older. DATA COLLECTION AND ANALYSIS: Two review authors assessed studies for eligibility. Two review authors independently extracted data and assessed the risk of bias in the included studies. We contacted trial authors for clarification or missing information whenever possible. All standard methodological procedures expected by Cochrane were used. MAIN RESULTS: We found six publications involving a total of 47 participants requiring maxillary advancement of 4 mm to 10 mm. All of them related to a single trial performed between 2002 and 2008 at the University of Hong Kong, but not all of the publications reported outcomes from all 47 participants. The study compared maxillary distraction osteogenesis with orthognathic surgery, and included participants from 13 to 45 years of age.Results and conclusions should be interpreted with caution given the fact that this was a single trial at high risk of bias, with a small sample size.The main outcomes assessed were hard and soft tissue changes, skeletal relapse, effects on speech and velopharyngeal function, psychological status, and clinical morbidities.Both interventions produced notable hard and soft tissue improvements. Nevertheless, the distraction group demonstrated a greater maxillary advancement, evaluated as the advancement of Subspinale A-point: a mean difference of 4.40 mm (95% CI 0.24 to 8.56) was recorded two years postoperatively.Horizontal relapse of the maxilla was significantly less in the distraction osteogenesis group five years after surgery. A total forward movement of A-point of 2.27 mm was noted for the distraction group, whereas a backward movement of 2.53 mm was recorded for the osteotomy group (mean difference 4.8 mm, 95% CI 0.41 to 9.19).No statistically significant differences could be detected between the groups in speech outcomes, when evaluated through resonance (hypernasality) at 17 months postoperatively (RR 0.11, 95% CI 0.01 to 1.85) and nasal emissions at 17 months postoperatively (RR 3.00, 95% CI 0.14 to 66.53), or in velopharyngeal function at the same time point (RR 1.28, 95% CI 0.65 to 2.52).Maxillary distraction initially lowered social self-esteem at least until the distractors were removed, at three months postoperatively, compared to the osteotomy group, but this improved over time and the distraction group had higher satisfaction with life in the long term (two years after surgery) (MD 2.95, 95% CI 014 to 5.76).Adverse effects, in terms of clinical morbidities, included mainly occlusal relapse and mucosal infection, with the frequency being similar between groups (3/15 participants in the distraction osteogenesis group and 3/14 participants in the osteotomy group). There was no severe harm to any participant. AUTHORS' CONCLUSIONS: This review found only one small randomised controlled trial concerning the effectiveness of distraction osteogenesis compared to conventional orthognathic surgery. The available evidence is of very low quality, which indicates that further research is likely to change the estimate of the effect. Based on measured outcomes, distraction osteogenesis may produce more satisfactory results; however, further prospective research comprising assessment of a larger sample size with participants with different facial characteristics is required to confirm possible true differences between interventions.

Evaluation of parental and surgeon stressors and perceptions of distraction osteogenesis in pediatric craniofacial patients: a cross-sectional survey study.

PURPOSE: There is a paucity of literature on how limitations of distraction osteogenesis (DO) are perceived by physicians and parents of pediatric patients. Specifically understanding which features of DO are most concerning to these two groups may better inform parent education, as well as direct improvements in distraction protocols and devices. METHOD: Parents/guardians of patients (between January 2016 and October 2017) being treated with craniofacial distraction were recruited to complete a survey regarding level of stress (1 = not stressful, 9 = maximally stressful) associated with eight features of DO. Craniofacial surgeons completed a survey asking them to report (1) their personal level of stress and (2) their perceptions of parental stress regarding these same eight features of DO. RESULTS: Thirty-five parents and 15 craniofacial surgeons completed the survey. The risk of the device getting infected was perceived as most stressful by parents (5.5 ± 2.3) followed by the device sticking through the skin (4.9 ± 2.6) and the second operation for removal (4.7 ± 2.3). These same three features also elicited the highest level of stress among surgeons. Surgeon-perceived parental stress regarding turning of the distractor (5.8 ± 1.5) was significantly higher than parent self-reported stress (4.2 ± 2.8, p = 0.042). CONCLUSIONS: Both parents and surgeons perceive risk of device-associated infection, the protrusion of the device through the skin, and the requirement of a second operation for removal as the most stressful drawbacks of distraction. Infection reduction protocols, less obtrusive devices, and devices that do not require removal are potential targets for stress reduction.

Internal Distraction Resulted in Improved Patient-Reported Outcomes for Midface Hypoplasia.

BACKGROUND: Both internal and external distraction devices have been used successfully in correcting midface hypoplasia. Although the indication for surgery and the osteotomy techniques may be similar, deciding when to use internal versus external devices has not been studied. The authors studied patient-reported outcomes with FACE-Q and functional surveys for internal and external devices for midface distraction patients. METHODS: Patients who underwent distraction advancement after Le Fort I and Le Fort III were surveyed using the FACE-Q survey and a functional survey. Equal groups of internal and external device patients were compared (n = 64). Data recorded included: sex, age, follow-up, diagnosis, operating room time, expected blood loss, length of stay, distraction length, consolidation time, and complications. RESULTS: Internal and external device groups were similar with regards to patient diagnosis, operative time, expected blood loss, distraction length but consolidation times differed (internal = 3.6 versus external = 1.1 months). For FACE-Q appearance appraisal, there were similarities in domain and scale. For the functional survey (airway/breathing, ocular/vision, occlusion/eating, speech/articulation), there was also similar scoring. However, internal device patients had superior FACE-Q scores for Quality of Life: Social Function (80.9 versus 68.9), Early Life Impact (92.9 versus 62.4), Dental Anxiety (70.2 versus 48.3), Psychological Well-being (87.8 versus 68.6); and Decision Satisfaction (81.2 versus 56.9) and Outcome Satisfaction (91.0 versus 84.7). CONCLUSIONS: Internal and external midface distraction patients had similar patient-reported outcomes for appearance and functional improvement; however, internal device patients were more satisfied with their quality of life and their decision to undergo the procedure.

Frontofacial advancement by distraction osteogenesis: a long-term review.

BACKGROUND: Frontofacial advancement by distraction osteogenesis is known to produce significant functional and aesthetic gains in the short and medium term. Little is known about the long-term effects of this technique. The authors present a long-term review of frontofacial advancement by distraction in patients with syndromic craniosynostosis. METHODS: In 2008, Witherow et al. presented functional outcomes in 20 consecutive patients who underwent frontofacial advancement by distraction osteogenesis using the rigid external distractor frame. This retrospective study has revisited the outcomes in these patients after a mean follow-up period of 10.2 years. RESULTS: Records for the original 20 patients were assessed. Cephalometric analysis demonstrated stable advancements over the long term. No patient achieved any anterior growth of the midface after frontofacial advancement. Long-term follow-up demonstrated a later deterioration of respiratory function in the younger group of patients (36.4 percent). Four patients required adenotonsillectomy. One patient has a persistent tracheostomy. Only one patient required further procedures to correct recurrent raised intracranial pressure. None of the patients in the older group experienced a deterioration of function. Other procedures included reduction of the supraorbital ridge, bimaxillary osteotomies, and rhinoplasties. CONCLUSIONS: Frontofacial distraction produced long-term stable advancement in all cases, and 15 of the 17 living patients (88.2 percent) achieved stable functional gains. Anterior growth of the midface does not occur following monobloc distraction. Deterioration in function is seen in those who undergo this procedure at a young age. The authors recommend regular follow-up of these patients. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

Family-centred care during midface advancement with a rigid external device: what do families need?

Midface advancement with distraction osteogenesis using the rigid external device (RED) is an effective but invasive treatment to correct the hypoplastic midface. This study draws up an inventory of the stressors, needs and coping strategies of families during this treatment, to determine the best conditions for family-centred care. Data were collected by reviewing the patients' files and administering semi-structured interviews. The data were analysed using the software program Atlas.ti and were re-analysed by an independent researcher. Parents and patients were interviewed separately. Fourteen families participated. Four patients had an absolute indication for surgery. All families were eager to have the patient's facial appearance improved. Nevertheless, despite psychological counselling, they experienced stress when confronted with the changed facial appearance. Another stressor was weight loss. Six patients were in a state of acute malnutrition and needed supplementary feeding. We conclude that the best conditions for family-centred care should be aligned to the different phases of treatment. Leading up to surgery it is important to screen families' expectations regarding aesthetic, functional and social outcomes and to assess their capacity to cope with the long treatment and effects of changed facial appearance. Peer contact and psychosocial training to increase self-esteem are tools to enhance co-operation and satisfaction. During the distraction and stabilisation phase, we advise the monitoring of nutritional intake and weight. During all phases of treatment easy accessibility to the team is recommended.

The comparison of psychological adjustment of patients with cleft lip and palate after maxillary distraction osteogenesis and conventional orthognathic surgery.

OBJECTIVE: The objective of this study was to compare the psychological changes of patients with cleft lip and palate (CLP) undergoing maxillary Le Fort I advancement by distraction osteogenesis (DO) and conventional orthognathic surgery (CO). STUDY DESIGN: Prospective randomized controlled study. Participants completed a set of questionnaires to measure their psychological states preoperatively and postoperatively. RESULT: When compared with CO, CLP patients treated with DO had lower social self-esteem and higher social avoidance and distress levels during the first 3 postoperative months. Their self-esteem, social avoidance, and distress levels improved after the distractors were removed. Both CO and DO patients had similar levels of self-esteem, social avoidance, and distress levels 2 years postoperatively. The CLP patients treated with DO were more satisfied with their lives after 2 years. CONCLUSIONS: DO may induce short-term distress to patients up to 3 months but CLP patients who received DO were more satisfied with their lives in the long term.

External craniofacial osteodistraction in complex craniosynostoses.

PURPOSE: The use of distraction osteogenesis is the first option in the treatment of craniofacial hypoplasia. Both internal craniofacial distractor devices (ICDD) and external craniofacial distractor devices (ECDD) can improve visual and respiratory functions in patients with craniofacial synostosis significantly. They also improve the cosmetic outcome. DISCUSSION: We reviewed the pertinent literature concerning the use of ECDD in complex craniostenosis. Surgical data as well as quality of life notes were analyzed and compared with those reported for ICDD. CONCLUSION: The advantage of ECDD compared with ICDD is the easier application and postoperative management as compared with ICDD, with comparable results for extent and quality of distraction osteogenesis. The disadvantage is the quality of life which most authors report is worse if compared to ICDD during the distraction osteogenesis period.

Five years experience with a new intraoral maxillary distraction device (RID).

Maxillary distraction osteogenesis is well established for the treatment of severe retromaxilla. We report our experience since 2004 of the treatment of 19 patients using a new intraoral maxillary distraction device. Maxillary advancement was successful in all patients with mean advancement of 9.6mm (range 4-17) measured at a point in lateral cephalograms. The new device limited surgical exposure and the amount of materials implanted, and improved control in every phase of the distraction. It was psychologically accepted by patients and was more comfortable than existing devices.

Bone transport and compression-distraction in the treatment of bone loss of the lower limbs.

A clinical series of 17 adult patients operated due to significant by bone loss of the long bones of the lower extremity (3 femurs and 11 tibias), is presented. Their management consisted of 6 bone transports (6 tibias) and 11 compression distraction procedures (3 femurs and 8 tibiae) using monolateral external fixators. Bone loss ranged from 3.9 cm to 14.7 cm. Mean healing time was 301 days with a mean healing index of 45.6 days for cm of lengthening achieved. The clinical and radiological results were excellent in 9, good in 6 and fair in 2 patients according to the utilised criteria of assessment. Consolidation was achieved in all but one patient who developed an aseptic stiff non-union. Two patients developed residual limb-length discrepancy less than 1.5 cm, three tibias ended up with less than 5° of valgus deviation. In two cases the half-pins were re-inserted due to early loosening. In two cases reoperation was needed for late bending of the callus after fixator removal. Three cases of bone transport and 1 case of compression distraction needed bone grafting at the docking site. Bone transport and compression-distraction are effective methods for treating bone loss in the lower extremity. It is suggested that the compression-distraction technique is preferable, since this is associated with a lower incidence of complications than bone transport procedures. The deciding factor, however, is the actual extent of the bone loss.

What the patients and parents do not tell you-recollections from families following external LeFort III midface distraction.

OBJECTIVE: The purpose of this study was to document the experience of patients and parents of patients who had recently undergone LeFort III midface distraction using an external halo-based device. DESIGN: Cross-sectional study. SETTING: A craniofacial center in a pediatric tertiary care medical center. SUBJECTS: Eight children who had undergone midface distraction within 1 year of the interview and their caregivers. INTERVENTION: Semi-structured interview. MAIN OUTCOME MEASURE: Transcripts of the interviews were rendered anonymous and analyzed by our multi-disciplinary team. Consistent themes in the subjects' experience during and after midface distraction were identified. RESULTS: (1) Family participation in the decision to undergo distraction and pre-operative preparation was recognized as valuable, but parents identified that there are inherent limitations; (2) home-care tasks seemed daunting pre-operatively but were easier than expected; (3) discomfort, sleeping, and interaction with peers were considered well accommodated, but feeding was challenging; (4) individualized pre-operative plans for community support was important; (5) parents and patients were impressed by the change in appearance, specifically in the peri-orbital region; (6) access to team members and to parents of patients who had participated in the distraction process was invaluable. CONCLUSION: External midface distraction is a valuable clinical technique, but requires intensive preparation and support from a multi-disciplinary team. We provide suggestions for consideration by centers initiating and refining patient care plans for this surgery.

[Psychological impact of alveolar mandibular distraction]. / Impact psychologique de la distraction alvéolaire mandibulaire.

INTRODUCTION: Implant supported dental prostheses are the most up-to-date solution for edentulous patients. This technique requires and adequate bone quantity and quality. Bone distraction may allow compensating for some bone deficit, especially mandibulary. Few studies have been dedicated to how patients adjusted to this therapy (Int J oral Maxillofac Surg 34 [2005] 238-42, Int J oral Maxillofac Surg 36 [2007] 896-9, Med Oral Pathol Oral Cir Bucal 12 [2007] E225-8). We evaluated the psychological impact of alveolar mandibular distraction. PATIENTS AND METHODS: Between 1999 and 2006, 31 patients aged 27 to 70 years underwent vertical alveolar mandibular distraction. Seventeen patients (54.8%) presented with complications. A questionnaire assessed the psychological impact by using notions used in healthcare psychology: perceived stress, perceived control, and social support. RESULTS: Twenty-three answers (74.2% of operated cases) were studied. In 87% of the cases, patients adjusted well the distraction procedure. Eighty-one percent felt no stress. Fifty-seven percent reported light to moderate pain, and 43% found the treatment painful. Confrontation to adverse events was mentioned only in 13% of the cases. In 17% of the cases, there was a slight alteration of sleep. Fifty-seven percent of the patients managed to forget the presence of the distractor. The most difficult stages were insertion of the distractor (48%) and the activation phase (17%). Seventy-one percent of the patients did not find the protocol restraining. The treatment length was not a problem for 65%. Two patients (9%) found it too long. Ninety-one percent of the patients activated the device on their own, for two (9% of the cases) the surgeon activated the device. Ninety-seven percent of the patients found supervision satisfactory. Medical information helped to adjust well to the procedure in 96% of the cases. Forty-three percent of the patients (10 cases) required specific help during the treatment: family support, attending physician, or surgical team. Among these, 70% considered the medical team's contribution as the most beneficial compared to family support, and 30% reported they were the same. For 96% of the patients, the medical team was the major actor of therapeutic success. Twenty-two percent of the patients considered they were partly responsible for treatment results (perceived internal control). Fifty-two percent of the cases reported they would be ready to undergo another distraction protocol if necessary. DISCUSSION: Alveolar mandibular distraction has only a weak psychological impact. It improves the patient's quality of life. It can be suggested that hope for improvement helped patients to better stand treatment. The information received contributed to better adjust to events. This unpredictable situation does not allow the patient to prepare himself and generates stress. Information in necessary before operating, to let the patient assess his own resources and prepare his own psychological adaptation mechanism. It is through this means that we can speak about true "informed consent or refusal".

Patient satisfaction related to rigid external distraction osteogenesis.

The aim of this study was to evaluate satisfaction with treatment among cleft lip and palate patients who underwent maxillary advancement using a rigid external distraction (RED) device. Nine patients (four boys, five girls), mean age 17.7 years (SD 4.0), were included in the study. Outcome measures included satisfaction with facial appearance and function (sensitivity/pain, discomfort during daily functioning, daily activities, speech, eating and/or drinking, expression of affection) before, during and after treatment with the RED device assessed by a self-administered questionnaire. Before treatment, the majority of patients were not satisfied with their facial appearance. Some received negative remarks about their appearance and experienced minor functional problems. Dissatisfaction with appearance, negative remarks and functional problems increased significantly during active treatment, and the majority of patients experienced pain or sensitivity. After treatment all patients but one were satisfied with their appearance and level of function. Overall patient satisfaction after treatment with a RED device is high, but the active treatment period, during which the frame is worn, significantly compromises function and may be painful. For most patients, satisfaction with the final result and appearance outweighs the negative factors they reported.

Patient's perception of the events during and after osteogenic alveolar distraction.

OBJECTIVE: The aim of the study was to evaluate the patient s perception of the events during and after an osteogenic alveolar distraction (OAD) procedure MATERIALS AND METHODS: A total of fifty-five (55) osteogenic alveolar distraction (OAD) procedures were performed in fifty (50) patients, who then were asked to answer ten (10) questions related to the treatment. Six (6) questions made reference to predefined values in a Visual Analogical Scale (VAS), three (3) questions could be answered by a predetermined answer, and only one (1) question had a free answer. RESULTS: In 76% of cases, the patient s description of the sensation felt during the surgery was good and bearable; 84% of the patients didn t feel pain after surgery. 4% of the patients felt pain during the activation period and 58% of the patients described the sensation during the activation period as pressure, felt most commonly, at the end of the period, and for about 20 minutes (66.6 %). In these cases the most frequently used analgesic was Paracetamol. Also, 46% expressed having had some difficulty to activate the device, with 10% of them in need of extra help. The presence of the activation rod caused discomfort in 52%. Finally, 78% of the patients treated with OAD would undergo this procedure again if it was necessary. A bone graft was performed in 27 out of the 50 treated patients, with 70% of them describing the bone graft surgery as more painful than the OAD. CONCLUSION: The OAD technique had a high degree of acceptance among the treated patients, however, some details as the interference of the activation rod continue to disturb them. The acceptance of the OAD technique is much better when compared with bone graft surgery technique as a second treatment.

The early psychological adjustment of cleft patients after maxillary distraction osteogenesis and conventional orthognathic surgery: a preliminary study.

PURPOSE: To compare the early psychological changes of cleft lip and palate (CLP) and noncleft patients after maxillofacial corrective surgery, including maxillary distraction osteogenesis and conventional orthognathic surgery. MATERIALS AND METHODS: Nine CLP patients were compared with a group of 9 non-CLP patients having similar dentofacial deformities in a prospective longitudinal cohort study. Five of the CLP patients underwent maxillary distraction osteogenesis and 4 underwent conventional orthognathic surgery. A control group of 9 noncleft patients received conventional orthognathic surgery. All patients completed a set of questionnaires to enable their psychological profile to be assessed. The data were collected immediately before surgery (T1), and at 3 weeks (T2) and 12 weeks (T3) after surgery. RESULTS: The CLP patients treated with distraction osteogenesis were happier, but had a higher level of social anxiety and distress than the CLP patients receiving conventional orthognathic surgery. On the other hand, the CLP patients overall were happier, with lower social anxiety and distress, than the noncleft control group. The CLP patients showed a higher level of parental self-esteem than the noncleft patients. CONCLUSION: This preliminary study shows that CLP patients were generally happier, and had a higher level of parental support, than normal patients suffering from dentofacial deformities. Maxillary distraction osteogenesis seemed to induce a higher level of anxiety and distress in CLP patients than conventional orthognathic surgery in both cleft and noncleft patients.

Patients' responses to distraction osteogenesis: a multi-centre study.

This investigation aimed to compare and contrast practical difficulties experienced by 54 children from three different centres (Mexico City, New York, Paris) treated with either extra-oral or intra-oral distraction devices. The possible effect of distraction osteogenesis on pain/sleeping difficulty, speech and eating problems and disturbance of recreational activities and alteration in sensation were investigated. It was expected that intra-oral devices would reduce the difficulties associated with the distraction process and their psychological effect on the child. Some benefits resulting from the use of intra-oral devices were identified, these included fewer sleeping problems (P = 0.006) and less disturbance of recreational activities (P = 0.002). However, eating and maintaining oral hygiene were more problematic with intra-oral devices the differences between the intra-oral and extra-oral groups was approaching significance at P = 0.07. A major disadvantage of the extra oral device was scarring. In both groups the alteration of lip sensation was temporary, pain was limited to the time of activation of the distraction device. A high level of patients' cooperation was mandatory for successful completion of the treatment.

Unexpected events during mandibular distraction osteogenesis.

Various unexpected events during mandibular distraction osteogenesis are described in 16 syndromic children with severely retrognathic or asymmetric lower jaws who were treated with unilateral or bilateral extraoral distraction. The mean total time with the distraction devices attached was 14 weeks (range 10-19). Although improvement in facial aesthetics and the desired occlusion was achieved in all but one case, there were unexpected events associated with mandibular distraction. These included pain not related to the operation, functional disturbance in movement of the jaw, dietary problems, weight loss, unfulfilled expectations of decannulation, temporary unilateral facial nerve palsy, and transient unilateral hypoaesthesia of the inferior alveolar nerve. Reoperation was required in 5 of the 16. In addition to these events, psychosocial problems within one family complicated the procedure. Patients and their families should be informed of the lesser-known aspects of distraction osteogenesis before the procedure is used.

Response of patients and families to lengthening of the facial bones by extraoral distraction osteogenesis: a review of 14 patients.

Fourteen patients or their immediate family were interviewed about their experiences of having either unilateral or bilateral external distraction osteogenesis of the mandible. The patients showed a high level of co-operation with treatment. Six of the 14 patients required repeat distractions, and had been informed and accepted that this was a possibility before the initial distraction. However, patients or their parents expressed some reservations about the extraoral distractors, which prevented them from practising their favourite sport and made them vulnerable to bullying by their friends and colleagues. Patients had moderate pain when the appliances were removed. They all expressed their satisfaction with the results and would recommend this treatment to others. Problems, including speech, eating, pain, and sleeping difficulties, were encountered by patients at all stages of treatment. Of considerable concern was the disruption of education when the child was treated during the school term.