Orthodontic and orthopedic management of dentofacial deformity from juvenile idiopathic arthritis: a systematic literature review.

BACKGROUND: An update on the knowledge regarding the orthopedic/orthodontic role in treating JIA-related dentofacial deformities is relevant. OBJECTIVES: This systematic review aimed to assess the level of evidence regarding the management of dentofacial deformity from juvenile idiopathic arthritis (JIA) with orthodontics and/or dentofacial orthopedics. SEARCH METHODS: The following databases were searched without time or language restrictions up to 31 January 2024 (Medline, Embase, Cochrane Central Register of Controlled Trials, Scopus, Web of Science, and Latin American and Caribbean Health Sciences Literature). SELECTION CRITERIA: Inclusion criteria were studies dealing with JIA subjects receiving treatment with orthodontic and/or dentofacial orthopedic functional appliances. DATA COLLECTION AND ANALYSIS: After the removal of duplicate studies, data extraction, and risk of bias assessment according to ROBINS-I guidelines were conducted. Data extraction was conducted by two independent authors. RESULTS: The electronic database search identified 397 eligible articles after the removal of duplicates. Following the application of the pre-defined inclusion and exclusion criteria, 11 articles were left for inclusion. Two trials were associated with a severe risk of bias, four trials were at moderate risk of bias, and the other five presented a low risk of bias. Various research groups employed and documented the effects of different types of appliances and methodologies. The study heterogeneity did not allow for meta-analyses. In addition, a lack of uniformity in treatment objectives was observed across the included studies. After treatment with dentofacial orthopedics skeletal improvement was demonstrated in 10 studies, and a decrease in orofacial signs and symptoms was reported in 7 studies. CONCLUSIONS: Across the available literature, there is minor evidence to suggest that dentofacial orthopedics may be beneficial in the management of dentofacial deformities from JIA. There is little evidence to suggest that it can reduce orofacial signs and symptoms in patients with JIA. Based on current evidence, it is not possible to outline clinical recommendations for specific aspects of orthopedic management in growing subjects with JIA-related dentofacial deformity. REGISTRATION: PROSPERO (CRD42023390746).

Long-Term Follow-Up of Orthognathic Surgery in 19 Patients with Juvenile Idiopathic Arthritis.

Objectives: Dentofacial deformity following juvenile idiopathic arthritis with temporomandibular joint involvement is associated with functional, aesthetic, and psychosocial impairment. Surgical treatment may involve combinations of orthognathic surgery. The aims of this retrospective study were to assess orofacial symptoms, functional and aesthetic status, and stability after orthognathic surgery. Material and Methods: Nineteen patients with juvenile idiopathic arthritis of the temporomandibular joint (TMJ) and dentofacial deformities were included. All patients were treated with combinations of bilateral sagittal split osteotomy, Le Fort I and/or genioplasty, between September 10, 2007 and October 17, 2017. Analysis of patient symptoms and clinical registrations, and frontal/lateral cephalograms was performed pre- and postoperative and long-term (mean: 3.8 and 2.6 years, respectively). Results: Patients experienced no changes in orofacial symptoms or TMJ function, and stable normalisation of horizontal and vertical incisal relations at long-term (horizontal overbite; vertical overbite: P < 0.05). Mandibular lengthening was achieved postoperatively (from mean 79.7 to 87.2 mm; P = 0.004) and was stable. Sella-nasion to A point (SNA) and sella-nasion to B point (SNB) angles increased postoperatively (SNA, mean 79.9° to 82.8°; P = 0.022 and SNB, mean 73.9° to 77.8°; P = 0.003), however, largely reverted to preoperative status at long-term. Conclusions: Orthognathic surgery normalized incisal relations while providing stable mandibular lengthening without long-term deterioration of temporomandibular joint function or orofacial symptoms. No long-term effect on jaw advancements was observed.

Zygomatic repositioning and Le Fort II distraction with intraoral devices in Apert syndrome: A case report.

The aim of this case report is to describe the surgical technique and outcome using internal intraoral distraction devices in LeFort II distraction with zygomatic repositioning (LF2ZR). In Apert syndrome the midface is characterized by a complex hypoplasia, with the central part being more affected than the lateral orbito-zygomatic complex. In LF2ZR, the zygomas are repositioned and internally fixated, and the central midface is further advanced through a LeFort II distraction. In previous publications, the distraction has been performed using external halo-based devices. It seems that the LF2ZR procedure can be planned and performed with adequate accuracy using virtual surgical planning tools. Knowledge about the possibility of using internal intraoral distraction devices in LF2ZR is important, as the inconspicuous placement of intraoral devices can be advantageous for some patients.

Initial radiological signs of dentofacial deformity in juvenile idiopathic arthritis.

Juvenile idiopathic arthritis (JIA) is the most common rheumatic disease in childhood and the temporomandibular joint (TMJ) is often involved. TMJ arthritis in growing individuals can cause deformation of facial skeleton (dentofacial deformity) and TMJ components (TMJ deformity). Treatment outcome hinges on early initiation of anti-inflammatory treatment and orthopaedic treatment with dental splints. The aim of the present study was to characterize the radiological signs of dentofacial deformity in patients with a JIA-induced need for orthopaedic treatment. We retrospectively studied 96 patients with JIA and 20 non-JIA controls to identify the initial radiological signs of JIA-induced dentofacial deformity leading to initiation of orthopaedic treatment. We found that initial radiological signs of dentofacial deformities were subtle and characterized by minor mandibular asymmetry and occlusal plane steepening. Radiological findings of TMJ deformity associated with initial dentofacial deformity were frequent and characterized by condylar articular surface flattening (OR 8.42), condylar subcortical cyst (OR 5.94), condylar surface erosion (OR 5.38) and condylar deviation in form (OR 25.39). Radiological signs of TMJ deformity were also documented in TMJs considered "healthy" during initial clinical and radiological examination. This study presents new knowledge of importance for early diagnosis of dentofacial deformity in JIA. Early diagnosis of dentofacial deformity is important as treatment outcome is greatly influenced by timely initiation.

Comparing Clinical and Radiological Manifestations of Adolescent Idiopathic Condylar Resorption and Juvenile Idiopathic Arthritis in the Temporomandibular Joint.

PURPOSE: This cross-sectional study compared orofacial manifestations at the time of diagnosis in 2 temporomandibular joint (TMJ) conditions: adolescent idiopathic condylar resorption (ICR) and TMJ involvement from juvenile idiopathic arthritis (JIA). MATERIALS AND METHODS: This retrospective study included 19 JIAs, 19 ICRs, and 19 control patients, all treated at the Section of Orthodontics, Aarhus University Craniofacial Clinic, Denmark. From patient files, we retrieved radiological data from cone-beam computed tomographies along with information on symptoms and orofacial function at the time of diagnosis. Validated methodologies were used to evaluate TMJ and dentofacial morphology. RESULTS: We found no statistically significant intergroup differences in severity of deformation of TMJ structures (TMJ deformity) between JIA and ICR patients. However, the ICR group showed significantly greater signs of dentofacial deformity on 4 outcome variables: mandibular inclination, posterior/anterior lower face height ratio, mandibular sagittal position, and mandibular occlusal plane inclination. Significant intergroup differences in clinical presentation were seen in 5 of 20 variables. Thus, the JIA group reported significantly more symptoms of TMJ pain, TMJ morning stiffness, and TMJ pain on palpation during the clinical examination, whereas the ICR group reported significantly more TMJ clicking during function and had a higher proportion of patients with anterior open bite. CONCLUSIONS: Cone-beam computed tomography examination showed a similar degree of TMJ deformity in ICR and JIA patients at the time of diagnosis. ICR patients presented with a significantly higher degree of dentofacial deformity than JIA patients and healthy controls, which relates to the timing of the diagnosis. Arthralgia, TMJ morning stiffness, and TMJ palpitation pain were more common in JIA patients, whereas TMJ clicking during function and anterior open bites were more common in ICR patients.

Influence of Infant Cleft Dimensions on Velopharyngeal Function in 5-Year-Old Danish Children Born With Unilateral Cleft Lip and Palate.

AIM: To examine the association of cleft severity at infancy and velopharyngeal competence in preschool children with unilateral cleft lip and palate operated with early or delayed hard palate repair. DESIGN: Subgroup analysis within a multicenter randomized controlled trial of primary surgery (Scandcleft). SETTING: Tertiary health care. One surgical center. PATIENTS AND METHODS: One hundred twenty-five infants received cheilo-rhinoplasty and soft palate repair at age 3 to 4 months and were randomized to hard palate closure at age 12 or 36 months. Cleft size and cleft morphology were measured 3 dimensionally on digital models, obtained by laser surface scanning of preoperative plaster models (mean age: 1.8 months). MAIN OUTCOME MEASUREMENTS: Velopharyngeal competence (VPC) and hypernasality assessed from a naming test (VPC-Sum) and connected speech (VPC-Rate). In both scales, higher scores indicated a more severe velopharyngeal insufficiency. RESULTS: No difference between surgical groups was shown. A low positive correlation was found between posterior cleft width and VPC-Rate (Spearman = .23; P = .025). The role of the covariate "cleft size at tuberosity level" was confirmed in an ordinal logistic regression model (odds ratio [OR] = 1.17; 95% confidence interval [CI]:1.01-1.35). A low negative correlation was shown between anteroposterior palatal length and VPC-Sum (Spearman = -.27; P = .004) and confirmed by the pooled scores VPC-Pooled (OR = 0.82; 95% CI: 0.69-0.98) and VPC-Dichotomic (OR = 0.82; 95% CI: 0.68-0.99). CONCLUSIONS: Posterior cleft dimensions can be a modest indicator for the prognosis of velopharyngeal function at age 5 years, when the soft palate is closed first, independently on the timing of hard palate repair. Antero-posterior palatal length seems to protect from velopharyngeal insufficiency and hypernasality. However, the association found was significant but low.

Interdisciplinary Management of Dentofacial Deformity in Juvenile Idiopathic Arthritis.

Temporomandibular joint (TMJ) arthritis impacts mandibular growth and development. This can result in skeletal deformity, such as facial asymmetry and/or malocclusion asymmetry. This article reviews the unique properties of TMJ and dentofacial growth and development in the setting of juvenile idiopathic arthritis (JIA). Specific orthopedic/orthodontic and surgical management of children with JIA and TMJ arthritis is discussed. The importance of interdisciplinary collaboration is highlighted.

Palatal morphology in unilateral cleft lip and palate patients: Association with infant cleft dimensions and timing of hard palate repair.

OBJECTIVES: To assess the impact of cleft severity and timing of hard palate repair on palatal dimensions in unilateral cleft lip and palate (UCLP) children. SETTING AND SAMPLE POPULATION: Single-centre analysis within a multicenter RCT of primary surgery; 122 UCLP randomized to early hard palate closure (EHPC) at 12 months or delayed hard palate closure (DHPC) at 36 months; 28 frequency-matched controls. METHODS: Linear measurements of palatal height, width and length were performed on 116 digital models of UCLP subjects (8.21 years, SD = 0.53) and 28 models of non-cleft individuals (8.44 years SD = 0.72). Cleft dimensions at infancy (mean 1.8 months) were considered. In a pilot study, shell-to-shell distances between the 3D cleft palate objects and a reference mesh were calculated and differences between the groups assessed. Morphological differences were visualized using colour mapping. RESULTS: Compared to controls, UCLP subjects presented a higher palate at the level of the anterior scar (P = 0.002), but generally a lower palate in the middle region (P < 0.001). Comparing UCLP subgroups, the DHPC subjects showed a flatter palate posteriorly (P = 0.048) and the EHPC group exhibited more transversal constriction (P = 0.003 at M1 level). 3D analysis revealed a shallower palate in the DHPC group both in the middle (P = 0.002) and the posterior part (P = 0.008). Anterior cleft severity correlated negatively with palatal height (P = 0.01). CONCLUSIONS: Unilateral cleft lip and palate palates differ from controls in width and height. DHPC may represent an advantage for the transversal dimension, but a disadvantage for palatal height. Infant cleft dimensions partially explain differences in palatal height.

An Algorithm for Management of Dentofacial Deformity Resulting From Juvenile Idiopathic Arthritis: Results of a Multinational Consensus Conference.

PURPOSE: Juvenile idiopathic arthritis (JIA) frequently affects the temporomandibular joints and may cause dentofacial deformity and dysfunction. The adverse effects of JIA on dentofacial growth, morphology, and function may be due to erosion of the existing mandibular condyle(s), the inhibitory effect of the arthritis on the growing mandible, or both. No algorithm exists for management of JIA-induced skeletal and dental abnormalities; treatment varies widely. MATERIALS AND METHODS: On the basis of the available literature and expert opinion obtained by a consensus conference held by the Temporomandibular Joint Juvenile Arthritis (TMJAW) group-a multidisciplinary and multinational clinical and research network dedicated to the diagnosis and management of temporomandibular joint arthritis caused by JIA-we present an algorithm to be used as a conceptual framework for management of dentofacial deformity resulting from JIA. RESULTS: An algorithm for management of dentofacial deformity resulting from JIA is presented and exemplified by 5 clinical cases. CONCLUSIONS: A standardized algorithm will improve clinical decision making and facilitate outcome research by allowing valid comparisons between published research studies. We emphasize the importance of multidisciplinary evaluation, management, and long-term follow-up.

Moebius sequence -a multidisciplinary clinical approach.

BACKGROUND: Moebius Sequence (MS) is a rare disorder defined by bilateral congenital paralysis of the abducens and facial nerves in combination with various odontological, craniofacial, ophthalmological and orthopaedic conditions. The aetiology is still unknown; but both genetic (de novo mutations) and vascular events in utero are reported. The purpose of present study was through a multidisciplinary clinical approach to examine children diagnosed with Moebius-like symptoms. Ten children underwent odontological, ophthalmological, obstetric, paediatric, orthopaedic, genetic, radiological and photographical evaluation. Five patients maintained the diagnosis of MS according to the diagnostic criteria. RESULTS: All five patients had bilateral facial and abducens paralysis confirmed by ophthalmological examination. Three of five had normal brain MR imaging. Two had missing facial nerves and one had missing abducens nerves. The Strengths and Difficulties Questionnaire (SDQ) showed normal scores in three of five patients. Interestingly, two of five children were born to mothers with uterine abnormalities (unicornuate/bicornuate uterus). In the odontological examination three of five showed enamel hypomineralisation. All five had abnormal orofacial motor function and maxillary prognathism. Two patients had adactyly, syndactyly and brachydactyly. None of the five patients had Poland anomaly, hip dislocation or dysplasia but all had a mild degree of scoliosis. We observed congenital club-feet, calcaneovalgus deformities, macrodactyly of one or more toes or curly toes. Pedobarography showed plantar pressures within normal ranges. CONCLUSIONS: Adherence to standard diagnostic criteria is central in the diagnosis of MS. An accurate diagnosis is the basis for correct discussion of other relevant concomitant symptoms of MS, genetic testing and evaluation of prognosis. The multidisciplinary approach and adherence to diagnostic criteria taken in present study increases the knowledge on the relationship between genotype, phenotype and symptomatology of MS.

Stability after Cleft Maxillary Distraction Osteogenesis or Conventional Orthognathic Surgery.

OBJECTIVES: To compare stability of maxillary advancements in patients with cleft lip and palate following distraction osteogenesis or orthognathic surgery. INCLUSION CRITERIA: 1) cleft lip and palate, 2) advancement > 8 mm. Eleven patients comprised the distraction osteogenesis group (DOG). Seven patients comprised the orthognathic treatment group (CONVG). Skeletal and soft tissue points were traced on lateral cephalograms: T1 (preoperatively), T2 (after surgery), T3 (follow-up). Group differences were analyzed using Students t-test. RESULTS: At T1-T2, advancement of 6.98 mm (P = 0.002) was observed in DOG. Horizontal overjet increased 11.62 mm (P = 0.001). A point-nasion-B point (ANB) angle increased 8.82° (P = 0.001). Aesthetic plane to upper lip was reduced 5.44 mm (P = 0.017) and the naso-labial angle increased 16.6° (P = 0.001). Vertical overbite (VOB) increased 2.27 mm (P = 0.021). In T2-T3, no significant changes were observed in DOG. In T1-T2, horizontal overjet increased 8.45 mm (P = 0.02). The ANB angle, 9.33° (P = 0.009) in CONVG. At T2-T3, VOB increased, 2.35 mm (P = 0.046), and the ANB angle reduced, 3.83° (P = 0.003). In T2-T3, no parameters changed in CONVG. At follow-up (T3), VOB increased in CONVG compared with DOG, (P = 0.01). Vertical position of A point differed between the groups (P = 0.04). No significant intergroup differences between soft tissue parameters occurred. CONCLUSIONS: Distraction osteogenesis resulted in a stable position of the maxilla and movement upwards in vertical plane, however in case of orthognathic treatment sagittal relapse and a continued postoperatively downward movement was registered.

Effect of mandibular distraction osteogenesis on the temporomandibular joint: a systematic review of animal experimental studies.

OBJECTIVE: The present systematic review aimed to test the hypothesis of no effect of mandibular distraction osteogenesis on the temporomandibular joint. STUDY DESIGN: Animal experimental studies from January 1985 to August 2013 were included. Studies were searched in PubMed, Embase, Scopus, and the Cochrane Library. A total of 289 articles were identified, and 17 were included. RESULTS: Included studies were characterized by a high risk of bias and by inhomogeneity related to animal species, experimental procedures, and evaluation methods. Mandibular distraction osteogenesis within physiologic limits may be followed by adaptive changes in bone, disk, and cartilage. Increased daily rates and total activation length may influence the severity of the adaptive changes. CONCLUSIONS: Animal experimental studies indicate that mandibular distraction osteogenesis may induce adaptive changes in the temporomandibular joint. Adaptive changes may be influenced by increased daily rates and total length of distraction osteogenesis. Well-designed studies are needed before final conclusions can be drawn.

Complications after mandibular distraction osteogenesis: a retrospective study of 131 patients.

OBJECTIVES: The objectives of this study were to evaluate the occurrence and severity of complications after mandibular distraction osteogenesis (DO) with internal devices. STUDY DESIGN: The study was a retrospective analysis of 131 patients (mean age: 16.2 years) consecutively treated by mandibular DO from 1998 to 2009. Ninety-two patients had unilateral and 39 had bilateral distraction, yielding a total of 170 procedures. The mean follow-up period was 21 months. Severity of complications was ranked in terms of need of intervention and risk of a compromised outcome. RESULTS: Minor, moderate, and severe complications occurred in 58%, 8%, and 3% of the patients, respectively. Most minor complications were related to device activation or temporary hypesthesia. Moderate complications often related to hardware, whereas severe complications occurred in 4 patients with sensory deficit or temporomandibular joint problems. CONCLUSIONS: Although minor complications were frequent, they did not compromise treatment outcome, so mandibular DO is considered a safe method for correction of mandibular deformities. Moreover, some of the complications could be prevented by proper precautions.

Stereolithographic models for simulation and transfer of vector in vertical distraction of the mandibular ramus: a technical note.

Mandibular distraction osteogenesis is, despite impressive results, compromised as a treatment modality by difficulties with vector control independent of whether intra- or extraoral devices are used. Resistance of the masticatory muscles and the remaining soft tissue envelope toward elongation and incorrect planning and transfer of the vector cause lack of vector control. To ensure predictability in correct placement of intraoral distraction devices and thereby treatment outcome, a combined planning, simulation, and transfer method was developed. The purpose of the simulation and transfer technique is to duplicate the planned and simulated position and orientation of the distraction device during surgery. The preoperative plan is based on frontal and lateral head films, and the planned position and orientation of the distraction apparatus was applied on the stereolithographic model. The model was cut according to the planned osteotomy, and the distraction was simulated on the stereolithographic model. Based on plaster models transferred to the stereolithographic model, an individual guiding splint was fabricated to reproduce the planned and simulated distraction treatment in the patient. The details of the simulation and transfer technique are presented and discussed in two cases.

Unilateral mandibular ramus elongation by intraoral distraction osteogenesis.

Successful correction of facial asymmetry by mandibular distraction osteogenesis relies on mastering vector control. Lack of necessary vector control continues to compromise the treatment modality. The purpose of this study was to describe a new simplified method for vector transfer and to evaluate this method according to the outcome and the efficacy of distraction osteogenesis in the correction of unilateral mandibular hypoplasia and asymmetry. Twenty-seven patients with unilateral hypoplasia of the mandibular ramus underwent unilateral mandibular distraction osteogenesis with intraoral distraction devices. Posterior-anterior and lateral cephalograms were analyzed by digitalization. Changes in sagittal, vertical, and transversal linear and angular dimensions after the distraction treatment were evaluated by measurements performed on headfilms taken before and after surgery. Means and variances were calculated for selected cephalometric variables for each time point. The differences between the control and the treatment side were calculated, statistically described, and compared with a paired Student t test. Correction of the mandibular asymmetry, chin position, and the canting of the occlusal plane was obtained in all patients clinically, as well as radiographically, by the use of intraoral unidirectional distraction osteogenesis.

Reliability of distraction vector transfer in unilateral vertical distraction of the mandibular ramus.

Mandibular distraction osteogenesis fails in correcting the skeletal deformities if the vector of distraction is incorrect. Lack of vector control is caused mainly by two factors: first, deficient vector planning and transfer, and, second, resistance of the masticatory muscles and the remaining soft tissue envelope toward elongation. To enhance predictable placement of intraoral distraction devices during surgery and thereby the treatment outcome, a method combining planning, simulation, and transfer was developed. The presurgical planning was based on frontal and lateral head films, and the planned position and orientation of the distraction device was applied on the stereolithographic model. This model was cut according to the planned osteotomy, and the distraction was simulated. The transfer procedure copied the planned and simulated position of the distraction device during surgery by use of an individual guiding splint. The guiding splint was fabricated on plaster models, and transferred to the stereolithographic model to reproduce the planned and simulated distraction treatment during surgery. The reliability of the vector-transfer method was evaluated cephalometrically in a homogenous group of 13 patients and their respective stereolithographic models in comparison with the planned, simulated, and transferred distraction vectors.