Complications of mandibular distraction osteogenesis for congenital deformities: a systematic review of the literature and proposal of a new classification for complications

Abstract

A systematic review of English and non-English language articles on the complications of mandibular distraction osteogenesis (MDO) for patients with congenital deformities was performed, in accordance with the PRISMA statement. Search terms expressing distraction osteogenesis were used in ‘AND’ combination with search terms comprising ‘mandible’ and terms for complication, failure, and morbidity. A search using PubMed (National Library of Medicine, NCBI), EMBASE, and the Cochrane Central Register of Controlled Trials yielded 644 articles published between 1966 and mid October 2013. Clinical articles that reported complications related to MDO were included. Finally 81 articles on MDO in congenital deformities were eligible and were screened in detail. Complications including minor infection (6.0%), device-related problems (7.3%), skeletal open bite (2.4%), hypertrophic scar formation (2.1%), facial nerve palsy (1.8%), neurosensory disturbances of the inferior alveolar nerve (1.9%), and (fibrous) non-union (0.7%) were seen. A new index for more detailed classification of complications in MDO is proposed based on six categories that indicate the impact of the complication and its further treatment or final results. The proposed complication index may be a useful tool to classify complications related to MDO.

Craniomaxillofacial distraction osteogenesis (DO) is a well-described surgical reconstructive technique that was first reported in the peer-reviewed literature by McCarthy et al.; the technique was applied in the treatment of the hypoplastic mandible in four patients. Since then, several systematic reviews on the clinical application of craniomaxillofacial DO and DO in infancy have been published. Master et al. published an article on complications in mandibular DO (MDO). Nevertheless, evidence-based reports on the long-term results, relapse, and complications of MDO are limited.

Paley introduced a classification in which complications arising in the orthopaedic application of DO are divided into problems, obstacles, and complications. This classification was adopted by Neyt et al. for transpalatal DO. Mofid et al. grouped the complications of craniofacial DO into five major categories: technical failure of the distraction process, injury to a vital structure, failure to guide the distraction process along the appropriate vector, infection, and ‘other’. Shetye et al. reported a stratification system for MDO in which incidents related to hardware or hard and soft tissue were subdivided into minor, moderate, and major. In 2010, Davidson et al. developed a similar classification for complications in MDO. However, the authors think there is a need for a standard classification that is more detailed with regard to the relevant clinical situation and possible further treatment, and is more widely applicable for use by clinicians.

The aims of this study were (1) to perform a systematic review of the literature on complications of MDO for congenital deformities, and (2) to introduce an index for the classification of complications in (mandibular) DO in general.

Materials and methods

Literature search

A comprehensive systematic review of the literature was performed in the bibliographic databases PubMed (National Library of Medicine, NCBI), EMBASE, and the Cochrane Central Register of Controlled Trials from inception to 15 October 2013; the review was performed in accordance with the PRISMA statement. Search terms included controlled terms from medical subject headings (MeSH) in PubMed and Emtree in EMBASE, as well as free text terms. We used free text terms only in the Cochrane register. Search terms expressing distraction osteogenesis were used in ‘AND’ combination with search terms comprising ‘mandible’ and terms for complication, failure, and morbidity ( Table 1 ). The references of the identified articles were searched for additional relevant publications.

Table 1
Primary and secondary key words used for the systematic research.
Primary key words Secondary key words
Distraction Mandible
Distraction osteogenesis Mandibular
Lengthening Alveolar
Complication
Complicated
Failure
Morbidity

Study selection and inclusion criteria

Two reviewers independently screened all potentially relevant titles and abstracts for pre-specified eligibility criteria. If necessary, the full text article was checked for the eligibility criteria. Differences in judgement were resolved through a consensus procedure. The full text of articles was then obtained for further review.

The articles were included if they met the following eligibility criteria: (1) clinical article, (2) mandibular distraction osteogenesis (MDO), (3) congenital deformity, and (4) a report on complications. Studies were excluded if data on complications were insufficient, no translation was available, or the publication was a non-clinical article ( Table 2 ).

Table 2
Inclusion and exclusion criteria.
Condition Article types Number of papers ( n )
Excluded from the systematic review Non-congenital deformities 124
Insufficient or no information on complications and/or methods 57
Non-clinical articles (experimental, scientific, synopsis) 24
Non-(mandibular) distraction osteogenesis 4
No translation available 11
Publication type, e.g. letter to the editor, discussion 5
Not available in international libraries 3
Included in the systematic review Clinical articles on complications in mandibular distraction osteogenesis for congenital deformities 81

Articles that were found clinically relevant to the study subject were included in the systematic review. According to their emphasis, these relevant papers were included if they described MDO in the treatment of congenital mandibular deformities. The articles were screened for the following data: type of deformity, number of patients, type of DO, distraction device, vector, and type and number of complications. The latter were classified according to the proposed classification index shown in Fig. 1 . This classification emphasizes on the severity and clinical consequences of a complication by dividing events according to spontaneous resolving or permanent complications, hospitalization or general anesthesia required for correction of the complication.

Fig. 1
Distraction osteogenesis complication index, based on this systematic review.

The initial literature search identified a total of 973 references: 521 in PubMed, 437 in EMBASE, and 15 in the Cochrane register. After removing duplicate references ( n = 329) that were selected from more than one database, 644 papers remained. Titles and abstracts were screened for eligibility by the two reviewers; 335 articles were excluded from the review based on the abstract. The full text was obtained for 309 papers and analyzed thoroughly. Subsequent categorization produced the following clusters ( Table 2 ): (1) 124 articles concerned non-congenital deformities; (2) 57 had insufficient or no information on complications and/or methods; (3) 24 papers were non-clinical (eight scientific, 16 synopsis); (4) four papers were not relevant (three non-DO, one maxilla); (5) 11 papers had no available translation (one Russian, eight Chinese, one Japanese, one Polish); (6) five articles had an edited publication type (three discussion, two letters to the editor/authors); (7) three papers were not available in the international libraries. These seven groups were excluded from further evaluation. In the case of a paper that reported complications in a mixed population (congenital, developmental, or acquired), in which the complications could not be traced back to the exact patient subgroup, the article was excluded on the basis of insufficient data. In total 228 articles were excluded based on the eligibility criteria. Eighty-one articles on MDO for congenital deformities were included. The flowchart of the literature search and selection process through the different phases of the systematic review (PRISMA) is shown in Fig. 2 .

Fig. 2
Flowchart of the search and selection procedure using PubMed (National Library of Medicine, NCBI), EMBASE, and the Cochrane Central Register of Controlled Trials.

Results

The study included 81 publications reviewing a total of 1258 patients. The majority of the group consisted of paediatric patients. The eligible articles displayed a broad spectrum of congenital deformities. Hemifacial microsomia patients accounted for the majority of this group (HFM, n = 717, 57.0%). Detailed information on the remaining congenital deformities is shown in Table 3 .

Table 3
Indications for mandibular distraction osteogenesis.
Congenital deformity Number %
Hemifacial microsomia (HFM) 717 57.0
Pierre Robin sequence (PRS) 341 27.1
Treacher Collins syndrome (TCS) 61 4.8
Goldenhar syndrome (GHS) 23 1.8
Nager syndrome 20 1.6
Stickler syndrome 7 0.6
Down’s syndrome 2 0.2
Cerebral palsy 2 0.2
Cornelia de Lange syndrome 2 0.2
Hanhart syndrome 1 0.1
Other/non-specified 82 6.5

Mandibular lengthening was carried out in 99.8% ( n = 1255) of the cases. The MDO was performed bilaterally in 665 patients (52.9%) and unilaterally in 590 patients (46.9%). Bioresorbable distraction devices were used in 127 patients (10.1%). Seventy-one patients (5.6%) underwent DO after mandibular reconstruction with a bone graft (mainly costocartilaginous grafts in HFM). Characteristics of the DO for all included patients are shown in Table 4 .

Table 4
Characteristics of distraction osteogenesis in congenital deformities ( N = 1258).
Distraction type Number
Mandibular lengthening 1255
Bilateral 665
Unilateral 590
External, unidirectional 682
External, multidirectional 84
Internal, unidirectional 398
Internal, multidirectional 91
Mandibular widening 3
Graft DO 71
Site
Native 5
Graft 26
Junction 3

No complications were reported in 27 papers on mandibular lengthening involving 146 patients with congenital deformities. The remaining 54 papers listed 433 complications. Hence the overall incidence of complications in the total population of 1258 patients was 34.4%. The most common complications in this group are listed in Table 5 .

Table 5
Complications in mandibular distraction osteogenesis for the treatment of congenital deformities, classified according to the proposed index.
Complications n (%)
Type I (complications resolving spontaneously within 6 months)
Temporary IAN neurosensory disturbances 24
Pain 21
Trismus 12
Temporary facial nerve palsy 9
Minor occlusal disturbances 7
Parotid gland injury 2
Periodontal damage 1
Asymmetrical distraction 1
Subtotal 77 (6.1%)
Type II (medically or technically manageable complication, without hospitalization)
Local infection 73
Incorrect vector 55
Device-related problems (pin loosening or extrusion) 39
Device back-up/inadequate length 11
Dehiscence/pin exposure 5
Cellulitis 3
Contralateral open bite 2
Subtotal 188 (14.9%)
Type III (surgically manageable complication requiring local anaesthesia only, without hospitalization)
Soft tissue dehiscence 2 (0.2%)
Type IV (technical complication, necessitating general anaesthesia for correction)
Device failure (requiring replacement) 31
Pin loosening (requiring re-fixation) 10
Incomplete osteotomy/premature ossification 8
Fracture of a transport disc 1
Subtotal 50 (4.0%)
Type V (medically/surgically manageable complication with hospitalization or general anaesthesia)
Hypertrophic scar formation (requiring revision) 8
Relapse or skeletal anterior open bite 3
Dentigerous cyst formation 3
Carotid artery injury 1
Mandible fracture 1
Subtotal 16 (1.3%)
Type VI (permanent sequelae, functionally and/or psychosocially disabling, and unachieved goal or unsatisfactory result)
Skeletal open bite 30
Hypertrophic scar formation 18
Permanent damage to teeth or follicles 14
Permanent facial nerve palsy 14
(Fibrous) non-union 9
TMJ ankylosis 7
Relapse of distracted bone 5
Pseudo-arthrosis 2
Permanent IAN neurosensory disturbance 1
Subtotal 100 (7.9%)
Total 433 (34.4%)
IAN, inferior alveolar nerve; TMJ, temporomandibular joint.

Results

The study included 81 publications reviewing a total of 1258 patients. The majority of the group consisted of paediatric patients. The eligible articles displayed a broad spectrum of congenital deformities. Hemifacial microsomia patients accounted for the majority of this group (HFM, n = 717, 57.0%). Detailed information on the remaining congenital deformities is shown in Table 3 .

Table 3
Indications for mandibular distraction osteogenesis.
Congenital deformity Number %
Hemifacial microsomia (HFM) 717 57.0
Pierre Robin sequence (PRS) 341 27.1
Treacher Collins syndrome (TCS) 61 4.8
Goldenhar syndrome (GHS) 23 1.8
Nager syndrome 20 1.6
Stickler syndrome 7 0.6
Down’s syndrome 2 0.2
Cerebral palsy 2 0.2
Cornelia de Lange syndrome 2 0.2
Hanhart syndrome 1 0.1
Other/non-specified 82 6.5
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Jan 17, 2018 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Complications of mandibular distraction osteogenesis for congenital deformities: a systematic review of the literature and proposal of a new classification for complications
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