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MAXILLOFACIAL DISTRACTION OSTEOGENESIS Experiences, results & dilemmas Charlotte R.A. Verlinden ABOUT THE AUTHOR Charlotte Verlinden (1978) werd geboren te Leiden als oudste dochter van Willem en Harmke Verlinden. Het doktersvak werd haar met de paplepel ingegoten in de huisartspraktijk aan huis van haar vader. Na haar middelbare schoolperiode studeerde zij Geneeskunde aan de Vrije Universiteit. Tijdens haar vierde jaar combineerde zij dit met Tandheelkunde aan het Academisch Centrum Tandheelkunde Amsterdam (ACTA). In 2005 behaalde zij haar artsexamen en in 2007 het tandartsexamen. In 2008 begon zij haar specialisatie tot mond-, kaak- en aangezichtschirurg in het VUmc o.l.v. professor van der Waal en professor Schulten. In 2012 vestigde zij zich samen met Jantine Bremmer als MKA-chirurg in Reinier de Graaf Gasthuis te Delft, waar zij met veel plezier werkt. Charlotte is de trotse moeder van Nylah (2010), Liv (2012) en Loïs (2014) en samen met haar grote liefde Maurice wonen zij in Bloemendaal. ISBN# 978-90-825959-0-1

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  • MAXILLOFACIAL DISTRACTION OSTEOGENESIS Experiences, results & dilemmas

    Charlotte R.A. Verlinden

    ABOUT THE AUTHOR Charlotte Verlinden (1978) werd geboren te Leiden als oudste dochter van Willem en Harmke Verlinden. Het doktersvak werd haar met de paplepel ingegoten in de huisartspraktijk aan huis van haar vader. Na haar middelbare schoolperiode studeerde zij Geneeskunde aan de Vrije Universiteit. Tijdens haar vierde jaar combineerde zij dit met Tandheelkunde aan het Academisch Centrum Tandheelkunde Amsterdam (ACTA). In 2005 behaalde zij haar artsexamen en in 2007 het tandartsexamen. In 2008 begon zij haar specialisatie tot mond-, kaak- en aangezichtschirurg in het VUmc o.l.v. professor van der Waal en professor Schulten. In 2012 vestigde zij zich samen met Jantine Bremmer als MKA-chirurg in Reinier de Graaf Gasthuis te Delft, waar zij met veel plezier werkt. Charlotte is de trotse moeder van Nylah (2010), Liv (2012) en Loïs (2014) en samen met haar grote liefde Maurice wonen zij in Bloemendaal.

    ISBN# 978-90-825959-0-1

  • ACADEMISCH PROEFSCHRIFT

    Charlotte R.A. Verlinden

    MAXILLOFACIAL DISTRACTION OSTEOGENESIS

    Experiences, results & dilemmas

    4

  • The studies presented in this thesis were conducted at the Department of Oral and Maxillofacial Surgery, VU University Medical Center / Academic Centre for Dentistry Amsterdam (ACTA), Amsterdam, the Netherlands.

    The printing and distribution of this thesis has been financially supported by:• VU Medical Center, Amsterdam• Nederlandse Vereniging voor Mondziekten, Kaak- en Aangezichtschirurgie (NVMKA)• ZorgApotheek 't Hart van Lelystad• Straumann B.V.• Pothof Ventures B.V.• KLS Martin Group• Dam Medical B.V.

    All proceeds from this thesis will be donated to the Children of Sumatra, helping children suffering from cleft lip and palate living on the island of Sumatra, Indonesia, run by Katie Pavett. www.childrenofsumatra.org

    COVER & LAYOUT Marjolein Verlinden, www.design-a-la-carte.netISBN 978-90-825959-0-1

    Copyright 2016 Charlotte R.A. VerlindenAll rights reserved. No parts of this publication may be reproduced, stored in a retrieval system or transmitted in any form or by any means without prior permission from the author.

    VRIJE UNIVERSITEIT

    Maxillofacial distraction osteogenesis :

    experiences, results & dilemmas

    ACADEMISCH PROEFSCHRIFT

    ter verkrijging van de graad Doctor aan

    de Vrije Universiteit Amsterdam,

    op gezag van de rector magnificus

    prof.dr. V. Subramaniam,

    in het openbaar te verdedigen

    ten overstaan van de promotiecommissie

    van de Faculteit der Geneeskunde

    op maandag 31 oktober 2016 om 13.45 uur

    in de aula van de universiteit,

    De Boelelaan 1105

    door

    Charlotte Rosalina Anna Verlinden

    geboren te Leiden

    5 6

  • Promotoren: prof.dr. T. Forouzanfar prof.dr. D.B. Tuinzing

    7 8

  • Paranimfen: Dr. J.F. Bremmer Drs. A.E. Dijckmeester

    Dare to err and to dream

    ~ F. Schiller

    9 10

  • CONTENT

    Chapter 1 General introduction 13

    Chapter 2 Complications of mandibular distraction osteogenesis in 23 congenital deformities: a systematic review of the literature and proposal of a new classification for complications

    Chapter 3 Complications of mandibular distraction osteogenesis in 43 developmental deformities: a systematic review of the literature

    Chapter 4 Complications of mandibular distraction osteogenesis in 59 acquired deformities: a systematic review of the literature

    Chapter 5 Long-lasting neurosensory disturbance following 81 advancement of the retrognathic mandible: distraction osteogenesis versus bilateral sagittal split osteotomy

    Chapter 6 Symptomatic venous thromboembolism in orthognathic 99 surgery and distraction osteogenesis: a retrospective cohort study of 4127 patients

    Chapter 7 Infections in intraoral distraction osteogenesis for mandibular 111 lengthening: clinical results and systematic review of the literature

    Chapter 8 Complications in transpalatal distraction osteogenesis: 123 a retrospective clinical study

    Chapter 9 General discussion & Summary 141

    Chapter 10 Conclusion & future perspectives 151 Acknowledgements About the author

    11 12

  • 1GENERAL INTRODUCTION1413

  • GENERAL INTRODUCTION

    History of distraction osteogenesis

    Gavriil Abramovich Ilizarov (1921-1992), a Soviet physician, took orthopaedic surgery to a new level by introducing his life’s work: a technique called distraction osteogenesis (also described as callus distraction, osteodistraction). In 1951, distraction osteogenesis (DO) was used to reconstruct skeletal deformities by lengthening the long bones1,2. Whilst inventing the Ilizarov apparatus (external distractor) for this innovative technique, his work gave rise to a new spectrum of orthopaedic treatments. Patients were treated for indications such as limb lengthening, non-unions, bone and soft tissue defects, osteomyelitis, and (post-traumatic) skeletal deformations2,3.

    The main principles of the Ilizarov technique involve2:

    • Preservation of the blood supply • Preservation of the osteogenic tissue • Complete anatomic reduction • Stable fixation • Functional activity of the muscles and joints • Early patient mobilization

    Furthermore, Ilizarov established the “law of tension stress” model, implying that controlled, mechanically applied tensile stress can lead to osteogenesis and histiogenesis1. Ilizarov continued to improve his technique during his career.

    Distraction protocol

    A corticotomy of the affected bone is performed, followed by a sequence of three phases. Preservation of the periosteum (vascularization) is believed to be important for more rapid consolidation2,3. First, in the latency phase lasting 5-10 days, a neutral fixation is maintained, allowing callus and microvascularisation to be formed. During the following distraction phase, bone segments are gradually distracted. These forces stimulate osteo- and histiogenesis, allowing new bone to be formed in the distraction gap4. The ideal distraction rate is 1 mm/day, and a gradual rhythm of distraction (number of distractions/day) produces superior results compared to distraction performed once a day4,5. A neutral fixation is again held during the consolidation phase, to allow healing, remodeling, and consolidation of the new bone, prior to removal of the distraction device2. This phase usually requires up to 6 months for sufficient formation of the neocortex.

    15 16Chapter 1 Chapter 1

  • Distraction osteogenesis of the craniofacial skeleton

    DO of the craniofacial skeleton was not applied until 1972, when it was used by Snyder et al for lengthening a canine mandible, using an external fixator6. Michieli and Miotti introduced the intraoral distractor7. Only in the year Ilizarov passed away, in 1992, was the first article on DO of the human mandible published by McCarthy et al8. Since then, DO of the craniofacial bones has been proven to be effective in treating various skeletal defects of different origins. Molina et al optimistically bade a “farewell” to major osteotomies, advocating mandibular elongation by distraction osteogenesis in patients suffering from hemifacial microsomia. This minor surgery (corticotomy of the mandible) allowed maximal preservation of nerve structures and vascular supply9.

    In many patients, individual planning is required, depending on the severity of the skeletal deformation, and therefore in many cases it has a patient/specific or casuistic character9. Nada et al published on the lack of consensus amongst surgeons about a variety of parameters, such as the timing of DO, surgical planning (conventional surgery versus DO), device selection, and the retention period10.

    DO is applied worldwide for various craniofacial deformities, but there remains a lack of evidence, and therefore there is no consensus on the success of DO and the appropriate treatment protocol10-12.

    Indications

    DO has a wide spectrum of indications. Congenital deformities, such as hemifacial microsomia, Treacher-Collins syndrome, Pierre Robin sequence, and cleft palate patients. Patients requiring bone transplants can be treated by mandibular elongation by DO, combined with maxillary DO in some cases9,13,14. Developmental deformities, such as Class II mandibular hypoplasia or transverse mandibular hypoplasia are successfully treated by mandibular elongation15 or transverse widening using DO16,17. Acquired defects, such as atrophy of the mandibular process, segmental bone defects after oncology-related surgery or post-traumatic bone defects are treated using this technique13. Transverse maxillary expansion can also be treated by DO using a transpalatal distraction device, which provides widening of the maxilla by applying direct forces on the palatinal bone18.

    Evolution of technique

    The distraction hardware has evolved from a bulky external device to an invisible, internal version, which can be driven intraorally19,20. Advantages of the intraoral DO technique are absence of disfiguring scars (especially in young patients with congenital deformities), the external presence of the distractor and the lower risk of loosening or dislocation. However, intraoral devices also have their disadvantages, including a risk of damage to tooth buds and increased periosteal stripping, leading to other types of complications. Intraoral devices usually need to be removed under general anaesthesia, which can be challenging when they are fully extended19. Another innovation was made by improving the vector control. In some patients, a unidirectional vector led to an undesired (severe) anterior open bite, requiring surgical correction. Mandibular DO can be divided in a horizontal vector leading to a horizontal elongation, while a vertical vector of distraction can produce a vertical elongation of the ramus. With multidirectional devices, vector selection and pre-operative planning are believed to contribute to a better outcome9,19,20

    Complications

    The success of this approach by the “Michelangelo of Orthopaedics” was unfortunately also accompanied by complications21. Paley et al described muscle contractures, joint luxation, axial deviation, neurologic injury, vascular injury, premature consolidation, delayed consolidation, non-union, pin site problems, bone and soft tissues defects, pseudoarthrosis, and problems related to the distraction device 3,22. He proposed a classification that divided difficulties into “problems”, resolved without surgical intervention, and “obstacles”, representing difficulties that required operative intervention. Intraoperative injuries and problems persisting until the end of DO were considered true complications22. This classification was adopted by Neyt et al for classifying the difficulties during another form of DO: (maxillary) transpalatal distraction osteogenesis23.

    The number of publications on craniofacial DO has increased rapidly. However, the reports on complications have not increased simultaneously. We performed an initial PubMed search using the terms ‘mandibular’ and ‘distraction osteogenesis’, which yielded approximately 5500 articles. With the addition of the search terms ‘complications’ and ‘failure’, the number of eligible articles was reduced to 521. Therefore, complications in mandibular DO may be underreported14.

    Swennen et al13 reported the clinical parameters of craniofacial DO in 2001. The authors found that a complication occurred in 22% of the patients, mostly due to mechanical distractor-related problems and local infections. In 2001, Mofid et al published a review of 3278 cases of

    17 18Chapter 1 Chapter 1

  • craniofacial DO24. Complications in all types of craniofacial DO were classified 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’. Despite the large number of cases involved, a possible limitation of this classification could be the fact that there is no subdivision by type of DO (midfacial, maxillary, or mandibular) or deformity (congenital, developmental, or acquired) and also no differentiation of the impact of the complications and whether further treatment was required or whether permanent injury resulted. Some groups are more prone to complications due to the surgical technique (patients treated with grafts), medical history (prior treatment), type of deformity (syndromic), or age (infancy). Even after the publication of the review by Mofid et al, there remains a need for objective reporting of complications. A structural classification of complications is also lacking in DO, which ultimately needs to be more detailed for wider application in the clinical environment.

    AIM OF THESIS

    The aim of this thesis was to investigate adverse outcomes during treatment with maxillofacial DO and to categorize these outcomes in accordance with different indications as congenital, developmental, and acquired deformities of the mandible.

    This was performed by:

    u investigating the complications of mandibular DO in congenital deformities and introducing a general index for classification of complications (chapter 2)

    v investigating complications of mandibular DO in developmental deformities (chapter 3)

    w investigating complications of mandibular DO in acquired deformities (chapter 4)

    General complications of mandibular distraction osteogenesis were studied, by:

    x investigating neurosensory disturbances of mandibular DO patients in comparison with conventional surgery for mandibular advancement (bilateral sagittal split osteotomy) (chapter 5)

    y investigating general complications, viz., symptomatic venous thromboembolism, of mandibular DO (chapter 6)

    z investigating general complications, viz., post-operative infections, of mandibular DO (chapter 7)

    The use of distraction osteogenesis for transverse widening of the maxilla was performed by:

    { investigating complications of transpalatal (bone-borne) distraction osteogenesis of the maxilla (chapter 8)

    19 20Chapter 1 Chapter 1

  • REFERENCES1. Ilizarov GA. The principles of the Ilizarov method. Bull Hosp Jt Dis Orthop Inst 1988:48(1):1-11.

    2. Ilizarov GA. Clinical application of the tension-stress effect for limb lengthening. Clin Orthop Relat Res 1990

    Jan:(250):8-26

    3. Paley D. Ilizarov Technology. 1[1993 (oct)], 243-287. 1-10-0993. In Stauffer, Richard. Advances in operative

    orthopaedics.

    4. Ilizarov GA. The tension-stress effect on the genesis and growth of tissues. Part I. The influence of stability

    of fixation and soft-tissue preservation. Clin Orthop Relat Res 1989 Jan:(238):249-81.

    5. Ilizarov GA. The tension-stress effect on the genesis and growth of tissues: Part II. The influence of the rate

    and frequency of distraction. Clin Orthop Relat Res 1989 Feb:(239):263-85.

    6. Snyder CC, Levine GA, Swanson HM. Mandibular lengtening by gradual distraction. Plast Reconstr Surg

    1973 Jan 1:51:506.

    7. Michieli S. Lengthening of mandibular body by gradual surgical-orthodontic distraction. J Oral Surg 1977

    Jan 1:35(3):187-92.

    8. McCarthy JG, Schreiber J, Karp N, Thorne CH, Grayson BH. Lengthening the human mandible by gradual

    distraction. Plast Reconstr Surg 1992 Jan:89(1):1-8.

    9. Molina F, Ortiz Monasterio F. Mandibular elongation and remodeling by distraction: a farewell to major

    osteotomies. Plast Reconstr Surg 1995 Sep 1:96(4):825-40.

    10. Nada RM, Sugar AW, Wijdeveld MG, Borstlap WA CLHBK-JA, Eurocran Distraction Osteogenesis Group.

    Current practice of distraction osteogenesis for craniofacial anomalies in Europe: a web based survey. J

    Craniomaxillofac Surg 2010 Mar 1:38(2):83-9.

    11. Adolphs N, Ernst N, Menneking H, Hoffmeister B. Significance of distraction osteogenesis of the

    craniomaxillofacial skeleton - a clinical review after 10 years of experience with the technique. 42 2014 Sep

    1:6:966-75.

    12. Swennen GR, Schliephake H, Dempf R, Schierle H, Malevez C. Craniofacial distraction osteogenesis: a review

    of the literature: Part 1: clinical studies. Int J Oral Maxillofac Surg 2001 Apr:30(2):89-103.

    13. Verlinden CR, van de Vijfeijken SE, Jansma EP, Becking AG, Swennen GR. Complications of mandibular

    distraction osteogenesis for congenital deformities: a systematic review of the literature and proposal of a

    new classification for complications. Int J Oral Maxillofac Surg 2015 Jan:44(1):37-43.

    14. van Strijen PJ. Distraction osteogenesis for mandibular advancement. Int J Oral Maxillofac Implants 2000

    Apr 1:29(2):81-5.

    15. Mommaerts MY. Anterior transmandibular osteodistraction: clinical and model observations. J Craniomaxillofac

    Surg 2005 Oct 1:33(5):318-25.

    16. Uckan S. Mandibular midline distraction using a simple device. Oral Surg Oral Med Oral Pathol 2015 Nov

    1:100(5):85-91.

    17. Mommaerts MY. Transpalatal distraction as a method of maxillary expansion. Br J Oral Maxillofac Surg 1999

    Jan 1:37:268-72.

    18. Robinson RC, Knapp TR. Distraction osteogenesis in the craniofacial skeleton. Otolaryngol Clin North Am

    2005 Apr:38(2):333-59, vii.

    19. Davidson EH, Brown D, Shetye PR, Greig AV. The evolution of mandibular distraction: device selection. Plast

    Reconstr Surg 2016:126(6):2061-7.

    20. Ilizarov S, Rozbruch SR. The Ilizarov Method: History and Scope. CRC Press; 2006.

    21. Paley D. Problems, obstacles, and complications of limb lengthening by the Ilizarov technique. Clin Orthop

    Relat Res 1990 Jan:(250):81-104.

    22. Neyt NM, Mommaerts MY, Abeloos JV, De Clercq CA, Neyt LF. Problems, obstacles and complications with

    transpalatal distraction in non-congenital deformities. J Craniomaxillofac Surg 2002 Jun:30(3):139-43.

    23. Mofid MM, Manson PN, Robertson BC, Tufaro AP, Elias JJ, Vander Kolk CA. Craniofacial distraction

    osteogenesis: a review of 3278 cases. Plast Reconstr Surg 2001 Oct:108(5):1103-14.

    21 22Chapter 1 Chapter 1

  • 2COMPLICATIONS IN MANDIBULAR DISTRACTION OSTEOGENESIS IN CONGENITAL DEFORMITIES: a systematic review of the literature

    and proposal of a new classification for complications

    Verlinden CRA, van de Vijfeijken SECM, Jansma EP, Becking AG, Swennen GRJ International Journal of Oral and Maxillofacial Surgery. 2015 Jan; 44(1): 37-43

    2423

  • ABSTRACTA 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) nonunion (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.

    Keywords: distraction osteogenesis, lengthening, complication, complicated, congenital, syndromic, failure, morbidity, mandible, mandibular, systematic review, PRISMA statement

    INTRODUCTIONCraniomaxillofacial 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 patients1. Since then, several systematic reviews on the clinical application of craniomaxillofacial DO2–4 and DO in infancy5 have been published. Master et al6 published an article on complications in mandibular DO (MDO). Nevertheless, evidence-based reports on the longterm 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 complications7. This classification was adopted by Neyt et al for transpalatal DO8. Mofid et al3 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 major9. In 2010, Davidson developed a similar classification for complications in MDO10. However, we believe there is the 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 provide a systematic review of the literature on complications in MDO for congenital deformities, and (2) to introduce an index for the classification of complications in (mandibular) DO in general.

    25 26Chapter 2 Chapter 2

  • MATERIALS AND METHODS

    Literature search

    A comprehensive systematic review of the literature was performed in the bibliographic databases Pubmed, EMBASE, Cochrane Controlled Trials Register (National Library of Medicine, NCBI) from inception to 15 October 2013; the review was performed in accordance with the PRISMA statement11. 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 Library. 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 keywords used for the systematic research.

    PRIMARY KEYWORDS SECONDARY KEYWORDS

    Distraction

    Distraction osteogenesis

    Lengthening

    Complication

    Complicated

    Failure

    Morbidity

    Mandible

    Mandibular

    Alveolar

    Study selection and inclusion criteria

    Two reviewers independently screened all potentially relevant titles and abstracts for pre-specified eligibility criteria11. 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 review11. 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 there was insufficient data on complications, 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

    27 28Chapter 2 Chapter 2

  • 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 congenital 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 Figure 1.

    FIGURE 1 DO complication index, based on this SR

    Type I. Spontaneous resolving complication within 6 months after the retention period.

    Type II. Medically or technically manageable complication, without hospitalisation

    Type III. Surgically manageable complication under local anaesthesia, without hospitalisation.

    Type IV. Technical complication, necessitating general anaesthesia for correction.

    Type V. Medically or surgically manageable complication with hospitalisation or general anaesthesia

    Type VI. Permanent sequellae, functionally and/or psychosocially disabling and unachieved goal or unsatisfactory result

    The initial literature search (Table 1) identified a total of 973 references: 521 in PubMed, 437 in EMBASE, and 15 in The Cochrane Library. 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 Figure 211

    FIGURE 2 Flowchart of the search and selection procedure, PubMed, EMBASE, Cochrane.

    INC

    LUD

    ED

    ELI

    GIB

    ILIT

    YSC

    RE

    EN

    ING

    IDE

    NT

    IFIC

    AT

    ION

    Full-text articles excluded:

    - Non-congenital deformity

    (n=124)

    - No information (n=57 )

    - Non-clinical (n=24 ) - No relevance (n=4 )

    - Publication type (n=5 )

    - No translation (n=11)

    - Not available (n=3) (n=22)

    - Not available in International

    libraries (n=8)

    Records excluded based on abstract (n=335 )

    Full-text articles assessed for eligibility (n=309 )

    Studies included in qualitative synthesis ( n=81 )

    Records screened (n=644)

    Records identified after initial search (n=973)

    Records after removal duplicates and update (n=644)

    29 30Chapter 2 Chapter 2

  • 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 in 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 syndrome (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 72 5.7

    Mandibular lengthening was carried out in 99.8% (n = 1255) of the cases. The MDO was performed bilaterally in 668 patients (53.1%) and unilaterally in 590 patients (46.9%). Bioresorbable distraction devices were used in 127 patients (10.1%)12–14. Seventy-one patients (5.6%) underwent DO after mandibular reconstruction with a bone graft (mainly costocartilaginous grafts in HFM)10,15–18. 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 deformities13,19–44. 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 510,15,45-90.

    31 32Chapter 2 Chapter 2

  • TABLE 5 Complications in mandibular distraction osteogenesis for the treatment of congenital deformities, classified according to the proposed index.

    COMPLICATIONS NUMBER

    Type I complications resolving spontaneously

    Temporary IAN neurosensory disturbances10,15,59,77,78 24

    Pain10,61,80 21

    Trismus10,58 12

    Temporary facial nerve palsy49,59,65,68,77,87 9

    Minor occlusal disturbances48,59,87 7

    Parotid gland injury10 2

    Periodontal damage61 1

    Asymmetrical distraction67 1

    Subtotal 77 (6.1%)

    Type II medically or technically manageable complication, without hospitalization

    Local infection10,12,12,14,15,47,50,54,56,57,60,62,63,65,66,70;71,75,77,81,88 73

    Incorrect vector10,70 55

    Device-related problems (pin loosening or extrusion) 10,63,70,77,79 39

    Device back-up/inadequate length10 11

    Dehiscence/pin exposure69,72,81 5

    Cellulitis18,86 3

    Contralateral open bite77 2

    Subtotal 188 (14.9%)

    Type III surgically manageable complication requiring local anaesthesia only, without hospitalization

    Soft tissue dehiscence12 2 (0.2%)

    Type IV technical complication, necessitating general anaesthesia for correction

    Device failure (requiring replacement) 10,12,13,15,16,18,51,52,56,57,65,69,70,86 31

    Pin loosening (requiring refixation) 49,50,57,66,71 10

    Incomplete osteotomy/premature ossification10,45,52,63,84 8

    Fracture of a transport disc10 1

    Subtotal 50 (4.0%)

    Type V medically/surgically manageable complication with hospitalization or general anaesthesia

    Hypertrophic scar formation (requiring revision)10,58,63 8

    Relapse or skeletal anterior open bite55,58,76 3 Dentigerous cyst formation73,89 3

    Carotid artery injury85 1

    Mandible fracture10 1

    Subtotal 16 (1.27%)

    Type VI permanent sequelae, functionally and/or psychosocially disabling, and unachieved goal or unsatisfactory result

    Skeletal open bite64,69,75,83,89 30

    Hypertrophic scar formation10,15,69,72,83,87,89 18

    Permanent damage to teeth or follicles10,74,75,83,89 14

    Permanent facial nerve palsy56,63,77,83,89,89 14

    (Fibrous) non-union16,18,53,65 9

    TMJ ankylosis10,65,82 7

    Relapse of distracted bone18,48,82 5

    Pseudo-arthrosis15,17 2

    Permanent IAN neurosensory disturbance90 1

    Subtotal 100 (7.9%)

    Total 433 (34.4%)

    IAN, inferior alveolar nerve; TMJ, temporomandibular joint.

    33 34Chapter 2 Chapter 2

  • DISCUSSIONMandibular distraction osteogenesis (MDO) is a relatively new technique that has had a major impact on the correction of various craniofacial deformities2. The number of publications on this subject continues to increase. Nevertheless, evidence is essential and is required for further validation of this broad spectrum of treatments. Our initial PubMed search yielded approximately 5500 articles using the terms ‘mandibular’ and ‘distraction osteogenesis’. With the addition of the search terms ‘complications’ and ‘failure’, the number of eligible articles was limited to 521. Therefore, complications in MDO may be under-reported3,4,9.

    Swennen et al reported the clinical parameters of craniofacial DO in 2001. The authors found that a complication occurred in 22% of the patients, mostly due to mechanical distractor-related problems and local infections2. In the present population, an overall complication incidence of 34.4% was found. Infection was seen in 5.8% and device-related problems were present in 7.3% of all patients.

    DO in infancy has been a useful technique for the treatment of upper airway obstruction in syndromal patients, e.g. those with Pierre Robin sequence. Complications in infant DO occur, and since DO is in its infancy itself, long-term results and complications in mandibular growth, TMJ disorders, and relapse rates are not known5.

    In 2001 Mofid et al published a review of 3278 cases of craniofacial DO, in which 274 respondents completed a questionnaire on treatment parameters, outcomes, and complications3. Complications in all types of craniofacial DO were classified 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’. Despite the large number of cases involved, a possible limitation of this classification could be the fact that there is no subdivision by type of DO (midfacial, maxillary, or mandibular) or deformity (congenital, developmental, or acquired) and also no differentiation of the impact of the complications and further treatment required or resulting permanent injury. Some groups are more prone to complications due to the surgical technique (patients treated with grafts), medical history (prior treatment), type of deformity (syndromic), or age (infancy).

    More than 10 years have passed since the publication of the review by Mofid et al and there is still a need for objective reporting of complications3. A structural classification is also lacking for complications in DO, which ultimately needs to be more detailed for the clinical environment in order to attain wider applicability. Shetye et al mentioned a possible under-reporting of complications and suggested a new stratification system that divides complications into minor, moderate, and major complications9. Major complications lead to negative treatment outcomes. This general system is simple and efficient, but lacks details or a description that is directly related to the DO procedure

    and the related treatment or correction. We developed an index that focuses on the effects of DO. By dividing these effects into six categories, the impact of the complications is classified into a spectrum of type I complications, i.e. those that resolve spontaneously, to type VI complications, i.e. a permanent negative outcome. The authors believe that current indices do not provide this insight. HFM is the second most common congenital craniofacial deformity in children and is believed to be especially suitable for DO due to the hypoplastic nature of the mandible and the morbidity caused by alternative treatment4. The majority of these patients are usually treated during early childhood (mid-mixed dentition), when optimal results in the interceptive treatment of the deformity can be achieved. Mandibular elongation can minimize the secondary growth deformities of vertical maxillary excess and contralateral mandibular overgrowth. However, the long-term stability and occurrence of relapse and mandibular overgrowth in these patients are not known4,18. In Pruzansky-Kaban types IIb/III, the deformity is usually more progressive and more severe4. Unfortunately, injury to tooth germs, facial nerve palsy, non-union, and hypertrophic scarring at the pin entry sites are not uncommon in these patients. Advancements have been made in the design of distraction devices in the last decade. Whereas, multivector control was initially only possible with external devices, internal devices have undergone changes in design, resulting in internal multivector and curvilinear devices with a similar effect10;90;91 . The application of internal devices is more challenging and requires more extensive degloving of the periosteum92. External devices are more prone to pin loosening and extrusion, and can be difficult to maintain, especially in young infants. Device-related complications requiring revisional surgery (type IV) were seen in 41 patients (3.3%) with congenital deformities. Pin loosening or extrusion was seen in 39 patients (3.1%) and device back-up or inadequate length without further effects on the outcome was seen in 11 patients (0.9%). This is in agreement with the cumulative incidences reported in the literature of up to 7.9%2,3,6. Local infection is reported in 8.3% to 9.5% of patients. In this review an incidence of 6.0% (including cellulitis) was found. More severe complications such as fusion errors including (fibrous) non-union and pseudo-arthrosis were noted in 11 patients (0.9%). Not surprisingly this involved more patients requiring graft DO16,18. In particular, graft DO at the rib–mandible junction was found to lead to more complications such as resorption and non-union18. The results of this systematic review show that there remains a clear need for objective reporting of complications of MDO. Our initial search yielded 309 eligible clinical articles of which 57 articles (18.4%) had to be excluded due to insufficient or absent data on complications. There might be a tendency to report only the severe complications9. However, it is essential to have full knowledge of all types of complications and especially their incidences.

    35 36Chapter 2 Chapter 2

  • Our proposed complication index for MDO is based on six categories that classify the impact of the complications and their further treatment and final results. Each category divides the complications into those of surgical or technical origin and differentiates between treatments under local or general anaesthesia. Also, temporary and permanent complications are differentiated from one another. For example, an impaired vector control leading to anterior open bite may be mild and controlled orthodontically with minimal invasive measures. In other cases it may be severe and only treatable with a secondary orthognathic surgical procedure, which has a greater impact on the patient. These two types should to be differentiated from one another for clinical reasons and patient consent. The suggested classification enables differentiation of these types of complication and can be applied to any type of DO. In conclusion, this systematic review shows that MDO can be applied to different indications of DO in congenital deformities and that treatment and treatment outcomes vary widely. A unifying index that meets the criteria of a good complication classification is challenging. The authors have developed a detailed index for classifying the outcomes of MDO.

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    41 42Chapter 2 Chapter 2

  • 3COMPLICATIONS OF MANDIBULAR DISTRACTION OSTEOGENESIS IN DEVELOPMENTAL DEFORMITIES: a systematic review of the literature.

    Verlinden CRA, van de Vijfeijken SECM, Tuinzing DB, Jansma EP, Becking AG, Swennen GRJ International Journal of Oral and Maxillofacial Surgery. 2015 Jan; 44(1): 44-49

    4443

  • ABSTRACTA systematic review of English and non-English articles on the complications of mandibular distraction osteogenesis (MDO) for patients with developmental 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 Cochrane Controlled Trials Register yielded 644 articles published between 1966 and mid October 2013. Clinical articles that reported complications related to MDO in developmental deformities were included. Two hundred and fifty articles were eligible and were screened in detail. A total of 32 articles reporting the cases of 565 patients were finally included. Patients underwent mandibular lengthening and transverse widening. A total of 211 complications were reported (37.4%); these were classified according to an index that indicates the clinical impact. Inferior alveolar nerve (IAN) neurosensory disturbances, minor infection, device failure, anterior open bite, permanent dental damage, and skeletal relapse were most represented. Complications that resolved spontaneously (type I) were seen in 11.0%, medically or technically manageable complications, without hospitalization, were seen in 10.8% (type II), and permanent complications (type VI) were seen in 9.6%.

    Keywords: distraction, distraction osteogenesis, lengthening, complication, complicated, failure, morbidity, mandible, mandibular hypoplasia, retrognathia, orthognathic surgery, systematic review

    INTRODUCTION More than two decades after its introduction by McCarthy et al mandibular distraction osteogenesis (MDO) has become an established technique alongside orthognathic surgery for the correction of non-syndromic mandibular hypoplasia1. Corpus lengthening using MDO transcends the believed limitations of 10 mm lengthening of the bilateral sagittal split osteotomy2. Clinical experience and improvements in the technique have led to an evolution in surgical planning and devices. A spectrum of external, unidirectional, and semi-buried/hybrid devices, progressing to miniaturized internal, multidirectional, and custom-made devices has been employed. Multidirectional MDO requires more detailed pre-surgical planning directed at the individual anatomical needs3. The increased use of this technique and development of devices has given rise to a wide variety of complications2,4,5. Several reviews on clinical parameters (including complications) in MDO for varying indications have been published4–6. At the same time, several different systematic reviews on the clinical application of craniomaxillofacial DO5–7 have been published. Nevertheless, evidence-based reports on the long-term results, relapse, and complications of MDO are limited.

    The aims of this study were (1) to perform a systematic review of the literature on complications of MDO for developmental deformities, in accordance with the PRISMA statement, and (2) to classify all complications using a recently devised classification8.

    45 46Chapter 3 Chapter 3

  • 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 statement9. 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 Library. Search terms expressing distraction osteogenesis were used in ‘AND’ combination with search terms comprising ‘mandible’ and terms for complication, failure, and morbidity. The references of the articles identified were searched for additional relevant publications.

    Study selection and inclusion criteria

    Two reviewers independently screened all potentially relevant titles and abstracts for pre-specified eligibility criteria9. If necessary, the full text article was checked for the eligibility criteria. Differences in judgement were resolved through a consensus procedure. Full text articles were then obtained for further review9. Articles were included if the following eligibility criteria were met: (1) clinical article, (2) mandibular distraction osteogenesis (MDO), (3) developmental deformities, 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 1).

    TABLE 1 Inclusion and exclusion criteria.

    CONDITION ARTICLE TYPES NUMBER OF PAPERS (N)

    Excluded from the systematic review

    Non-developmental deformities 180

    Insufficient or no information on complications and/or methods

    20

    Non-clinical articles (experimental, scientific, synopsis)

    9

    No translation available 3

    Publication type, e.g. discussion 1

    Included in the systematic review

    Clinical articles on complications in mandibular distraction osteogenesis for developmental deformities

    32

    The remaining clinically relevant articles were included in this systematic review. According to their emphasis, these relevant papers were included if they described MDO in developmental 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 figure 1.

    47 48Chapter 3 Chapter 3

  • FIGURE 2. Flowchart of the search and selection procedure using PubMed, EMBASE, and The Cochrane Library

    INC

    LUD

    ED

    ELI

    GIB

    ILIT

    YSC

    RE

    EN

    ING

    IDE

    NT

    IFIC

    AT

    ION

    Full-text articles excluded:

    - Non-congenital deformity (n=124)

    - No information (n=57 )

    - Non-clinical (n=24 )

    - No relevance (n=4 )

    - Publication type (n=5 )

    - No translation (n=11)

    - Not available (n=3) (n=22)

    - Not available in

    International libraries (n=8)

    Records excluded based on abstract (n=394)

    Full-text articles assessed for eligibility (n=250 )

    Studies included in qualitative synthesis ( n=32 )

    Records screened (n=644)

    Records identified after initial search (n=973)

    Records after removal duplicates and update (n=611)

    FIGURE 1. Distraction osteogenesis complication index 8

    Type I. Spontaneous resolving complication within 6 months after the retention period.

    Type II. Medically or technically manageable complication, without hospitalisation

    Type III. Surgically manageable complication under local anaesthesia, without hospitalisation

    Type IV. Technical complication, necessitating general anaesthesia for correction

    Type V. Medically or surgically manageable complication with hospitalisation or general anaesthesia

    Type VI. Permanent sequellae, functionally and/or psychosocially disabling and unachieved goal or unsatisfactory result

    The initial literature search yielded a total of 973 references: 521 in PubMed, 437 in EMBASE, and 15 in The Cochrane Library. After removing duplicate references (n = 329) that were selected from more than one database, 644 papers remained. Titles and abstracts were screened according to the eligibility criteria; 394 articles were excluded from the review based on the abstract. The full text was obtained for 250 papers and analyzed thoroughly. The following groups were identified (Table 1): (1) 185 articles reported non-developmental mandibular deformities, (2) 20 articles contained insufficient or no information on complications and/or methods; (3) nine papers were non-clinical (seven scientific, one cephalometric, one synopsis); (4) three papers had no available translation (two Chinese, one Hungarian); (5) one article had an edited publication type (discussion). These five groups were excluded from further evaluation. In the case of a paper that reported on complications in a mixed population in which the complications could not be traced back to the exact patient subgroup, the article was excluded on the basis of insufficient data. A total of 32 articles on MDO for developmental deformities met the inclusion criteria. The flowchart of the literature search and selection process through the different phases of the systematic review (PRISMA) is shown in figure 29.

    49 50Chapter 3 Chapter 3

  • RESULTSThe study group covered two deformities, class II mandibular hypoplasia and transverse mandibular hypoplasia, involving a total 565 patients. Sixteen publications reported mandibular lengthening in 273 patients with class II mandibular hypoplasia, and 16 publications reported mandibular widening for transverse mandibular deficiency in 292 patients. The 32 publications reported 211 complications, leading to an overall complication incidence of 37.4%. The majority of complications was identified in index types I, II, and VI (31.3%). Eight publications reported no complications in a total of 44 patients.10–17 An overview of all reported complications is presented in Table 2.

    TABLE 2 Complications in mandibular distraction osteogenesis for the treatment of congenital deformities, classified according to DO-classification index8.

    COMPLICATIONS NUMBERS

    Type I complications resolving spontaneously within 6 months

    Temporary IAN neurosensory disturbances16–20,29 40

    TMJ-related complaints/pain17,27,29,30 7

    Hardware-related pressure ulcers24,31,32 7

    Tooth mobility20,24 4

    Delayed union24 2

    Posterior open bite31,33 2

    Subtotal 62 (11.0%)

    Type II medically or technically manageable complication, without hospitalization

    Minor infection (including pin tract infection)31,34–44 45

    Anterior open bite/incorrect vector31,32 14

    Non-compliance 2

    Subtotal 61 (10.8%)

    Type III surgically manageable complication requiring local anaesthesia only, without hospitalization

    0

    Type IV technical complication, necessitating general anaesthesia for correction

    Device failure (requiring replacement)17,18,25 10

    Broken distraction rod31,32 9

    Fracture of device26,45 4

    Insufficient length38 1

    Subtotal 24 (4.2%)

    Type V medically/surgically manageable complication with hospitalization or general anaesthesia

    Premature consolidation25,46 6

    Incomplete osteotomy18,31,32 3

    Surgical correction of severe open bite32 1

    Subtotal 10 (1.8%)

    Type VI permanent sequelae, functionally and/or psychosocially disabling, and unachieved goal or unsatisfactory result

    Permanent IAN neurosensory disturbance18,21,29 17

    Condylar resorption16,47 12

    Skeletal relapse16,47 12

    Permanent (periodontal) damage to central incisors23–28 10

    Chin ptosis26 1

    Anterior open bite39 1

    Lingual nerve neurosensory disturbance31 1

    Subtotal 54 (9.6%)

    Total 211 (37.3%)

    IAN, inferior alveolar nerve; TMJ, temporomandibular joint.

    51 52Chapter 3 Chapter 3

  • The remaining 24 articles listed the following complications: the group of type I complications comprised 62 minor complications (11.0%), including temporary inferior alveolar nerve (IAN) hypoesthesia (7.1%)18–23 temporomandibular joint (TMJ) related pain/complaints (1.2%)19,20,24,25 and complications such as hardware-related pressure ulcers26–28 tooth mobility18,26 delayed union26 and posterior open bite27,29 which resolved spontaneously within 6 months after the retention period. The group of type II complications comprised minor infections (pin tract infection, local infection, cellulitis) (8.0%)20,21,25–28,30–34 anterior open bite (2.5%)27,28 and non-compliance27 which could be treated without further surgical intervention, e.g. antibiotics treatment, elastic band treatment. No type III complications were reported in this group of patients with developmental deformities. Type IV complications included mainly device-related problems, such as device failure (1.8%)20,23,30 and fracture of the distraction device or rod (2.3%),27,28,31,35 and there was one case of insufficient length of the device.32 Surgical revision (type V) was required for premature consolidation of the osteotomy (1.1%)30,36 and incomplete osteotomies (0.5%).23,27,28 One patient required a surgical correction of an anterior open bite.28 The permanent complications (type VI) observed most frequently were IAN neurosensory disturbances (3.0%),19,23,37 skeletal relapse (2.1%),22,38 condylar resorption (2.1%),22,38,39 and permanent (periodontal) damage to the central incisors (1.8%).25,26,30,31,40,41 Single cases of chin ptosis31 lingual nerve neurosensory disturbance27 and severe anterior open bite33 were also reported incidentally

    DISCUSSIONSwennen et al5 published a review of 109 clinical articles on craniofacial DO, in which 70% of all patients underwent mandibular lengthening or widening. Complications were reported in 72.3% of these articles, which indicates a possible under-reporting. An overall complication rate of 20% was seen in the mandibular lengthening group and 29.7% in the mandibular widening group. This differs slightly from the incidence of 37.4% reported herein. In the review by Swennen et al, the most common complications were device-related problems, IAN neurosensory disturbances, and infection; however their review covered both congenital and non-congenital mandibular hypoplasia.

    Thirty-two eligible clinical publications on MDO for developmental deformities were included in this systematic review further analysis of the details of complications. Angle class II mandibular hypoplasia and transverse mandibular deficiency were almost equally represented. The leading reported complication was a minor or local infection, which occurred in 45 patients (8.0%). This mild complication could be resolved with the administration of antibiotics and/or topical application of chlorhexidine. No failure of DO was seen in these patients. Overall incidences vary from 2.4% to 9.5%.5,6 In the class II mandibular hypoplasia group, a temporary IAN neurosensory disturbance was identified in 1.1%18,20–23 and a permanent disturbance in 3.0%.23,37 In the literature, incidences of (temporary) IAN hypoesthesia vary between 2.4% and 14.4%.4–6,42 In most publications these incidences were calculated in groups in which congenital, developmental, and acquired deformity patients were combined, which might have led to a higher incidence, due to the more complex surgery required for congenital and acquired (oncology, trauma, severe atrophic mandible) deformities. In a previous systematic review on MDO for congenital deformities, a total IAN neurosensory disturbance of

  • Complications were classified according to a previously introduced index for MDO,8 which is based on six categories classifying the impact of the complications and their further treatment and final results. Each category divides complications into those of surgical or technical origin and differentiates between treatments performed under local and general anaesthesia. Type I and II complications comprised 60% of all complications. Nevertheless, a permanent (type VI) complication was reported in more than 25% of all complications. The overall incidence of type VI complications was 9.6%. This is slightly higher than the 7.9% in MDO congenital deformities,8 which is unexpected, since MDO in congenital deformities is known to be more challenging and complication-prone due to the small and abnormal mandibular anatomy. In conclusion, this systematic review shows that there is a lack of homogeneous reporting on different indications for and types of MDO, hindering the calculation of the actual complication rate. This study, however, provides an overview of all complications of MDO performed for developmental, orthognathic deformities and has classified these complications using an index that will lead to more insight into the morbidity resulting from these MDO procedures.

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    6. Mofid MM, Manson PN, Robertson BC, Tufaro AP, Elias JJ, Vander Kolk CA. Craniofacial distraction

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    7. Nagy K, Kuijpers-Jagtman AM, Mommaerts MY. No evidence for long-term effectiveness of early osteodistraction

    in hemifacial microsomia. Plast Reconstr Surg 2009; 124:2061–71.

    8. Verlinden CR, van de Vijfeijken SE, Jansma EP, Becking AG, Swennen GR. Complications of mandibular

    distraction osteogenesis for congenital deformities: a systematic review of the literature and proposal of a

    new classification for complications. Int J Oral Maxillofac Surg. 2015 Jan;44(1):37-43

    9. Liberati A, Altman DG, Tetzlaff J, Mulrow C, Gotzsche PC, Ioannidis JP, et al The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: explanation

    and elaboration. Ann Intern Med 2009;151: W65–94.

    10. Azumi Y, Sugawara J, Nagasaka H, Kawamura H. Mandibular widening by intraoral distraction osteogenesis

    for severe telescopic bite correction: a report of 2 cases. World J Orthod 2007;8:221–31.

    11. Guilleminault C, Li KK. Maxillomandibular expansion for the treatment of sleepdisordered breathing:

    preliminary result. Laryngoscope 2004;114:893–6.

    12. Meyer U, Kleinheinz J, Joos U. Biomechanical and clinical implications of distraction osteogenesis in craniofacial

    surgery. J Craniomaxillofac Surg 2004;32:140–9.

    13. Ploder O, Kohnke R, Klug C, Kolk A, Winsauer H. Three-dimensional measurement of the mandible after

    mandibular midline distraction using a cemented and screw fixated tooth-borne appliance: a clinical study.

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    14. Sukurica Y, Gurel HG, Mutlu N. Six year follow-up of a patient treated with mandibular symphyseal distraction

    osteogenesis. J Craniomaxillofac Surg 2010;38:26–31.

    15. Takato T, Horii K, Komuro Y, Yonehara Y, Miyamoto M, Susami T. Bilateral mandibular lengthening by using

    an external lengthening device: a case report. Jpn J Plast Reconstr Surg 1994;37:519–24.

    16. Whitesides LM, Meyer RA. Effect of distraction osteogenesis on the severely hypoplastic mandible and

    inferior alveolar nerve function. J Oral Maxillofac Surg 2004;62:292–7.

    17. Yamauchi K, Takahashi T, Kaneuji T, Nogami S, Miyamoto I, Lethaus B. Pivot technique combined with

    mandibular backward distraction osteogenesis for the patient with high risk for relapse. J Craniofac Surg

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    18. Kewitt GF, Van Sickels JE. Long-term effect of mandibular midline distraction osteogenesis on the status of

    the temporomandibular joint, teeth, periodontal structures, and neurosensory function. J Oral Maxillofac

    Surg 1999;57: 1419–25.

    19. Merli M, Merli M, Triaca A, Esposito M. Segmental distraction osteogenesis of the anterior mandible for

    improving facial esthetics. Preliminary results. World J Orthod 2007;8:19–29.

    20. Raoul G, Wojcik T, Ferri J. Outcome of mandibular symphyseal distraction osteogenesis with bone-borne

    devices. J Craniofac Surg 2009;20:488–93.

    21. Rubio-Bueno P, Villa E, Carreno A, Naval L, Sastre J, Manzanares R, et al Intraoral mandibular distraction osteogenesis: special attention to treatment planning. J Craniomaxillofac Surg 2001;29:254–62.

    22. Sahoo NK, Rangarajan H. Comparative evaluation of vertical body osteotomy and sagittal split osteotomy

    for mandibular corpus distraction. J Oral Maxillofac Surg 2011; 69:381–9.

    23. van Strijen PJ, Breuning KH, Becking AG, Perdijk FB, Tuinzing DB. Complications in bilateral mandibular

    distraction osteogenesis using internal devices. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;

    96:392–7.

    24. Guerrero CA, Bell WH, Contasti GI, Rodriguez AM. Mandibular widening by intraoral distraction osteogenesis.

    Br J Oral Maxillofac Surg 1997;35:383–92.

    25. Gunbay T, Akay MC, Aras A, Gomel M. Effects of transmandibular symphyseal distraction on teeth, bone,

    and temporomandibular joint. J Oral Maxillofac Surg 2009; 67:2254–65.

    26. Mommaerts MY, Spaey YJ, Soares Correia PE, Swennen GR. Morbidity related to transmandibular distraction

    osteogenesis for patients with developmental deformities. J Craniomaxillofac Surg 2008;36: 192–7.

    27. Ow A, Cheung LK. Skeletal stability and complications of bilateral sagittal split osteotomies and mandibular

    distraction osteogenesis: an evidence-based review. J Oral Maxillofac Surg 2009;67: 2344–53.

    28. van Strijen PJ, Perdijk FB, Becking AG, Breuning KH. Distraction osteogenesis for mandibular advancement.

    Int J Oral Maxillofac Surg 2000;29:81–5.

    29. Hodge TM. American orthodontics Maurice Berman Prize 2005. J Orthod 2006;33: 160–71.

    30. Von Bremen J, Schafer D, Kater W, Ruf S. Complications during mandibular midline distraction: the first 100

    patients. Angle Orthod 2008;78:20–4.

    31. Alkan A, Ozer M, Bas B, Bayram M, Celebi N, Inal S, et al Mandibular symphyseal distraction osteogenesis: review of three techniques. Int J Oral Maxillofac Surg 2007;36:111–7.

    32. Ow A, Cheung LK. Bilateral sagittal split osteotomies versus mandibular distraction osteogenesis: a

    prospective clinical trial comparing inferior alveolar nerve function and complications. Int J Oral Maxillofac

    Surg 2010;39:756–60.

    33. Ow AT, Cheung LK. Bilateral sagittal split osteotomies versus mandibular distraction osteogenesis: which is

    better? Ann R Australas Coll Dent Surg 2008;19:55–7.

    34. Savoldelli C, Lesne V, Ciszek E, Lebeau J, Bettega G. [Symphyseal distraction: a simplified procedure]. Rev

    Stomatol Chir Maxillofac 2010;111:259–69.

    35. Uckan S, Veziroglu F, Arman A. Unexpected breakage of mandibular midline distraction device: case report.

    Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006; 102:21–5

    36. Weil TS, Van Sickels JE, Payne CJ. Distraction osteogenesis for correction of transverse mandibular deficiency:

    a preliminary report. J Oral Maxillofac Surg 1997;55:953–60.

    37. Wijbenga JG, Verlinden CR, Jansma J, Becking AG, Stegenga B. Long-lasting neurosensory disturbance

    following advancement of the retrognathic mandible: distraction osteogenesis versus bilateral sagittal split

    osteotomy. Int J Oral Maxillofac Surg. 2009 Jul;38(7):719-25.

    38. van Strijen PJ, Breuning KH, Becking AG, Tuinzing DB. Stability after distraction osteogenesis to lengthen

    the mandible: results in 50 patients. J Oral Maxillofac Surg 2004;62:304–7.

    39. Hamada T, Ono T, Otsuka R, Honda E, Harada K, Kurabayashi T, et al Mandibular distraction osteogenesis in a skeletal Class II patient with obstructive sleep apnea. Am J Orthod Dentofacial Orthop 2007;131: 415–25.

    40. Mommaerts MY, Polsbroek R, Santler G, Correia PE, Abeloos JV, Ali N. Anterior transmandibular osteodistraction:

    clinical and model observations. J Craniomaxillofac Surg 2005;33:318–25.

    41. Uckan S, Guler N, Arman A, Mutlu N. Mandibular midline distraction using a simple device. Oral Surg Oral

    Med Oral Pathol Oral Radiol Endod 2005;100:85–91.

    42. Shetye PR, Warren SM, Brown D, Garfinkle JS, Grayson BH, McCarthy JG. Documentation of the incidents

    associated with mandibular distraction: introduction of a new stratification system. Plast Reconstr Surg

    2009;123:627–34.

    57 58Chapter 3 Chapter 3

  • 4COMPLICATIONS OF MANDIBULAR DISTRACTION OSTEOGENESIS IN ACQUIRED DEFORMITIES: a systematic review of the literature

    Verlinden CRA, van de Vijfeijken SECM, Tuinzing DB, Becking AG, Swennen GRJ International Journal of Oral and Maxillofacial Surgery. 2015; 44: 956–964

    6059

  • ABSTRACTA systematic review of English and non-English language articles on the complications of all forms of mandibular distraction osteogenesis (MDO) 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 Controlled Trials Register yielded 644 articles published between 1966 and mid October 2013. Three hundred and twenty-one eligible articles were screened in detail. Complications related to MDO in acquired deformities were reported in 105 clinical articles, involving 1332 patients. Treatments included alveolar distraction osteogenesis (ADO), mandibular lengthening, DO in bone grafts, and bi-/trifocal transport disc DO (TDDO) for segmental mandibular defects. A high incidence of complications was seen in MDO for acquired deformities (ADO 44.4%; residual group 43.9%). An index for classifying complications in mandibular distraction osteogenesis, based on the impact and further treatment or final results, was used. In the ADO-group, soft tissue complications (8.0%), insufficient vector control (7.6%), temporary inferior alveolar nerve (IAN) neurosensory disturbances (6.5%), device-related problems (3.5%), mandible fractures (2.8%), insufficient bone formation (2.5%), and fracture of the transport disc (1.3%) were seen. In the residual group, temporary IAN neurosensory disturbances (13.4%), minor infection (5.3% ), DO failure (4.0%), and device-related problems (3.8%) were reported.

    Keywords: distraction osteogenesis, complication, complicated, failure, morbidity, mandible, mandibular, alveolar process, obstructive sleep apnoea, TMJ ankylosis, systematic review, PRISMA Statement

    INTRODUCTIONMandibular distraction osteogenesis (MDO) is a versatile technique that is applied in patients with congenital1, developmental2, and acquired mandibular deformities. After its introduction for the lengthening of the human mandible by McCarthy in 1992, a broad spectrum of indications and applications has arisen3.

    Vertical distraction osteogenesis of the alveolar bone (ADO) for dental implant placement as an alternative to bone grafting is a common indication4,5. This challenging technique is prone to a variety of complications6–9.

    Indications such as reconstructive surgery for segmental mandibular defects after ablative oncology surgery and post-traumatic patients and those with complications following prior surgery to the mandible have been treated successfully using a form of DO. Transport disc DO (TDDO) can be applied in a bifocal or trifocal manner, enabling DO in more than one location in the patient10. DO can also be used for bone regeneration in free vascularized bone grafts (e.g. fibula)11–14. Temporomandibular joint (TMJ) ankylosis can be treated successfully by means of DO for mandibular lengthening, and this can be done in combination with a gap arthroplasty in some patients15–17.

    The aims of this study were (1) to perform a systematic review of the literature on complications of MDO for acquired deformities, and (2) to classify all complications using a new classification1.

    61 62Chapter 4 Chapter 4

  • 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 statement18. 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 Library. Search terms expressing distraction osteogenesis were used in ‘AND’ combi