ABOUT THE AUTHOR MAXILLOFACIAL DISTRACTION OSTEOGENESIS. Experiences, results & dilemmas ISBN# Charlotte R.A

ABOUT THE AUTHOR Charlotte Verlinden (1978) werd geboren te Leiden als oudste dochter van Willem en Harmke Verlinden. Het doktersvak werd haar met de ...
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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

MAXILLOFACIAL DISTRACTION OSTEOGENESIS Experiences, results & dilemmas

Charlotte R.A. Verlinden

4

ACADEMISCH PROEFSCHRIFT

MAXILLOFACIAL DISTRACTION OSTEOGENESIS Experiences, results & dilemmas

Charlotte R.A. Verlinden

5

6

VRIJE UNIVERSITEIT

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.

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 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

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,

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

Copyright 2016 Charlotte R.A. Verlinden All 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.

De Boelelaan 1105

door Charlotte Rosalina Anna Verlinden geboren te Leiden

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Promotoren:

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prof.dr. T. Forouzanfar prof.dr. D.B. Tuinzing

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Paranimfen:

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Dr. J.F. Bremmer Drs. A.E. Dijckmeester

Dare to err and to dream ~ F. Schiller

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CONTENT

Chapter 1

General introduction

Chapter 2

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

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Chapter 3

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

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Chapter 4

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

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Chapter 5

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

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Chapter 6

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

99

Chapter 7

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

111

Chapter 8

Complications in transpalatal distraction osteogenesis: a retrospective clinical study

123

Chapter 9

General discussion & Summary

141

Chapter 10

Conclusion & future perspectives Acknowledgements About the author

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1 14

GENERAL INTRODUCTION

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Chapter 1

Chapter 1

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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, nonunions, 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.

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Chapter 1

Chapter 1

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Distraction osteogenesis of the craniofacial skeleton

Evolution of technique

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.

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

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.

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 distractorrelated problems and local infections. In 2001, Mofid et al published a review of 3278 cases of

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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.

Chapter 1

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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)

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Chapter 1

Chapter 1

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REFERENCES 1.

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

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

Jan:(250):8-26

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

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.

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.

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COMPLICATIONS 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

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Chapter 2

Chapter 2

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ABSTRACT

INTRODUCTION

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) 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.

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 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, evidencebased reports on the longterm results, relapse, and complications of MDO are limited.

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

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.

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Chapter 2

Chapter 2

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MATERIALS AND METHODS Literature search

Study selection and inclusion criteria

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.

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.

TABLE 1

TABLE 2

Primary and secondary keywords used for the systematic research.

PRIMARY KEYWORDS

SECONDARY KEYWORDS

Distraction

Mandible

Distraction osteogenesis

Mandibular

Lengthening

Alveolar

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).

Inclusion and exclusion criteria.

CONDITION

Non-congenital deformities

Complication Complicated

ARTICLE TYPES

Excluded from the systematic review

Failure

Included in the systematic review

124

Insufficient or no information on complications and/ or methods

57

Non-clinical articles (experimental, scientific, synopsis)

24

Non-(mandibular) distraction osteogenesis No translation available

Morbidity

NUMBER OF PAPERS (N)

4 11

Publication type, e.g. letter to the editor, discussion

5

Not available in international libraries

3

Clinical articles on complications in mandibular distraction osteogenesis for congenital deformities

81

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Chapter 2

Chapter 2

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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 1

FIGURE 2

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.

IDENTIFICATION

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.



SCREENING

 Records screened (n=644)

’

Records excluded based on abstract (n=335 )

’

Full-text articles excluded:

ELIGIBILITY

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

- 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)

INCLUDED

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

Records identified after initial search (n=973)

Records after removal duplicates and update (n=644)

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

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

- Not available (n=3) (n=22) Studies included in qualitative synthesis ( n=81 )

- Not available in International

libraries (n=8)

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Chapter 2

Chapter 2

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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 4 Characteristics of distraction osteogenesis in congenital deformities (n = 1258). DISTRACTION TYPE Mandibular lengthening

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.

NUMBER 1255

Bilateral

665

Unilateral

590



External, unidirectional

682



External, multidirectional



Internal, unidirectional



Internal, multidirectional

Mandibular widening Graft DO Site: Native Graft Junction

84 398 91 3 71 5 26 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.

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Chapter 2

Chapter 2

TABLE 5

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

COMPLICATIONS

NUMBER

Type I complications resolving spontaneously

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)

10

49,50,57,66,71

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

24

Incomplete osteotomy/premature ossification

Pain10,61,80

21

Fracture of a transport disc

Trismus10,58

12

Subtotal

10,45,52,63,84

8 1

10

50 (4.0%)

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

9

Minor occlusal disturbances48,59,87

7

Parotid gland injury10

2

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

Periodontal damage61

1

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

8

Asymmetrical distraction67

1

Relapse or skeletal anterior open bite55,58,76

3

Dentigerous cyst formation73,89

3

Carotid artery injury85

1

Mandible fracture10

1

Subtotal

77 (6.1%)

Type II medically or technically manageable complication, without hospitalization Local infection

Incorrect vector

55

10,70

Device-related problems (pin loosening or extrusion)

10,63,70,77,79

Device back-up/inadequate length10 Dehiscence/pin exposure Cellulitis

69,72,81

18,86

Contralateral open bite

Subtotal

16 (1.27%)

73

10,12,12,14,15,47,50,54,56,57,60,62,63,65,66,70;71,75,77,81,88

77

Subtotal

39 11

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

5

Skeletal open bite64,69,75,83,89

3

Hypertrophic scar formation

2

Permanent damage to teeth or follicles

14

Permanent facial nerve palsy

14

188 (14.9%)

Soft tissue dehiscence12

18

10,74,75,83,89

56,63,77,83,89,89

(Fibrous) non-union Type III surgically manageable complication requiring local anaesthesia only, without hospitalization

30 10,15,69,72,83,87,89

TMJ ankylosis

9

16,18,53,65

7

10,65,82

Relapse of distracted bone

5

18,48,82

2 (0.2%)

Pseudo-arthrosis

2

15,17

Permanent IAN neurosensory disturbance

90

Subtotal Total IAN, inferior alveolar nerve; TMJ, temporomandibular joint.

1 100 (7.9%) 433 (34.4%)

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Chapter 2

Chapter 2

36

DISCUSSION Mandibular 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, nonunion, 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-union 18. 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.

37

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.

Chapter 2

REFERENCES 1.

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.

2.

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.

3.

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;108(5):1103-14.

4.

Nagy K, Kuijpers-Jagtman AM, Mommaerts MY. No evidence for long-term effectiveness of early osteodistraction in hemifacial microsomia. Plast Reconstr Surg 2009 Dec;124(6):2061-71.

5.

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.

38

Zim S. Treatment of upper airway obstruction in infants with micrognathia using mandibular distraction osteogenesis. Facial Plast Surg 2007 May;23(2):107-12.

6.

Master DL, Hanson PR, Gosain AK. Complications of mandibular distraction osteogenesis. J Craniofac Surg 2010 Sep;21(5):1565-70.

7.

Paley D. Problems, obstacles, and complications of limb lengthening by the Ilizarov technique. Clin Orthop Relat Res 1990 Jan;(250):81-104.

8.

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.

9.

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 Feb;123(2):627-34.

10. Davidson EH, Brown D, Shetye PR, Greig AVH, Grayson BH, Warren SM, McCarthy JG. The evolution of mandibular distraction: device selection. Plast Reconstr Surg 2010 Dec;126(6):2061-70. 11. Liberati A, Altman DG, Tetzlaff J, Mulrow C, Gotzsche PC, Ioannidis JP, Ioannidis JP, Clarke M, Devereaux PJ, Kleijnen J, Moher D. The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: explanation and elaboration. Ann Intern Med 2009 Aug 18;151(4):W65-W94. 12. Burstein FD. Resorbable distraction of the mandible: technical evolution and clinical experience. J Craniofac Surg 2008 May;19(3):637-43. 13. Margulis A, Patel PK, Daw JL, Bauer BS. Distraction osteogenesis of the mandible with an internal bioresorbable device. J Craniofac Surg 2003 Sep;14(5):791-6. 14. Breugem C, Paes E, Kon M, van der Molen ABM. Bioresorbable distraction device for the treatment of airway problems for infants with Robin sequence. Clin Oral Investig 2012 Aug;16(4):1325-31. 15. Corcoran J, Hubli EH, Salyer KE. Distraction osteogenesis of costochondral neomandibles: a clinical experience. Plast Reconstr Surg 1997 Aug;100(2):311-5. 16. Stelnicki EJ, Hollier L, Lee C, Lin WY, Grayson B, McCarthy JG. Distraction osteogenesis of costochondral bone grafts in the mandible. Plast Reconstr Surg 2002 Mar;109(3):925-33. 17. Thomas DJ, Rees MJ. Fibrous ankylosis after distraction osteogenesis of a costochondral neomandible in a patient with grade III hemifacial microsomia. J Craniofac Surg 2001 Sep;12(5):469-74. 18. Wan DC, Taub PJ, Allam KA, Perry A, Tabit CJ, Kawamoto HK, Bradley JP. Distraction osteogenesis of

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costocartilaginous rib grafts and treatment algorithm for severely hypoplastic mandibles. Plast Reconstr Surg 2011 May;127(5):2005-13. 19. Denny A, Amm C. New technique for airway correction in neonates with severe Pierre Robin sequence. J Pediatr 2005;147(1):97-101. 20. Diner PA, Kollar E, Martinez H, Vazquez MP. Submerged intraoral device for mandibular lengthening. J Craniomaxillofac Surg 1997 Jun;25(3):116-23. 21. Eppley BL. Distraction lengthening of the mandibular costochondral graft: A precautionary note. J Craniofac Surg 2000;11(4):350-3. 22. Farina R, Castellon L, Nagelash E, Valladares S. A new way to anchor the external device in mandibular distraction: Three case reports with a Pierre Robin sequence. Int J Oral Maxillofac Surg 2011;40(5):471-4. 23. Iatrou I, Theologie-Lygidakis N, Schoinohoriti O. Mandibular distraction osteogenesis for severe airway obstruction in Robin Sequence. Case report. J Craniomaxillofac Surg 2010 Sep;38(6):431-5. 24. Labbe D, Badie Modiri B, Kaluzinski E, Compere JF. [Mandibular reconstruction of gunshot wounds by progressive bone distraction. Report of five cases]. Ann Chir Plast Esthet 1998 Apr;43(2):141-8. 25. Mahatumarat C, Chokrungvaranont P, Rojvachiranonda N. Mandibular distraction osteogenesis in unilateral craniofacial microsomia: preliminary report. J Med Assoc Thai 2001 Jun;84(6):811-20. 26. Ortiz Monasterio F, Molina F, Andrade L, Rodriguez C, Sainz Arregui J. Simultaneous mandibular and maxillary distraction in hemifacial microsomia in adults: avoiding occlusal disasters. Plast Reconstr Surg 1997 Sep;100(4):852-61. 27. Ortiz Monasterio F, Molina Montalva F, Sainz Arregui J. Maxilo-mandibular simultaneous distraction. Cir Plast Ibero-Latinoam 2002;28(3):187-94. 28. Preciado DA, Sidman JD, Sampson DE, Rimell FL. Mandibular distraction to relieve airway obstruction in children with cerebral palsy. Arch Otolaryngol Head Neck Surg 2004 Jun;130(6):741-5. 29. Rhee ST, Buchman SR. Pediatric mandibular distraction osteogenesis: the present and the future. J Craniofac Surg 2003 Sep;14(5):803-8. 30. Satoh K, Suzuki T, Uemura T, Hosaka Y. Maxillo-mandibular distraction osteogenesis for hemifacial microsomia in children. Ann Plast Surg 2002;49(6):572-8. 31. Schendel SA, Linck DW. Mandibular distraction osteogenesis by sagittal split osteotomy and intraoral curvilinear distraction. J Craniofac Surg 2004 Jul;15(4):631-5. 32. Shen W, Jie C, Chen J, Zou J, Ji Y. Mandibular distraction osteogenesis to relieve Pierre Robin severe airway obstruction in neonates: indication and operation. J Craniofac Surg 2009 Sep;20 Suppl 2:1812-6. 33. Stelnicki EJ, Boyd JB, Nott RL, Barnavon Y, Uecker C, Henson T. Early treatment of severe mandibular hypoplasia with distraction mesenchymogenesis and bilateral free fibula flaps. J Craniofac Surg 2001 Jul;12(4):337-48. 34. Sugihara T, Kamashima K, Igawa H, Ohura T, Yamamura M, Ohata N. Mandibular lengthening by gradual distraction in humans. Eur J Plast Surg 1995;18(1):7-10. 35. Scolozzi P, Herzog G, Jaques B. Simultaneous maxillo-mandibular distraction osteogenesis in hemifacial microsomia: a new technique using two distractors. Plast Reconstr Surg 2006 Apr 15;117(5):1530-41.

Chapter 2

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37. Wang C, Zeng Rs, Wang Jn, Huang Hz, Liu X, Wang A. Simultaneous maxillomandibular distraction osteogenesis in severe progressive hemifacial atrophy with two distractors. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011 Mar;111(3):292-7. 38. Williams JK, Maull D, Grayson BH, Longaker MT, McCarthy JG. Early decannulation with bilateral mandibular distraction for tracheostomy-dependent patients. Plast Reconstr Surg 1999 Jan;103(1):48-57. 39. Wittenborn W, Panchal J, Marsh JL, Sekar KC, Gurley J. Neonatal distraction surgery for micrognathia reduces obstructive apnea and the need for tracheotomy. J Craniofac Surg 2004 Jul;15(4):623-30. 40. Zenha H, Azevedo L, Rios L, Pereira A, Pinto A, Barroso ML, Barroso ML, Costa H. Bilateral mandibular distraction osteogenesis in the neonate with pierre robin sequence and airway obstruction: a primary option. Craniomaxillofac Trauma Reconstr 2012 Mar;5(1):25-30. 41. Brevi B, Lagana F, Piazza F, Sesenna E. Mandibular distraction osteogenesis with a small semiburied device in neonates: report of 2 cases. Ear Nose Throat J 2006 Feb;85(2):102-5. 42. Choi JY, Hwang KG, Baek SH, Lee JH, Kim TW, Kim MJ, Chang YI. Original sagittal split osteotomy revisited for mandibular distraction. J Craniomaxillofac Surg 2001 Jun;29(3):165-73. 43. Santamaria E, Morales C, Taylor JA, Hay A, Ortiz-Monasterio F. Mandibular microsurgical reconstruction in patients with hemifacial microsomia. Plast Reconstr Surg 2008 Dec;122(6):1839-49. 44. Judge B, Hamlar D, Rimell FL. Mandibular distraction osteogenesis in a neonate. Arch Otolaryngol Head Neck Surg 1999 Sep;125(9):1029-32. 45. Ayoub AF, Lalani Z, Moos KF, Wood GA. Complications following orthognathic surgery that required early surgical intervention: fifteen years’ experience. Int J Adult Orthodon Orthognath Surg 2001;16(2):138-44. 46. Burstein FD, Williams JK. Mandibular distraction osteogenesis in Pierre Robin sequence: Application of a new internal single-stage resorbable device. Plast Reconstr Surg 2005;115(1):61-7. 47. Cavaliere CM, Buchman SR. Mandibular distraction in the absence of an ascending ramus and condyle. J Craniofac Surg 2002 Jul;13(4):527-32. 48. Cho BC, Shin DP, Park JW, Baik BS. Bimaxillary osteodistraction for the treatment of facial asymmetry in adults. Br J Plast Surg 2001 Sep;54(6):491-8. 49. Cohen SR, Simms C, Burstein FD. Mandibular distraction osteogenesis in the treatment of upper airway obstruction in children with craniofacial deformities. Plast Reconstr Surg 1998 Feb;101(2):312-8. 50. Denny AD, Talisman R, Hanson PR, Recinos RF. Mandibular distraction osteogenesis in very young patients to correct airway obstruction. Plast Reconstr Surg 2001 Aug;108(2):302-11. 51. Denny AD, Kalantarian B. Mandibular distraction in neonates: a strategy to avoid tracheostomy. Plast Reconstr Surg 2002 Mar;109(3):896-904. 52. Diner PA, Tomat C, Soupre V, Martinez H, Vazquez MP. Intraoral mandibular distraction: indications, technique and long-term results. Ann Acad Med Singapore 1999 Sep;28(5):634-41. 53. Franco J, Carstens M. Mandibular distraction in infancy. Oper Tech Otolaryngol Head Neck Surg 2009;20(4):241-4. 54. Franco J, Coppage J, Carstens MH. Mandibular distraction using bone morphogenic protein and rapid distraction in neonates with Pierre Robin syndrome. J Craniofac Surg 2010 Jul;21(4):1158-61.

36. Tharanon W, Sinn DP. Mandibular distraction osteogenesis with multidirectional extraoral distraction device

55. Gabbay JS, Heller JB, Song YY, Wasson KL, Harrington H, Bradley JP. Temporomandibular joint bony ankylosis:

in hemifacial microsomia patients: Three-dimensional treatment planning, prediction tracings, and case

comparison of treatment with transport distraction osteogenesis or the matthews device arthroplasty. J

outcomes. J Craniofac Surg 1999;10(3):202-13.

Craniofac Surg 2006 May;17(3):516-22.

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56. Genecov DG, Barcelo CR, Steinberg D, Trone T, Sperry E. Clinical experience with the application of distraction osteogenesis for airway obstruction. J Craniofac Surg 2009 Sep;20 Suppl 2:1817-21. 57. Gifford T, Park A, Muntz H. Description of a novel technique to perform neonatal mandibular distraction. Laryngoscope 2008 Jun;118(6):1063-5. 58. Hollier LH, Kim JH, Grayson B, McCarthy JG. Mandibular growth after distraction in patients under 48 months of age. Plast Reconstr Surg 1999 Apr;103(5):1361-70. 59. Hurmerinta K, Peltomaki T, Hukki J. Unexpected events during mandibular distraction osteogenesis. Scand J Plast Reconstr Surg Hand Surg 2004;38(4):209-14. 60. Izadi K, Yellon R, Mandell DL, Smith M, Song SY, Bidic S, Bidic S, Bradley JP. Correction of upper airway obstruction in the newborn with internal mandibular distraction osteogenesis. J Craniofac Surg 2003 Jul;14(4):493-9.

Chapter 2

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75. Pluijmers BI, Koudstaal MJ, Wolvius EB, van der Wal KGH. Custom-made intraoral mandibular distraction as treatment for neonatal airway obstruction. Int J Oral Maxillofac Surg 2012 Feb;41(2):186-91. 76. Politi M, Sembronio S, Robiony M, Costa F. The floating bone technique of the vertical ramus in hemifacial microsomia: case report. Int J Adult Orthodon Orthognath Surg 2002;17(3):223-9. 77. Rachmiel A, Aizenbud D, Eleftheriou S, Peled M, Laufer D. Extraoral vs. Intraoral distraction osteogenesis in the treatment of hemifacial microsomia. Ann Plast Surg 2000;45(4):386-94. 78. Rachmiel A, Manor R, Peled M, Laufer D. Intraoral distraction osteogenesis of the mandible in hemifacial microsomia. J Oral Maxillofac Surg 2001 Jul;59(7):728-33. 79. Rachmiel A, Srouji S, Emodi O, Aizenbud D. Distraction osteogenesis for tracheostomy dependent children with severe micrognathia. J Craniofac Surg 2012 Mar;23(2):459-63.

61. Kita H, Kochi S, Yamada A, Imai Y, Konno N, Saitou C, Saitou C, Mitani H.. Mandibular widening by distraction

80. Ramanathan M, Parameshwaran AA, Jayakumar N, Raghaviah AM. Reactivation of trigeminal neuralgia

osteogenesis in the treatment of a constricted mandible and telescopic bite. Cleft Palate Craniofac J 2004

following distraction osteogenesis in an 8-year-old child: report of a unique case. J Indian Soc Pedod Prev

Nov;41(6):664-73. 62. Kollar EM, Diner PA, Vazquez MP, Accart G, Pirollo M. [Bone distraction using an external fixator: a new mandibular lengthening technic. A preliminary study apropos of 2 cases of children with mandibular hypoplasia]. Rev Stomatol Chir Maxillofac 1994;95(6):411-6. 63. Kolstad CK, Senders CW, Rubinstein BK, Tollefson TT. Mandibular distraction osteogenesis: at what age to proceed. Int J Pediatr Otorhinolaryngol 2011 Nov;75(11):1380-4. 64. Kunz C, Hammer B, Prein J. Manipulation of callus after linear distraction: A ‘lifeboat’ or an alternative to multivectorial distraction osteogenesis of the mandible? Plast Reconstr Surg 2000;105(2):674-9. 65. Lin SJ, Roy S, Patel PK. Distraction osteogenesis in the pediatric population. Otolaryngol Head Neck Surg 2007 Aug;137(2):233-8. 66. Maranes Galvez C, Martinez Plaza A, Fernandez Valades R, Liceras Liceras E, Martin Cano F, Cortes Sanchez R, Martínez Tellería A, Cabello A, García Medina B. [Mandibular distraction osteogenesis in patients with craniofacial malformation]. Cir Pediatr 2011 Apr;24(2):102-8. 67. Margaride LA, Breuer J, Traine E. Hemifacial Microsomia. Treatment by osteogenic mandibular distraction. Cir Plast Ibero-Latinoam 2002;28(3):179-85. 68. Miller JJ, Kahn D, Lorenz HP, Schendel SA. Infant mandibular distraction with an internal curvilinear device. J Craniofac Surg 2007 Nov;18(6):1403-7. 69. Miloro M. Mandibular Distraction Osteogenesis for Pediatric Airway Management. J Oral Maxillofac Surg 2010;68(7):1512-23. 70. Molina F, Ortiz Monasterio F, Yudovich M. Hemifacial microsomia. Treatment with mandibular distraction. Cir Plast Ibero-Latinoam 2002;28(3):163-78. 71. Morovic CG, Monasterio L. Distraction osteogenesis for obstructive apneas in patients with congenital craniofacial malformations. Plast Reconstr Surg 2000 Jun;105(7):2324-30. 72. Morovic I. Current management of Pierre Robin syndrome. Rev Chil Pediatr 2004;75(1):36-42. 73. Murray DJ, Chong DK, Sandor GKB, Forrest CR. Dentigerous cyst after distraction osteogenesis of the mandible. J Craniofac Surg 2007 Nov;18(6):1349-52. 74. Parashar SY, Anderson PJ, David DJ. An unusual complication of mandibular distraction. Int J Paediatr Dent 2006 Jan;16(1):55-8.

Dent 2007 Mar;25(1):49-51. 81. Roy S, Patel PK. Mandibular lengthening in micrognathic infants with the internal distraction device. Arch Facial Plast Surg 2006 Jan;8(1):60-4. 82. Sadakah AA, Elshall MA, Farhat AA. Bilateral intra-oral distraction osteogenesis for the management of severe congenital mandibular hypoplasia in early childhood. J Craniomaxillofac Surg 2009 Jun;37(4):216-24. 83. Scott AR, Tibesar RJ, Lander TA, Sampson DE, Sidman JD. Mandibular distraction osteogenesis in infants younger than 3 months. Arch Facial Plast Surg 2011 May;13(3):173-9. 84. Sidman JD, Sampson D, Templeton B. Distraction osteogenesis of the mandible for airway obstruction in children. Laryngoscope 2001 Jul;111(7):1137-46. 85. Sigler A, Gutierrez M, Harris A, Capiz M, Uriarte F. Carotid artery injury during mandibular distraction. Br J Oral Maxillofac Surg 2008 Jul;46(5):419-20. 86. Singh DJ, Glick PH, Bartlett SP. Mandibular deformities: single-vector distraction techniques for a multivector problem. J Craniofac Surg 2009 Sep;20(5):1468-72. 87. Spring MA, Mount DL. Pediatric feeding disorder and growth decline following mandibular distraction osteogenesis. Plast Reconstr Surg 2006 Aug;118(2):476-82. 88. Stelnicki EJ, Lin WY, Lee C, Grayson BH, McCarthy JG. Long-term outcome study of bilateral mandibular distraction: A comparison of Treacher Collins and Nager syndromes to other types of micrognathia. Plast Reconstr Surg 2002;109(6):1819-25. 89. Tibesar RJ, Scott AR, McNamara C, Sampson D, Lander TA, Sidman JD. Distraction osteogenesis of the mandible for airway obstruction in children: long-term results. Otolaryngol Head Neck Surg 2010 Jul;143(1):906. 90. Robinson RC, Knapp TR. Distraction osteogenesis in the craniofacial skeleton. Otolaryngol Clin North Am 2005 Apr;38(2):333-59, vii. 91. Kaban LB, Seldin EB, Kikinis R, Yeshwant K, Padwa BL, Troulis MJ. Clinical application of curvilinear distraction osteogenesis for correction of mandibular deformities. J Oral Maxillofac Surg 2009 May;67(5):996-1008. 92. Tibesar RJ, Sidman JD. Mandibular distraction osteogenesis in the pediatric patient. Curr Opin Otolaryngol Head Neck Surg 2008 Dec;16(6):548-54.

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3 44

COMPLICATIONS 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

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Chapter 3

46

ABSTRACT

INTRODUCTION

A 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%.

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.

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

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.

47

Chapter 3

Chapter 3

48

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

Excluded from the systematic review

Non-developmental deformities

Included in the systematic review

Insufficient or no information on complications and/or methods

NUMBER OF PAPERS (N) 180 20

Non-clinical articles (experimental, scientific, synopsis)

9

No translation available

3

Publication type, e.g. discussion

1

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.

Chapter 3

FIGURE 1. Type I.

Chapter 3

Distraction osteogenesis complication index 8

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

FIGURE 2. IDENTIFICATION

49

Records identified after initial search (n=973)



SCREENING

 Records screened (n=644)

’

Records excluded based on abstract (n=394)

’

Full-text articles excluded:

ELIGIBILITY

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

- 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)

INCLUDED

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.

Flowchart of the search and selection procedure using PubMed, EMBASE, and The Cochrane Library

Records after removal duplicates and update (n=611)

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

50

- Not available (n=3) (n=22) - Not available in Studies included in qualitative synthesis ( n=32 )

International libraries (n=8)

51

Chapter 3

Chapter 3

RESULTS The 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

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

52

0

Type IV technical complication, necessitating general anaesthesia for correction 10



Device failure (requiring replacement)17,18,25



Broken distraction rod31,32

9



Fracture of device26,45

4



Insufficient length38

1

Subtotal

24 (4.2%)

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

Type I complications resolving spontaneously within 6 months



Premature consolidation25,46

6

40



Incomplete osteotomy18,31,32

3

Surgical correction of severe open bite32

1



Temporary IAN neurosensory disturbances16–20,29



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

7





Hardware-related pressure ulcers24,31,32

7

Subtotal



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

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



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

45



Chin ptosis26

1



Anterior open bite/incorrect vector31,32

14



Anterior open bite39

1

2



Lingual nerve neurosensory disturbance31

1

Non-compliance Subtotal

61 (10.8%)

Subtotal Total IAN, inferior alveolar nerve; TMJ, temporomandibular joint.

54 (9.6%) 211 (37.3%)

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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

Chapter 3

54

DISCUSSION Swennen 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 1 year) neuro-sensory disturbance of the IAN varies. The risk of neurosensory disturbance seems to be lower in DO compared with BSSO. The variation in reported prevalence of neurosensory disturbance after orthognatic surgery depends on whether objective measurements or subjective self-reports are used8,46,50. The results from objective clinical neurosensory tests do not always correspond with patients’ subjective reports of neurosensory disturbance5. Patients seem to accept a mild neurosensory disturbance and might report sensory function as being normal, despite a slightly altered sensa-tion5,20,32,49. 87–100% of patients who underwent orthognathic surgery were satisfied with the result and would recommend the treatment to others, independent of a neurosensory disturbance5,9,13.

Distraction osteogenesis (DO) of the mandible using intraoral devices has proved to be a reliable alternative method for advancement of the retrognathic mandible11,17,19,22–25,38,39. DO has specific clinical benefits over BSSO, but complications still exist and may involve injury of the IAN. The incidence of neurosensory deficits after DO ranges from 0% to 52% of patients21,45.

The aim of this retrospective study was to assess the occurrence of long-lasting (>1 year) neurosensory disturbance and overall patient satisfaction. Based on the results, an indication can be given whether DO is a more suitable procedure for the surgical correction of the retrognathic mandible compared with BSSO. This study also compared the results of a sub-jective patient questionnaire with the results of a recently developed objective neurosensory ‘quick test’.

85

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Chapter 5

86

MATERIAL AND METHODS A retrospective clinical study was per-formed in 91 patients (38 males and 53 females) who had undergone mandibular advancement surgery. Group 1 consisted of 45 patients (10 males, 35 females; mean age 26.4 years (SD 10.4), age range 15–59 years) in which a BSSO had been performed at the University Medical Centre Groningen. Group 2 consisted of 46 patients (28 males, 18 females; mean age 15.0 years (SD 1.5), age range 11–18 years who had undergone DO of the mandible in the VU Medical Centre in Amsterdam and the Kennemer Gasthuis in Haarlem. Patients were selected based on the information on the operative procedure in their medical records and available surgery reports. Patients were included if either a BSSO or a bimaxillary procedure (group 1) or bilateral mandibular DO (group 2) had been performed. Within each group, surgery was always performed in a residency program under the super-vision of the same surgeon (JJ in group 1; AB in group 2). Patients who had previously undergone orthognathic surgery and patients in whom a genioplasty was concomitantly performed were excluded. In all patients in the BSSO group, rigid fixation was carried out using monocortical bone-screws and titanium miniplates. Exact data about the distance of advancement were not available in all surgery reports, but the indications for advancement did not exceed 7 mm in group 1 and 12 mm in group 2. Of the 126 patients who were invited for this study, 91 patients (72%) accepted the invitation and gave their consent for participation. The patients were requested to complete a questionnaire and to undergo a clinical examination. Clinicians who per-formed the interviews and examinations were not involved in the surgical treatments. The subjects were interviewed with special focus on their subjective experience of neurosensory disturbance after the surgical procedure. They were asked about perceived neurosensory disturbance in the distribution area of the IAN, duration of these changes, and any altered quality of life caused by these changes. A 10 cm visual analogue scale graded from 0 (no discomfort) to 10 (intolerable discomfort) was used to estimate the degree of disturbance. The grades were interpreted as follows: 0–2 mild discomfort; 2–4 mild to moderate discomfort; 4–6 moderate dis-comfort; 6–8 moderate to severe discom-fort; and 8–10 severe discomfort1. Patients were also asked about their satisfaction with the result of the operation in relation to the neurosensory disturbance.

After completion of the questionnaire, a recently developed objective clinical neurosensory test was performed to assess long-lasting neurosensory distur-bance36,40. This neurosensory test consisted of a mechanoceptive test (light touch sensation; large myelinated A-a and A-b afferent fibres) and a nociceptive test (cold sensation; small myelinated A-d and unmyelinated C afferent fibers) and was performed by examiners who were unaware of the outcome of the question-naire (JW in group 1; CV in group 2). The cutaneous areas of the lower lip and chin (both left and right side) were selected as test sites for assessment of the neurosen-sory function of the IAN. The upper lip was tested as a control site. The mechanoceptive test was per-formed using a Semmes-Weinstein Pressure Aesthesiometer Monofilament #3.61 (North Coast Medical, Inc., San Jose, CA, USA), which applies a calibrated force of 455 mg40,41. The real stimulus was touching the test site, whereas approaching the test site with the filament turned away was the fake stimulus (Figure 1). Touching the test site with monofilament #3.61 was detected in 99% of cases41. The nociceptive test was performed using a heat-conducting aluminium stick (shaft diameter 7.0 mm, tip diameter 2.0 mm) as a real stimulus (22 8C; cold sensation) (Figure 2). The fake stimulus was produced by a non-heatconducting Perspex1 stick of neutral temperature. At touching, the difference in temperature between the aluminium and Perspex1 sticks was distinguished by 99% of healthy subjects36,40,41. FIGURE. 1.

FIGURE. 2.

Test stimulus with SW Pressure Aesthesiometer

Test stimulus with a heat-conducting aluminium

Monofilament #3.61 (North Coast Medical, Inc., San Jose,

stick (shaft diameter 7.0 mm, tip diameter 2.0 mm)

CA, USA), which applies a calibrated force of 455 mg.

at 22 8C.

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Chapter 5

In both the mechanoceptive and the nociceptive neurosensory tests, the patients were offered a randomised series of seven successive pairs of stimuli. While the patients kept their eyes closed, either a real stimulus or a fake stimulus was pre-sented after announcement of the first or second interval. After each pair, the patients were asked to report if the real stimulus was perceived at either the first or the second delivery. The subjects were forced to answer this question, regardless of whether the real stimulus was felt or not (2-alternate forced-choice method). The outcome of the neurosensory test was negative (no neurosensory disturbance) if the real stimulus was correctly detected in each of the seven consecutive pairs of applications. If a wrong answer was given (i.e. false detection of the real stimulus) the outcome of the test was positive, indi-cating neurosensory disturbance. By applying seven pairs of stimuli, the chance of a false-negative outcome is 1 year after surgery

27

21

48

Speaking/eating

1

2

3

NSD >1 year after surgery and still bothering

23

12

35

Touching/speaking

2

0

2

Touching/eating

4

2

6

Touching/kissing

2

0

2

Touching/other

2

0

2

Touching/speaking/kissing

2

0

2

Touching/speaking/eating/kissing

1

0

1

Always

4

2

6

23

12

35

TABLE 2. Location of long-lasting neurosensory disturbance after surgery Lower lip + chin

Number of IANs

BSSO

DO

Total

19

14

33

Lower lip

2

1

3

Chin

3

0

3

Other areas

2

7

9

26

22

48

Total

Total

TABLE 5. Degree of discomfort caused by the long lasting neurosensory disturbance

Number of IANs

BSSO

DO

No discomfort

2

-

Mild discomfort

9

0

9

Mild to moderate discomfort

8

7

15

Moderate discomfort

0

2

2

Moderate to severe discomfort

3

2

5

Severe discomfort Total

Total 2

1

1

2

23

12

35

91

Chapter 5

Chapter 5

92

Degree of satisfaction

DISCUSSION

44 of 45 patients in group 1 (98%) were satisfied with the final result of the BSSO. 98% would undergo the surgery again if they were in the same situation or recommend the surgery to another person. In group 2, all patients (100%) were satisfied with the final result of the DO procedure. 41 patients (89%) would undergo the procedure again or recommend the surgery to others.

Neurosensory disturbance following orthognathic surgery has been extensively described for BSSO, but its reported prevalence varies. Immediate postoperative paraesthesia is common, with reports of 80–100% incidence. Follow-up of the patients has shown a prevalence of 34– 97% in the first week after surgery, and a prevalence of 0–85% 1 or 2 years postoperatively6,16,20,26,29,44,47–49. Few studies reported the prevalence of neurosensory disturbance after DO, but it ranges from 0% to 52% of patients21,45. The prevalence of long-lasting neurosensory disturbance after BSSO and DO found in this study (8% in group 1; 10% in group 2, P > 0.05) is relatively low within the limits found in the literature.

All of the 34 patients (100%) representing the 48 nerves with long-lasting neurosensory disturbance were satisfied with the result. One patient in group 1 would not undergo the BSSO again or recommend it to others, whereas 3 patients in group 2 would not undergo the DO procedure again or recommend it to others.

Neurosensory ‘quick test’ Of the 90 IANs in the BSSO group, seven nerves (7.8%) in six patients tested positive, which means that in these cases longlasting (>1 year postoperative) neurosensory disturbance existed at the time of testing. Nine nerves (9.8%) in seven patients tested positive in the DO group. In 27 cases in group 1 (18 patients) the neurosensory disturbance was reported to have existed for over a year and was still present. In 24 cases (89% of these 27) the neurosensory deficit was reported in the cutaneous area of the lower lip, the chin or both. Of these 24 cases, only 5 could be objectively measured (21%). In two cases, long-lasting sensory disturbance was not subjectively reported, but nociceptive disturbance was found positive with the objective test. In 15 of the 21 nerves (71%) in group 2 that were reported to show long-lasting neurosensory disturbance, the reported disturbance was present in the cutaneous area of the lower lip, the chin or both. Of these 15 nerves, 4 could be measured (27%), while 11 could not be objectified. In 5 cases, long-lasting neurosensory disturbance was not subjectively reported, but sensory disturbance tested positive (Table 6).

There could have been false-negative test results due to the materials used. The mechanoceptive test as a part of the neurosensory ‘quick test’ was performed using Semmes-Weinstein monofilament #3.61. It was found, that the touch of a monofilament #3.61 could be detected in 99% of cases41. The touch detection threshold corresponding to 95% positive responses are calibrated to be accomplished by using Semmes-Weinstein monofilament #2.44 in the lower lip and #2.83 in the mental region of female subjects, and by using #2.83 in the lower lip and #3.22 in the mental region of male subjects. Thus, the monofilament #3.61, as used in this study, could have led to a higher number of false-negative test results of the mechanoceptive sensory modality. The authors findings accord with these observations. A similar difference between the 99th and 95th percentiles may mean that the outcome of the nociceptive test may also include false-negative test results. The nociceptive test using a heat-conducting aluminium stick uses the temperature (skin temperature vs. stick temperature) as an index. Kabasawa et al reported a novel evaluation method for neurosensory disturbance using a heat flux technique. This method allows the accurate measurement of thermal sensation in a short time and the quantitative measurement of warm and cold sense thresholds by using a thermostimulator. This thermode consists of Peltier elements that are either cooled or warmed linearly by 0.1 8C/s until the subject feels a cold or warm sensation18.

TABLE 6. Long lasting neurosensory disturbance: subjective report vs. objective measurement

BSSO

Number of IANs DO

Total

Reported and measured

5

4

9

Reported, not measured

19

11

30

Measured, not reported

2

5

7

Total reported

24

15

39

Total measured

7

9

16

In group 1, a concomitantly performed BSSO and Le Fort I procedure may have resulted in a neurosensory disturbance of the upper lip, although it was not reported by the patients and it was not objectively tested positive.

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There is little information in the literature on whether long lasting sensory disturbance after orthognathic surgery influences patient satisfaction with the outcome of the procedure. For BSSO, Westermark et al found an overall patient satisfaction of 84%42. Al-Bishri et al found that 91% of patients were satisfied with the result of the operation, and of the 4 dissatisfied patients only one patient was dissatisfied because of neurosensory disturbance1. In general, the longterm satisfaction rate in patients who underwent orthognathic surgery is 87–100%5,9,13. In cases where a neurosensory disturbance of the lower lip and chin existed, it was reported normal or not uncomfortable by the patients5,20,28,31,49 In this study, a very high satisfaction rate was found after BSSO (98%) and DO (100%). This indicates that the discomfort of the nerve damage seems to be outweighed by the positive functional and aesthetic results. Together with the relatively low prevalence of long-lasting neurosensory disturbance, the high degree of patient satisfaction confirms the idea that both procedures are highly suitable for the surgical correction of the retrognathic mandible. In group 1, 44 subjects would undergo the BSSO procedure again if they were in the same situation, whereas 43 patients in group 2 would do so with regard to the DO procedure (98% vs. 89%, respectively; P > 0,05). This finding could be attributed to factors that contribute to the level of discomfort experienced by patients under-going DO, such as the duration of the distraction period and the disadvantage of requiring a second surgical intervention to remove the distraction device, and the procedure itself37. The relationship between objective assessments and the patients’ subjective evaluation of neurosensory disturbance is unclear. Patients reported normal neuro-sensory function even though objective testing indicated continued neurosensory deficit14. Conversely, patients appeared to over-report (mechanoceptive) neurosen-sory problems when compared with the objectively tested level of disturbance8. Pepersack et al found a reasonably high prevalence (61% of 123 patients) of per-manent sensory alteration at least 5 years after BSSO for mandibular prognathism, testing with both tactile and thermal stimuli and sharp/blunt discrimination. Sub-jectively, only 42% of the patients reported sensory disturbances in the lower lip30. Coghlan et al reported a higher prevalence of neurosensory disturbance among 19 patients in clinical neurosensory testing (66% of the nerves) than of subjective symptoms (26% of the nerves) two years after BSSO7. Fridrich et al concluded that patients seem to adapt to a neurosensory disturbance and report nor-mal neurosensory function even though objective testing indicates continued sensory deficit14.

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94

Leira et al objectively found a sensory disturbance in 34% of the operated sides 4 days after BSSO and in 8% at 6 months after BSSO, whereas subjective sensory disturbance was present in 54% immedi-ately after the operation, and in 34% at 6 months after BSSO20. Cunningham et al found that more than 70% of 101 patients subjectively reported neurosensory pro-blems, but objective assessment identified neurosensory deficits in less than 60% of the patients. They concluded that patients seem to over-report neurosensory distur-bance8, a conclusion that is supported by the results of the present study. In earlier studies, a significant association between patient’s age and neurosen-sory disturbance was reported2,28,29,35,43. The results of these studies showed significantly more neurosensory disturbance in patients aged 30 years or older than in patients younger than 30 years. Upton et al suggested that the higher prevalence of neurosensory disturbance in older patients may not be due to a greater risk of nerve damage, but to poorer regeneration of damaged nerves35. Some studies have not revealed significant differences in the incidence of neurosensory disturbance by age, but this may be due to a small age range14. The subjects in group 1 (mean age 26.4 years) were significantly older than the patients in group 2 (mean age 15.0 years; P < 0.05). The results show that the dif-ference in the mean age of the subjects in the two groups was significant with regard to the subjectively reported neurosensory disturbance (29.8 years (BSSO), 14.8 years (DO); P < 0.05) as well as with regard to the objectively measured sensory deficit (32.5 years (BSSO), 15.1 years (DO); P < 0.05). There was no significant difference between group 1 and 2 with regard to the prevalence of both subjectively reported neurosensory disturbance (30% and 23%, respectively; P > 0.05) and of the objectively measured sensory deficit (8% and 10%; P > 0.05). If the mean age was about the same in both groups, the results of this study would imply that the prevalence of long-lasting neurosensory disturbance was relatively higher in the DO group, although there was no significant difference between the results. The magnitude of mandibular advancement also influences the prevalence of neurosensory disturbance. Ylikontiola et al found significantly higher rates of neurosensory disturbance in patients with mandibular movements larger than 7 mm49. Westermark et al did not find a significant correlation between the neurosensory deficit and the magnitude of mandibular movement44.

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Whitesides et al studied the function of the inferior alveolar nerve after mandibular DO of more than 10 mm advancement (rate 1 mm/ 24 h). They found that all nerves recovered to preoperative (40% of the nerves) or near preoperative (60% of the nerves) values within 1 year 45.

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and satisfaction with outcome. Br J Oral Maxillofac Surg 1995: 33: 9–14. 28. Ylikontiola L. Neurosensory disturbance after bilateral sagittal split osteotomy. Thesis University of Oulu 2002: p. 53–61. 29. Fridrich K, Holton T, Pansegrau K, Buckley M. Neurosensory recovery following the mandibular bilateral sagittal split osteotomy. J Oral Maxillofac Surg 1995: 53: 1300– 1306. 30. Ylikontiola L, Kinnunen J, Oikarinen K. Comparison of different tests assessing neurosensory disturbances after bilateral sagittal split osteotomy. Int J Oral Maxillofac Surg 1998: 27: 417–421. 31. Hunsuck EE. A modified intraoral sagittal splitting technic for correction of mandibular prognathism. J Oral Surg 1968: 26: 250–253. 32. van Strijen PJ, Breuning KH, Becking AG, Perdijk FB, Tuinzing DB. Cost, operation and hospitalization times in distraction osteogenesis versus sagittal split osteotomy. J Craniomaxillofac Surg 2003: 31: 42–45. 33. Coghlan KM, Irvine GH. Neurological damage after sagittal split osteotomy. Int J Oral Maxillofac Surg 1986: 15: 369– 371. 34. Cunningham LL, Tiner BD, Clark GM, Bays RA, Keeling SD, Rugh JD. A comparison of questionnaire versus monofilament assessment of neurosensory deficit. J Oral Maxillofac Surg 1996: 54: 454–459. 35. Zuniga J, Meyer R, Gregg J, Miloro M, Davis L. The accuracy of clinical neurosensory testing for nerve injury diagnosis. J Oral Maxillofac Surg 1998: 56: 2–8. 36. Vriens JP, van der Glas HW, Bosman F, Koole R, Moos KF. Information on infraorbital nerve damage from multitesting of sensory function. Int J Oral Maxillofac Surg 1998: 27: 20–26. 37. Ylikontiola L, Kinnunen J, Laukkanen P, Oikarinen K. Prediction of recovery from neurosensory deficit after bilateral sagittal split osteotomy. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000: 90: 275–281. 38. Nishioka GJ, Zysset MK, Van Sickels JE. Neurosensory disturbance with rigid fixation of the bilateral sagittal split osteotomy. J Oral Maxillofac Surg 1987: 45: 20–26.

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SYMPTOMATIC VENOUS THROMBOEMBOLISM IN ORTHOGNATHIC SURGERY AND DISTRACTION OSTEOGENESIS: a retrospective cohort study of 4127 patients

Verlinden CRA, Tuinzing DB, Forouzanfar T British Journal of Oral and Maxillofacial Surgery. 2014; 52: 401–404

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Chapter 6

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ABSTRACT

INTRODUCTION

Venous thromboembolism is a common postoperative complication, and orthopaedic procedures are particularly at risk. We designed a retrospective, single centre, observational, cohort study of 4127 patients (mean (SD) age 27 (11) years) who had elective orthognathic operations or distraction osteogenesis between January 1970 and February 2012 at the VU University Medical Centre, Amsterdam, to investigate the incidence in this group over the 42-year period, 2 patients developed symptomatic venous thromboembolism (1 woman had a deep vein thrombosis (DVT) and 1 man had a DVT and pulmonary embolus) postoperatively. In relatively young patients with low to moderate risk factors and short hospital stay this type of operation is associated with a particularly low risk of developing thrombosis. It could be advisable to limit the use of thromboprophylaxis to patients at high risk or according to hospital guidelines.

Venous thromboembolism comprises deep venous thrombosis (DVT) and pulmonary embolism (PE), and is a common complication after operation, trauma, and prolonged immobilization. It can lead to further morbidity that comprises post-thrombotic syndrome, pulmonary hypertension and mortality.1–5 It is often asymptomatic and misdiagnosed, and can there-fore cause appreciable morbidity and mortality. Cohen et al designed an epidemiological model to estimate the number of community and hospital-related incidents and recurrence of non-fatal venous thromboses and thrombosis-related deaths in Europe. According to the model an estimated 465 715 cases of DVT, 295 982 cases of PE, and 370 012 related deaths occur yearly.5 This makes it one of the major health issues in the European Union.5,6 The reports on incidence vary and are influenced by factors as age, race, sex, necropsy evidence, and recurrent episodes.6 Strong clinical risk factors associated with thromboembolic disease are orthopedic trauma (hip or leg), major orthopedic operations (hip or knee replacement), major general surgical operations, major trauma, and injury to the spinal cord. Age (>40 years), obesity, recurrent disease, cancer, pro-longed immobilization (>5 days) and hormone replacement are moderate risk factors (Table 1).1,4,13

Keywords: Oral and maxillofacial surgery; Orthognathic surgery; Distraction osteogenesis; Pulmonary embolism; Thrombosis; Deep vein thrombosis; Venous thromboembolism (VTE)

TABLE 1.

Risks of venous thromboembolism in surgical patients without prophylaxis, according to Geerts et al1 Data are given as percentage risk.

LEVEL OF RISK

DVT (CALF)

DVT (PROXIMAL)

PE (CLINICAL)

PE (FATAL)

2

0.4

0.2