Is there any determinant for successful distraction osteogenesis of the mandible in hemifacial microsomia patients?

구순구개열

Vol. 7, No. 2 2004

쑤

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^ Is there any determinant for successful

더혜벼

distraction osteogenesis of the mandible in

&지引

hemifacial microsomia patients?

Seung-Hak Baek

Dept, of Orthodontics, College of Dentistry, Seo 나 National University

厂

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반안면왜소증 환자의 하악골 신장술시 치료결과의 차이에 기여하는 인자

백승학

서울대학교 치과대학 치과교정 학교실

본 연구의 목적은 반안면왜소증 환자의 하악골 신장술시 초기 치아골격 특성들 중에서 치료결과의 차이에 기여하는 인자를 찾는 것이다. 치료전의 치아골격 특성, 골신장술의 효과와 그 유지상태를 관찰하기 위하여 골신장술 직전(T0), 직후(T1), 추적 2년후(T2)에 측모와 정모 두부방사선 계측사진을 촬영하여 전후방, 수직, 치아, 비대칭 항목들을 계측하였다. T2 시기의 계측 결과에 따라서 환자들을 1군(양호군, 10명)과 2군(불량 군, 9명)으로 분류하였다. 두 군에서 각 시기와 TO-T1, T1-T2 동안의 변화량의 차이를 Mann-Whitney U test, Wilcoxon signed independent t-test, rank test, ANOVA test를 사용하여 분석하였다. Pruzansky type이 골신 장술의 성공과 실패 여부와 관계가 깊게 나타났다. T0 시기에 2군은 1군에 비하여 하악골이 후방위치되었고, 하악지 고경(ramus height)이 짧았고, 하악각(gonial angle)이 컸으며, 이환측 하악지가 내측경사되었고, 이환 측으로의 이부변위 (chin point deviation)가 크게 나타났다. 1군에서 골신장술의 주요한 효과는 하악지 고경 의 증가, 하악골의 전방위치, 하악각의 증가, articular an이e의 감소에 따른 하악골의 반시계방향 회전, 이환 측의 하악지 경사의 증가, 교합면경사와 이부변위의 개선으로 나타났다. 그러나 2군에서는 골신장술을 시행 했을 때 1군에 비하여 하악골이 시계방향으로 회전되었고 하악지 고경의 증가량이 작게 나타났다. T2 시기

에 2군에서는 하악골의 반시계방향 회전이 나타났고, 하악지 성장이 일어나지 않았으나, 1군은 반대의 경향

을 보였다. 이러한 인자들이 골신장술 결과의 차이에 기여하는 것으로 생각된다.

Key word: 골신장술, 반안면왜 소증 , 하악골 , 종단적 결과

구순구개

7:77-91, 2004

77

Seung-Hak Baek

Introduction

Hemifacial microsomia (HFM) is the second most prevalent congenital craniofacial anomaly,13 which involves skeletal, soft tissue, and neuromuscular components of the first and second branchial arches.1’

4-6 Twenty to thirty percent of these patients show some degree of bilateral deformities.7

HFM represented a spectrum of clinical findings such as hypoplasia of the mandibular ramus and condyle, confinement of the maxillary vertical growth, external and/or middle ear defects, involvement of some cranial suture, buccal soft tissue, facial nerve, and muscles in the affected side.8" Several classification systems have been developed in an attempt to both qualify and quantify the severity of the skeletal and soft tissue deformity seen in these patients.3’6’9-,3'20

The spectrum of mandibular malformation ranges from a small but normally shaped ramus and temporomandibular joint to complete absence of these structures.4 Asymmetric growth of the mandible results in a canting of the occlusal plane superiorly toward the affected side. Tooth development is delayed on the affected side and abnormality of tooth dimension, especially the posterior teeth, in the affected side is proportionate to the degree of mandibular deformity.21

Bone lengthening by osteotomy (corticotomy) and distraction osteogenesis (DO) of long bones was first described by Codvilla22 and popularized by Ilizarov.23 Since McCarthy et al. 24 successfully elongated the

human mandible by DO, it has been advocated as an effective and safe technique to increase both ramus height and mandibular body length in young patients with HFM.

Advantages of DO in comparison to conventional surgical techniques are minimalization of surgery and admission time, no bone graft, no donor morbidity, no intermaxillary fixation, remodelling of the mandible, simultaneous increase of soft tissues, and possible improvement of symmetry at an early age.25’27 Disadvantages and complications of DO are pin tract infection and scar formation in extraoral device case, osteomyelitis, pain, nerve injury, tooth germ injury, angular deviation, failed or delayed ossification, possible relapse, and the frequent need for a second operation after growth has completed.28

After DO, most cases still require placement of the removable mandibular occlusal splint and/or the fixed orthodontic appliance to complete treatment.29 31

Although there are several follow up studies about results of facial asymmetry correction using DO,32’33 there are a few studies about the sagittal and vertical relationships between the maxilla and the mandible.

Since the same treatment protocol may produce a good result in one but other patient, it is necessary to divide samples according to retention results. Purpose of the present study was to investigate sagittal, vertical, and asymmetric skeletal characteristics that could determine success or failure of DO of the mandible in HFM patients using longitudinal follow­

up data.

78

구순구개열

Vol. 7, No. 2 2004

Materals and methods

The records of 19 HFM patients (10 m시es, 9 females) who were treated with DO of the mandible in the Department of Plastic Surgery, Seoul National University Hospital and orthodontic therapy in the Department of Orthodontics, Seoul National University Dental Hospital from 1998 to 2002 were

examined. ,

Posteroanterior (PA) cephalometric radiograph was used for asymmetry analysis and lateral cephalometric radiograph for sagittal and vertical relationships between the maxilla and the mandible. The patient was positioned in cephalostat so that the Frankfort horizontal (FH) and interpupillary lines were parallel to the floor. To locate ear rode accurately and reliably, the amounts of distance from the alar base of the nose to the pina of the ear and from distal canthus of the eye to pina of the ear in the non-affected side were measured. In the affected side, the same amounts of distance were copied along with FH and interpupillary lines.

All patients were treated by extraoral bi-directional distractor appliance (Orthopix Pennig Minifixator, Orthopix, Germany) because it can control vector during distraction period. The oblique osteotomy line was designed in Pruzansky type I and Ila and the horizontal osteotomy line in Pruzansky type lib and III. The direction of pin fixation is defined by the technique described by Losken et a/.34

Presurgical orthodontic preparation consisted of the lingual arch at the maxillary and the mandibular

dentition in order to use elastic traction during distraction. The distraction protocol was as follows:

latency period, 5 days; distraction amount, 1 or 2mm a day; distraction rhythm, 0.5mm twice a day or 1.0mm twice a day; consolidation period, 8~10 weeks.

Overcorrection of the mandibular dental midline to the non-affected side as much as the width of the mandibular central incisor was done. Postsurgical orthodontic treatment started with the removable mandibular occlusal splint to preclude loading of the distraction site by maintaining the posterior vertical relationship. Three months after stabilization, the affected maxillary dentition was allowed to extrude by selective reduction of this splint and the fixed orthodontic treatment.

Lateral and PA cephalometric radiographs before DO (TO), after DO (Tl), and after retention (T2) were traced. The affected side of the mandible in unilateral HFM and more severe side of the mandible in bilateral HFM cases were analyzed to get more objective change of cephalometric measurements. From the measurements at T2, patients were classified into Group 1 showing good retention results (N=10, 5 males and 5 females) and Group 2 showing poor results (N=9, 6 males and 3 females). Relationship among group, Pruzansky type, gender, and lateralism were analyzed by bivariate analysis. Sagittal, vertical, dental and soft tissue, and asymmetric variables were measured (Fig.1-4). Differences at each stage and during T0-T1 and T1-T2 between two groups were compared by Mann-Whitney U test, Wilcoxon signed rank test and ANOVA test.

79

Seung-Hak Baek

Fig. 1, Sagittal variables

1. SNA (°), 2. SNB (°), 3. ANB (°); 4. Facial plane angle (°), 5. Facial convexity angle (°), 6. Facial angle (Downs) (°), 7. A-B plane angle (°), 8. Palatal plane angle (°), 9. Y-axis to SN (°), 10. Facial axis (Ricketts) (°), 11. A-N perpendicular (mm), 12. Pog- N perpendicular (mm), 13. Ramus height (mm), 14.

Mandibular body length (mm), 15. APDI (°)； The dotted line is the affected side and the solid line is the non-affected side of the mandible.

Fig. 3. Dental and soft tissue variables

1. IMPA (°), 2. FMIA (°), 3. LI to facial plane (mm), 4. LI to NB (°), 5. LI to NB (mm), 6. LI to A-Pog (mm), 7. Pog to NB (mm), 8. Esthetic line to upper lip (mm), 9. Esthetic line to lower lip (mm)； The dotted line is the affected side and the solid line is the non-affected side of the mandible.

Fig. 2. Vertical variables

1. Articular angle (°), 2. Gonial angle (°): 3. Saddle angle (°), 4. Upper gonial angle (°), 5. Lower gonial angle (°), 6. Posterior facial height (mm), 7. Anterior facial height (mm), 8. Facial height ratio (%), 9. SN- GoMe (°), 10. Occlusal plane to GoMe (°), 11. FMA (°), 12. Bjork sum (°)； The dotted line is the affected side and the solid line is the non-affected side of the mandible.

Fig. 4. Asymmetric variables

1. Ramus inclination of the affected side (°) : angle between ramus line of the affected side and zygomaticofrontai suture line, 2. Ramus inclination of the non-affected side (°) ： angle between ramus line of the non-affected side and zygomaticofrontai suture line, 3. Occlusal plane canting (°) : angle between occlusal plane and zygomaticofrontai suture line, 4.

Chin point deviation (°) ： angle between midpoint of the chin and midsagittal line.

80

구순구개열

Vol. 7, No. 2 2004

Results

The ratio of Pruzansky type I, Ila, lib, and III was approximately 2.6 : 3.2 : 3.2 : 1.0 and the prevalence of male and female was approximately 6 : 4 (Table 1).

Gender did not significantly relate with the prevalence of Pruzansky type (Table 2). The overall distributions of unilateral and bilateral HFM were approximately 9 : 1 (Table 3). Although Pruzansky type did not significantly influence the prevalence of literalism, Group 2 had more tendency of bilateral involvement than Group 1 (Table 4). Group 1 and 2 did not 아low statistically significant differences in the stages (Table 5). Bivariate analysis showed that Pruzansky type significantly influenced the success or failure of DO (p<0.01) (Table 6). When the degree of ramus involvement increased, so did the incidence of failure

of DO result.

Cephalometric measurements that showed significant differences between two groups at TO were: SNB, ANB, facial plane angle, facial convexity angle, Ricketts’ facial axis angle, gonial angle, upper gonial angle, ramus height, ramus inclination of the affected side (p<0.01), APDI, Y-axis to SN, Pog-N perpendicular, articular angle, occlusal plane to GoMe angle, ramus inclination of the non-affected side, chin point deviation (p<0.05). (Table 7-10). Those mean that, at TO, Group 2 showed more posterior positioning of the mandible, smaller ramus height (Table 7), more vertical growth pattern of the mandible, smaller articular angle (Table 8), more labioversion of the mandibular incisor (Table 9), more inward ramus inclination of the affected side, more outward ramus inclination of the non-affected side,

Table 1. Distribution of hemifacial microsomia patients by Pruzansky type and gender.

Pruzansky type Male Female Total

unilateral 2 3 5

I

bilateral 0 0 0

total 2 (40%) 3 (60%) 5 (26.3%)

unilateral 3 3

6

Ila

bilateral 0 0 0

total 3 (50%) 3 (50%) 6(31.6%)

unilateral 3 2 5

lib

bilateral 1 b 1

total 4 (66.7%) 2 (33.3%) 6(31.6%)

unilateral 1 0 1

III

bilateral 1 0 1

total 2(100%) .0 (0%) 2(10.5%)

Total 11 (57.9%) 8(42.1%) 19

81

Seung-Hak Baek

Table 2. Bivariate analysis of Pruzansky type by gender.

Pruzansky type

I na nbm

Male Female % of Female Spearman’s rho (p) p-value

2 3 60.0

3 3 50.0

4 2 33.3

2 0 0.0 -0.335 0.161

Pruzansky type Unilateral Bilateral

Table 3. Distribution of hemifaciarmicrosomia patients by Pruzansky type and lateralism.

Left 3

I

Right 2 0

total 5(100%) 0(0%)

Left 3

Da Right 3 0

total 6 (100%) 0(0%)

Left 5 1

nb Right 0 1

total 5 (83.3%) 1 (16.7%)

Left 1

m Right 0

ⁱ

total 1(50%) 1 (50%)

Sum 17 (89.5%) 2 (10.5%)

Table 4. Bivariate analysis of Pnjzansky type by lateralism

Pruzansky type Unilateral Bilateral % of bilateral Spearman s rho (p) p-value

I 5 0 0.0

na 6 0 0.0

nb 5 1 16.7

m 1 1 50.0 0.424 0.070

Table 5. Distribution of hemifacial microsomia patients’ group by stages.

Stages Group 1

Mean

(N=10) SD

Group 2 Mean

’ (N=9)

SD Sig.

Before DO (TO) 5Y 9M 1Y 10M 7Y 11M 3Y IM NS

After DO (Tl) 6Y 6M 1Y6M 8Y 3M 3Y 3M NS

After retention (T2) 8Y 7M 10M 10Y 0M 2Y 9M NS

SD means standard deviation; Sig., Significance; NS : not significant.

Mann-Whitney U test, * p<0.05, ** p<0.01, *** p<0.001

82

구순구개열

Vol. 7, No. 2 2004

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구순구개열

Vol. 7, No. 2 2004

and more deviation of chin point to the affected side (Table 10) than Group 1. Especially, ramus inclination of the affected side and non-affected side of Group 2 at TO showed reverse tendency when compared with Group 1 (Table 10).

The major effects of DO on Group 1 were lengthening of the ramus height, forward positioning of the mandible (Table 7, 11), increase of the gonial angle, counterclockwise rotation due to decrease of articular angle (Table 8, 12), increase of ramus inclination of the affected side, and improvement of occlusal canting and chin point deviation (Table 10, 14). However, after DO (Tl), Group 2 showed still retrognathic mandible and less increase of ramus height (Table 7, 11), clockwise rotation of the mandible due to increase of articular angle (Table 8, 12), and more labioversion of the mandibular incisor and less improvement of lip protrusion to E-line (Table 9,13), less improvement of occlusal plane canting and chin point deviation (Table 10, 14) than Group 1.

Although improvement of the sagittal relationship of the retrognathic mandible was maintained after retention (T2), Group 2 at T2 showed still retrognathic mandible and little growth of ramus height (Table 7), counter-clockwise rotation of the mandible due to decrease of articular angle (Table 8) and more labioversion of the mandibular incisor than Group 1 (Table 9).

Both Group 1 and Group 2 showed significant difference in ramus inclination between the affected and non-affected side before DO (TO), after DO (Tl), after retention (T2) (Table 10). Occlusal plane canting showed significant change in Group 1 among before

DO (TO), after DO (Tl), and after retention (T2). In Group 2, occlusal plane canting showed significant change between before DO (TO) and after retention (T2) (Table 10).

Cephalometric measurements such as ramus height, articular angle, ramus inclination of the affected side, occlusal plane canting, chin point deviation (p<0.01), facial convexity angle, Pog-N perpendicular, gonial angle, upper gonial angle (p<0.05) showed significant differences during DO (T0-T1) and occlusal plane canting (p<0.01), articular angle, upper gonial angle (p<0.05) during retention (T1-T2) between the two groups (Table 11-14).

Since Group 2 showed more severe sagittal, vertical, and asymmetric skeletal relationships than Group 1 at TO, effect of DO on Group 2 was less than Group 1 during DO (T0-T1). Group 1 showed more forward positioning of the mandible, more lengthening of the ramus height rather than body length (Table 11), more increase of gonial angle, counterclockwise rotation of the mandible due to decrease of articular angle (Table 12), uprighting of the ramus inclination of the affected side, improvement of occlusal plane canting and chin point deviation (Table 14) than Group 2.

During retention (T1-T2) Group 2 showed counterclockwise rotation of the mandible due to decrease of articular angle and more increase of upper gonial angle (Table 12) than Group 1. Decrease of occlusal plane canting in Group 2 during retention (T1-T2) (Table 10, 14) seems to relate with bilateral cases which occlusal plane canting is not severe and less vertical growth of the ramus of both sides.

85

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86