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阿拉吉尔综合征中髁突发育不全的治疗——一例报告

Treatment of Condylar Hypoplasia in Alagille Syndrome - A Case Report.

作者信息

Nardini Luca Guarda, Val Matteo, Colonna Anna, Cagidiaco Edoardo Ferrari, Ferrari Marco, Manfredini Daniele

机构信息

Unit of Oral and Maxillofacial Surgery, Ca' Foncello Hospital, Treviso, Italy.

Department of Biomedical Technologies, School of Dentistry, University of Siena, Siena, Italy.

出版信息

Ann Maxillofac Surg. 2024 Jan-Jun;14(1):85-88. doi: 10.4103/ams.ams_220_23. Epub 2024 Apr 19.

DOI:10.4103/ams.ams_220_23
PMID:39184415
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11340841/
Abstract

RATIONALE

Alagille syndrome is a rare genetic disorder with dental and facial abnormalities in the head-and-neck area. It is autosomal dominant and occurs in approximately 1 in 100,000 people. No cases of Alagille Syndrome (ALGS) with mandibular hypoplasia and temporomandibular joint ankyloses (TMJa) have been reported to date.

PATIENT CONCERNS AND DIAGNOSIS

A 3-year-old female patient suffering from ALGS came to our hospital affected by unilateral mandibular hypoplasia and TMJa with severe limitation of mouth opening (maximal interincisal distance [MID] of 2 mm).

TREATMENT

A two-phase surgical management approach was undertaken based on computed tomography scans and the patient's age. The first phase involved mandibular distraction, followed by arthroplasty with amniotic human membrane placement.

OUTCOMES

After immediate post-surgery rehabilitation, the patient showed good mandibular function with no complications after 15 months. MID was 20 mm.

TAKE-AWAY LESSONS: This is a rare and interesting case with no previous literature reports. The use of amniotic membranes in surgical management adds further significance.

摘要

理论依据

阿拉吉列综合征是一种罕见的遗传性疾病，在头颈部区域存在牙齿和面部异常。它是常染色体显性遗传，发病率约为十万分之一。迄今为止，尚未有下颌发育不全和颞下颌关节强直（TMJa）的阿拉吉列综合征（ALGS）病例报告。

患者情况与诊断

一名患有ALGS的3岁女性患者因单侧下颌发育不全和TMJa前来我院就诊，伴有严重的张口受限（最大切牙间距离[MID]为2毫米）。

治疗

基于计算机断层扫描和患者年龄，采取了两阶段手术治疗方法。第一阶段进行下颌骨牵张成骨术，随后进行关节成形术并植入人羊膜。

结果

术后立即进行康复治疗，15个月后患者下颌功能良好，无并发症。MID为20毫米。

经验教训

这是一个罕见且有趣的病例，此前无文献报道。在手术治疗中使用羊膜具有更大的意义。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/993380f322b6/AMS-14-85-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/8581689bded1/AMS-14-85-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/5fbe13ac9a0b/AMS-14-85-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/b488148e6ca3/AMS-14-85-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/993380f322b6/AMS-14-85-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/8581689bded1/AMS-14-85-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/5fbe13ac9a0b/AMS-14-85-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/b488148e6ca3/AMS-14-85-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0957/11340841/993380f322b6/AMS-14-85-g004.jpg

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本文引用的文献

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J Oral Biol Craniofac Res. 2021 Oct-Dec;11(4):581-595. doi: 10.1016/j.jobcr.2021.07.007. Epub 2021 Jul 16.
2
Childhood TMJ Ankylosis: A Surgical Challenge.儿童颞下颌关节强直：外科挑战。
J Long Term Eff Med Implants. 2021;31(2):9-11. doi: 10.1615/JLongTermEffMedImplants.2021037143.
3
Computer-assisted surgery with custom prostheses and human amniotic membrane in a patient with bilateral class IV TMJ reankylosis: a case report.
计算机辅助手术联合定制假体和人羊膜治疗双侧 TMJ Ⅳ类强直患者：病例报告。
Cell Tissue Bank. 2022 Jun;23(2):395-400. doi: 10.1007/s10561-021-09940-w. Epub 2021 Jun 27.
4
Cost-Effectiveness Analysis, Psychosocial, and Utility Outcomes of Early Mandibular Distraction in Craniofacial Microsomia.颅面骨发育不全中早期下颌骨牵引的成本效益分析、心理社会和效用结果。
J Craniofac Surg. 2020 Oct;31(7):1888-1894. doi: 10.1097/SCS.0000000000006622.
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Temporomandibular Joint Ankylosis in Pediatric Patients With Craniofacial Differences: Causes, Recurrence and Clinical Outcomes.患有颅面差异的儿科患者的颞下颌关节强直：病因、复发情况及临床结果
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