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先天性多发性关节挛缩症与张口受限:1例病例报告及文献复习

Arthrogryposis multiplex congenita and limitation of mouth opening: Presentation of a case and review of the literature.

作者信息

Bénard M, Sesqué A, Barthélémy I, Depeyre A

机构信息

Oral and Maxillofacial Department, Estaing Hospital, CHU de Clermont-Ferrand, 63003 Clermont-Ferrand, France; Faculty of Medicine, université d'Auvergne, 63001 Clermont-Ferrand cedex 1, France.

Oral and Maxillofacial Department, Estaing Hospital, CHU de Clermont-Ferrand, 63003 Clermont-Ferrand, France; Faculty of Medicine, université d'Auvergne, 63001 Clermont-Ferrand cedex 1, France; Inserm U1107 Neuro-Dol, Trigeminal Pain and Migraine, Faculty of Dental Surgery, 63100 Clermont-Ferrand, France.

出版信息

J Stomatol Oral Maxillofac Surg. 2021 Feb;122(1):101-106. doi: 10.1016/j.jormas.2020.05.017. Epub 2020 May 23.

DOI:10.1016/j.jormas.2020.05.017
PMID:32450320
Abstract

Arthrogryposis multiplex congenita (AMC) is a rare entity gathering many diseases, isolated or included in more global syndromes. It affects symmetrically joints of lower limbs, back and maxillofacial area with temporomandibular joint (TMJ) impairment leading to deterioration of oral functions and mouth opening resulting in poor quality of life. Therapeutic strategy varies according to type and severity of impairment and could involve physical therapy and/or surgery. This article reported a case of AMC followed in our unit and highlighting various maxillo-facial impairments, especially restricted mouth opening and its consequences due to fibrotic TMJ and hypertrophic coronoid processes. We described our therapeutic approach based on surgery and postoperative physical therapy. A stable improvement of mouth opening was obtained, and patient's quality of life was increased. We performed a review of the literature and suggested a therapeutic management of mouth opening limitation in AMC.

摘要

先天性多发性关节挛缩症(AMC)是一种罕见的病症,涵盖多种疾病,这些疾病既可以是孤立存在的,也可以包含在更广泛的综合征中。它对称地影响下肢、背部和颌面区域的关节,伴有颞下颌关节(TMJ)功能障碍,导致口腔功能恶化和张口受限,从而使生活质量下降。治疗策略因功能障碍的类型和严重程度而异,可能包括物理治疗和/或手术。本文报告了我们科室收治的一例AMC病例,重点介绍了各种颌面功能障碍,尤其是由于颞下颌关节纤维化和冠状突肥大导致的张口受限及其后果。我们描述了基于手术和术后物理治疗的治疗方法。患者的张口情况得到了稳定改善,生活质量也有所提高。我们对文献进行了综述,并提出了AMC张口受限问题的治疗管理方法。

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引用本文的文献

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Arthrogryposis multiplex congenita with maxillofacial involvement: a case report.伴有颌面受累的先天性多发性关节挛缩症:病例报告
Maxillofac Plast Reconstr Surg. 2023 Feb 8;45(1):10. doi: 10.1186/s40902-023-00378-6.
2
Clinical and Genetic Findings in a Series of Eight Families with Arthrogryposis.关节挛缩症八家系的临床和遗传学发现。
Genes (Basel). 2021 Dec 23;13(1):29. doi: 10.3390/genes13010029.