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线状硬皮病:一种不寻常的口腔表现。

Morphea "en coup de sabre": An unusual oral presentation.

作者信息

Niklander Sven, Marín Constanza, Martínez René, Esguep Alfredo

机构信息

DDS, MDent, MSc, Department of Oral Medicine and Oral Pathology, Universidad Andres Bello, Viña del Mar, Chile. Avenida Valparaíso 1560, Viña del Mar, Chile.

DDS, Department of Oral Medicine and Oral Pathology, Universidad Andres Bello, Viña del Mar, Chile. Avenida Valparaíso 1560, Viña del Mar, Chile.

出版信息

J Clin Exp Dent. 2017 Feb 1;9(2):e315-e318. doi: 10.4317/jced.53151. eCollection 2017 Feb.

DOI:10.4317/jced.53151
PMID:28210455
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5303337/
Abstract

Morphea, or localized scleroderma, is an inflammatory disease that leads to sclerosis of the skin and underlying tissues due to excessive collagen deposition. Oral involvement is unusual and it may produce white linear fibrotic areas with a scar-like appearance, atrophy of tongue papillae, gingival recession and alveolar bone resorption. We report a case of a 13-year-old girl who consulted for progressive recession on the attached gingiva of her upper left incisors. She also presented a hypopigmented line on the left side skin of her upper lip, which continued through the vermilion and the lip mucosa, including the gingiva of the affected teeth. Clinical examination, blood tests, computerized axial tomography, echo-Doppler ultrasound and histopathological evaluation confirmed the diagnosis of morphea. Treatment with methotrexate and systemic corticosteroids was conducted. After 24 months, no other lesions appeared. No adverse side effects have been reported so far. Localized scleroderma, oral morphea, linear scleroderma, oral involvement, intraoral lesions.

摘要

硬斑病,即局限性硬皮病,是一种炎症性疾病,由于胶原蛋白过度沉积导致皮肤及皮下组织硬化。口腔受累情况并不常见,可能会产生白色线状纤维化区域,外观类似瘢痕,舌乳头萎缩、牙龈退缩及牙槽骨吸收。我们报告一例13岁女孩,因左上切牙附着龈渐进性退缩前来就诊。她在上唇左侧皮肤还有一条色素减退线,该线穿过唇红及唇黏膜,包括患牙的牙龈。临床检查、血液检查、计算机断层扫描、超声多普勒及组织病理学评估确诊为硬斑病。采用甲氨蝶呤和全身性皮质类固醇进行治疗。24个月后,未出现其他病变。目前尚未报告有不良反应。局限性硬皮病、口腔硬斑病、线状硬皮病、口腔受累、口腔内病变

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本文引用的文献

1
A rare cause of gingival recession: morphea with intra-oral involvement.牙龈退缩的罕见原因:硬斑病伴口腔内受累。
Oral Surg Oral Med Oral Pathol Oral Radiol. 2015 May;119(5):e257-64. doi: 10.1016/j.oooo.2015.02.002. Epub 2015 Feb 14.
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Histological features of localized scleroderma 'en coup de sabre': a study of 16 cases.局限性硬皮病“剑伤状”的组织学特征:一项16例病例的研究
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Update on the classification and treatment of localized scleroderma.
颅面硬斑病患者口颌系统紊乱及其矫正可能性
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Clinical and therapeutic course in head variants of linear morphea in adults: a retrospective review.成人线状硬斑病头部变异型的临床和治疗过程:回顾性研究。
Arch Dermatol Res. 2023 Jul;315(5):1161-1170. doi: 10.1007/s00403-022-02478-1. Epub 2022 Dec 2.
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Not every white spot is vitiligo.并非每个白斑都是白癜风。
Paediatr Child Health. 2021 Aug 31;27(1):1-3. doi: 10.1093/pch/pxab050. eCollection 2022 Mar.
6
Oral manifestations of morphea en plaque: Case report.斑块状硬斑病的口腔表现:病例报告。
Ann Med Surg (Lond). 2021 Oct 9;71:102891. doi: 10.1016/j.amsu.2021.102891. eCollection 2021 Nov.
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Therapeutic and Reconstructive Management Options in Scleroderma (Morphea) en Coup de Sabre in Children and Adults. A Systematic Literature Review.儿童和成人剑伤性硬皮病(局限性硬皮病)的治疗与重建管理方案。系统文献综述
J Clin Med. 2021 Sep 29;10(19):4517. doi: 10.3390/jcm10194517.
8
Morphea with Oral Mucosa Involvement and Unilateral Nevoid Telangiectasia as an Early Presentation of Morphea: A Case Report and Review of the Literature.硬斑病累及口腔黏膜并伴有单侧痣样毛细血管扩张作为硬斑病的早期表现:一例病例报告及文献复习
J Clin Aesthet Dermatol. 2020 Jan;13(1):38-40. Epub 2020 Jan 1.
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A case of linear morphea involving the oral cavity.一例累及口腔的线状硬皮病。
JAAD Case Rep. 2019 Jan 25;5(2):144-146. doi: 10.1016/j.jdcr.2018.10.024. eCollection 2019 Feb.
10
Progressive Hemifacial Atrophy and Linear Scleroderma En Coup de Sabre: A Spectrum of the Same Disease?进行性半侧颜面萎缩和线性硬皮病剑伤样损害:是同一种疾病的不同表现吗?
Front Med (Lausanne). 2018 Jan 31;4:258. doi: 10.3389/fmed.2017.00258. eCollection 2017.
局限性硬皮病的分类与治疗进展
Actas Dermosifiliogr. 2013 Oct;104(8):654-66. doi: 10.1016/j.adengl.2012.10.012. Epub 2013 Aug 13.
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Morphea in adults and children cohort III: nested case-control study--the clinical significance of autoantibodies in morphea.成人和儿童硬斑病队列 III:巢式病例对照研究——硬斑病中自身抗体的临床意义。
JAMA Dermatol. 2013 Oct;149(10):1159-65. doi: 10.1001/jamadermatol.2013.4207.
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Linear morphea: a case series with long-term follow-up of young, methotrexate-treated patients.线性硬斑病:接受甲氨蝶呤治疗的年轻患者的长期随访病例系列。
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A long-term follow-up study of methotrexate in juvenile localized scleroderma (morphea).甲氨蝶呤治疗儿童局限性硬皮病(硬斑病)的长期随访研究。
J Am Acad Dermatol. 2012 Dec;67(6):1151-6. doi: 10.1016/j.jaad.2012.03.036. Epub 2012 May 30.
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Oral mucosal morphea: a new variant.口腔黏膜硬斑病:一种新的变异型。
Dermatology. 2012;224(3):215-20. doi: 10.1159/000337554. Epub 2012 Apr 25.
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Methotrexate and corticosteroids in the treatment of localized scleroderma: a standardized prospective longitudinal single-center study.甲氨蝶呤和皮质类固醇治疗局限性硬皮病:一项标准化的前瞻性纵向单中心研究。
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