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进行性骨化性纤维发育不良的头颈部表现:2例临床及影像学表现

Head and neck manifestations of fibrodysplasia ossificans progressiva: Clinical and imaging findings in 2 cases.

作者信息

Jo Gyu-Dong, Kang Ju-Hee, Kim Jo-Eun, Yi Won-Jin, Heo Min-Suk, Lee Sam-Sun, Huh Kyung-Hoe

机构信息

Department of Oral and Maxillofacial Radiology, Seoul National University Dental Hospital, Seoul, Korea.

Department of Oral and Maxillofacial Radiology, School of Dentistry, Seoul National University, Seoul, Korea.

出版信息

Imaging Sci Dent. 2023 Sep;53(3):257-264. doi: 10.5624/isd.20230069. Epub 2023 Jun 20.

Abstract

Fibrodysplasia ossificans progressiva is a rare hereditary disorder characterized by progressive heterotopic ossification in muscle and connective tissue, with few reported cases affecting the head and neck region. Although plain radiographic findings and computed tomography features have been well documented, limited reports exist on magnetic resonance findings. This report presents 2 cases of fibrodysplasia ossificans progressiva, one with limited mouth opening due to heterotopic ossification of the lateral pterygoid muscle and the other with restricted neck movement due to heterotopic ossification of the platysma muscle. Clinical findings of restricted mouth opening or limited neck movement, along with radiological findings of associated heterotopic ossification, should prompt consideration of fibrodysplasia ossificans progressiva in the differential diagnosis. Dentists should be particularly vigilant with patients diagnosed with fibrodysplasia ossificans progressiva to avoid exposure to diagnostic biopsy and invasive dental procedures.

摘要

进行性骨化性纤维发育不良是一种罕见的遗传性疾病,其特征为肌肉和结缔组织中进行性异位骨化,累及头颈部区域的报道病例较少。尽管X线平片表现和计算机断层扫描特征已有详细记录,但关于磁共振成像表现的报道有限。本报告介绍2例进行性骨化性纤维发育不良病例,1例因翼外肌异位骨化导致张口受限,另1例因颈阔肌异位骨化导致颈部活动受限。张口受限或颈部活动受限的临床表现,以及相关异位骨化的影像学表现,应促使在鉴别诊断中考虑进行性骨化性纤维发育不良。牙科医生应对诊断为进行性骨化性纤维发育不良的患者格外警惕,避免进行诊断性活检和侵入性牙科操作。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b120/10548149/ef8fca0d6f95/isd-53-257-g001.jpg

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