{"title":"颌间肌纤维增生症并发创伤后和感染性骨化性肌炎:病例报告。","authors":"Yian Guan,Dongyang Ma","doi":"10.1097/md.0000000000039648","DOIUrl":null,"url":null,"abstract":"RATIONALE\r\nMyositis ossificans (MO) is characterized by benign heterotopic ossificans in soft tissues like muscles, which can be classified into nonhereditary MO and fibrodysplasia ossificans progressiva (FOP). Nonhereditary MO is characterized by ossification of the soft tissues after acute or repetitive trauma, burns, or surgical intervention. FOP is a rare and crippling disease characterized by congenital malformation of the big toe and heterotopic ossification in muscle. The majority of FOP's musculoskeletal traits are associated with dysregulated chondrogenesis. The diagnosis is mainly based on clinical manifestation, imaging examination, and genetic analysis. There is still no effective treatment to cure or slow its progression. The best approach remains early diagnosis, conservative drug treatment, and injury prevention to avoid local ossification.\r\n\r\nPATIENT CONCERNS\r\nA 34-year-old male presented at our hospital because of trismus caused by ossification of the masseter muscle. In addition, he had serious stiffness and multiple bony masses throughout the body, which led to limited movement.\r\n\r\nDIAGNOSES\r\nBased on the clinical manifestation of movement restriction, characteristic radiographic images of ossification of soft tissues, the genetic test showing a heterozygous molecule (c.974G > C, p.G325A) of the activin A receptor type I, the patient was diagnosed as FOP complicated with localized MO in masseter after trauma and infection.\r\n\r\nINTERVENTIONS\r\nThe patient underwent the surgical resection of ossification in the masseter muscle, he was instructed to insist on mouth-opening exercises and take glucocorticoids and nonsteroidal anti-inflammatory medications after surgery.\r\n\r\nOUTCOMES\r\nThe symptoms of trismus are relieved, and eating can be basically achieved after surgery, while the symptoms of trismus recurred 2 years later.\r\n\r\nLESSONS\r\nAlthough FOP has unique clinical manifestations, its diagnosis may be difficult because of its rarity. Gene analysis is the main standard for diagnosis, while patients with different genotypic variations may show different clinical symptoms. Therapeutic interventions are still supportive and preventive, and surgery is not recommended except under certain circumstances.","PeriodicalId":18549,"journal":{"name":"Medicine","volume":null,"pages":null},"PeriodicalIF":1.3000,"publicationDate":"2024-09-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Fibrodysplasia ossificans progressiva complicated with post traumatic and infectious myositis ossificans in masseter: A case report.\",\"authors\":\"Yian Guan,Dongyang Ma\",\"doi\":\"10.1097/md.0000000000039648\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"RATIONALE\\r\\nMyositis ossificans (MO) is characterized by benign heterotopic ossificans in soft tissues like muscles, which can be classified into nonhereditary MO and fibrodysplasia ossificans progressiva (FOP). Nonhereditary MO is characterized by ossification of the soft tissues after acute or repetitive trauma, burns, or surgical intervention. FOP is a rare and crippling disease characterized by congenital malformation of the big toe and heterotopic ossification in muscle. The majority of FOP's musculoskeletal traits are associated with dysregulated chondrogenesis. The diagnosis is mainly based on clinical manifestation, imaging examination, and genetic analysis. There is still no effective treatment to cure or slow its progression. The best approach remains early diagnosis, conservative drug treatment, and injury prevention to avoid local ossification.\\r\\n\\r\\nPATIENT CONCERNS\\r\\nA 34-year-old male presented at our hospital because of trismus caused by ossification of the masseter muscle. In addition, he had serious stiffness and multiple bony masses throughout the body, which led to limited movement.\\r\\n\\r\\nDIAGNOSES\\r\\nBased on the clinical manifestation of movement restriction, characteristic radiographic images of ossification of soft tissues, the genetic test showing a heterozygous molecule (c.974G > C, p.G325A) of the activin A receptor type I, the patient was diagnosed as FOP complicated with localized MO in masseter after trauma and infection.\\r\\n\\r\\nINTERVENTIONS\\r\\nThe patient underwent the surgical resection of ossification in the masseter muscle, he was instructed to insist on mouth-opening exercises and take glucocorticoids and nonsteroidal anti-inflammatory medications after surgery.\\r\\n\\r\\nOUTCOMES\\r\\nThe symptoms of trismus are relieved, and eating can be basically achieved after surgery, while the symptoms of trismus recurred 2 years later.\\r\\n\\r\\nLESSONS\\r\\nAlthough FOP has unique clinical manifestations, its diagnosis may be difficult because of its rarity. Gene analysis is the main standard for diagnosis, while patients with different genotypic variations may show different clinical symptoms. Therapeutic interventions are still supportive and preventive, and surgery is not recommended except under certain circumstances.\",\"PeriodicalId\":18549,\"journal\":{\"name\":\"Medicine\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":1.3000,\"publicationDate\":\"2024-09-13\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Medicine\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1097/md.0000000000039648\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"MEDICINE, GENERAL & INTERNAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Medicine","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1097/md.0000000000039648","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"MEDICINE, GENERAL & INTERNAL","Score":null,"Total":0}
引用次数: 0
摘要
理论依据骨化性肌炎(MO)的特征是肌肉等软组织的良性异位骨化,可分为非遗传性骨化性肌炎和进行性纤维性骨化症(FOP)。非遗传性 MO 的特征是软组织在急性或反复创伤、烧伤或手术干预后发生骨化。FOP 是一种罕见的致残性疾病,以大脚趾先天性畸形和肌肉异位骨化为特征。FOP 的大多数肌肉骨骼特征与软骨生成失调有关。诊断主要依据临床表现、影像学检查和基因分析。目前还没有有效的治疗方法来治愈或延缓其进展。最好的方法仍然是早期诊断、药物保守治疗和预防损伤,以避免局部骨化。此外,他还伴有严重的僵硬感和全身多处骨性肿块,导致活动受限。诊断根据活动受限的临床表现、软组织骨化的特征性影像学图像、基因检测显示的杂合分子(c.974 G > C, p.G3干预措施患者接受了手术切除咀嚼肌骨化,术后嘱其坚持张口运动,并服用糖皮质激素和非甾体抗炎药物。结论虽然 FOP 有独特的临床表现,但由于其罕见性,诊断可能比较困难。基因分析是诊断的主要标准,而不同基因型变异的患者可能表现出不同的临床症状。治疗干预措施仍以支持性和预防性为主,除非在特定情况下,否则不建议进行手术治疗。
Fibrodysplasia ossificans progressiva complicated with post traumatic and infectious myositis ossificans in masseter: A case report.
RATIONALE
Myositis ossificans (MO) is characterized by benign heterotopic ossificans in soft tissues like muscles, which can be classified into nonhereditary MO and fibrodysplasia ossificans progressiva (FOP). Nonhereditary MO is characterized by ossification of the soft tissues after acute or repetitive trauma, burns, or surgical intervention. FOP is a rare and crippling disease characterized by congenital malformation of the big toe and heterotopic ossification in muscle. The majority of FOP's musculoskeletal traits are associated with dysregulated chondrogenesis. The diagnosis is mainly based on clinical manifestation, imaging examination, and genetic analysis. There is still no effective treatment to cure or slow its progression. The best approach remains early diagnosis, conservative drug treatment, and injury prevention to avoid local ossification.
PATIENT CONCERNS
A 34-year-old male presented at our hospital because of trismus caused by ossification of the masseter muscle. In addition, he had serious stiffness and multiple bony masses throughout the body, which led to limited movement.
DIAGNOSES
Based on the clinical manifestation of movement restriction, characteristic radiographic images of ossification of soft tissues, the genetic test showing a heterozygous molecule (c.974G > C, p.G325A) of the activin A receptor type I, the patient was diagnosed as FOP complicated with localized MO in masseter after trauma and infection.
INTERVENTIONS
The patient underwent the surgical resection of ossification in the masseter muscle, he was instructed to insist on mouth-opening exercises and take glucocorticoids and nonsteroidal anti-inflammatory medications after surgery.
OUTCOMES
The symptoms of trismus are relieved, and eating can be basically achieved after surgery, while the symptoms of trismus recurred 2 years later.
LESSONS
Although FOP has unique clinical manifestations, its diagnosis may be difficult because of its rarity. Gene analysis is the main standard for diagnosis, while patients with different genotypic variations may show different clinical symptoms. Therapeutic interventions are still supportive and preventive, and surgery is not recommended except under certain circumstances.
期刊介绍:
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