{"title":"一例罕见的伴有慢性前臂疼痛的右尺骨单骨质疏松症--病例报告。","authors":"Thinley Ugyen, Letho Letho","doi":"10.1016/j.ijscr.2024.110603","DOIUrl":null,"url":null,"abstract":"<div><h3>Introduction</h3><div>Melorheostosis is extremely rare non-cancerous lesion of bone mainly affecting the long bones and soft tissues. The incidence is 0.9 per million population. It affects male and female equally and reported among children and early adulthood. Common symptoms include pain, deformity and restricted range of movement of affected joint.</div></div><div><h3>Case presentation</h3><div>A 28-year-old female presented with pain at right forearm for nine months. The pain was of insidious onset and progressive in nature with severity of VAS 7/10. On examination, no gross deformity of right forearm was noted, no tenderness along the bones and range of movement of right elbow and wrist joints were within normal limit. Radiograph of right forearm revealed sclerosis of entire right ulna with dripping candle wax appearance with bowing deformity of ulna. She underwent osteoplasty of right ulna and biopsy. Oral bisphosphonate was started.</div></div><div><h3>Discussion</h3><div>Due to the rarity of the condition, there is still no standard guideline of the treatment of Melorheostosis, the widely practiced mode of treatment is symptomatic with analgesics, contracture release and deformity correction in extreme cases. If the diagnosis is doubtful it is important to rule out other sinister causes such as infection and malignancy. Biopsy is indicated only to confirm diagnosis.</div></div><div><h3>Conclusion</h3><div>Early diagnosis of Melorheostosis still remains challenge due to paucity of the condition. Melorheostosis should be included in one of the differential diagnosis of any hypersclerotic conditions of bone.</div></div>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"125 ","pages":"Article 110603"},"PeriodicalIF":0.6000,"publicationDate":"2024-11-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"A rare case of monostotic Melorheostosis of right ulna presenting with chronic forearm pain - Case report\",\"authors\":\"Thinley Ugyen, Letho Letho\",\"doi\":\"10.1016/j.ijscr.2024.110603\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><h3>Introduction</h3><div>Melorheostosis is extremely rare non-cancerous lesion of bone mainly affecting the long bones and soft tissues. The incidence is 0.9 per million population. It affects male and female equally and reported among children and early adulthood. Common symptoms include pain, deformity and restricted range of movement of affected joint.</div></div><div><h3>Case presentation</h3><div>A 28-year-old female presented with pain at right forearm for nine months. The pain was of insidious onset and progressive in nature with severity of VAS 7/10. On examination, no gross deformity of right forearm was noted, no tenderness along the bones and range of movement of right elbow and wrist joints were within normal limit. Radiograph of right forearm revealed sclerosis of entire right ulna with dripping candle wax appearance with bowing deformity of ulna. She underwent osteoplasty of right ulna and biopsy. Oral bisphosphonate was started.</div></div><div><h3>Discussion</h3><div>Due to the rarity of the condition, there is still no standard guideline of the treatment of Melorheostosis, the widely practiced mode of treatment is symptomatic with analgesics, contracture release and deformity correction in extreme cases. If the diagnosis is doubtful it is important to rule out other sinister causes such as infection and malignancy. Biopsy is indicated only to confirm diagnosis.</div></div><div><h3>Conclusion</h3><div>Early diagnosis of Melorheostosis still remains challenge due to paucity of the condition. Melorheostosis should be included in one of the differential diagnosis of any hypersclerotic conditions of bone.</div></div>\",\"PeriodicalId\":48113,\"journal\":{\"name\":\"International Journal of Surgery Case Reports\",\"volume\":\"125 \",\"pages\":\"Article 110603\"},\"PeriodicalIF\":0.6000,\"publicationDate\":\"2024-11-13\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"International Journal of Surgery Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S2210261224013841\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"SURGERY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"International Journal of Surgery Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S2210261224013841","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"SURGERY","Score":null,"Total":0}
引用次数: 0
摘要
简介骨质疏松症(Melorheostosis)是一种极为罕见的非癌症性骨病,主要影响长骨和软组织。发病率为每百万人口中 0.9 例。男女发病率相同,多见于儿童和成年早期。常见症状包括疼痛、畸形和受累关节活动范围受限:一名 28 岁的女性因右前臂疼痛 9 个月前来就诊。疼痛起病隐匿,呈进行性,严重程度为 VAS 7/10。检查时未发现右前臂严重变形,骨骼无压痛,右肘和腕关节活动范围在正常范围内。右前臂 X 光片显示整个右尺骨硬化,呈滴蜡状,尺骨呈弓形畸形。她接受了右尺骨骨整形术和活组织检查。开始口服双膦酸盐:讨论:由于这种疾病非常罕见,目前还没有治疗骨质疏松症的标准指南,广泛采用的治疗模式是对症止痛、挛缩松解和极端情况下的畸形矫正。如果诊断有疑问,必须排除感染和恶性肿瘤等其他险恶原因。活组织检查仅适用于确诊:结论:由于Melorheostosis病例较少,因此其早期诊断仍是一项挑战。Melorheostosis应被列为任何骨硬化症的鉴别诊断之一。
A rare case of monostotic Melorheostosis of right ulna presenting with chronic forearm pain - Case report
Introduction
Melorheostosis is extremely rare non-cancerous lesion of bone mainly affecting the long bones and soft tissues. The incidence is 0.9 per million population. It affects male and female equally and reported among children and early adulthood. Common symptoms include pain, deformity and restricted range of movement of affected joint.
Case presentation
A 28-year-old female presented with pain at right forearm for nine months. The pain was of insidious onset and progressive in nature with severity of VAS 7/10. On examination, no gross deformity of right forearm was noted, no tenderness along the bones and range of movement of right elbow and wrist joints were within normal limit. Radiograph of right forearm revealed sclerosis of entire right ulna with dripping candle wax appearance with bowing deformity of ulna. She underwent osteoplasty of right ulna and biopsy. Oral bisphosphonate was started.
Discussion
Due to the rarity of the condition, there is still no standard guideline of the treatment of Melorheostosis, the widely practiced mode of treatment is symptomatic with analgesics, contracture release and deformity correction in extreme cases. If the diagnosis is doubtful it is important to rule out other sinister causes such as infection and malignancy. Biopsy is indicated only to confirm diagnosis.
Conclusion
Early diagnosis of Melorheostosis still remains challenge due to paucity of the condition. Melorheostosis should be included in one of the differential diagnosis of any hypersclerotic conditions of bone.