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危及生命的暴发性紫癜存活后双侧异位骨化的发生

Development of Bilateral Heterotopic Ossification After Survival of Life Threatening Purpura Fulminans.

作者信息

Asif Mohammed, Klifto Kevin M, Lagziel Tomer, Caffrey Julie

机构信息

Plastic Surgery, The Johns Hopkins University School of Medicine, Baltimore, USA.

出版信息

Cureus. 2020 Jan 21;12(1):e6731. doi: 10.7759/cureus.6731.

Abstract

Heterotopic ossification has been reported in patients who have undergone traumatic amputations, burn injuries, and total hip arthroplasty; however, the incidence of heterotopic ossification following purpura fulminans has only been reported in one case with unilateral involvement. Here we present a bilateral lower extremity case of heterotopic ossification as sequelae of purpura fulminans.  A 34-year-old male smoker with a past medical history of stab wounds to the chest and abdomen requiring emergent exploratory laparotomy, diaphragmatic repair, and splenectomy 15 years ago presented to the emergency department with a rapid onset of high fevers, chills and myalgia. He did not receive post-splenectomy prophylactic vaccinations for , , and . The patient presented clinically in septic shock with disseminated intravascular coagulation. The patient was admitted to the Medical Intensive Care Unit and subsequent workup suggested  bacteremia. Over the next 48 hours, the patient developed extensive necrosis of the bilateral upper and lower extremities concerning for purpura fulminans. The decision was made to perform a right transradial forearm amputation as well as bilateral transtibial amputations. He tolerated these procedures and was discharged to an inpatient rehabilitation facility. Approximately four months following his bilateral below knee amputations, the patient had difficulty wearing the prosthetics secondary to pain and eventually discontinued use altogether. At home, he continued to ambulate by bearing weight on his knees while wearing kneepads. He continued to report significant tenderness and pain along the bilaterally, below knee amputation stumps. His physical examination was concerning for significant distal bone formation in his bilateral amputation stump sites without evidence of skin breakdown. Intraoperatively, extensive bony formation was found bilaterally within his soleus muscle flaps, concerning for heterotopic ossification. Postoperatively, the patient was refitted for lower extremity prosthetics.  Similar to burns and trauma, the development of heterotopic ossification in patients with purpura fulminans may be directly related to the inflammatory process and amount of tissue damage. In some cases, heterotopic ossification could be caused from daily living activities, so the timing of diagnostic imaging techniques and clinical intervention is crucial.

摘要

创伤性截肢、烧伤和全髋关节置换术后的患者中曾有异位骨化的报道;然而,暴发性紫癜后异位骨化的发生率仅有单侧受累的一例报道。在此,我们报告一例双侧下肢异位骨化作为暴发性紫癜后遗症的病例。一名34岁有吸烟史的男性,15年前曾因胸腹部刺伤接受急诊剖腹探查术、膈肌修补术和脾切除术,现因高热、寒战和肌痛迅速发作就诊于急诊科。他未接受脾切除术后针对 、 和 的预防性疫苗接种。患者临床诊断为感染性休克伴弥散性血管内凝血。患者被收入医学重症监护病房,后续检查提示 菌血症。在接下来的48小时内,患者双上肢和双下肢出现广泛坏死,疑似暴发性紫癜。决定行右桡骨远端截肢术及双侧胫骨截肢术。他耐受了这些手术,并出院至住院康复机构。双侧膝下截肢术后约四个月,患者因疼痛难以佩戴假肢,最终完全停止使用。在家中,他继续通过佩戴护膝用膝盖承重行走。他持续报告双侧膝下截肢残端有明显压痛和疼痛。体格检查发现双侧截肢残端部位有明显的远端骨形成,无皮肤破损迹象。术中,在双侧比目鱼肌皮瓣内发现广泛的骨形成,疑似异位骨化。术后,为患者重新安装下肢假肢。与烧伤和创伤类似,暴发性紫癜患者异位骨化的发生可能与炎症过程和组织损伤程度直接相关。在某些情况下,异位骨化可能由日常生活活动引起,因此诊断性影像学技术和临床干预的时机至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6da9/7034744/23b28a7259c3/cureus-0012-00000006731-i01.jpg

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