Department of Clinical-Surgical, Diagnostic, and Pediatric Science, Institute of Dermatology, Fondazione IRCCS Policlinico San Matteo, Pavia, Italy.
Department of Rheumatology, Orthopedic Institute Galeazzi, Milan, Italy.
Dermatol Ther. 2019 Mar;32(2):e12821. doi: 10.1111/dth.12821. Epub 2019 Feb 5.
We report the case of a female patient who developed a firm, wooden-like, nonpitting edema of the left lower leg after a dermo-hypodermitis. The clinical picture was accompanied by intense pain, strongly impacting the patient's quality of life. A soft-tissue ultrasound demonstrated several millimetric hyperechoic linear lesions whose histopathological examination was conclusive for panniculitis ossificans. A conservative medical management with compression stockings associated with pentoxifylline 800 mg/day was prescribed with improvement of the edema and, in particular, a good pain control. To date, after a 2-year therapy with pentoxifylline, the leg wooden-like edema has substantially improved, despite the persistence of the well-known foci of ossification, and the pain has resolved, conditioning a substantial improvement of the patient's quality of life. No side effect has been observed during the routine follow up. Although there is no unanimous opinion in the literature about the effect of pentoxifylline on bone formation and osteogenic differentiation, pentoxifylline treatment proved to be beneficial in our patient both for the heterotopic ossification process and the pain control. We collected some of the data in literature about pentoxifylline effects and advanced some hypotheses to explain our results. Finally, we suggest that an anti-inflammatory and vasodilators drug such as pentoxifylline could be a possible alternative in heterotopic ossification disorders.
我们报告了一例女性患者,在发生皮肤-皮下组织炎后,左小腿出现坚硬如木的非凹陷性水肿。临床表现伴有剧烈疼痛,严重影响患者的生活质量。软组织超声显示数毫米的高回声线性病变,其组织病理学检查提示为骨化性脂膜炎。采用压迫性弹力袜联合己酮可可碱 800mg/天的保守药物治疗,水肿得到改善,特别是疼痛得到良好控制。迄今为止,在接受己酮可可碱治疗 2 年后,尽管骨化的已知病灶持续存在,但腿部木样水肿得到了显著改善,疼痛得到缓解,患者的生活质量得到了实质性改善。在常规随访期间未观察到任何副作用。尽管文献中对于己酮可可碱对骨形成和成骨分化的影响尚无一致意见,但己酮可可碱治疗对我们的患者在异位骨化过程和疼痛控制方面都显示出了有益作用。我们收集了一些关于己酮可可碱作用的数据,并提出了一些假设来解释我们的结果。最后,我们认为,像己酮可可碱这样的抗炎和血管扩张药物可能是异位骨化疾病的一种可行替代治疗方法。