Ahmmed M F, Shazzad M N, Ferdous S, Azad A K, Haq S A
Dr Md Faysal Ahmmed, Resident, Department of Rheumatology, Bangabandhu Sheikh Mujib Medical University (BSMMU), Dhaka, Bangladesh.
Mymensingh Med J. 2017 Oct;26(4):939-943.
Pachydermoperiostosis or primary hypertrophic osteoarthropathy, also known as Touraine-Solente-Gole syndrome, is a rare process, frequently inherited. In its complete form it is characterized by pachydermia (thickening of the skin), skeletal changes (periostosis) and acropachia (digital clubbing). Diagnosis can be made considering the typical clinical features and the histological feature. We report a patient of 25-year old man presented with joint pain involving in multiple joints for last 7 years and progressive enlargement of his hands and feet with profuse sweating of palms and soles for last 4 years. Physical examination revealed thickened skin with excessive furrowing of his forehead, dropping of both eyelids, clubbing of all fingers, toes and enlargement of his hands and feet with pamoplantar hyperhidrosis. Laboratory investigation shows raised CRP, X-ray feet lateral view showed normal heel pad thickness, new bone formation and periosteal elevation in lower end of tibia and fibula and skin biopsy showed dense fibrocollagenous tissue in dermis and subcutis and mild acanthosis. With this scenario he was diagnosed as primary hypertrophic osteoarthropathy (Pachydermoperiostosis). After diagnosis he was treated with cholchicine (0.6mg) twice daily, naproxen (500mg) twice daily, and risedronate (150mg) monthly. After one year his skin texture became less thickened, joint pain improved, there was no further enlargement of acral part of fore arm. In this report we review the characteristic features of this syndrome. We highlight the importance of ruling out secondary forms of hypertrophic osteoarthropathy and of a close follow-up of these patients because of complications that might develop on long-term. Although no treatment was satisfactory, we wanted to emphasize that NSAIDs, Cholchicine and risedronate could be an effective treatment option.
厚皮性骨膜病或原发性肥大性骨关节病,也称为图赖讷-索伦特-戈勒综合征,是一种罕见的、常为遗传性的病症。其典型表现为厚皮症(皮肤增厚)、骨骼改变(骨膜增生)和杵状指(趾)。可根据典型的临床特征和组织学特征进行诊断。我们报告一名25岁男性患者,过去7年一直存在多关节疼痛,过去4年双手和双脚逐渐肿大,伴有手掌和足底大量出汗。体格检查发现皮肤增厚,前额有过多皱纹,双眼睑下垂,所有手指、脚趾均呈杵状,双手和双脚肿大,伴有掌跖多汗。实验室检查显示C反应蛋白升高,足部X线侧位片显示足跟垫厚度正常,胫骨和腓骨下端有新骨形成和骨膜抬高,皮肤活检显示真皮和皮下组织有致密的纤维胶原组织,并有轻度棘皮症。根据这些情况,他被诊断为原发性肥大性骨关节病(厚皮性骨膜病)。诊断后,他接受了秋水仙碱(0.6毫克)每日两次、萘普生(500毫克)每日两次和利塞膦酸盐(150毫克)每月一次的治疗。一年后,他的皮肤质地变薄,关节疼痛改善,前臂远端没有进一步肿大。在本报告中,我们回顾了该综合征的特征。我们强调排除继发性肥大性骨关节病的重要性,以及由于可能出现的长期并发症而对这些患者进行密切随访的重要性。尽管没有令人满意的治疗方法,但我们想强调非甾体抗炎药、秋水仙碱和利塞膦酸盐可能是有效的治疗选择。