Zhang Qing, Shen Min, Yang Bo, Yu Keyi
Department of Rheumatology, Peking Union Medical College Hospital, Chinese Academy of Medical Science & Peking Union Medical College, Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education, Beijing 100730, China.
J Med Case Rep. 2013 Dec 13;7:268. doi: 10.1186/1752-1947-7-268.
Pachydermoperiostosis is a rare, hereditary disease commonly presenting with digital clubbing, pachydermia and periosteal hypertrophy. Therapeutic options for pachydermoperiostosis are few because of the unknown pathogenesis. Here, we report the complicated case of a patient with pachydermoperiostosis combined with spondyloarthritides, who was refractory to steroids and tumor necrosis factor alpha antagonists. We treated this patient with zoledronic acid and performed an arthroscopic synovectomy, with a satisfactory outcome. To the best of our knowledge, this is the first report in English on the combination of zoledronic acid administration and synovectomy for the treatment of a patient with pachydermoperiostosis and spondyloarthritides.
An 18-year-old Han Chinese man was diagnosed with pachydermoperiostosis in the presence of digital clubbing, periostitis and swollen limbs. Combined spondyloarthritides was also considered based on his lower back pain, lower limbs synovitis, bilateral sacroiliac sclerosis and a positive test for human leukocyte antigen B27, as well as immunoglobulin A nephropathy. He was refractory to steroids and tumor necrosis factor alpha antagonists, but treated with intravenous zoledronic acid and an arthroscopic synovectomy, with a satisfactory outcome.
This is a rare, complicated case of pachydermoperiostosis with spondyloarthritides. Combination therapy of zoledronic acid administration with synovectomy is a novel, convenient and effective option for patients with pachydermoperiostosis with remarkable synovitis.
厚皮性骨膜病是一种罕见的遗传性疾病，通常表现为杵状指、皮肤增厚和骨膜增生。由于发病机制不明，厚皮性骨膜病的治疗选择有限。在此，我们报告一例厚皮性骨膜病合并脊柱关节炎的复杂病例，该患者对类固醇和肿瘤坏死因子α拮抗剂治疗无效。我们用唑来膦酸治疗该患者并进行了关节镜下滑膜切除术，取得了满意的效果。据我们所知，这是英文文献中首例关于唑来膦酸给药与滑膜切除术联合治疗厚皮性骨膜病合并脊柱关节炎患者的报告。
一名18岁的汉族男性，因出现杵状指、骨膜炎和肢体肿胀被诊断为厚皮性骨膜病。基于他的下背部疼痛、下肢滑膜炎、双侧骶髂关节硬化以及人类白细胞抗原B27检测呈阳性，同时伴有免疫球蛋白A肾病，还考虑合并脊柱关节炎。他对类固醇和肿瘤坏死因子α拮抗剂治疗无效，但接受了静脉注射唑来膦酸和关节镜下滑膜切除术，取得了满意的效果。
这是一例罕见的厚皮性骨膜病合并脊柱关节炎的复杂病例。唑来膦酸给药与滑膜切除术联合治疗是厚皮性骨膜病伴明显滑膜炎患者的一种新颖、便捷且有效的选择。
