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强直性脊柱炎患者急性髓系白血病骨髓移植后韧带骨赘消退:一例报告

Regression of syndesmophyte after bone marrow transplantation for acute myeloid leukemia in a patient with ankylosing spondylitis: a case report.

作者信息

Yang Hae Kyung, Moon Su-Jin, Shin Jae Ho, Kwok Seung-Ki, Park Kyung Su, Park Sung-Hwan, Kim Ho-Youn, Ju Ji Hyeon

机构信息

Division of Rheumatology, Department of Internal Medicine, Seoul St, Mary's Hospital, College of Medicine, Catholic Institute of Medical Science, The Catholic University of Korea, 505 Banpo-dong, Seocho-gu, Seoul, South Korea.

出版信息

J Med Case Rep. 2012 Aug 21;6:250. doi: 10.1186/1752-1947-6-250.

Abstract

INTRODUCTION

Disease progression of ankylosing spondylitis has been considered irreversible. However, we report a case of spontaneous regression of syndesmophyte development following allogeneic peripheral blood stem cell transplantation in a patient with acute myeloid leukemia, who was also diagnosed as having ankylosing spondylitis. To the best of our knowledge, this is the first case report presenting the partial radiologic regression of syndesmophytes.

CASE PRESENTATION

A 39-year-old man with active ankylosing spondylitis achieved clinical remission and partial radiological regression of cervical spine syndesmophytes following a peripheral blood stem cell transplantation for acute myeloid leukemia. Our patient received an allogeneic peripheral blood stem cell transplantation following a pre-transplantation conditioning regimen of total body irradiation and cyclophosphamide. The donor was a human leukocyte antigen-matched 29-year-old man. Our patient has remained asymptomatic and has received no medication for ankylosing spondylitis for nearly three years.

CONCLUSIONS

Several explanations are proposed for the regression of syndesmophytes and clinical improvement in active ankylosing spondylitis observed in our patient, including changes in bone remodeling and immune reconstitution following stem cell transplantation, the effect of immunosuppressive agents, or fluctuation in the natural course of ankylosing spondylitis although further studies are required. The regression of syndesmophytes in ankylosing spondylitis in this case raises the possibility that stem cell transplantation might contribute to the development of a novel therapeutic strategy for treatment of the disease.

摘要

引言

强直性脊柱炎的疾病进展一直被认为是不可逆的。然而,我们报告了一例急性髓系白血病患者在接受异基因外周血干细胞移植后,其韧带骨赘发育出现自发消退的病例,该患者同时也被诊断为患有强直性脊柱炎。据我们所知,这是首例报道韧带骨赘出现部分影像学消退的病例。

病例介绍

一名39岁的活动性强直性脊柱炎男性患者,在接受急性髓系白血病外周血干细胞移植后,实现了临床缓解以及颈椎韧带骨赘的部分影像学消退。我们的患者在接受全身照射和环磷酰胺的预处理方案后,接受了异基因外周血干细胞移植。供者是一名29岁、人类白细胞抗原匹配的男性。我们的患者近三年一直无症状,且未接受过治疗强直性脊柱炎的药物。

结论

对于我们患者中观察到的韧带骨赘消退以及活动性强直性脊柱炎的临床改善,提出了几种解释,包括干细胞移植后骨重塑和免疫重建的变化、免疫抑制剂的作用,或者强直性脊柱炎自然病程的波动,不过还需要进一步研究。该病例中强直性脊柱炎韧带骨赘的消退增加了干细胞移植可能有助于开发该疾病新治疗策略的可能性。

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