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伴有肾上腺功能不全的SAPHO综合征:一例报告

SAPHO syndrome with adrenal deficiency: a case report.

作者信息

Eyigör Sibel, Karapolat Hale, Adanur Hilal, Kirazli Yeşim

机构信息

Physical Therapy and Rehabilitation Department, Ege University Faculty of Medicine, 35100 Bornova, Izmir, Turkey.

出版信息

Cases J. 2009 Aug 14;2:6281. doi: 10.1186/1757-1626-0002-0000006281.

DOI:10.1186/1757-1626-0002-0000006281
PMID:20184675
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC2827074/
Abstract

INTRODUCTION

The SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis, osteomyelitis) is a rare painful disorder, usually with a good long-term prognosis. Its etiology remains unclear, and various treatment regimens frequently fail to control the disease.

CASE PRESENTATION

A 46-year-old Caucasian female was referred for anterior chest wall and back pain. Physical examination was unremarkable except for skin lesions noted on soles of both feet, extremities and the face. A thoracic magnetic resonance imaging study demonstrated a lesion characterized with bone marrow edema and proliferation of soft tissue in the sternum. A brain MRI was requested secondary to the elevated prolactin level which was compatible with empty sella syndrome.

CONCLUSION

The case presented here has the unique feature of adrenal deficiency presenting alongside the SAPHO syndrome and is presented as the first case reported. This syndrome could become complicated with different organ system involvement other than bone and skin. There is a need further studies that will explore the weak relationship between SAPHO syndrome and adrenal deficiency.

摘要

引言

滑膜炎、痤疮、脓疱病、骨肥厚、骨髓炎综合征(SAPHO综合征)是一种罕见的疼痛性疾病,通常具有良好的长期预后。其病因尚不清楚,各种治疗方案常常无法控制该病。

病例报告

一名46岁的白种女性因前胸壁和背部疼痛前来就诊。体格检查无异常,仅在双足底、四肢及面部发现皮肤病变。胸部磁共振成像研究显示胸骨有骨髓水肿和软组织增生的病变。因催乳素水平升高并符合空蝶鞍综合征,故进行了脑部MRI检查。

结论

此处报告的病例具有肾上腺功能减退与SAPHO综合征同时出现的独特特征,是首例报告病例。该综合征可能会因骨骼和皮肤以外的不同器官系统受累而变得复杂。有必要进一步开展研究,探索SAPHO综合征与肾上腺功能减退之间的微弱关系。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fcb/2827074/3d5f82f1f6d6/1757-1626-0002-0000006281-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fcb/2827074/3d5f82f1f6d6/1757-1626-0002-0000006281-1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6fcb/2827074/3d5f82f1f6d6/1757-1626-0002-0000006281-1.jpg

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本文引用的文献

1
Valuable lessons from SAPHO syndrome.SAPHO综合征的宝贵经验教训。
Joint Bone Spine. 2007 Mar;74(2):123-6. doi: 10.1016/j.jbspin.2006.06.006. Epub 2007 Jan 31.
2
A study of musculoskeletal manifestations in 12 patients with SAPHO syndrome.
J Clin Rheumatol. 2002 Feb;8(1):13-22. doi: 10.1097/00124743-200202000-00005.
3
Pulmonary adenocarcinoma associated with SAPHO syndrome difficult to differentiate from multiple bone metastasis.与SAPHO综合征相关的肺腺癌难以与多发骨转移相鉴别。
Intern Med. 2006;45(8):543-6. doi: 10.2169/internalmedicine.45.1628. Epub 2006 May 15.
4
A hidradenitis suppurativa related SAPHO case associated with features resembling spondylarthropathy and proteinuria.1例与类似脊柱关节病及蛋白尿特征相关的化脓性汗腺炎相关性SAPHO病例
Clin Rheumatol. 2007 May;26(5):789-91. doi: 10.1007/s10067-005-0199-5. Epub 2006 May 6.
5
Is SAPHO syndrome a target for antibiotic therapy?滑膜炎、痤疮、脓疱病、骨肥厚、骨髓炎综合征(SAPHO综合征)是抗生素治疗的靶点吗?
Clin Rheumatol. 2007 May;26(5):817-20. doi: 10.1007/s10067-006-0274-6. Epub 2006 Apr 7.
6
In SAPHO syndrome anti-TNF-alpha therapy may induce persistent amelioration of osteoarticular complaints, but may exacerbate cutaneous manifestations.在SAPHO综合征中,抗TNF-α治疗可能会使骨关节症状持续改善,但可能会加重皮肤表现。
Rheumatology (Oxford). 2006 Jun;45(6):730-3. doi: 10.1093/rheumatology/kei221. Epub 2006 Jan 10.
7
Isolated thoracic spine lesion: is this the presentation of a SAPHO syndrome? A case report.孤立性胸椎病变:这是SAPHO综合征的表现吗?一例病例报告。
Eur Spine J. 2005 Sep;14(7):711-5. doi: 10.1007/s00586-004-0791-4. Epub 2004 Oct 12.
8
Successful treatment of SAPHO syndrome with zoledronic acid.唑来膦酸成功治疗SAPHO综合征。
Arthritis Rheum. 2004 Sep;50(9):2970-3. doi: 10.1002/art.20464.
9
Diagnostic management of patients with SAPHO syndrome: use of MR imaging to guide bone biopsy at CT for microbiological and histological work-up.SAPHO综合征患者的诊断管理:利用磁共振成像指导计算机断层扫描下的骨活检以进行微生物学和组织学检查。
Eur Radiol. 2003 Oct;13(10):2304-8. doi: 10.1007/s00330-003-1849-0. Epub 2003 Mar 13.
10
Pamidronate: a novel treatment for the SAPHO syndrome?帕米膦酸盐:一种治疗滑膜炎、痤疮、脓疱病、骨肥厚、骨炎综合征(SAPHO综合征)的新疗法?
Rheumatology (Oxford). 2002 Feb;41(2):231-3. doi: 10.1093/rheumatology/41.2.231-a.