• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

干燥综合征合并范科尼综合征及骨软化症

Sjögren's Syndrome Associated with Fanconi's Syndrome and Osteomalacia.

作者信息

Gutierrez Jorge O, Zurita Maria F, Zurita Luis A

机构信息

School of Medicine, Universidad Espíritu Santo (Holy Spirit University), Samborondón, Ecuador.

Department of Rheumatology, Unidad de Enfermedades Reumáticas y Autoinmunes [UNERA] (Unit of Rheumatology and Autoimmune Diseases), Guayaquil, Ecuador.

出版信息

Am J Case Rep. 2018 Apr 3;19:392-396. doi: 10.12659/ajcr.907503.

DOI:10.12659/ajcr.907503
PMID:29610453
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5900466/
Abstract

BACKGROUND Sjögren's syndrome is a chronic inflammatory autoimmune disease, which is also known as sicca syndrome, due to the symptoms of dry eyes and dry mouth, and is associated with other connective tissue diseases and autoimmune diseases. Sjögren's syndrome can also be associated with renal involvement. Fanconi's syndrome is associated with impaired reabsorption in the proximal renal tubule associated with tubulointerstitial nephritis and is associated with renal tubular acidosis and hypophosphatemia. Osteomalacia is a rare association with Sjögren's syndrome, which may result from renal disease. CASE REPORT We report the case of a 34-year-old woman who presented with xerostomia, xerophthalmia, bone fractures, and osteomuscular pain. A Schirmer test showed reduced tear production, and a biopsy of a minor salivary gland of the lip, with high titers of antinuclear antibodies (ANA), and positive anti-SSA/Ro and anti-SSB/La antibodies confirmed the diagnosis of Sjögren's syndrome. Serum and urinary laboratories tests and clinical manifestations confirmed Fanconi's syndrome associated with osteomalacia. The patient was treated with potassium supplements, 25-hydroxyvitamin D (25(OH)D), hydroxychloroquine, mycophenolate mofetil, and prednisone, with a favorable response. CONCLUSIONS This case is of a rare association between Sjögren's syndrome, Fanconi's syndrome, and osteomalacia. Even though these are rare clinical associations, early detection can improve the quality of life and prevent further complications.

摘要

背景 干燥综合征是一种慢性炎症性自身免疫性疾病,因其干眼和口干症状也被称为口眼干燥症,且与其他结缔组织疾病和自身免疫性疾病相关。干燥综合征也可伴有肾脏受累。范科尼综合征与近端肾小管重吸收受损有关,伴有肾小管间质性肾炎,并与肾小管酸中毒和低磷血症相关。骨软化症是干燥综合征一种罕见的关联疾病,可能由肾脏疾病引起。病例报告 我们报告一例34岁女性患者,其表现为口干、眼干、骨折和骨肌肉疼痛。施密特试验显示泪液分泌减少,唇部小唾液腺活检显示抗核抗体(ANA)滴度高,抗SSA/Ro和抗SSB/La抗体阳性,确诊为干燥综合征。血清和尿液实验室检查及临床表现确诊为与骨软化症相关的范科尼综合征。该患者接受了补钾、25-羟维生素D(25(OH)D)、羟氯喹、霉酚酸酯和泼尼松治疗,反应良好。结论 本病例为干燥综合征、范科尼综合征和骨软化症之间罕见的关联。尽管这些是罕见的临床关联,但早期检测可改善生活质量并预防进一步并发症。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cdfc/5900466/6b0f263ba8f6/amjcaserep-19-392-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cdfc/5900466/3c35b32f23e1/amjcaserep-19-392-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cdfc/5900466/6b0f263ba8f6/amjcaserep-19-392-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cdfc/5900466/3c35b32f23e1/amjcaserep-19-392-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cdfc/5900466/6b0f263ba8f6/amjcaserep-19-392-g002.jpg

相似文献

1
Sjögren's Syndrome Associated with Fanconi's Syndrome and Osteomalacia.干燥综合征合并范科尼综合征及骨软化症
Am J Case Rep. 2018 Apr 3;19:392-396. doi: 10.12659/ajcr.907503.
2
Acquired hypophosphatemia osteomalacia associated with Fanconi's syndrome in Sjögren's syndrome.干燥综合征中与范科尼综合征相关的获得性低磷血症性骨软化症。
Rheumatol Int. 2007 Apr;27(6):593-7. doi: 10.1007/s00296-006-0257-6. Epub 2006 Nov 9.
3
Osteomalacia complicating renal tubular acidosis in association with Sjogren's syndrome.骨软化症合并肾小管酸中毒及干燥综合征
Saudi J Kidney Dis Transpl. 2014 Sep;25(5):1072-7. doi: 10.4103/1319-2442.139944.
4
Osteomalacia associated with adult Fanconi's syndrome: clinical and diagnostic features.成人范科尼综合征相关的骨软化症:临床及诊断特征
Clin Endocrinol (Oxf). 1995 Oct;43(4):479-90. doi: 10.1111/j.1365-2265.1995.tb02621.x.
5
Tubulointerstitial nephritis-induced hypophosphatemic osteomalacia in Sjögren's syndrome: a case report and review of the literature.干燥综合征肾小管间质性肾炎致低血磷性骨软化症 1 例报告并文献复习
Clin Rheumatol. 2018 Jan;37(1):257-263. doi: 10.1007/s10067-017-3762-y. Epub 2017 Jul 20.
6
Fanconi's syndrome and distal (type 1) renal tubular acidosis in a patient with primary Sjögren's syndrome with monoclonal gammopathy of undetermined significance.原发性干燥综合征合并意义未明单克隆丙种球蛋白病患者出现范科尼综合征和远端(1型)肾小管酸中毒。
Clin Nephrol. 2006 Jun;65(6):427-32. doi: 10.5414/cnp65427.
7
Multiple bone fracture due to Fanconi's syndrome in primary Sjögren's syndrome complicated with organizing pneumonia.原发性干燥综合征合并机化性肺炎致范可尼综合征引起多处骨折。
Rheumatol Int. 2009 Dec;30(2):265-7. doi: 10.1007/s00296-009-0924-5.
8
Hypokalemic paralysis as primary presentation of Fanconi syndrome associated with Sjögren syndrome.低钾性麻痹为干燥综合征合并范可尼综合征的首发表现
J Clin Rheumatol. 2010 Jun;16(4):178-80. doi: 10.1097/RHU.0b013e3181df903f.
9
[The clinical characteristics of 26 cases of hypophosphatemia osteomalacia misdiagnosed as spondyloarthritis].[26例误诊为脊柱关节炎的低磷性骨软化症临床特征分析]
Zhonghua Nei Ke Za Zhi. 2014 Nov;53(11):847-51.
10
[Sjögren's syndrome - case report].[干燥综合征——病例报告]
Rev Chil Pediatr. 2015 Jan-Feb;86(1):47-51. doi: 10.1016/j.rchipe.2015.04.009.

引用本文的文献

1
Musculoskeletal Manifestations in Sjogren's Syndrome: An Orthopedic Point of View.干燥综合征的肌肉骨骼表现:骨科视角
J Clin Med. 2021 Apr 8;10(8):1574. doi: 10.3390/jcm10081574.
2
Renal Tubular Acidosis and Management Strategies: A Narrative Review.肾小管酸中毒与治疗策略:一篇叙述性综述
Adv Ther. 2021 Feb;38(2):949-968. doi: 10.1007/s12325-020-01587-5. Epub 2020 Dec 26.
3
Biomarkers and Diagnostic Testing for Renal Disease in Sjogren's Syndrome.干燥综合征相关肾脏疾病的生物标志物与诊断性检测。

本文引用的文献

1
A Case of Kidney Involvement in Primary Sjögren's Syndrome.原发性干燥综合征累及肾脏一例。
Am J Case Rep. 2017 Jun 3;18:622-626. doi: 10.12659/ajcr.903476.
2
Crucial Role of Vitamin D in the Musculoskeletal System.维生素D在肌肉骨骼系统中的关键作用。
Nutrients. 2016 Jun 1;8(6):319. doi: 10.3390/nu8060319.
3
Sjögren's syndrome complicated with Fanconi syndrome and Hashimoto's thyroiditis: Case report and literature review.干燥综合征合并范科尼综合征和桥本甲状腺炎:病例报告及文献复习
Front Immunol. 2020 Sep 17;11:562101. doi: 10.3389/fimmu.2020.562101. eCollection 2020.
J Int Med Res. 2016 Jun;44(3):753-9. doi: 10.1177/0300060515593767. Epub 2016 Mar 9.
4
Tubulointerstitial nephritis in primary Sjögren syndrome: clinical manifestations and response to treatment.原发性干燥综合征中的肾小管间质性肾炎:临床表现及治疗反应
BMC Musculoskelet Disord. 2016 Jan 5;17:2. doi: 10.1186/s12891-015-0858-x.
5
Renal involvement in primary Sjögren syndrome.原发性干燥综合征的肾脏受累。
Nat Rev Nephrol. 2016 Feb;12(2):82-93. doi: 10.1038/nrneph.2015.174. Epub 2015 Nov 16.
6
Osteomalacia as inaugural manifestation of Sjögren syndrome.骨软化症作为干燥综合征的首发表现
BMJ Case Rep. 2013 Oct 9;2013:bcr2013201052. doi: 10.1136/bcr-2013-201052.
7
Sjögren syndrome presenting with hypopotassemic periodic paralysis due to renal tubular acidosis.干燥综合征合并肾小管酸中毒所致低钾性周期性麻痹。
Am J Case Rep. 2012;13:187-90. doi: 10.12659/AJCR.883326. Epub 2012 Aug 16.
8
Metabolic bone disease as a presenting manifestation of primary Sjögren's syndrome: Three cases and review of literature.代谢性骨病作为原发性干燥综合征的首发表现:三例报告并文献复习
Indian J Endocrinol Metab. 2011 Oct;15(4):341-5. doi: 10.4103/2230-8210.85599.
9
Hypokalemic paralysis as primary presentation of Fanconi syndrome associated with Sjögren syndrome.低钾性麻痹为干燥综合征合并范可尼综合征的首发表现
J Clin Rheumatol. 2010 Jun;16(4):178-80. doi: 10.1097/RHU.0b013e3181df903f.
10
Multiple bone fracture due to Fanconi's syndrome in primary Sjögren's syndrome complicated with organizing pneumonia.原发性干燥综合征合并机化性肺炎致范可尼综合征引起多处骨折。
Rheumatol Int. 2009 Dec;30(2):265-7. doi: 10.1007/s00296-009-0924-5.