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库欣综合征手术缓解后出现的伴有凹陷性水肿的缓解期血清阴性对称性滑膜炎。

Remitting seronegative symmetrical synovitis with pitting oedema after surgical remission of Cushing's syndrome.

作者信息

Iwasaki Hiroaki, Kanno Hitomi, Jiang Shi-Xu

机构信息

Department of Internal Medicine, Toshiba Rinkan Hospital, Sagamihara, Kanagawa, Japan

Department of Urology, Toshiba Rinkan Hospital, Sagamihara, Kanagawa, Japan.

出版信息

BMJ Case Rep. 2020 Mar 31;13(3):e232959. doi: 10.1136/bcr-2019-232959.

Abstract

A 64-year-old woman with refractory cellulitis in the lower legs was referred for inadequate glycaemic control. Physical examination revealed cushingoid features including central obesity. CT of the abdomen revealed a right adrenal mass that was positive on I-adosterol imaging. Findings on endocrine evaluation confirmed a diagnosis of Cushing's syndrome, which was cured with a right adrenalectomy. Two months after surgery, the patient complained of pain and marked swelling of the hands during hydrocortisone replacement therapy (20 mg per day) given for postoperative adrenal insufficiency. Laboratory examination was unremarkable. However, contrast-enhanced T2-weighted MRI of the hands revealed enhanced signals surrounding the flexor tendons, leading to a diagnosis of remitting seronegative symmetrical synovitis with pitting oedema. Prednisolone (15 mg per day) was then initiated, and the symptoms disappeared within a few days. This case illustrates the possibility that successful treatment of Cushing's syndrome may trigger emergence of a glucocorticoid-responsive disease.

摘要

一名64岁患有难治性小腿蜂窝织炎的女性因血糖控制不佳前来就诊。体格检查发现有库欣样特征,包括向心性肥胖。腹部CT显示右侧肾上腺肿块,碘胆固醇成像呈阳性。内分泌评估结果确诊为库欣综合征,通过右侧肾上腺切除术治愈。术后两个月,患者在接受术后肾上腺功能不全的氢化可的松替代治疗(每日20毫克)时,抱怨手部疼痛并明显肿胀。实验室检查无异常。然而,手部对比增强T2加权磁共振成像显示屈肌腱周围信号增强,诊断为缓解型血清阴性对称性滑膜炎伴凹陷性水肿。随后开始使用泼尼松龙(每日15毫克),症状在几天内消失。该病例表明成功治疗库欣综合征可能引发糖皮质激素反应性疾病。

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N Engl J Med. 2017 Apr 13;376(15):1451-1459. doi: 10.1056/NEJMra1505550.
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Nat Rev Rheumatol. 2014 Nov;10(11):662-70. doi: 10.1038/nrrheum.2014.138. Epub 2014 Aug 19.
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Endocrinol Metab Clin North Am. 2013 Dec;42(4):747-87. doi: 10.1016/j.ecl.2013.06.001.
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