ループス膀胱炎を合併した小児期発症全身性エリテマトーデスの一例 A case report of childhood systemic lupus erythematosus complicated with lupus cystitis

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  症例は13歳,女児.2000年8月発熱,下痢,嘔吐,関節痛,鼻出血,全身倦怠感あり,前医を受診した.血小板減少,抗核抗体,抗DNA抗体,PAIgGが陽性のため特発性血小板減少性紫斑病,全身性エリテマトーデス(SLE)の疑いで経過観察されていた.2001年1月麻疹に罹患後より,腹痛が出現し,尿蛋白・潜血陽性,左水腎,左尿管狭窄・拡張を認めた.症状は輸液,NSAIDsで軽快したが,2月発熱,両頬部紅斑認めSLEと診断され,当科入院となった.患児はシェーグレン症候群を併発したSLEで,経静脈性腎盂造影で左水腎,左尿管狭窄・拡張,膀胱内視鏡で左尿管口付近に発赤を認めた.同部位の病理所見で,SLEにみられる血管炎の所見が存在し,ループス膀胱炎と診断した.外科的治療は行わず,経静脈的シクロホスファミド(IVCY)パルス療法(計8回,1年間)を導入し,維持治療をプレドニゾロン,アザチオプリンとした.IVCYパルス療法1年間終了後,左水腎症,尿管拡張・狭窄は著明に改善した.副作用も認めておらず,現在内服治療のみで寛解維持ができている.小児期発症SLEにループス膀胱炎を合併し,IVCYパルス療法が著効した症例を経験したため報告した.<br>

  The patient was a 13-year-old girl. In August 2000, she presented with a fever, together with diarrhea, vomiting, arthralgia, nasal bleeding and malaise, and was examined by another physician. Because her platelet count was low, and there were positive reactions for anti-nuclear antibodies, anti-DNA antibodies and platelet-associated IgG, idiopathic thrombopenic purpura, and systemic lupus erythematosus (SLE) was suspected. From January 2001, when she caught measles, she reported abdominal pain, and urinalysis indicated urinary protein and occult blood, and the left kidney was found hydronephrotic. At the same time left ureter stenosis and dilatation were demonstrated. Symptoms were disappeared by hydration and treatment with NSAIDs, but 2 months later fever and erythematous patches seen on both cheeks led to the proper diagnosis of SLE, and she was admitted to our hospital. Intravenous pyelography revealed hydronephrosis on left kidney, constriction and dilatation of the left ureter, and intracystic endoscopy showed erythema at the orifice of the left ureter. The pathological examination indicated the presence of vasculitis, and finally lupus cystitis was diagnosed. Intravenous cyclophosphamide (IVCY)-pulse therapy was introduced to a total of 8 times over the period of a year, and maintenance therapy with predonisolone and azathioprin was also used. After completion of the IVCY-pulse therapy, the hydronephrosis and constriction of the ureter were disappeared. No side effects of IVCY-pulses were observed, and the patient is now in remission. We reported a case of childhood SLE complicated with lupus cystitis and successfully treated by IVCY-pulse therapy and maintenance predonisolone and azathioprin.<br>

収録刊行物

  • 日本臨床免疫学会会誌 = Japanese journal of clinical immunology  

    日本臨床免疫学会会誌 = Japanese journal of clinical immunology 29(3), 154-159, 2006-06-30 

    The Japan Society for Clinical Immunology

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各種コード

  • NII論文ID(NAID)
    10018057772
  • NII書誌ID(NCID)
    AN00357971
  • 本文言語コード
    JPN
  • 資料種別
    NOT
  • ISSN
    09114300
  • データ提供元
    CJP書誌  CJP引用  J-STAGE 
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