Successful treatment of steroid resistant ulcerative colitis associated with severe autoimmune hemolytic anemia with oral microemulsion cyclosporin - A brief case report [6]

Research output: Contribution to journalLetter

5 Citations (Scopus)

Abstract

DosageDuration:Oral microemulsion at 5 mg/kg (serum concentrations, 81-83 ng/ml), 10 months later, dose was reduced to 4 mg/kg. Duration: over 10 months.

TypeofStudy:The successful treatment of steroid resistant ulcerative colitis (UC) associated with severe autoimmune hemolytic anemia with oral Sandimmun Neoral is reported as a case report in a letter. Letters to the editor.

Results:Following administration of Neoral at an initial dosage of 5 mg/kg, the activity of UC started to decrease, and the hemolysis resolved after 1 week of treatment. 10 months later, she received combined azathioprine (2.5 mg/kg) and Neoral (4 mg/kg) therapy. Under this therapeutic regimen, there are still mild signs of UC activity but without hemolysis (Coombs test is consequently negative).

AdverseEffects:No adverse events were mentioned.

AuthorsConclusions:Our case suggests that CsA [cyclosporine A] may have beneficial effect in the treatment of another extraintestinal manifestation, namely, autoimmune haemolytic anemia. The reduction of UC activity is surely a key factor of this mechanism, but CsA may also influence the autoimmune hemolysis itself in a favorable manner. The rare association of autoimmune hemolysis and UC probably makes placebo-controlled studies unlikely; therefore, on the basis of these few reported cases, the use of CsA in the treatment of these associated conditions is worth considering.

FreeText:Patient was admitted because of a severe flare-up of UC associated with mild jaundice and poor general well-being. Her pancolitis was diagnosed according to standard endoscopic and histological criteria in 1988. An autoimmune Coombs positive hemolytic anemia with cold and warm autoantibodies developed in July, 1998. 12 units of erythrocyte concentration were transfused. A high dose steroid (methylprednisone, 2 mg/kg intravenously) and azathioprine (2 mg/kg) therapy was then introduced. After a 9-month remission achieved with azathioprine, UC and hemolysis reactivated simultaneously. She underwent transfusion again, steroid use was restarted (methylprednisone, 1 mg/kg), and danazol was started (400 mg daily) in combination with azathioprine. After a partial response, splenectomy was performed because of the therapy resistant hemolysis in May, 2000. 3 months before the patient's admission, bloody diarrhea started (number of stools, 10-20/day). She had a weight loss of 12 kg until October, 2001. Her physical examination showed a moonface caused by steroid therapy, as well as anemia, mild jaundice, and abdominal tenderness at the time of admission. Laboratory values were as follows: erythrocyte sedimention rate 77 mm/h, hemoglobin 84 g/L, hematocrit 27%; serum bilirubin total 22.37 mol/L, lactate dehydrogenase 819 g/ml, and C reactive protein (CRP) 68 mg/L. Direct and indirect Coombs tests were both highly positive (+ + + +). As an ultimate preoperative therapeutic possibility, Neoral was given.

Indications:1 patient with steroid resistant ulcerative colitis associated with severe autoimmune hemolytic anemia.

Patients:One 35-year-old female (inpatient).

Original languageEnglish
Pages (from-to)1207-1208
Number of pages2
JournalAmerican Journal of Gastroenterology
Volume98
Issue number5
DOIs
Publication statusPublished - May 1 2003

ASJC Scopus subject areas

  • Hepatology
  • Gastroenterology

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