Recurrent autoimmune hypophysitis successfully treated with glucocorticoids plus azathioprine: a report of three cases

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  • Yang Guo-Qing
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Lu Zhao-Hui
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Gu Wei-Jun
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Du Jin
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Guo Qing-Hua
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Wang Xian-Ling
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Ba Jian-Ming
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Dou Jing-Tao
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Mu Yi-Ming
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China
  • Lu Ju-Ming
    Department of Endocrinology, Chinese PLA General Hospital, Beijing, PR China

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抄録

Recurrent autoimmune hypophysitis is a rare autoimmune endocrine disease involving lymphocytic infiltration and chronic pituitary inflammation. It is even more rare than primary hypophysitis. The objective of the study was to evaluate the efficacy of glucocorticoid treatment combined with azathioprine for treating three cases of recurrent autoimmune lymphocytic hypophysitis encountered within a two-year period. The clinical features and follow-up data of these cases were analyzed, including results of treatment with glucocorticoids combined with azathioprine. All three patients were female and presented with the following clinical characteristics: case 1 was a 22-year-old with headache and diplopia; case 2 was a 70-year-old with dry mouth, polydipsia, and polyuria; case 3, a 32-year-old, with polydipsia, polyuria and menstrual disorders with headache and dizziness. Regarding recurrence, case 1 recurred 4 months after surgery and again 14 months after discontinuing prednisone; case 2 relapsed 16 months after receiving high-dose methylprednisolone pulse therapy; and case 3 recurred during the period of prednisone dose reduction. The patients were treated with glucocorticoids plus azathioprine, and positive responses were seen in all three cases. Symptoms were relieved, and MRI revealed significant reduction of lesions during follow-up. Pituitary function resumed in cases 1 and 3; permanent hypopituitarism was present in case 2. At last follow-up, MRI showed no further recurrence of disease in any patient. Treatment and responses of these patients with autoimmune hypophysitis suggest that glucocorticoid therapy combined with azothioprine is effective treatment for recurrent autoimmune hypophysitis. Endocrine and radiologic studies are an essential part of follow-up.

収録刊行物

  • Endocrine Journal

    Endocrine Journal 58 (8), 675-683, 2011

    一般社団法人 日本内分泌学会

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