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自身免疫性卵巢早衰和艾迪生病女性溃疡性结肠炎应用硫唑嘌呤治疗后妊娠:HLA 单倍型特征。

Pregnancy after azathioprine therapy for ulcerative colitis in a woman with autoimmune premature ovarian failure and Addison's disease: HLA haplotype characterization.

机构信息

Section of Endocrinology, Department of Medicine and Pharmacology, University of Messina, Messina, Italy.

出版信息

Fertil Steril. 2011 Jun;95(7):2430.e15-7. doi: 10.1016/j.fertnstert.2011.01.170. Epub 2011 Mar 2.

Abstract

OBJECTIVE

To present a case of fertility restored by azathioprine treatment in a woman with autoimmune premature ovarian failure, Addison's disease, and ulcerative colitis, and to study the genetic background of the three autoimmune diseases.

DESIGN

Case report.

SETTING

Endocrinology and Immunology Units of an university hospital.

PATIENT(S): A 30-year-old woman with autoimmune premature ovarian failure, Addison's disease, and ulcerative colitis.

INTERVENTION(S): Azathioprine has been administered as immunosuppressive treatment.

MAIN OUTCOME MEASURE(S): We performed analysis of human leukocyte antigens expression on lymphocytes and genomic haplotype of the patient.

RESULT(S): The human leukocyte antigen haplotype of the patient was consistent with the haplotypes predisposing for the three autoimmune diseases, as reported in the literature. The administration of azathioprine restored regular menses and allowed uneventful pregnancy.

CONCLUSION(S): This is the first clinical evidence of association of immunosuppressive azathioprine treatment and restored ovarian function and fertility in a woman with autoimmune premature ovarian failure. In this patient, the haplotype was associated with susceptibility to autoimmune premature ovarian failure, Addison's disease, and ulcerative colitis.

摘要

目的

介绍一例自身免疫性卵巢早衰、艾迪生病和溃疡性结肠炎妇女经巯嘌呤治疗后恢复生育能力的病例,并研究三种自身免疫性疾病的遗传背景。

设计

病例报告。

地点

一家大学医院的内分泌学和免疫学系。

患者

一名 30 岁女性,患有自身免疫性卵巢早衰、艾迪生病和溃疡性结肠炎。

干预措施

给予巯嘌呤作为免疫抑制治疗。

主要观察指标

我们对患者的淋巴细胞人类白细胞抗原表达和基因组单倍型进行了分析。

结果

患者的人类白细胞抗原单倍型与文献报道的易患三种自身免疫性疾病的单倍型一致。巯嘌呤的应用恢复了正常月经周期并使妊娠顺利进行。

结论

这是首例关于免疫抑制性巯嘌呤治疗和恢复自身免疫性卵巢早衰妇女卵巢功能和生育能力的临床证据。在该患者中,单倍型与自身免疫性卵巢早衰、艾迪生病和溃疡性结肠炎的易感性相关。

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