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孕期意外接触环磷酰胺的男孩患克莱佩尔-费尔综合征:一例报告

Klippel-Feil syndrome in a boy exposed inadvertently to cyclophosphamide during pregnancy: a case report.

作者信息

Lazalde Brissia, Grijalva-Flores Jesús, Guerrero-Romero Fernando

机构信息

Biomedical Research Unit, Mexican Social Security Institute, Durango, Dgo., México.

出版信息

Birth Defects Res A Clin Mol Teratol. 2012 Apr;94(4):249-52. doi: 10.1002/bdra.23004. Epub 2012 Mar 27.

DOI:10.1002/bdra.23004
PMID:22450872
Abstract

BACKGROUND

Cyclophosphamide (CPA) is an alkylating agent widely used as an immunosuppressive agent in the treatment of several autoimmune diseases, including systemic lupus erythematosus. Its teratogenic effect has been well studied in different experimental mammalian and non-mammalian animal models. In humans, 11 cases of CPA teratogenesis have been documented.

CASE

We present a case of a patient with Klippel-Feil syndrome inadvertently exposed to CPA and prednisone in utero during the first trimester. CONCLUSIONS This case of possible cyclophosphamide embryopathy provides evidence of teratogenesis as an etiologic agent in developmental field defects such as Klippel-Feil syndrome.

摘要

背景

环磷酰胺(CPA)是一种烷化剂,广泛用作免疫抑制剂,用于治疗包括系统性红斑狼疮在内的多种自身免疫性疾病。其致畸作用已在不同的实验性哺乳动物和非哺乳动物动物模型中得到充分研究。在人类中,已有11例CPA致畸的记录。

病例

我们报告一例在孕早期子宫内意外接触CPA和泼尼松的Klippel-Feil综合征患者。结论:这例可能的环磷酰胺胚胎病病例为致畸作用作为Klippel-Feil综合征等发育性场缺陷的病因提供了证据。

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Klippel-Feil syndrome in a boy exposed inadvertently to cyclophosphamide during pregnancy: a case report.孕期意外接触环磷酰胺的男孩患克莱佩尔-费尔综合征:一例报告
Birth Defects Res A Clin Mol Teratol. 2012 Apr;94(4):249-52. doi: 10.1002/bdra.23004. Epub 2012 Mar 27.
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