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子宫内暴露于霉酚酸酯:一种特征性表型?

In utero exposure to mycophenolate mofetil: a characteristic phenotype?

作者信息

Perez-Aytes Antonio, Ledo Ana, Boso Virginia, Sáenz Pilar, Roma Eva, Poveda José Luis, Vento Maximo

机构信息

Servicio de Neonatologia, Hospital Universitario Materno-Infantil La Fe, Valencia, Spain.

出版信息

Am J Med Genet A. 2008 Jan 1;146A(1):1-7. doi: 10.1002/ajmg.a.32117.

Abstract

Mycophenolate mofetil (MMF) is a widely prescribed immunosuppressive agent after solid organ transplantation. Potential teratogenic effects after in utero exposure to MMF in experimental studies and clinical observations in humans has been postulated in recent literature. However, a specific pattern of malformation has not been identified yet. We present a newborn patient, born to a recipient of renal transplantation, who became pregnant while taking MMF as immunosuppressive therapy. The newborn exhibited cleft lip and palate, bilateral microtia and atretic external auditory canals, chorioretinal coloboma, hypertelorism, and micrognathia. An extensive review of the literature documented six other cases with similar malformations after in utero exposure to MMF. A consistent pattern of malformations comprising cleft lip and palate, microtia and external auditory canals could be observed in five of the six cases. A different malformative pattern observed in one of the patients could be attributed to a different agent rather than MMF. The possible teratogenic effects of other immunosuppressive drugs, such as tacrolimus and prednisone, to which this patient was also exposed, are discussed herein. In addition, the differential diagnosis with other dysmorphic syndromes that can present with a similar phenotype, such as CHARGE syndrome, 18q deletion and hypertelorism-microtia-clefting (HMC) syndrome, is presented. We conclude that in utero exposure to MMF can cause a characteristic phenotype and propose the existence of a mycophenolate-associated embryopathy whose main features are: cleft lip and palate, microtia with atresia of external auditory canal, micrognathia and hypertelorism. Ocular anomalies, corpus callosum agenesis, heart defects, kidney malformations, and diaphragmatic hernia may be part of the phenotypic spectrum of MMF embryopathy. The human teratogenicity of MMF is reinforced by this report, and the current contraceptive recommendations about its use in fertile women are stressed.

摘要

霉酚酸酯(MMF)是实体器官移植后广泛使用的免疫抑制剂。近期文献推测,实验研究和人体临床观察显示,子宫内接触MMF后可能存在致畸作用。然而,尚未确定具体的畸形模式。我们报告了一名肾移植受者所生的新生儿,其母亲在接受MMF免疫抑制治疗期间怀孕。该新生儿表现为唇腭裂、双侧小耳畸形及外耳道闭锁、脉络膜视网膜缺损、眼距增宽和小颌畸形。广泛的文献回顾记录了另外6例子宫内接触MMF后出现类似畸形的病例。在这6例病例中的5例中,可以观察到包括唇腭裂、小耳畸形和外耳道在内的一致畸形模式。在其中1例患者中观察到的不同畸形模式可能归因于另一种药物而非MMF。本文还讨论了该患者同时接触的其他免疫抑制药物(如他克莫司和泼尼松)可能的致畸作用。此外,还介绍了与其他可能表现出类似表型的畸形综合征(如CHARGE综合征、18q缺失和眼距增宽-小耳畸形-腭裂(HMC)综合征)的鉴别诊断。我们得出结论,子宫内接触MMF可导致特征性表型,并提出存在一种与霉酚酸相关的胚胎病,其主要特征为:唇腭裂、外耳道闭锁的小耳畸形、小颌畸形和眼距增宽。眼部异常、胼胝体发育不全、心脏缺陷、肾脏畸形和膈疝可能是MMF胚胎病表型谱的一部分。本报告强化了MMF对人类的致畸性,并强调了目前关于其在育龄妇女中使用的避孕建议。

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