Abstract

We report on a patient with malformations following long-term and high-dose maternal fluconazole use during pregnancy. The patient had exorbitism, a large pear-shaped nose, “dysplastic” ears, and radiohumeral synostosis. He is similar to three previously reported patients with in utero exposure to fluconazole and also shares some characteristics with the Antley-Bixler syndrome. The four fluconazole-exposed and malformed infants are compared with one another and with the Antley-Bixler syndrome. We conclude that fluconazole is a teratogen and results in a specific pattern of malformations following first trimester use. Am. J. Med. Genet. 72:253–256, 1997. © 1997 Wiley-Liss, Inc.

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