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Licensed Unlicensed Requires Authentication Published by De Gruyter April 14, 2011

Aplasia cutis congenita in surviving co-twin after propylthiouracil exposure in utero

  • Ruth M. Löllgen , Anne-Marie Calza , Valérie M. Schwitzgebel and Riccardo E. Pfister EMAIL logo

Abstract

Aim: Aplasia cutis congenita (ACC) has been observed after fetal exposure to the antithyroid drug methimazole (MMI), but not reported after propylthiouracil (PTU), the current antithyroid drug of choice during pregnancy. This occurrence has implications for patient information and causal research.

Case report: We describe a surviving term co-twin to a mother with hyperthyroidism exposed to PTU from conception to 34 weeks of gestation presenting with ACC at birth.

Discussion: The association between PTU exposure and ACC is clinically relevant and allows speculation on the etiology. A similar mechanism to the classical MMI-induced ACC is postulated, unless a vascular etiology suggested by a vanishing twin or maternal hyperthyroidism itself is causal. Coincidence of PTU exposure and ACC seems unlikely.

Conclusion: ACC in a newborn after PTU exposure during pregnancy hitherto observed only after MMI strongly encourages further reports of similar cases that may remain clinically underdiagnosed or unreported. Such confirmation could have significant implications for maternal treatment of hyperthyroidism, common in women of childbearing age.


Corresponding author: Dr. R.E. Pfister, Neonatology Unit, University Children’s Hospital, 6, Rue Willy-Donzé, 1211 Geneva, Switzerland Phone: +41-22-3824351, Fax: +41-22-3824365

Published Online: 2011-04-14
Published in Print: 2011-04-01

©2011 by Walter de Gruyter Berlin New York

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