Truncal aplasia cutis congenita following prenatal exposure to misoprostol

Introduction

Aplasia cutis congenital (ACC) is a rare and heterogeneous dermatosis. It can be syndromic, familial or in most cases isolated and sporadic. The pathogeny remains unclear: different etiologic factors are considered such as genetic, vascular and traumatic factors, or drug induced. More often, ACC is localized on the scalp, as a unique lesion. Truncal ACC represents 15% of the cases and is usually extensive and symmetric.

Case report

A newborn baby boy presented at birth with multiple areas of aplasia cutis localized in the periumbilical region, on the sides and on the hips facing the femoral heads. Wound management was performed using low adherent silicone dressing (Mepitel®) and resulted in a complete healing after one month of treatment.

Relevant obstetrical history was marked by a failure of voluntary termination of pregnancy consisting of a non-effective use of intravaginal misoprostol (Cytotec®) one shot, followed by a failed curettage at 9 weeks of amenorrhea. The baby was born by caesarean delivery at 32 weeks of pregnancy after a premature rupture of membranes complicated by an acute chorioamnionitis. Placenta histological examination showed no sign of vascular infarctus and no sign of a papyraceous fetus. The karyotype of the newborn child was normal. No other birth defect was present. The mother had not taken any other medication during her pregnancy.

Today, at the corrected age of three months, the newborn has a normal neurological and psychomotor development.

Discussion

The case report was submitted to the Belgian Centre for Pharmacovigilance and the experts established a causal relationship between the prenatal exposure to misoprostol and the truncal aplasia cutis congenita.

Cases of cephalic aplasia cutis after prenatal exposure to misoprostol are reported in literature. Misoprostol is a well-known teratogenic agent: exposure to misoprostol during the first trimester of pregnancy increases the risk of severe birth defect for example Moebius syndrome, limb defect and central nervous system abnormalities.

In conclusion, this is the first case report of aplasia cutis congenita involving multiple and non-cephalic areas caused by a prenatal exposure to misoprostol.

Alexia Degraeuwe1, Audrey Bulinckx1, Antoine Bachy ², Pierre-Paul Roquet-Gravy1.
1 Dermatology, 2 Neonatology, Grand Hôpital de Charleroi, Charleroi, Belgium.