PIP: This article is the first account of an infant malformation in a case where a mother had been receiving injected medroxyprogesterone. The D-trisomic (47,XY, D+) infant, born in February 1970 and dead 1 hour after birth, had cyclopia and multiple extracerebral congenital defects. However, a look at the relevant literature suggests the association between the abnormalities and the injected medroxyprogesterone was probably coincidental. The 32-year-old woman has received 50 mg of parenteral medroxyprogesterone every 3 months for 3 years. The last injection was administered about 1/2 of a year before conception. The neurological and physiological abnormalities of the infant are detailed.