Objective: Cyclopia is a rare form of holoprosencephaly and is a congenital disorder characterised by the failure of the embryonic prosencephalon to properly divide the orbits of the eye into two cavities 
occurring between the 18th and the 28th day of gestation. It is the most severe facial expression of the holoprosencephaly syndrome. Its incidence is 1 in 100 000 in newborns. In Indonesia, similar case was reported on 2018 at Panyabungan, North Sumatera.
Case: A baby weighing 6300 gram was born by sectio caesarean to a 37 years old woman, G10P9L8A1 at 41 weeks of gestation. The baby was not breathing nor crying immediately after birth, receiving initial steps of resuscitation, baby started to cry weakly with labored breathing.  On physical examination the newborn was macrocephal with head circumference of 66cm, dysmorphic face with a median single eye, absence of nose, low set ears and presence of proboscis above the eye, all of which made cyclopia the possible initial diagnosis. Lower limb examination showed sign of clubfoot. Laboratory examination showed no abnormal findings. Imaging showed left pulmonary hypoplasia, spina bifida on thoracal 12, lumbal 3 and 4, lumbarisation of S1, bilateral congenital talipes equinovarus (CTEV), and macrocephaly which was revealed by ultrasonography to be suggestive of alobar holoprosencephaly. Patient received continuous positive airway pressure (CPAP) through mouth for breathing support, eventually died of respiratory failure 10 hours after born. 
Conclusion: Holoprosencephaly with cyclopia is rare condition that should be detected as early as possible in pregnancy. The etiology is still largely unknown. Possible risk factors include chromosomal and genetic causes, infections during pregnancy, usage of drugs, maternal diabetes, and advanced mother age.  In this case, the most possible risk factor for this congenital defects is advanced maternal age.