A CASE REPORT OF ISOLATED INFANTILE HEMANGIOMA WITH MICROPHTHALMOS Purpose: To report a rare case of infantile facial hemangioma with orbital extension associated with microphthalmos and high intraocular pressure of the left eye. Case: A full term 1-day-old girl presented with left sided facial hemangiomas since birth. Observations: Findings include large left-sided facial hemangiomas with orbital extension enveloping the left lateral rectus and optic nerve. Clinical examination showed no involvement of the upper and lower eyelid. Cycloplegic refraction showed anisometropia between the two eyes. The axial length of the left eye was found to be significantly shorter compared to the right eye, and intraocular pressure of the left eye was high. Conclusions: Infantile facial hemangioma may be present with orbital extension without any eyelid involvement, and associated with microphthalmos of the left eye with no other systemic abnormalities.