Introduction: Hyperviscosity syndrome is clinically manifested by oronasal bleeding, retinal haemorrhages, and variable neuro-logical symptoms. The most reliable clinical manifestations of serum hyperviscosity are visual disturbances and oronasal bleed. Its early recognition is important as it constitutes important morbidity and may lead to blindness. Case presentation: Herein, we present a case of a newborn female on day 3 of life who presented with lethargy, poor feeding, tachypnoea and cyanosis. Dilated fundoscopy showed extensive mid-peripheral retinal haemorrhages with dilated and tortuous retinal veins. The patient was investigated and haematocrit, blood counts, and bilirubin level was found to be raised due to hyperviscosity of blood. The patient was managed with phototherapy haemodilution to decrease blood viscosity which resulted in the uneventful recovery of the patient with normal blood count, haematocrit and other lab values. On follow up after two weeks, ophthalmoscopy showed normal retinal vessels with few retinal haemorrhages. Discussion: We describe a unique case of retinopathy associated with hyperviscosity syndrome. The retinal changes as seen in our case were due to increased haematocrit and blood viscosity. As there are scarcely a few studies about retinopathy associated with hyperviscosity syndrome, this case illustrates the prompt and accurate diagnosis of hyperviscosity syndrome for the institution of appropriate therapy.