Background: Kwashi shakes is a rare neurological complication of unknown aetiology in children with severe acute malnutrition. Previous reports show that it usually presents as coarse tremors one to eight weeks after commencement of nutritional rehabilitation in children with kwashiokor. We present a case of kwashi shakes with no prior nutritional rehabilitation. This is the first case of kwashi shakes managed in our facility. Methodology: A two year old male presented with a three day history of progressive body swelling and jerky body movements. He stopped breastfeeding at one year and was fed 2-3 times daily on mainly carbohydrates and beans. On examination, he was conscious, apathetic, wasted, moderately pale with sparse fluffy brown hair and pitting pedal oedema. His weight and length were 7kg and 69cm respectively (Z-score of <-3SD for age and sex). He had continuous coarse trembling of the upper limbs and head nodding. The trembling was worse during voluntary movements and reduced and stopped during rest and sleep. His tone was reduced in all limbs. Investigations showed a packed cell volume of 26%, elevated white cell counts, positive smear for plasmodium falciparum and normal findings of serum glucose, calcium, electrolytes, and cerebrospinal fluid analysis. He responded to nutritional rehabilitation, antibiotics, antimalarial and multivitamins. His symptoms subsided over two weeks. Conclusion: The case report shows an unusual presentation of kwashi shakes where the patient presented without any prior nutritional rehabilitation. Health care probiders should be on the lookout and should not confuse it with meningoencephalitis or seizure disorder.