Introduction: Carbon Monoxide (CO) is a colourless, odourless, tasteless gas. Mild poisoning by CO can be Mistaken for a non-specific headache or a viral illness headache or viral illness. Moderate to severe CO poisoning produces significant morbidity and mortality that provokes treatment controversy. Rhabdomyolysis, compartment syndrome, renal failure and peripheral neuropathy are unusual complications of CO That can be faced during practice and should be considered by physicians. Case presentation: A 34-years old Egyptian male was referred to the Toxicology Unit from the ED for evaluation of his lower limb weakness. Initial examination revealed a fully conscious patient with stable vital signs and arterial blood gases. However, the patient cannot stand steadily or walk. Neurologic examination revealed hypotonia and diminished reflexes in both lower limbs. No history of toxin exposure but a condition of sudden acute illness affecting him and his parents and upon which his old-age parents, were transferred to ICUin coma. Investigations revealed increased serum alanine transaminase and serum creatinine. A work-up that involved appropriate imaging and serum creatinekinase (CK) measurement revealed extremely elevated serum CK, normal appearance of liver and increased echogenicity of both kidneys with preserved cortico-medullary differentiation. Based on the above-mentioned data, a diagnosis of rhabdomyolysis complicating acute CO exposure with secondary renal insult was made. The patient begins haemodialysis with follow up of his serum K+, creatinine and CK. After one month, levels of serum creatinine and alanine transaminase became normal. The patient underwent rehabilitation therapy to improve his neuromuscular state. Conclusion: Carbon monoxide poisoning should be suspected in patients presented with acute illness without prior medical or surgical cause, and rare complications of CO poisoning like rhabdomyolysis should be suspected particularly in patients with delayed seek of medical care. Timely prompt medical care involving team therapy is necessary to prevent further complications like RF and muscle wasting.