| Summary: | Methemoglobinemia is an uncommon cause of cyanosis that may present at any age. Although acquired methemoglobinemia secondary to exposure to oxidative stressors is the most common cause of methemoglobinemia in adults, hereditary methemoglobinemia has to be considered in patients presenting with cyanosis during infancy and childhood. We report the case of a patient who was investigated for cyanosis that was only noted at the age of 15 years. Despite the negative family history, he was diagnosed with hereditary autosomal recessive methemoglobinemia with no other factors that can be identified to cause such an elevation of methemoglobin level. This case illustrates that hereditary methemoglobinemia has to be considered in such patients even if they were completely asymptomatic for many years of their lives.
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