Nitrous oxide (NO) is a widely used anesthetic agent. We report two patients with sickle cell disease (SCD) who presented with complications following the use of NO. Patient 1, a 15-year-old girl, presented severe hyperhomocysteinemia, pancytopenia, vitamin B deficiency, and peripheral polyneuropathy after massive use of NO for pain management. At the 1-year follow-up, hyperhomocysteinemia and B deficiency had resolved, but she had persisting mild symptoms of polyneuropathy. Patient 2, a 17-year-old boy, presented only severe hyperhomocysteinemia, only partially corrected by initial B supplementation. Careful monitoring of NO use, especially in patients with SCD, is mandatory to prevent complications.
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