• Zenghua Cai, Yu Shao, and Yu Wu.
• Department of Anesthesiology, Bethune International Peace Hospital, Shijiazhuang, China.
• Medicine (Baltimore). 2023 Apr 14; 102 (15): e33561e33561.

RationaleCongenital methemoglobinemia is a rare cyanosis cause that can be manifested in affected individuals. We report a case of congenital methemoglobinemia after middle ear cholesteatoma resection under general anesthesia.Patient ConcernsThe primary concern of the patient is to safely perform cholesteatoma resection of the middle ear to reduce the pain associated with years of surgery and to survive the perioperative period.DiagnosesCongenital methemoglobinemia type 1.InterventionsThe patient underwent general anesthesia and cholesteatoma resection of the middle ear.OutcomesThe patient successfully underwent cholesteatoma resection in the middle ear under general anesthesia and went through the perioperative period smoothly, and successfully returned to society.LessonsFor patients requiring general anesthesia complicated with rare methemoglobinemia, we improve the awareness of crisis and make comprehensive preparation and monitoring, learn the pathophysiological mechanism related to the disease, so as to protect the operation of methemoglobin patients under general anesthesia.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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