Rationale: Operative hysteroscopy intravascular absorption syndrome (OHIAS) results from systemic absorption of hypotonic solution during hysteroscopy, which may induce severe hyponatremia within hours. Depending on the serum sodium (Na + ) level, this can be life-threatening and requires prompt and careful remedial treatment.Patient concerns and diagnoses: A 53-year-old woman underwent hysteroscopic myomectomy for submucosal leiomyoma. Approximately 3 hours postoperatively, the serum Na + level decreased to 82 mM/L, accompanied by pulmonary edema and lactic acidosis. The patient was strongly suspicious of OHIAS.Interventions and outcomes: A rapid correction was made using 3% NaCl to prevent brain edema as an initial response. After the serum Na + level reached 120 mM/L, gradual correction was performed considering osmotic demyelination syndrome, and desmopressin was administered to prevent overcorrection caused by excessive water diuresis. Serum Na + level normalized in 4 days and the patient recovered without any specific sequelae.Lessons: The detection of OHIAS may be delayed under general anesthesia, and prior vigilance is important if the operation time is prolonged. In severe hyponatremia with an apparently rapid onset, such as OHIAS, a two-step correction process may be safe and useful: rapid correction followed by more gradual correction.