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Introduction: Severe anion gap (AG) acidosis associated with intravenous sodium thiosulfate (STS) administration has not been previously described in nondialysis chronic kidney disease (CKD) patients. Case Report: We present a CKD patient with a baseline creatinine 1.8 mg/dL (eGFR 28 ml/min/1.73 m) who developed sustained and life-threatening AG acidosis associated with intravenous STS treatment for calciphylaxis. Discussion: Although marketed as a safe drug, STS can cause life-threatening acidosis as illustrated in this case. STS-induced AG acidosis should be considered in the differential diagnosis of severe acidosis in patients receiving STS. Dosage adjustment and close follow-up of patients' acid-base status after STS initiation is necessary. [ABSTRACT FROM AUTHOR]