Can therapeutic plasma exchange be life-saving in life-threatening manganese intoxication?

Transfus Apher Sci. 2022 Aug;61(4):103417. doi: 10.1016/j.transci.2022.103417. Epub 2022 Mar 3.

Abstract

We present a pediatric patient presenting with life-threatening severe neurological signs, chronic liver disease, and manganese intoxication who fully recovered from neurological signs and symptoms following chelation therapy and therapeutic plasma exchange (TPE). A 13-year-old female patient was admitted with abdominal pain. Loss of consciousness and decorticate posture (GCS;M:1,V:1,M:3) developed at the 5th hour of admission. She admitted to the intensive care unit intubated. No infectious etiology that could explain acute encephalopathy was detected. Abdominal ultrasound showed granular, heterogeneous liver parenchyma suggesting chronic hepatic disease, and TPE was administered for two days since Wilson's disease and autoimmune encephalitis could not be ruled out. Cranial MRI findings were consistent with a diagnosis of manganese intoxication. On Day 3 after admission, chelation therapy and TPE were administered based on a diagnosis of manganese intoxication. Blood manganese levels at admission, day 2, and day 5 were 46, 22, and 17.5 μg/dL (NR:4.7-18.3). Control MRI results showed reduced intracranial manganese deposition, and the patient regained full consciousness. TPE as an adjunct to chelation therapy may represent an effective therapeutic option in manganese intoxication.

Keywords: Chelation therapy; Manganese intoxication; Pediatric patient; Therapeutic plasma exchange.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Child
  • Female
  • Hepatolenticular Degeneration* / therapy
  • Humans
  • Manganese
  • Plasma Exchange* / methods
  • Plasmapheresis

Substances

  • Manganese