Abstract
BackgroundOsmotic demyelination syndrome (ODS) primarily occurs after rapid correction of severe hyponatremia. There are no proven effective therapies for ODS, but we describe the first case showing the successful treatment of central pontine myelinolysis (CPM) by plasma exchange, which occurred after rapid development of hypernatremia from intravenous sodium bicarbonate therapy.Case presentationA 40-year-old woman presented with general weakness, hypokalemia, and metabolic acidosis. The patient was treated with oral and intravenous potassium chloride, along with intravenous sodium bicarbonate. Although her bicarbonate deficit was 365 mEq, we treated her with an overdose of intravenous sodium bicarbonate, 480 mEq for 24 hours, due to the severity of her acidemia and her altered mental status. The next day, she developed hypernatremia with serum sodium levels rising from 142.8 mEq/L to 172.8 mEq/L. Six days after developing hypernatremia, she exhibited tetraparesis, drooling, difficulty swallowing, and dysarthria, and a brain MRI revealed high signal intensity in the central pons with sparing of the peripheral portion, suggesting CPM. We diagnosed her with CPM associated with the rapid development of hypernatremia after intravenous sodium bicarbonate therapy and treated her with plasma exchange. After two consecutive plasma exchange sessions, her neurologic symptoms were markedly improved except for mild diplopia. After the plasma exchange sessions, we examined the patient to determine the reason for her symptoms upon presentation to the hospital. She had normal anion gap metabolic acidosis, low blood bicarbonate levels, a urine pH of 6.5, and a calyceal stone in her left kidney. We performed a sodium bicarbonate loading test and diagnosed distal renal tubular acidosis (RTA). We also found that she had Sjögren’s syndrome after a positive screen for anti-Lo, anti-Ra, and after the results of Schirmer’s test and a lower lip biopsy. She was discharged and treated as an outpatient with oral sodium bicarbonate and potassium chloride.ConclusionThis case indicates that serum sodium concentrations should be carefully monitored in patients with distal RTA receiving intravenous sodium bicarbonate therapy. We should keep in mind that acute hypernatremia and CPM can be associated with intravenous sodium bicarbonate therapy, and that CPM due to acute hypernatremia may be effectively treated with plasma exchange.
Highlights
Osmotic demyelination syndrome (ODS) primarily occurs after rapid correction of severe hyponatremia
This case indicates that serum sodium concentrations should be carefully monitored in patients with distal renal tubular acidosis (RTA) receiving intravenous sodium bicarbonate therapy
We should keep in mind that acute hypernatremia and central pontine myelinolysis (CPM) can be associated with intravenous sodium bicarbonate therapy, and that CPM due to acute hypernatremia may be effectively treated with plasma exchange
Summary
Our case indicates that serum sodium concentrations should be carefully monitored in patients with distal RTA who are receiving intravenous sodium bicarbonate therapy. We should keep in mind that acute hypernatremia and CPM can be associated with intravenous sodium bicarbonate therapy, and CPM due to acute hypernatremia may be effectively treated with plasma exchange. Consent Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. Authors’ contributions KYC, IHL, GJK, KWC, HSP and HWK treated the patient and provided data about the history and laboratory results in this report. The manuscript was prepared by KYC and HWK. All authors participated in discussions about the manuscript and approved the final version. Acknowlegements The authors would like to thank Young-Soo Kim, MD (Uijeongbu St. Mary’s Hospital, The Catholic University of Korea) for helpful comments on the manuscript.
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