We report a unique case of prolonged acute rhabdomyolysis in a 2-year-old male with known TANGO-2 mutation with delayed peak in creatinine kinase levels secondary to human metapneumovirus infection. Creatinine kinase peaked at 424,760 U/l on day 9 of hospitalization. Resolution of rhabdomyolysis was achieved using aggressive management with intravenous fluids with optimal urine output and no kidney injury. TANGO-2 patients may develop severe and prolonged rhabdomyolysis with a delayed peak suggesting the need for prolonged inpatient hospitalization to prevent life-threatening complications.
Background: The interest in the anti-seizure efficacy of cannabinoids has significantly increased over the past decade. Trials have shown a remarkable reduction in the frequency of convulsive seizures in Dravet and Lennox-Gastaut syndromes patient population. Studies have shown that CBD reacts with clobazam leading to a significant increase in the levels of N- desmethylclobazam, the active metabolite of clobazam. We present a case of a patient with Dravet Syndrome who presented to the ED with altered mental status secondary to clobazam toxicity related to CBD and clobazam drug-drug interaction. Methods and Findings: We present a case of a patient with Dravet Syndrome who presented to the ED with altered mental status. Patient was hospitalized and CBD was held off. Clobazam level and N-desmethylclobazam levels were obtained and came back elevated at 512 and 15020 respectively. Studies have reported clobazam toxicity secondary to CBD and clobazam drug- drug interaction since both drugs are metabolized by cytochrome P450 enzymes and glucuronyl transferases. Conclusion: Studies have shown the drug interactions of CBD with co-anti-epileptics, including a remarkable increase in the N-desmethylclobazam levels, the active metabolite of clobazam,which confirms the significant importance of monitoring blood levels for clobazam and norclobazam in patients using both CBD and clobazam, in addition for the need to reduce the Clobazam doses for this group of patients. Additional well-controlled studies are essential to establish the recommended doses and the management of the doses of other concomitant anti- epileptics medications with CBD use.
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