Immensely high Creatine Kinase levels in a case of rhabdomyolysis due to Legionnaires’ disease in a patient on tofacitinib: a case report and literature review

Abstract

A 58-year-old female patient presented with altered mental status, diarrhea, and fever. She was hospitalized for acute kidney injury [AKI] and a patchy right lower lobe infiltrates on chest X-ray. Subsequent testing revealed rhabdomyolysis and a positive urinary Legionella antigen test. Creatinine kinase [CK] level peaked at 512,820 U/L and was managed with aggressive intravenous hydration and appropriate antibiotic treatment. With clinical signs of resolution of pneumonia, the CK level declined rapidly, however renal function returned to baseline only after 2 months requiring hemodialysis in the meantime. The patient was also on tofacitinib which can rarely contribute to rhabdomyolysis. Legionella infection can cause severe rhabdomyolysis and AKI. Timely diagnosis of Legionella-associated rhabdomyolysis, and prompt treatment with aggressive IV hydration and appropriate antibiotics is required to prevent morbidity and mortality.

Reply to the letter to the editor titled “Before blaming Legionella for severe rhabdomyolysis, consider alternative triggers”

“… 2 , 3 But in our patient, hypokalemia (potassium of 3 mmol/L) was mild and was immediately corrected to normal reference range and so is unlikely to trigger rhabdomyolysis with CK elevation up to 512,820 IU/L. 1 Hyponatremia can also trigger rhabdomyolysis and Legionella pneumonia is known to cause hyponatremia, a retrospective analysis by Fiumefreddo et al showed that about 46% of patients with legionella pneumonia had hyponatremia with sodium levels less than 131 mmol/L 4 In an observational study by Lim et al looking into association between hyponatremia and rhabdomyolysis showed that prevalence of rhabdomyolysis in mild (sodium between 130 and 134 mmol/L), moderate (sodium between 125 and 129mmol/L) and severe (sodium <125 mmol/L) hyponatremia was 9.4%, 2.5%, and 2.1%, respectively. The peak CK elevation seen in this study in moderate hyponatremia was 40,000 IU/ml.…”

“…Even though losartan could be a possible culprit, dose at which it can cause rhabdomyolysis is not certain. 6 Our patient was on low dose losartan 25 mg once daily monotherapy for hypertension and no other diuretics 1 making it less likely the cause for rhabdomyolysis. Even though Tofacitinib was immediately discontinued after admission, it could have played a role to potentiate the degree rhabdomyolysis seen in our patient as mentioned in the title of the case report.…”

“…We appreciate the interest shown by the reviewers and their comments regarding our article titled “Immensely High Creatine Kinase Levels in a Case of Rhabdomyolysis Due to Legionnaires’ Disease in a Patient on Tofacitinib: A Case Report and Literature Review”. 1 …”

“…Even though Tofacitinib was immediately discontinued after admission, it could have played a role to potentiate the degree rhabdomyolysis seen in our patient as mentioned in the title of the case report. 1 On initial presentation patient was lethargic and slow to respond to commands possibly due to underlying sepsis, hypoxemia and electrolyte abnormalities. However, as mentioned in case report she did not have any focal neurological deficits, neck rigidity or signs of meningeal irritation.…”

“…Even though there was initial increase in creatinine kinase on azithromycin, she completed 14 days course of azithromycin with subsequent decline and eventual resolution of rhabdomyolysis. 1 This makes azithromycin induced CK elevation unlikely. This patient was admitted and treated in 2019 before the first case of COVID 19 was identified in USA.…”