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Abstract
Introduction5-oxoproline (pyroglutamic acid), an organic acid intermediate of the gamma-glutamyl cycle, is a rare cause of high anion gap metabolic acidosis. Acetaminophen and several other drugs have been implicated in the development of transient 5-oxoprolinemia in adults. We believe that reporting all cases of 5-oxoprolinemia will contribute to a better understanding of this disease. Here, we report the case of a patient who developed transient 5-oxoprolinemia following therapeutic acetaminophen use.Case presentationA 75-year-old Caucasian woman was initially admitted for treatment of an infected hip prosthesis and subsequently developed transient high anion gap metabolic acidosis. Our patient received 40g of acetaminophen over a 10-day period. After the more common causes of high anion gap metabolic acidosis were excluded, a urinary organic acid screen revealed a markedly increased level of 5-oxoproline. The acidosis resolved completely after discontinuation of the acetaminophen.Conclusion5-oxoproline acidosis is an uncommon cause of high anion gap metabolic acidosis; however, it is likely that it is under-diagnosed as awareness of the condition remains low and testing can only be performed at specialized laboratories. The diagnosis should be suspected in cases of anion gap metabolic acidosis, particularly in patients with recent acetaminophen use in combination with sepsis, malnutrition, liver disease, pregnancy or renal failure. This case has particular interest in medicine, especially for the specialties of nephrology and orthopedics. We hope that it will add more information to the literature about this rare condition.
Highlights
Transient 5-oxoprolinemia is a rare clinical condition caused by glutathione deficiency
It usually presents as severe anion gap metabolic acidosis
A less frequent cause is the temporary accumulation of the organic acid 5-oxoproline [1,2]. 5-oxoproline is primarily metabolized to glutamate by the enzyme 5-oxoprolinase and is eliminated by the renal system
Summary
Transient 5-oxoprolinemia is a rare clinical condition caused by glutathione deficiency It usually presents as severe anion gap metabolic acidosis. She started vancomycin (1g intravenously twice daily), along with acetaminophen (1g every six hours) for pain control Her other blood work, including liver and kidney function tests, matched her baseline results, with normal liver function test results and a serum creatinine of 152μmol/L. Arterial blood gas showed metabolic acidosis with respiratory compensation (pH, 7.18; partial pressure of carbon dioxide, 1.7kPa; partial pressure of oxygen, 16.7kPa; bicarbonate, 8.3mmol/L; base excess, −22.7mmol/L; SaO2, 98.6% in room air). The osmolar gap was mildly elevated at 21 mOsm/kg Because her serum toxicology screen was negative, our patient’s urine was sent for organic acid detection by gas chromatography–mass spectrometry, which demonstrated a markedly increased excretion of 5-oxoproline at the peak of her acidosis, 10 days into her admission. The diagnosis of 5-oxoprolinemia was confirmed on receipt of organic acid test results two weeks later
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