A case report of Paracetamol related pyroglutamic acidosis: mind the gap in a malnourished patient.


Journal

BMC nephrology
ISSN: 1471-2369
Titre abrégé: BMC Nephrol
Pays: England
ID NLM: 100967793

Informations de publication

Date de publication:
13 Aug 2024
Historique:
received: 23 08 2023
accepted: 18 07 2024
medline: 14 8 2024
pubmed: 14 8 2024
entrez: 13 8 2024
Statut: epublish

Résumé

Pyroglutamic acidosis is a rare cause of high anion gap metabolic acidosis. Most cases of paracetamol related pyroglutamic acidosis are described in malnourished women and patients with kidney/liver failure, alcohol use or severe sepsis. In this report, we describe how pyroglutamic acidosis could be related to the use of chronic therapeutic paracetamol with only malnutrition as an associated risk factor. We report a case of a 67-year-old male patient developing a pyroglutamic acidosis. The patient was initially admitted to hospital for infectious osteoarthritis and developed a metabolic acidosis during his hospital stay. Analgesics included daily therapeutic doses of paracetamol. What makes our case unusual is that our malnourished male patient did not have renal or hepatic failure. The diagnosis of paracetamol related pyroglutamic acidosis was made after ruling out the main causes of metabolic acidosis. It was further confirmed by urine organic acids measurement showing a markedly elevated level of pyroglutamic aciduria. Paracetamol was discontinued allowing a prompt correction of the anion gap. This case is a representative of pyroglutamic acidosis related to chronic therapeutic paracetamol with only malnutrition as an associated risk factor. Physicians should be aware of such unusual cause of metabolic acidosis, which may be more common than expected in hospitalized patients. A high clinical suspicion is needed when urine organic acids analysis is not available.

Sections du résumé

BACKGROUND BACKGROUND
Pyroglutamic acidosis is a rare cause of high anion gap metabolic acidosis. Most cases of paracetamol related pyroglutamic acidosis are described in malnourished women and patients with kidney/liver failure, alcohol use or severe sepsis. In this report, we describe how pyroglutamic acidosis could be related to the use of chronic therapeutic paracetamol with only malnutrition as an associated risk factor.
CASE PRESENTATION METHODS
We report a case of a 67-year-old male patient developing a pyroglutamic acidosis. The patient was initially admitted to hospital for infectious osteoarthritis and developed a metabolic acidosis during his hospital stay. Analgesics included daily therapeutic doses of paracetamol. What makes our case unusual is that our malnourished male patient did not have renal or hepatic failure. The diagnosis of paracetamol related pyroglutamic acidosis was made after ruling out the main causes of metabolic acidosis. It was further confirmed by urine organic acids measurement showing a markedly elevated level of pyroglutamic aciduria. Paracetamol was discontinued allowing a prompt correction of the anion gap.
CONCLUSION CONCLUSIONS
This case is a representative of pyroglutamic acidosis related to chronic therapeutic paracetamol with only malnutrition as an associated risk factor. Physicians should be aware of such unusual cause of metabolic acidosis, which may be more common than expected in hospitalized patients. A high clinical suspicion is needed when urine organic acids analysis is not available.

Identifiants

pubmed: 39138387
doi: 10.1186/s12882-024-03678-9
pii: 10.1186/s12882-024-03678-9
doi:

Substances chimiques

Acetaminophen 362O9ITL9D
Analgesics, Non-Narcotic 0
Pyrrolidonecarboxylic Acid SZB83O1W42

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

260

Informations de copyright

© 2024. The Author(s).

Références

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doi: 10.7861/clinmedicine.16-6-524
Brooker G, Jeffery J, Nataraj T, Sair1, Ayling M. High anion gap metabolic acidosis secondary to pyroglutamic aciduria (5-oxoprolinuria): associations with prescription drugs and malnutrition. Ann Clin Biochem. 2007;44:406–9. https://doi.org/10.1258/000456307780945769 .
doi: 10.1258/000456307780945769 pubmed: 17594793
Mehta AN, Emmet JB, Emmett M. GOLD MARK: an anion gap mnemonic for the 21st century. Lancet. 2008;372:892. https://doi.org/10.1016/S0140-6736(08)61398-7 .
doi: 10.1016/S0140-6736(08)61398-7 pubmed: 18790311
Humphreys BD, Forman JP, Zandi-nejad K, Bazari H, Seifter J, Magee CC. 5-Oxoprolinuria (Pyroglutamic Aciduria) Acquired in Hospital. Am J Kidney Dis. 2005;46(1):143–6. https://doi.org/10.1053/j.ajkd.2005.04.010 .
doi: 10.1053/j.ajkd.2005.04.010 pubmed: 15983968
Emmett M. Mini-review Acetaminophen Toxicity and 5-Oxoproline (pyroglutamic acid): a tale of two cycles, one an ATP-Depleting futile cycle and the other a useful cycle. Clin J Am Soc Nephrol. 2014;9:191–201. https://doi.org/10.2215/CJN.07730713 .
doi: 10.2215/CJN.07730713 pubmed: 24235282
Green T, Bijlsma JJ, Sweet D. Profound metabolic acidosis from pyroglutamic acidemia. CJEM. 2010;12(5):449–52. https://doi.org/10.1017/S148180350001263X .
doi: 10.1017/S148180350001263X pubmed: 20925168
Reed Poysen C. Pyroglutamic acidosis: a rare cause of metabolic acidosis in a malnourished patient. Anesth Cases. 2013;0166. https://doi.org/10.21466/ac.PAAUCOM.2013 .

Auteurs

Rita Eid (R)

Department of Nephrology, Dialysis, and Transplantation, Bicêtre University Hospital, APHP, Paris Saclay University, 78 rue du Général Leclerc, Le Kremlin-Bicêtre, 94270, France.

Emmanuel Zamparini (E)

Department of Infectious Diseases, Bicêtre University Hospital, APHP, Le Kremlin-Bicêtre, France.

Younes Ouchrif (Y)

Department of Orthopedic Surgery, Bicêtre University Hospital, APHP, Le Kremlin-Bicêtre, France.

Renaud Snanoudj (R)

Department of Nephrology, Dialysis, and Transplantation, Bicêtre University Hospital, APHP, Paris Saclay University, 78 rue du Général Leclerc, Le Kremlin-Bicêtre, 94270, France.
Paris-Saclay University, Le Kremlin-Bicêtre, France.

Chris Ottolenghi (C)

Department of Metabolic Biochemistry, Necker University Hospital for Sick Children, APHP, Paris, France.

Mohamad Zaidan (M)

Department of Nephrology, Dialysis, and Transplantation, Bicêtre University Hospital, APHP, Paris Saclay University, 78 rue du Général Leclerc, Le Kremlin-Bicêtre, 94270, France. mohamad.zaidan@aphp.fr.
Paris-Saclay University, Le Kremlin-Bicêtre, France. mohamad.zaidan@aphp.fr.

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