Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence

Ainsworth, James; Pillai, Suresh Gopala

doi:10.1155/crcc/5740399

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Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence

James Ainsworth

, Suresh Gopala Pillai

Case Reports in Critical Care, Volume: 2025, Issue: 1

Swansea University Author: Suresh Gopala Pillai

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DOI (Published version): 10.1155/crcc/5740399

Abstract

Introduction: Methamoglobinemia is a condition characterized by impaired oxygen delivery to the tissues due to the formation of methemoglobin (Met-Hb). Diagnosis is established when Met-Hb levels exceed 5%. Levels over 30% are associated with severe symptoms and can be life-threatening. Pulse oximet...

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Published in:	Case Reports in Critical Care
ISSN:	2090-6420 2090-6439
Published:	Wiley 2025
Online Access:	Check full text
URI:	https://cronfa.swan.ac.uk/Record/cronfa70777

first_indexed	2025-10-28T16:01:36Z
last_indexed	2025-11-14T12:47:32Z
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spelling	2025-11-13T12:55:48.5010532 v2 70777 2025-10-28 Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence f567f8d5db61d62ef08e811676fd8430 0000-0002-9753-6949 Suresh Gopala Pillai Suresh Gopala Pillai true false 2025-10-28 MEDS Introduction: Methamoglobinemia is a condition characterized by impaired oxygen delivery to the tissues due to the formation of methemoglobin (Met-Hb). Diagnosis is established when Met-Hb levels exceed 5%. Levels over 30% are associated with severe symptoms and can be life-threatening. Pulse oximetry may provide misleading readings, often reporting oxygen saturation around 85%, irrespective of actual oxygenation levels. Management involves discontinuing offending agents, providing supportive care, and use of specific treatment agents. Most commonly, cases report a quick and effective recovery following treatment.Case Presentation: We describe a case of a patient requiring four separate admissions over a 9-month period, all for methemoglobinemia, where the cause remained uncertain. A 25-year-old female was admitted to the intensive care unit (ICU) with sudden onset shortness of breath and cyanosis, and Met-Hb level 55.5%, suspected due to dapsone. She was treated with 1 mg/kg of methylene blue intravenously. Met-Hb levels remained persistently above 20% for a number of days, and reaccumulated following initial decline. Repeated doses of methylene blue were required, and activated charcoal was given. She was readmitted 3 days later, with reaccumulation of Met-Hb. This was again thought secondary to dapsone, given the long half-life of dapsone and potential for enterohepatic recirculation. She had a third admission with methemoglobinemia almost 2 months after the first admission, and again 8 months later. Suggestion was that azathioprine could be the causative agent; however, this has not been reported previously.Discussion: We report a case of a 25-year-old female with repeated admissions with methemoglobinemia. This case highlights the potential for refractory methemoglobinemia requiring repeated treatment, and the importance of longer periods of close observation. It is important to consider the diagnosis in patients without a clear trigger for methemoglobinemia, as an identifiable cause may not always be clear. Journal Article Case Reports in Critical Care 2025 1 Wiley 2090-6420 2090-6439 cyanosis; intensive care; methemoglobin; methemoglobinemia; methylene blue 17 6 2025 2025-06-17 10.1155/crcc/5740399 Case Report COLLEGE NANME Medical School COLLEGE CODE MEDS Swansea University Other None 2025-11-13T12:55:48.5010532 2025-10-28T09:38:41.1429002 Faculty of Medicine, Health and Life Sciences Swansea University Medical School - Biomedical Science James Ainsworth 0000-0001-5571-1983 1 Suresh Gopala Pillai 0000-0002-9753-6949 2 70777__35619__27b76c37e8a24e65a3369f74cd9b3357.pdf 70777.VoR.pdf 2025-11-13T12:52:49.2993639 Output 696896 application/pdf Version of Record true © 2025 James Ainsworth and Suresh Pillai. This is an open access article under the terms of the Creative Commons Attribution License. true eng http://creativecommons.org/licenses/by/4.0/
title	Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence
spellingShingle	Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence Suresh Gopala Pillai
title_short	Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence
title_full	Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence
title_fullStr	Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence
title_full_unstemmed	Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence
title_sort	Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence
author_id_str_mv	f567f8d5db61d62ef08e811676fd8430
author_id_fullname_str_mv	f567f8d5db61d62ef08e811676fd8430_***_Suresh Gopala Pillai
author	Suresh Gopala Pillai
author2	James Ainsworth Suresh Gopala Pillai
format	Journal article
container_title	Case Reports in Critical Care
container_volume	2025
container_issue	1
publishDate	2025
institution	Swansea University
issn	2090-6420 2090-6439
doi_str_mv	10.1155/crcc/5740399
publisher	Wiley
college_str	Faculty of Medicine, Health and Life Sciences
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hierarchy_top_title	Faculty of Medicine, Health and Life Sciences
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hierarchy_parent_title	Faculty of Medicine, Health and Life Sciences
department_str	Swansea University Medical School - Biomedical Science{{{_:::_}}}Faculty of Medicine, Health and Life Sciences{{{_:::_}}}Swansea University Medical School - Biomedical Science
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description	Introduction: Methamoglobinemia is a condition characterized by impaired oxygen delivery to the tissues due to the formation of methemoglobin (Met-Hb). Diagnosis is established when Met-Hb levels exceed 5%. Levels over 30% are associated with severe symptoms and can be life-threatening. Pulse oximetry may provide misleading readings, often reporting oxygen saturation around 85%, irrespective of actual oxygenation levels. Management involves discontinuing offending agents, providing supportive care, and use of specific treatment agents. Most commonly, cases report a quick and effective recovery following treatment.Case Presentation: We describe a case of a patient requiring four separate admissions over a 9-month period, all for methemoglobinemia, where the cause remained uncertain. A 25-year-old female was admitted to the intensive care unit (ICU) with sudden onset shortness of breath and cyanosis, and Met-Hb level 55.5%, suspected due to dapsone. She was treated with 1 mg/kg of methylene blue intravenously. Met-Hb levels remained persistently above 20% for a number of days, and reaccumulated following initial decline. Repeated doses of methylene blue were required, and activated charcoal was given. She was readmitted 3 days later, with reaccumulation of Met-Hb. This was again thought secondary to dapsone, given the long half-life of dapsone and potential for enterohepatic recirculation. She had a third admission with methemoglobinemia almost 2 months after the first admission, and again 8 months later. Suggestion was that azathioprine could be the causative agent; however, this has not been reported previously.Discussion: We report a case of a 25-year-old female with repeated admissions with methemoglobinemia. This case highlights the potential for refractory methemoglobinemia requiring repeated treatment, and the importance of longer periods of close observation. It is important to consider the diagnosis in patients without a clear trigger for methemoglobinemia, as an identifiable cause may not always be clear.
published_date	2025-06-17T05:25:48Z
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Severe Treatment‐Resistant Methemoglobinemia of Unknown Etiology With Recurrence

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