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Secondary hemophagocytic lymphohistiocytosis associated with heat stroke: A case report and review of literature

Hemophagocytic lymphohistiocytosis (HLH) is a syndrome with potentially fatal consequences that results from an excessive immune response caused by malfunctioning natural killer cells and cytotoxic T lymphocytes. Secondary HLH, which is the predominant type in adults, is associated with various medi...

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Autores principales: Wi, Wongook, Yoon, Kyoung Won, Kim, Hyo Jin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10219742/
https://www.ncbi.nlm.nih.gov/pubmed/37233425
http://dx.doi.org/10.1097/MD.0000000000033842
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author Wi, Wongook
Yoon, Kyoung Won
Kim, Hyo Jin
author_facet Wi, Wongook
Yoon, Kyoung Won
Kim, Hyo Jin
author_sort Wi, Wongook
collection PubMed
description Hemophagocytic lymphohistiocytosis (HLH) is a syndrome with potentially fatal consequences that results from an excessive immune response caused by malfunctioning natural killer cells and cytotoxic T lymphocytes. Secondary HLH, which is the predominant type in adults, is associated with various medical conditions, including infections, malignancies, and autoimmune diseases. Secondary HLH associated with heat stroke has not been reported. PATIENT CONCERNS: A 74-year-old male was admitted to the emergency department after being unconscious in a 42°C hot public bath. The patient was witnessed to be in the water for more than 4 hours. The patient’s condition was complicated by rhabdomyolysis and septic shock, which were managed with mechanical ventilation, vasoactive agents, and continuous renal replacement therapy. The patient also showed evidence of diffuse cerebral dysfunction. DIAGNOSES: While the patient’s condition initially improved, the patient developed a fever, anemia, thrombocytopenia, and an acute rise in total bilirubin, which, we suspected, was caused by HLH. Further investigations revealed elevated serum ferritin and soluble interleukin-2 receptor levels. INTERVENTIONS: The patient received 2 cycles of serial therapeutic plasma exchange to lower the endotoxin burden. To manage HLH, high-dose glucocorticoid therapy was done. OUTCOMES: Despite the best efforts, the patient did not recover and expired from progressive hepatic failure. LESSONS: We report a novel case of secondary HLH associated with heat stroke. Diagnosing secondary HLH can be difficult since clinical manifestations of the underlying disease and HLH may present simultaneously. Early diagnosis and prompt initiation of treatment is required to improve the prognosis of the disease.
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spelling pubmed-102197422023-05-27 Secondary hemophagocytic lymphohistiocytosis associated with heat stroke: A case report and review of literature Wi, Wongook Yoon, Kyoung Won Kim, Hyo Jin Medicine (Baltimore) 3900 Hemophagocytic lymphohistiocytosis (HLH) is a syndrome with potentially fatal consequences that results from an excessive immune response caused by malfunctioning natural killer cells and cytotoxic T lymphocytes. Secondary HLH, which is the predominant type in adults, is associated with various medical conditions, including infections, malignancies, and autoimmune diseases. Secondary HLH associated with heat stroke has not been reported. PATIENT CONCERNS: A 74-year-old male was admitted to the emergency department after being unconscious in a 42°C hot public bath. The patient was witnessed to be in the water for more than 4 hours. The patient’s condition was complicated by rhabdomyolysis and septic shock, which were managed with mechanical ventilation, vasoactive agents, and continuous renal replacement therapy. The patient also showed evidence of diffuse cerebral dysfunction. DIAGNOSES: While the patient’s condition initially improved, the patient developed a fever, anemia, thrombocytopenia, and an acute rise in total bilirubin, which, we suspected, was caused by HLH. Further investigations revealed elevated serum ferritin and soluble interleukin-2 receptor levels. INTERVENTIONS: The patient received 2 cycles of serial therapeutic plasma exchange to lower the endotoxin burden. To manage HLH, high-dose glucocorticoid therapy was done. OUTCOMES: Despite the best efforts, the patient did not recover and expired from progressive hepatic failure. LESSONS: We report a novel case of secondary HLH associated with heat stroke. Diagnosing secondary HLH can be difficult since clinical manifestations of the underlying disease and HLH may present simultaneously. Early diagnosis and prompt initiation of treatment is required to improve the prognosis of the disease. Lippincott Williams & Wilkins 2023-05-26 /pmc/articles/PMC10219742/ /pubmed/37233425 http://dx.doi.org/10.1097/MD.0000000000033842 Text en Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY) (https://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle 3900
Wi, Wongook
Yoon, Kyoung Won
Kim, Hyo Jin
Secondary hemophagocytic lymphohistiocytosis associated with heat stroke: A case report and review of literature
title Secondary hemophagocytic lymphohistiocytosis associated with heat stroke: A case report and review of literature
title_full Secondary hemophagocytic lymphohistiocytosis associated with heat stroke: A case report and review of literature
title_fullStr Secondary hemophagocytic lymphohistiocytosis associated with heat stroke: A case report and review of literature
title_full_unstemmed Secondary hemophagocytic lymphohistiocytosis associated with heat stroke: A case report and review of literature
title_short Secondary hemophagocytic lymphohistiocytosis associated with heat stroke: A case report and review of literature
title_sort secondary hemophagocytic lymphohistiocytosis associated with heat stroke: a case report and review of literature
topic 3900
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10219742/
https://www.ncbi.nlm.nih.gov/pubmed/37233425
http://dx.doi.org/10.1097/MD.0000000000033842
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