Hemophagocytic lymphohistiocytosis and histiocytic necrotizing lymphadenitis secondary to hemodialysis catheter-related bloodstream infection caused by Corynebacterium Striatum.
Catheter-related bloodstream infection
Corynebacterium striatum
Cytokine
Hemophagocytic lymphohistiocytosis
Kikuchi disease
Maintenance hemodialysis
Journal
BMC nephrology
ISSN: 1471-2369
Titre abrégé: BMC Nephrol
Pays: England
ID NLM: 100967793
Informations de publication
Date de publication:
06 10 2023
06 10 2023
Historique:
received:
20
05
2023
accepted:
30
09
2023
medline:
9
10
2023
pubmed:
7
10
2023
entrez:
6
10
2023
Statut:
epublish
Résumé
We herein described the coexistence of hemophagocytic lymphohistiocytosis (HLH) and histiocytic necrotizing lymphadenitis, alternatively known as the Kikuchi disease (KD), secondary to hemodialysis catheter-related bloodstream infection (BSI) caused by Corynebacterium striatum. A patient on maintenance hemodialysis had developed persistent fever and Corynebacterium striatum was subsequently identified from the culture of both catheter tip and peripheral blood. During mitigation of the BSI, however, his fever was unabated and ensuing workup further found thrombocytopenia, hyperferritinemia, hypertriglyceridemia, low NK cell activity and a surge in serum CD25 levels. Moreover, biopsy of the bone marrow and lymph node detected histopathological evidence of hemophagocytosis and KD, respectively. Upon these abnormalities, the title-bound diagnosis was considered and the patient was eventually recovered from the treatment of dexamethasone instead of antibiotics. Consistently, aberrations in his serum CD25 levels and NK cell activity had subsided two months after discharge. Arguably, this encounter offered a unique chance to unravel the principal pathogenic cascade in immunobiology that made the three entities one disease continuum. As such, our work may add new understandings of HLH and/or KD secondary to severe infections in general and excessive release of cytokines in particular among patients with kidney diseases. The resultant early diagnosis is crucial to initiate appropriate treatment and improve the survival of patients with these challenging and potentially life-threatening disorders.
Sections du résumé
BACKGROUND
We herein described the coexistence of hemophagocytic lymphohistiocytosis (HLH) and histiocytic necrotizing lymphadenitis, alternatively known as the Kikuchi disease (KD), secondary to hemodialysis catheter-related bloodstream infection (BSI) caused by Corynebacterium striatum.
CASE PRESENTATION
A patient on maintenance hemodialysis had developed persistent fever and Corynebacterium striatum was subsequently identified from the culture of both catheter tip and peripheral blood. During mitigation of the BSI, however, his fever was unabated and ensuing workup further found thrombocytopenia, hyperferritinemia, hypertriglyceridemia, low NK cell activity and a surge in serum CD25 levels. Moreover, biopsy of the bone marrow and lymph node detected histopathological evidence of hemophagocytosis and KD, respectively. Upon these abnormalities, the title-bound diagnosis was considered and the patient was eventually recovered from the treatment of dexamethasone instead of antibiotics. Consistently, aberrations in his serum CD25 levels and NK cell activity had subsided two months after discharge.
CONCLUSIONS
Arguably, this encounter offered a unique chance to unravel the principal pathogenic cascade in immunobiology that made the three entities one disease continuum. As such, our work may add new understandings of HLH and/or KD secondary to severe infections in general and excessive release of cytokines in particular among patients with kidney diseases. The resultant early diagnosis is crucial to initiate appropriate treatment and improve the survival of patients with these challenging and potentially life-threatening disorders.
Identifiants
pubmed: 37803308
doi: 10.1186/s12882-023-03356-2
pii: 10.1186/s12882-023-03356-2
pmc: PMC10559591
doi:
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
294Informations de copyright
© 2023. BioMed Central Ltd., part of Springer Nature.
Références
Ann Hematol. 2017 Aug;96(8):1241-1251
pubmed: 28497365
Clin Exp Nephrol. 2022 Jan;26(1):75-85
pubmed: 34436742
R I Med J (2013). 2020 Oct 1;103(8):46-49
pubmed: 33003679
Lancet Infect Dis. 2007 Dec;7(12):814-22
pubmed: 18045564
Blood. 2017 Jul 6;130(1):39-47
pubmed: 28515095
Kidney Int. 2020 May;97(5):829-838
pubmed: 32247631
Curr Drug Targets. 2021;22(1):52-67
pubmed: 33050860
Infect Immun. 2017 Jun 20;85(7):
pubmed: 28483856
Clin Infect Dis. 1997 Feb;24(2):185-91
pubmed: 9114145
Rheumatol Int. 2021 Jan;41(1):7-18
pubmed: 32588191
Curr Opin Rheumatol. 2021 Sep 1;33(5):419-430
pubmed: 34264880
Leuk Lymphoma. 2007 Dec;48(12):2447-51
pubmed: 18067022
Indian J Nephrol. 2017 Mar-Apr;27(2):133-135
pubmed: 28356667
Am J Blood Res. 2020 Dec 15;10(6):330-338
pubmed: 33489441
Clin Immunol. 2020 Sep;218:108515
pubmed: 32619646
Arch Microbiol. 2021 Jul;203(5):1863-1880
pubmed: 33625540
Curr Opin Hematol. 2008 Jul;15(4):359-67
pubmed: 18536575
Pediatr Blood Cancer. 2007 Feb;48(2):124-31
pubmed: 16937360
Pediatr Radiol. 2003 Jun;33(6):392-401
pubmed: 12768255
Pediatr Radiol. 2008 Jul;38(7):756-61
pubmed: 18496683
Medicine (Baltimore). 2020 Dec 18;99(51):e23500
pubmed: 33371072
Lancet. 2014 Apr 26;383(9927):1503-1516
pubmed: 24290661