The first pediatric case of hemophagocytic lymphohistiocytosis secondary to <scp>Crimean‐Congo</scp> haemorrhagic fever successfully treated with therapeutic plasma exchange accompanying ribavirin and intravenous immunoglobulin

Aydın, Zeynep Gökçe Gayretli; Yeşilbaş, Osman; Reis, Gökçe Pınar; Güven, Burcu

doi:10.1002/jca.21915

Cited by 7 publications

(4 citation statements)

References 13 publications

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“…One study reported HLH in 50% of adult CCHF patients ( 26 ); however, a case report describing an adult CCHF patient with HLH reported HLH as a rare condition ( 27 ). HLH is rarely reported among pediatric patients ( 28 ).…”

Section: Discussionmentioning

confidence: 99%

Changing Disease Course of Crimean-Congo Hemorrhagic Fever in Children, Turkey

Oygar¹,

Gürlevik²,

Sag³

et al. 2023

Emerg. Infect. Dis.

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Crimean-Congo hemorrhagic fever (CCHF), endemic in certain regions of the world, is listed as a priority disease with pandemic potential. Since CCHF was first identified in Turkey, children have been known to experience milder disease than adults. However, during the COVID-19 pandemic, we observed an unusually severe disease course, including hemophagocytic lymphohistiocytosis (HLH). We examined cytokine/chemokine profiles of 9/12 case-patients compared with healthy controls at 3 time intervals. Interferon pathway–related cytokines/chemokines, including interleukin (IL) 18, macrophage inflammatory protein 3α, and IL-33, were elevated, but tumor necrosis factor-α, IL-6, CXCL8 (formerly IL-8), and cytokines acting through C-C chemokine receptor 2 and CCR5 were lower among case-patients than controls. Interferon pathway activation and cytokines/chemokines acting through CCR2 and CCR5 improved health results among children with severe CCHF. Children can experience severe CCHF, including HLH, and HLH secondary to CCHF can be successfully treated with intravenous immunoglobulin and steroid therapy.

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Section: Discussionmentioning

confidence: 99%

Changing Disease Course of Crimean-Congo Hemorrhagic Fever in Children, Turkey

Oygar¹,

Gürlevik²,

Sag³

et al. 2023

Emerg. Infect. Dis.

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“…Pediatric cases of hemophagocytic lymphohistiocytosis (HLH) secondary to CCHF have been described ( 42 , 43 ). Secondary HLH, although rare, can be associated with malignancies, severe infections, medications, and autoimmune disorders and has been thought to be secondary to a hyperinflammatory syndrome ( 44 ).…”

Section: Clinical Featuresmentioning

confidence: 99%

Crimean-Congo Hemorrhagic Fever Virus for Clinicians—Epidemiology, Clinical Manifestations, and Prevention

Frank,

Weaver,

Raabe

2024

Emerg. Infect. Dis.

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Crimean-Congo hemorrhagic fever (CCHF) is a tickborne infection that can range from asymptomatic to fatal and has been described in >30 countries. Early identification and isolation of patients with suspected or confirmed CCHF and the use of appropriate prevention and control measures are essential for preventing human-to-human transmission. Here, we provide an overview of the epidemiology, clinical features, and prevention and control of CCHF. CCHF poses a continued public health threat given its wide geographic distribution, potential to spread to new regions, propensity for genetic variability, and potential for severe and fatal illness, in addition to the limited medical countermeasures for prophylaxis and treatment. A high index of suspicion, comprehensive travel and epidemiologic history, and clinical evaluation are essential for prompt diagnosis. Infection control measures can be effective in reducing the risk for transmission but require correct and consistent application.

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“…The Crimean Congo hemorrhagic fever-associated HLH has been resolved using antiviral therapy combined with IVIG therapy and plasma exchange ( 91 ).…”

Section: Treatmentmentioning

confidence: 99%

“…Dengue virus plays a significant role in tropical countries (73,(83)(84)(85)(86)(87)(88)(89)(90). Tick-borne diseases include the Crimean-Congo hemorrhagic fever nairovirus (91,92) and the Eastern equine encephalitis virus (93). Sporadic case reports include HLH related to HAV (90,94), HBV (95), HIV (89,96), Adenovirus (72,89,97,98), Parvovirus B19 (72), Measles (88) and Influenza virus (99)(100)(101).…”

Section: Classificationmentioning

confidence: 99%

Secondary hemophagocytic lymphohistiocytosis in children (Review)

et al. 2023

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Hemophagocytic lymphohistiocytosis (HLH) is a rare, life-threatening condition characterized by hyperinflammation in an uncontrolled and ineffective immune response. Despite great improvement in diagnosis and treatment, it still represents a challenge in clinical management, with poor prognosis in the absence of an aggressive therapeutic approach. The present literature review focuses on secondary HLH at pediatric age, which represents a heterogeneous group in terms of etiology and therapeutic approach. It summarizes the most recent evidence on epidemiology, pathophysiology, diagnosis, treatment and prognosis, and provides a detailed description and comparison of the major subtypes of secondary HLH. Finally, it addresses the open questions with a focus on diagnosis and new treatment insights.

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The first pediatric case of hemophagocytic lymphohistiocytosis secondary to Crimean‐Congo haemorrhagic fever successfully treated with therapeutic plasma exchange accompanying ribavirin and intravenous immunoglobulin

Cited by 7 publications

References 13 publications

Changing Disease Course of Crimean-Congo Hemorrhagic Fever in Children, Turkey

Changing Disease Course of Crimean-Congo Hemorrhagic Fever in Children, Turkey

Crimean-Congo Hemorrhagic Fever Virus for Clinicians—Epidemiology, Clinical Manifestations, and Prevention

Secondary hemophagocytic lymphohistiocytosis in children (Review)

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