Neonatal hemophagocytic lymphohistiocytosis associated with a vertical transmission of coxsackievirus B1

Fukazawa, Mitsuharu; Hoshina, Takayuki; Nanishi, Etsuro; Nishio, Hisanori; Doi, Takehiko; Ohga, Shouichi; Hara, Toshiro

doi:10.1007/s10156-013-0629-2

Cited by 13 publications

(7 citation statements)

References 9 publications

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“…Neonatal disseminated infections with HSV-1 or HSV-2 led to SIRS represented by high IL-6 and TNF-α levels, and occasionally to the rapid demise of multiple organ dysfunction syndrome or HLH. 9 10 11 12 Methylprednisolone therapy might be effective for the control of neonatal disseminated HSV infection with HLH. 10 11 In this reported case, 10 sequential changes in the declining pattern of IL-6, IL-10, and IFN-γ levels were consistent with the results of our observation, although HSV DNA copy number was not monitored.…”

Section: Discussionmentioning

confidence: 99%

“…On the contrary, appreciable levels of IL-10 and IFN-γ in the neonates with virus-associated HLH might be associated with a good prognosis. 9 10 Nagamori et al 11 have recently reported a successfully treated case of neonatal disseminate HSV infection with early anti-inflammatory therapy using prednisolone. On the basis of the kinetics of high-mobility group box protein1 (HMGB-1) and cytochrome c , they concluded that anti-inflammatory intervention may only be effective if it is undertaken during the early phase of disseminated neonatal HSV infections.…”

Section: Discussionmentioning

confidence: 99%

“… 4 Serum levels of IL-10 and IFN-γ were elevated in the present patient, differently from non-HSV–HLH in the newborns. 9 10 The distinct cytokine profiles may explain the clinical expressions and severity of neonatal HLH, according to the causative agents and genetic predispositions of HLH.…”

Section: Discussionmentioning

confidence: 99%

“…Serum cytokine levels during the acute phase of HSV–NLF (6–12 days after birth) were retrospectively measured by flow cytometry using a cytometric bead array kit (BD Pharmingen, San Diego, CA) according to the manufacturer's instructions. 9 Seven days after birth, serum levels of interferon (IFN)-γ, interleukin (IL)-6, and IL-10 were remarkably elevated ( Fig. 1B ).…”

Section: Case Reportmentioning

confidence: 99%

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Successful Treatment of Corticosteroid with Antiviral Therapy for a Neonatal Liver Failure with Disseminated Herpes Simplex Virus Infection

Maeba

Hasegawa

Shimomura

et al. 2015

AJP Rep

Self Cite

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Background Herpes simplex virus (HSV) infection carries one of the poorest outcomes of neonatal liver failure (NLF). Neonates with disseminated HSV infection can develop hemophagocytic lymphohistiocytosis (HLH), and occasionally need orthotopic liver transplantation. Early interventions may be critical for the cure of NLF. Case Report We describe herewith a 6-day-old neonate with fulminant hepatic failure due to disseminated HSV-1 infection, who successfully responded to high-dose corticosteroid therapy 72 hours after the onset of disease. Preceding acyclovir, gamma globulin, and exchange blood transfusion therapies failed to control the disease. Methylprednisolone pulse therapy led to a drastic improvement of liver function and cytokine storms, and prevented the disease progression to HLH. Sustained levels of plasma and cerebrospinal fluid HSV DNA declined after prolonged acyclovir therapy. Bilateral lesions of the periventricular white matter areas, assessed by magnetic resonance imaging, disappeared at 3 months of age. The infant showed normal growth and development at 4 years of age. Conclusion Early anti-hypercytokinemia therapy using corticosteroid, and prolonged antiviral therapy might only provide the transplantation-free cure of NLF with HSV dissemination.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Case Reportmentioning

confidence: 99%

See 2 more Smart Citations

Successful Treatment of Corticosteroid with Antiviral Therapy for a Neonatal Liver Failure with Disseminated Herpes Simplex Virus Infection

Maeba

Hasegawa

Shimomura

et al. 2015

AJP Rep

Self Cite

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“…Sometimes, this can result in secondary infections in organs, such as the heart or central nervous system causing for example polio, meningoencephalitis [4][5][6][7], sepsis [8], pancreatitis [9] or myocarditis [10]. Children younger than three years are the most susceptible to severe EV diseases and recent CVB1 epidemics have caused lifethreatening infections in neonates [10][11][12][13][14][15].…”

Section: Introductionmentioning

confidence: 99%

Optimized production and purification of Coxsackievirus B1 vaccine and its preclinical evaluation in a mouse model

Hankaniemi

Laitinen

Stone

et al. 2017

Vaccine

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Coxsackie B viruses are among the most common enteroviruses, causing a wide range of diseases. Recent studies have also suggested that they may contribute to the development of type 1 diabetes. Vaccination would provide an effective way to prevent CVB infections, and the objective of this study was to develop an efficient vaccine production protocol for the generation of novel CVB vaccines. Various steps in the production of a formalin-inactivated Coxsackievirus B1 (CVB1) vaccine were optimized including the Multiplicity Of Infection (MOI) used for virus amplification, virus cultivation time, type of cell growth medium, virus purification method and formulation of the purified virus. Safety and immunogenicity of the formalin inactivated CVB1 vaccine was characterized in a mouse model. Two of the developed methods were found to be optimal for virus purification: the first employed PEG-precipitation followed by gelatin-chromatography and sucrose cushion pelleting (three-step protocol), yielding 19-fold increase in virus concentration (0.06µg/cm) as compared to gold standard method. The second method utilized tandem sucrose pelleting without a PEG precipitation step, yielding 83-fold increase in virus concentration (0.24µg/cm), but it was more labor-intensive and cannot be efficiently scaled up. Both protocols provide radically higher virus yields compared with traditional virus purification protocols involving PEG-precipitation and sucrose gradient ultracentrifugation. Formalin inactivation of CVB1 produced a vaccine that induced a strong, virus-neutralizing antibody response in vaccinated mice, which protected against challenge with CVB1 virus. Altogether, these results provide valuable information for the development of new enterovirus vaccines.

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Disorders of platelets and coagulation, thrombosis and blood transfusion

2022

Neonatal Haematology

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Neonatal hemophagocytic lymphohistiocytosis associated with a vertical transmission of coxsackievirus B1

Cited by 13 publications

References 9 publications

Successful Treatment of Corticosteroid with Antiviral Therapy for a Neonatal Liver Failure with Disseminated Herpes Simplex Virus Infection

Successful Treatment of Corticosteroid with Antiviral Therapy for a Neonatal Liver Failure with Disseminated Herpes Simplex Virus Infection

Optimized production and purification of Coxsackievirus B1 vaccine and its preclinical evaluation in a mouse model

Disorders of platelets and coagulation, thrombosis and blood transfusion

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