Fatal missed case of hemophagocytic lymphohistiocytosis co-infected with parvovirus B19 and Epstein-Barr virus in an infant: Test hyperferritinaemia early

Kishore, Janak; Kishore, Divya

doi:10.4103/0255-0857.129819

Cited by 10 publications

(9 citation statements)

References 8 publications

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“…B19V targets erythroid progenitor cells such as erythroblast in the bone marrow and may causes lytic infection resulting in severe anemia1165, PRCA which may lead to myelodysplasia17. Non-erythroid cell68 infections by B19V involving platelets are uncommon but may occur as some of the thrombocytes are known to possess receptor for B19V and thus cause thrombocytopaenia with anaemia or bicytopaenias1819, while our report on acquired pure amegakaryocytic thrombocytopenia20 remains solitary. Other afflictions such as macrophage activation as in haemophagocytosis syndrome19 induced by B19V are also rare.…”

Section: Multiorgan Afflictions By Human Parvovirus B19 (B19v) and Itmentioning

confidence: 68%

“…Children with juvenile chronic arthropathy (n=69) now known as juvenile idiopathic arthropathy, were studied and B19V infection was observed in 27 per cent children16. Next, B19V-induced clinical cases ending fatally with pure red cell aplasia (PRCA), severe anaemia and thrombocytopaenia with hepatitis in a child and haemophagocytic syndrome in an infant were reported171819. Further three novel clinical associations of B19V were reported, namely B19V-induced pure amegakaryocytic thrombocytopaenia in a nine month old male infant (got cured by IVIG treatment)20, myositis21 as a complication of erythema infectiosum in a nine year old female child and a series of eight cases with non-occlusive ischaemic gangrene of stomach or bowel including four cases having extensive gangrene of either entire ileum or jejunum to right colon who died post-operatively due to short gut syndrome (mortality 50%)22.…”

Section: Approach To Unveil Clinical Manifestations Furthermentioning

confidence: 99%

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Clinical impact & pathogenic mechanisms of human parvovirus B19: A multiorgan disease inflictor incognito

Kishore

2018

Indian J Med Res

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Human parvovirus B19 (B19V) causes myriads of clinical diseases; however, owing to lack of awareness and undetermined clinical impact, it has failed to become a virus pathogen of global concern. Cryptically, B19V causes significant morbidity and mortality. Half of the world population and 60 per cent of Indians are known to be serologically naive and are at risk of acquiring B19V infections. Cumulatively, our data showed 21.3 per cent B19V-infected patients with juvenile chronic arthropathy, recurrent abortions, multi-transfused thalassaemia and leukaemia. In addition, B19V-infected cases that ended fatally included patients with pure red cell aplasia, fulminant hepatitis and haemophagocytic syndrome. Novel clinical associations of B19V observed were amegakaryocytic thrombocytopaenia, myositis and non-occlusive ischaemic gangrene of bowel. B19V possesses multiple receptors which are distributed widely in human tissues. Vascular endothelial cell infection by B19V causes endothelialitis and vasculitic injuries besides antibody-dependent enhancement which empowered B19V to cause multiorgan diseases. Owing to lack of suitable animal model for B19V, true causal role remains to be determined, but numerous reports on B19V infections substantiate a causal role in multiorgan diseases. Hence, B19V infections need to be recognized, investigated and treated besides making efforts on vaccine developments.

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Section: Multiorgan Afflictions By Human Parvovirus B19 (B19v) and Itmentioning

confidence: 68%

Section: Approach To Unveil Clinical Manifestations Furthermentioning

confidence: 99%

Clinical impact & pathogenic mechanisms of human parvovirus B19: A multiorgan disease inflictor incognito

Kishore

2018

Indian J Med Res

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“…There are 19 published cases of Parvovirus B19‐associated HLH , which are summarized in Table , and an additional three cases published in either German or Japanese, which were not included in this case series . Of these 19, 5 were female, 13 were male, and in one case, gender was not stated.…”

Section: Introductionmentioning

confidence: 99%

A review of blood diseases and cytopenias associated with human parvovirus B19 infection

Kerr

2015

Reviews in Medical Virology

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Parvovirus B19 is a single-stranded DNA virus which preferentially targets the erythroblast resulting in red cell aplasia, which is temporary in immunocompetent persons. Since the discovery of B19 virus in 1975, a wide variety of blood diseases and cytopenias affecting several blood cell lineages have been documented during or following B19 infection. These include cytopenias affecting the erythroid, megakaryoblastoid, myeloid and lymphoid lineages, as well as a variety of bicytopenias, pancytopenia, bone marrow necrosis / fat embolism syndrome, myelodysplastic syndrome, leucoerythroblastopenia, and hemophagocytic lymphohistiocytosis. B19 infection may also complicate and precede the course of acute leukemia, the significance of which remains to be determined. This review describes the current state of knowledge of the abnormalities of individual blood cell lineages encountered during parvovirus B19 infection, over the almost 40 years since its discovery, and reveals some very interesting themes, which improve our understanding of the pathogenesis of B19 infection with particular reference to the bone marrow.

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“…There are 32 cases of parvovirus B19 associated HLH reported so far in the literature, including our case (16)(17)(18)(19)(20)(21)(22)(23). Sixteen of the patients were adults while another sixteen patients, were children.…”

Section: Discussionmentioning

confidence: 89%

“…Other underlying conditions that were present in previous case reports include sickle cell anaemia and iron deficiency anaemia. Most of these patients had spontaneous recovery without specific therapy, while some of them were treated with corticosteroids or intravenous immunoglobulin (16)(17)(18)(19)(20)(21)(22)(23). This condition has showed a benign clinical course in more than 80% of the cases.…”

Section: Introductionmentioning

confidence: 99%

Parvovirus B19 Associated Haemophagocytic Lymphohistiocytisis in Hereditary Spherocytosis Patient: A Case Report

Cs¹,

Gan²,

Pc³

et al. 2016

JUMMEC

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Haemophagocytic lymphohistiocytosis (HLH) is a clinico-pathologic entity caused by increased proliferation and activation of benign macrophages with haemophagocytosis throughout the reticulo-endothelial system. Virus-associated HLH is a well-recognised entity. Although majority of parvovirus B19 associated HLH does not require any specific treatment and carries good prognosis, outcome of children is worse than adults. We report here a case of HLH associated with acute parvovirus B19 infection in a young healthy patient with underlying hereditary spherocytosis, with bone marrow findings typical of parvovirus infection. Although this patient had spontaneous recovery of cell counts, he succumbed due to complication from prolonged ventilation. Unexpectedly, his immunoglobulin levels were inappropriately normal despite ongoing ventilator associated pneumonia, which reflects inadequate humoral immune response towards infection.

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Fatal missed case of hemophagocytic lymphohistiocytosis co-infected with parvovirus B19 and Epstein-Barr virus in an infant: Test hyperferritinaemia early

Cited by 10 publications

References 8 publications

Clinical impact & pathogenic mechanisms of human parvovirus B19: A multiorgan disease inflictor incognito

Clinical impact & pathogenic mechanisms of human parvovirus B19: A multiorgan disease inflictor incognito

A review of blood diseases and cytopenias associated with human parvovirus B19 infection

Parvovirus B19 Associated Haemophagocytic Lymphohistiocytisis in Hereditary Spherocytosis Patient: A Case Report

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