Hemophagocytic lymphohistiocytosis caused by systemic herpes simplex virus type 1 infection: Successful treatment with dexamethasone palmitate

Otsubo, Kenji; Fukumura, Akiko; Hirayama, Mariko; Morimoto, Takahiro; Katô, Masahiko; Mochizuki, Hiroyuki

doi:10.1111/ped.12817

Cited by 10 publications

(5 citation statements)

References 10 publications

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“…Studies were excluded if they did not discuss nHLH ( n = 280); aggregated nHLH patient data with all ages ( n = 83); provided insufficient detail and/or data describing a case of nHLH ( n = 28); and for other reasons ( n = 8). Two‐hundred‐five cases of nHLH from 142 articles were included in our analysis 12–152 …”

Section: Resultsmentioning

confidence: 99%

Neonatal hemophagocytic lymphohistiocytosis: A meta‐analysis of 205 cases

Kranz,

Hahn,

Thompson

et al. 2024

Pediatric Blood & Cancer

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BackgroundNeonatal hemophagocytic lymphohistiocytosis (nHLH), defined as HLH that presents in the first month of life, is clinically devastating. There have been few large descriptive studies of nHLH.ObjectivesThe objective of this study was to perform a meta‐analysis of published cases of nHLH.MethodsA comprehensive literature database search was performed. Cases of HLH were eligible for inclusion if clinical analysis was performed at age ≤30 days. Up to 70 variables were extracted from each case.ResultsA total of 544 studies were assessed for eligibility, and 205 cases of nHLH from 142 articles were included. The median age of symptom onset was day of life 3 (interquartile range [IQR]: 0–11, n = 141). Median age at diagnosis was day of life 15 (IQR: 6–27, n = 87). Causes of HLH included familial HLH (48%, n = 99/205), infection (26%, n = 53/205), unknown (17%, n = 35/205), macrophage activation syndrome/rheumatologic (2.9%, n = 4/205), primary immune deficiency (2.0%, n = 5/205), inborn errors of metabolism (2.4%, n = 5/205), and malignancy (2.0%, n = 4/205). Fever was absent in 19% (n = 28/147) of all neonates and 39% (n = 15/38) of preterm neonates. Bicytopenia was absent in 26% (n = 47/183) of patients. Central nervous system (CNS) manifestations were reported in 63% of cases (n = 64/102). Liver injury (68%, n = 91/134) and/or liver failure (24%, n = 32/134) were common. Flow cytometry was performed in 22% (n = 45/205) of cases. Many patients (63%, n = 121/193) died within the period of reporting. Discernable values for HLH diagnostic criteria were reported between 30% and 83% of the time.ConclusionsEvaluation of nHLH requires rapid testing for a wide range of differential diagnoses. HLH diagnostic criteria such as fever and bicytopenia may not occur as frequently in the neonatal population as in older pediatric populations. Neurologic and hepatic manifestations frequently occur in the neonatal population. Current reports of nHLH suggest a high mortality rate. Future publications containing data on nHLH should improve reporting quality by reporting all clinically relevant data.

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Section: Resultsmentioning

confidence: 99%

Neonatal hemophagocytic lymphohistiocytosis: A meta‐analysis of 205 cases

Kranz,

Hahn,

Thompson

et al. 2024

Pediatric Blood & Cancer

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“…It is well known that systemic infections can lead to complications such as HLH, and several studies have reported viral infections as the leading infectious triggers of HLH [12], [13], [14], [15], [16]. Disseminated HHV-1 infection usually occurs in immunocompromised patients, but has also been described in immunocompetent hosts; reports have described four of five patients with HSV-related HLH as immunocompetent.…”

Section: Discussionmentioning

confidence: 99%

V348I mutation in UL23 gene of human herpesvirus 1 in a case of herpetic hepatitis and haemophagocytic lymphohistiocytosis

Corcione

Sidoti

Costa

et al. 2019

New Microbes and New Infections

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We herein report the case of a young immunocompetent adult patient with a rapidly fatal haemophagocytic lymphohistiocytosis syndrome related to human herpesvirus 1 (HHV-1) infection, with herpetic hepatitis and persistent high-level viraemia despite treatment with acyclovir. Haemophagocytic lymphohistiocytosis was confirmed in the patient's spleen and bone marrow. HHV-1 DNA was extracted from whole blood and liver biopsy and the UL23 gene was sequenced. A V348I natural polymorphism of the TK protein was found in blood and liver specimens. Further studies are needed to investigate the role of this polymorphism in the development of systemic immune dysregulation.

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“…As an effective anti-inflammatory drug, limethason is used for treating multiple disorders, including macrophage activation syndrome, hemophagocytic lymphohistiocytosis and idiopathic pulmonary hemosiderosis ( 15 , 17 , 18 ). In addition, Hoshi et al ( 19 ) demonstrated that this drug has a higher treatment rate of rheumatoid arthritis with lower risk of side effects compared with dexamethasone.…”

Section: Discussionmentioning

confidence: 99%

Limethason reduces airway inflammation in a murine model of ovalbumin-induced chronic asthma without causing side effects

Miao

Wang

et al. 2018

Exp Ther Med

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Airway inflammation is the major pathological feature of asthma. Thus, the current therapeutic strategy for asthma is to control inflammation. Limethason, an anti-inflammation drug, is widely used in rheumatoid arthritis treatment. The aim of the present study was to detect the anti-inflammatory effect and side effects of limethason on airways that were sensitized with ovalbumin in a murine model of chronic asthma. In the present study, BALB/c mice were sensitized with ovalbumin. Airway hyperresponsiveness was estimated, and hematoxylin and eosin staining, Periodic acid-Schiff staining and bronchoalveolar lavage were used to detect the effect on chronic asthma. Limethason effectively reduced airway hyperresponsiveness, and inhibited inflammatory cell infiltration and mucus secretion. Bronchoalveolar lavage fluid analysis revealed that limethason suppressed levels of airway eosinophils. In the period of treatment, limethason exhibited no influence on morphology of the femoral head, bone mineral content or bone mineral density, which were detected by histological studies and dual-energy X-ray absorptiometry. The index of liver, spleen, kidney, gastrocnemius and brown adipose tissue also demonstrated that limethason had no adverse effects on organs and tissues. The present study revealed that limethason could effectively reduce inflammation in an asthma mouse model without side effects. Therefore, limethason may have therapeutic potential for treating chronic asthma clinically.

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Hemophagocytic lymphohistiocytosis caused by systemic herpes simplex virus type 1 infection: Successful treatment with dexamethasone palmitate

Cited by 10 publications

References 10 publications

Neonatal hemophagocytic lymphohistiocytosis: A meta‐analysis of 205 cases

Neonatal hemophagocytic lymphohistiocytosis: A meta‐analysis of 205 cases

V348I mutation in UL23 gene of human herpesvirus 1 in a case of herpetic hepatitis and haemophagocytic lymphohistiocytosis

Limethason reduces airway inflammation in a murine model of ovalbumin-induced chronic asthma without causing side effects

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