Virus-associated hemophagocytic syndrome due to rubella virus and varicella-zoster virus dual infection in patient with adult idiopathic thrombocytopenic purpura

Takeoka, Yasunobu; Hino, Masayuki; Oiso, Naoki; Nishi, Shinichi; Kr, Koh; Yamane, Takahisa; Ohta, Kensuke; Nakamae, Hirohisa; Aoyama, Yasutaka; Hirose, Asao; Fujino, Hitoshi; Takubo, Takayuki; Inoue, Takeshi; Tatsumi, Noriyuki

doi:10.1007/s002770000282

Cited by 13 publications

(5 citation statements)

References 22 publications

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“…Of note, VZV was reported in all of the three fatal cases, while two of the cases also had co-infection with CMV; these cases also had a significant medical history, including autoimmune diseases such as ITP and SLE. Other studies reported VZV as the most common viral infection among ITP patients (39). SLE patients are on lifetime immunosuppressants and at high risk for viral infections, especially VZV (40).…”

Section: Discussionmentioning

confidence: 95%

Viruses Causing Aseptic Meningitis: A Tertiary Medical Center Experience With a Multiplex PCR Assay

et al. 2020

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Background: Central nervous system (CNS) infection is associated with high rates of morbidity and mortality, and despite advancements in molecular testing, aseptic meningitis remains challenging to diagnose. Aseptic meningitis cases are often underreported worldwide, which impacts the quality of patient care. Therefore, we aimed to assess the results of BioFire® FilmArray® meningitis/encephalitis (ME) PCR panel, clinical characteristics, and etiologies of aseptic meningitis patients.Methods: From January 2018 to January 2020, all pediatric and adult patients in a large tertiary medical center who underwent lumbar puncture and cerebrospinal fluid (CSF) testing by a ME multiplex PCR panel and who fit the aseptic meningitis definition were retrospectively reviewed.Results: Data were reviewed from 1,607 patients; 240 met the inclusion criteria (54.6% males; 68.8% <4 years of age). The rate of detected viral causes of aseptic meningitis was 40.4%; therefore, 59.6% of the patients remained with unidentified etiology. Among the identified viral meningitis, enterovirus and human herpesvirus 6 (HHV-6) were the most common (25 and 7.9%, respectively). The median length of hospital stay was 6 days, and it was longer in patients with unidentifiable aseptic meningitis (p < 0.0001).Conclusion: Aseptic meningitis is common among suspected meningitis patients, but most cases remained of unknown etiology. The most common identified viruses were enterovirus followed by HHV-6, and there is predominance in males and the pediatric age group. These results highlight that further research is needed to identify other etiologies and possible additional viral pathogens for aseptic meningitis in the current diagnostic methods.

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Section: Discussionmentioning

confidence: 95%

Viruses Causing Aseptic Meningitis: A Tertiary Medical Center Experience With a Multiplex PCR Assay

et al. 2020

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“…However, he finally made a full recovery and remained well during a 3-year follow-up period. In fact, there was another patient who survived from HLH and liver dysfunction secondary to rubella and varicella virus dual infection, but her liver damage did not reach the standard of ALF[ 21 ].…”

Section: Discussionmentioning

confidence: 99%

Successful rescue of acute liver failure and hemophagocytic lymphohistiocytosis following varicella infection: A case report and review of literature

Zhang

Guo

Zhu

et al. 2018

WJCC

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Herein we report a case of acute liver failure (ALF) and hemophagocytic lymphohistiocytosis (HLH) induced by varicella infection, successfully rescued by a combination therapy of acyclovir, supportive care, and immunosuppression with dexamethasone and etoposide. A previously healthy 16-year-old boy presented with generalized rash, fever, severe abdominal pain, and abnormal liver function within 4 d. Chickenpox was suspected, and acyclovir and intravenous immunoglobulin were started on admission. However, the patient’s condition deteriorated overnight with soaring transaminases, severe coagulopathy and encephalopathy. On the fourth day of admission, pancytopenia emerged, accompanied by hypofibrinogenemia and hyperferritinemia. The patient was diagnosed with ALF. He also met the diagnostic criteria of HLH according to the HLH-2004 guideline. Polymerase chain reaction (PCR) amplifications of varicella-zoster virus (VZV) were positive, confirming that VZV was a causative trigger for ALF and HLH. In view of the devastating immune activation in HLH, immunosuppression therapy with dexamethasone and etoposide was administered, in addition to high dose acyclovir. The patient’s symptoms improved dramatically and he finally made a full recovery. To our knowledge, this is only the second report of a successful rescue of ALF associated with HLH, without resorting to liver transplantation. The first case was reported in a neonate infected by herpes simplex virus-1. However, survival data in older children and adults are lacking, most of whom died or underwent liver transplantation. Our report emphasizes the clinical vigilance for the possible presence of HLH, and the necessity of extensive investigation for underlying etiologies in patients presenting with indeterminate ALF. Early initiation of specific therapy targeting the underlying etiology, and watchful immunosuppression such as dexamethasone and etoposide, together with supportive therapy, are of crucial importance in this life-threatening disorder.

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“…Systemic lupus erythematosus and adult-onset still's disease, which represent a slightly different condition known as macrophage activation syndrome or MAS (22), as well as four genes (PRF1, UNC13D, STX11, and STXBP2), which all encode proteins necessary for lymphocyte cytotoxicity, have so far been found to have disease-causing mutations in primary HLH, which is passed down in an autosomal recessive manner. (2,20,27) .…”

Section: Case Presentationmentioning

confidence: 99%

“…Instead, intubation with an ambo bag was performed when the patient was in a persistent shock state with CPR for 30 minutes on pulseless electrical activity. with the rubella virus and varicella zoster virus have also been reported (20), as has a case of Candida albicans and reactivated EBV infections (21), or from bacteria such as mycobacteria, mycoplasma, rickettsia, and murine typhus, particularly in patients returning from endemic or other tropical regions (18, 22), Chlamydia pneumonia (23), and Capnocytophaga canimorsus bacteremia (21). Protozoa and fungi are two additional infectious agents (13).…”

Section: Case Presentationmentioning

confidence: 99%

“…Protozoa and fungi are two additional infectious agents (13). Malignant diseases are frequently seen in malignant lymphoma (20), which oncologists believe is a good imitator of HLH and can cause HLH diseases (24), though a case report of melanoma has also been identi ed (7). Other important etiological factors that are considered rare but play an important role in the etiology of this disease are surgery, speci cally silicone implants surgery for breast augmentation and reconstruction (3), which were found to be associated with anaplastic large cell lymphoma, despite being extremely rare but continuing to be reported (25,26), proving that tumorogenesis and tumor progression have been linked to in ammation activating the pro in ammatory mediators as well as infections that are considered to be the key role for the activation of B and T cells (26).…”

Section: Case Presentationmentioning

confidence: 99%

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Haemophagocytic lymphocytosis (HLH) vicious cycle in a Cannabinoids addict patient case report and disease review.

Amer

Hamdar

Amer

et al. 2023

Preprint

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Haemophagocytic lymphocytosis (HLH) is a rare life-threatening syndrome caused by the uncontrolled activity of cytokines, natural killers, and macrophages, which can alter the activity of the organism, resulting in multiple organ dysfunction and mortality. Fever, splenomegaly, coagulopathies, dyspnea, changes in mental status, or irritability may be associated with HLH diseases. Depending on the underlying causes, such as bacterial or viral infections, HLH may be primary, hereditary, or secondary. The early diagnosis and treatment of patients are directly related to their prognosis and clinical outcome. On the other hand, HLH can present a number of obstacles, particularly for children and newborns, as well as hematological defects which might cause other autoimmune disorders. Case presentation: We present a case of a 28-year-old male patient admitted to the hospital with a history of persistent high grade fever for two weeks, right lower limb swelling three weeks prior to admission, and right side weakness for one month. Aside from being addicted to cannabis for three years, the patient has no prior medical history. On admission, the patient was awake, conscious, oriented, and hemodynamically stable. A complete blood count, ultrasound examinations, and a whole-body CT scan revealed that the results were favorable for multiple enlarged lymph nodes and hepatosplenomegaly. The patient was given acyclovir, Vfend, Colistin, Targocid, Tinam, Septrin, and anticoagulant during the follow-up. The patient's clinical condition was rapidly deteriorating; a bone marrow biopsy was performed, which revealed haemophagocytosis; and dexamethasone was started. The patient's clinical condition deteriorated during his hospital stay until he went into cardiac arrest and had to be resuscitated for 30 minutes with pulseless electrical activity. Background and Aim: This case suggests that we should be vigilant to the patient who is admitted to the hospital with symptoms for unknown reasons, in order to diagnose HLH as soon as possible and clarify its cause, and it also puts several theories regarding the pathogenicity of this disease in our hands, which will be described in this case, making this case a subject for discussion and research in the medical field. Conclusion: Haemophagocytic lymphocytosis (HLH) is a severe inflammatory disease that improperly controls the body's immune response. Viruses and bacterial infections are just two examples of the various etiological causes that can cause it. Lab results and symptom presentations that are particular to the primary infection can indicate it. Diagnosis and treatment must be provided as soon as possible in order to reduce morbidity and mortality.

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Virus-associated hemophagocytic syndrome due to rubella virus and varicella-zoster virus dual infection in patient with adult idiopathic thrombocytopenic purpura

Cited by 13 publications

References 22 publications

Viruses Causing Aseptic Meningitis: A Tertiary Medical Center Experience With a Multiplex PCR Assay

Viruses Causing Aseptic Meningitis: A Tertiary Medical Center Experience With a Multiplex PCR Assay

Successful rescue of acute liver failure and hemophagocytic lymphohistiocytosis following varicella infection: A case report and review of literature

Haemophagocytic lymphocytosis (HLH) vicious cycle in a Cannabinoids addict patient case report and disease review.

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