Browse
Submit Data
Databases
API
Help

Dataset Information

27 Views

0 Connections

0 Citations

0 Reanalyses

0 Downloads

Omics score: 0

First case report of bloodstream infection by Rhizomucor pusillus in a child with hemophagocytic lymphohistiocytosis.

ABSTRACT: We describe an unusual presentation of fatal infection due to Rhizomucor pusillus bloodstream infection in a 12-year old pediatric patient recently diagnosed with hemophagocytic lymphohistiocytosis. R. pusillus was isolated from one blood culture drawn on Day 11 of hospitalization.

SUBMITTER: Dien Bard J

PROVIDER: S-EPMC4066188 | biostudies-literature | 2014 Jul

REPOSITORIES: biostudies-literature

ACCESS DATA

Json Xml

Publications

First case report of bloodstream infection by Rhizomucor pusillus in a child with hemophagocytic lymphohistiocytosis.

Dien Bard Jennifer J Mangahis Aida A Hofstra Thomas C TC Bender Jeffrey M JM

Medical mycology case reports 20140604

We describe an unusual presentation of fatal infection due to Rhizomucor pusillus bloodstream infection in a 12-year old pediatric patient recently diagnosed with hemophagocytic lymphohistiocytosis. R. pusillus was isolated from one blood culture drawn on Day 11 of hospitalization. ...[more]

PMID: 24967155

Similar Datasets

Case Report: HAVCR2 mutation-associated Hemophagocytic lymphohistiocytosis

Project description: Not available

| S-EPMC10701531 | biostudies-literature

Hemophagocytic lymphohistiocytosis associated with Epstein-Barr virus infection: case report and literature review.

Project description:Hemophagocytic lymphohistiocytosis (HLH) is rare and life threatening syndrome. There are only a few reported cases of HLH with GI symptoms. We describe the case of an 18 months old boy who presented with a history of fever for 40 days, abdominal distention and hepatosplenomegaly. Abdominal x-ray showed a pneumoperitoneum. Urgent laparotomy was done which revealed an isolated cecal perforation. The histopathological findings in the subsequent resected bowel was HLH with evidence of positive EBV Barr infection.

| S-EPMC6598294 | biostudies-literature

Mycobacterium iranicum bacteremia and hemophagocytic lymphohistiocytosis: a case report.

Project description:BackgroundMycobacterium iranicum has recently been recognised as an opportunistic human pathogen. Although infectious conditions represent frequent triggers for hemophagocytic lymphohistiocytosis, non-tuberculous mycobacterial infections are rarely associated with this entity. To this date, M. iranicum infection has never been reported in France, has never been associated with hemophagocytic lymphohistiocytosis and has never been found to be multi-resistant on standardized antimicrobial susceptibility testing.Case presentationWe report a case of a French Caucasian man with secondary hemophagocytic lymphohistiocytosis in the context of M. iranicum bacteraemia and Hodgkin's disease. We review available data concerning M. iranicum antimycobacterial susceptibility testing and treatment outcomes. We also review the association between hemophagocytic lymphohistiocytosis and non-tuberculous mycobacterial infections.ConclusionInterpretation of M. iranicum positive cultures remains a clinical challenge and non-tuberculous mycobacterial infections need to be considered in secondary hemophagocytic lymphohistiocytosis differential diagnosis.

| S-EPMC5549388 | biostudies-literature

B-cell lymphoma-associated hemophagocytic lymphohistiocytosis: A case report.

Project description:Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening condition characterized by an exaggerated but dysregulated immune response resulting in hyperinflammation, with a potential for progression to multiple organ dysfunction and failure. Infectious diseases, inflammatory disorders, malignancies and immunodeficiency syndromes are known triggers of HLH in adults. The present study reported the case of a middle-aged man with HLH triggered by B-cell lymphoma who was successfully treated with dexamethasone; etoposide, prednisone, vincristine, cyclophosphamide, hydroxy-doxorubicin and rituximab chemotherapy; and multiple intrathecal methotrexate with a good outcome.

| S-EPMC9214690 | biostudies-literature

Case report: Hemophagocytic lymphohistiocytosis in a child with primary immunodeficiency infected with <i>Talaromyces marneffei</i>.

Project description:Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening immune-mediated disease that affects patients with known genetic defects and is increasingly found among those with autoimmune diseases and persistent infections. Talaromyces marneffei (TM) is a human opportunistic fungus that commonly infects immunodeficient or immunosuppressed individuals. Few TM-associated secondary HLH cases resulting from autoimmune deficiency have been reported previously. The current case study describes a pediatric patient hospitalized with recurrent fever and lymphadenopathy. The child had abnormal blood cell classification, and microscopy revealed mature granulocytes that phagocytized fungal spores. It was speculated that the patient was infected with TM. The pathogen was detected earlier than the blood culture and confirmed by metagenomic next-generation sequencing. Whole-exome sequencing revealed that the patient had complex mutations associated with immunodeficiency. This included a mutation in exon 3 of the CD40LG gene, c.346G>A, which may be linked to hyper-IgM syndrome, a primary immunodeficiency disease with immunoglobulin conversion recombination defects that could explain the patient's increased susceptibility to serious opportunistic infections. In addition, a heterozygous frameshift variant, c.820dup (p.Asp274GlyfsTer61), was detected in exon 6 of CARD9, a key gene associated with fungal immune surveillance. After 4 days of fungal treatment, the abnormal blood cell clusters disappeared, but other infections occurred in succession for 6 months after rehabilitation. The patient was followed with the aim of providing subsequent immunotherapy. This study found that infection can trigger HLH in HIV-negative individuals, highlighting the importance of early definitive identification of the causative agent and investigation of potential immunodeficiency.

| S-EPMC9755863 | biostudies-literature

Hemophagocytic lymphohistiocytosis and congenital factor VII deficiency: a case report.

Project description:BackgroundHemophagocytic lymfohistiocytosis (HLH) is a rare, life-threatening hyperinflammation, characterized by immune system over-activation resulting in hemophagocytosis. HLH could appear as a primary disease caused by mutations of immune-regulatory genes, or develop as a result of viral or bacterial infections, or malignancy. Congenital factor VII (FVII) deficiency is a rare autosomal recessive disorder characterized by prolonged prothrombin time (PT) and low FVII, which may increase bleeding risk.Case presentationA 50-year-old woman was admitted for a fever persisted for 20 days, presenting with cytopenia, high hyperferritinemia, low activity of NK cells. Bone marrow aspiration showed hemophagocytosis. CT scanning found pulmonary infection. EBV and CMV were not detected. Genetic scanning did not find pathogenic mutation of a HLH NGS panel including 26 genes. This patient was treated as recommended by the HLH 2004 Guidelines. Coagulation tests identified FVII deficiency. Genetic analysis of F7 gene in the patient and her family members identified recurrent compound heterozygous F7 c.64 + 5G > A and c.1224 T > G (p.His408Gln) mutations in this patient and her brother who showed postoperative hemorrhage after surgical resection of renal cell carcinoma. Heterozygotes in this family were asymptomatic.ConclusionsTo our knowledge, this is the first report of HLH in combination with congenital FVII deficiency in Chinese population.

| S-EPMC6134588 | biostudies-literature

Hemophagocytic lymphohistiocytosis and myopericarditis induced by campylobacter: a case report.

Project description:BackgroundHemophagocytic lymphohistiocytosis (HLH) is a severe disorder characterized by excessive activation of the immune system, leading to hypercytokinemia and damage to multiple organs. We report a rare case of HLH with myopericarditis caused by Campylobacter infection.Case presentationA 28-year-old male patient with a history of hypertension without medicine control presented at the hospital after a four-day fever, decreasing urine amount, rashes on his trunk and limbs, and other symptoms. He was admitted with a provisional diagnosis of atypical infection and allergic skin rash related to diclofenac. However, his condition deteriorated, and he developed shock, tachycardia, chest distress, and bilateral pleural effusion after admission. Further investigations revealed cardiogenic shock related to myopericarditis, and he was transferred to the ICU. In addition, a stool PCR panel subsequently revealed a positive result for Campylobacter. On day 6, he was diagnosed with HLH. Under Clarithromycin and dexamethasone infusion, leukocytosis, anemia and thrombocytopenia with cardiogenic shock status improved. Then, he was later discharged in stable condition.ConclusionsHLH and myopericarditis caused by Campylobacter are very rare. Early detection of Campylobacter-induced HLH and multiple organ failure, as well as prompt use of antibiotics and immunosuppressants, can be helpful for prognosis.

| S-EPMC11003181 | biostudies-literature

Case Report: Parvovirus B19 infection complicated by hemophagocytic lymphohistiocytosis in a heart-lung transplant patient.

Project description:Immunosuppressed patients can contract parvovirus B19, and some may experience hemophagocytic lymphohistiocytosis (HLH). Herein, we describe the first report of hemophagocytic lymphohistiocytosis in a heart-lung transplant patient with concomitant parvovirus B19 infection. The patient was treated with intravenous immune globulin (IVIG) and the features of HLH were remission. This instance emphasizes the significance of parvovirus B19 monitoring in transplant patients with anemia; if HLH complicates the situation, IVIG may be an adequate remedy. Finally, a summary of the development in diagnosing and managing parvovirus B19 infection complicated by HLH is provided.

| S-EPMC9941661 | biostudies-literature

Diagnostic challenges of hemophagocytic lymphohistiocytosis in child with multiorgan dysfunction in a low-resource setting: A case report.

Project description:Introductionand importance: Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening hyperinflammatory syndrome as a result of dysregulation of the immune system. Physicians in the intensive care unit (ICU), especially pediatricians, need to know how to recognize the diagnostic criteria and spectrum of HLH clinical presentations because early detection and timing of initial therapy affect the survival rate of the patient.Case presentationA 7-year-old female patient was referred to the pediatric ICU (PICU) at our tertiary hospital because of the suspicion of severe sepsis with shock and disseminated intravascular coagulation. On the fifth day of treatment, the patient was intubated and given a mechanical ventilator after experiencing respiratory failure. On the seventh day in the PICU, high fever persisted and the patient developed worsening acute kidney injury with oliguria. When worsening conditions continued, the patient experienced hypotension and cardiac arrest. The patient died on the 8th day of treatment at PICU due to severe shock and multiorgan failure.Clinical discussionHLH complications can be life-threatening with documented ICU mortality of 35%, even with an appropriate initial therapeutic approach. Patients with HLH can have rapid progression of disease and often require a significant amount of ICU supportive care, including vasopressor support, significant amount of blood products, ventilator support, and renal replacement therapy.ConclusionThe diagnosis of HLH should be considered if there are cases with persistent, prolonged fever, organomegaly, cytopenias and evolving Multi Organ Dysfunction Syndrome. It is important for pediatricians to know the diagnostic criteria and possible clinical presentations of HLH.

| S-EPMC8347814 | biostudies-literature

Project description:Adult hemophagocytic lymphohistiocytosis is a secondary immunopathologic phenomenon, mainly secondary to malignancy, infection, or autoimmune disorders. The performance of diagnostic criteria, studied in the pediatric population, is yet to be validated in the adult population. Some of the criteria include cytopenias and organomegaly that are inherent features to malignant processes, thus making the diagnosis of hemophagocytic lymphohistiocytosis a challenge in patients with cancer.We describe the case of a 54-year-old white man with history of metastatic maxillary sinus adenoid cystic carcinoma who had severe liver injury and cytopenias with progressive clinical deterioration. We performed an evaluation, by flow cytometry, of the expression of surface markers in his natural killer cells that revealed remarkable abnormalities. His syndrome eventually fulfilled criteria for hemophagocytic lymphohistiocytosis and he received therapy with steroids with interval clinical improvement. Unfortunately, he refused further cytotoxic treatment and died 2 weeks later.The conventional criteria for the diagnosis of hemophagocytic lymphohistiocytosis are suboptimal for adult patients with cancer resulting in delays in diagnosis and timely initiation of treatment. The diagnostic criteria have to be re-evaluated in patients with cancer; novel, easily available, and accurate diagnostic methods are needed.

| S-EPMC5485603 | biostudies-literature

OmicsDI is part of the ELIXIR infrastructure

OmicsDI is an Elixir interoperability service. Learn more ›

Tweets

OmicsDI Databases

PRIDE
PeptideAtlas
MassIVE
JPOST Repository
Physiome Model Repository

EGA
EVA
ENA
LINCS
PAXDB
Cell Collective

MetaboLights
Metabolomics Workbench
MetabolomeExpress
GNPS
BioModels
FAIRDOMHub

ArrayExpress
dbGaP
ExpressionAtlas
GEO
NODE

Information

Databases
Help
API
Contact us
Code on GitHub
Terms of use
Submit Data