Coxiella burnetii causing haemophagocytic syndrome: a rare complication of an unusual pathogen

Harris, Patrick N A; Dixit, R.; Norton, Robert

doi:10.1007/s15010-011-0142-4

Cited by 16 publications

(5 citation statements)

References 28 publications

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“…Hemophagocytic syndrome can be a consequence of bone marrow involvement. This clinical presentation was reported for the first time in 1984 by Estrov et al (510), and since then fewer than 10 cases have been reported (511)(512)(513), with the most recent involving an asplenic patient (514). 18 F-FDG PET/CT, which is less invasive than bone marrow biopsy, can detect bone marrow involvement.…”

Section: Clinical Manifestationsmentioning

confidence: 77%

From Q Fever to Coxiella burnetii Infection: a Paradigm Change

et al. 2017

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Coxiella burnetii is the agent of Q fever, or "query fever," a zoonosis first described in Australia in 1937. Since this first description, knowledge about this pathogen and its associated infections has increased dramatically. We review here all the progress made over the last 20 years on this topic. C. burnetii is classically a strict intracellular, Gram-negative bacterium. However, a major step in the characterization of this pathogen was achieved by the establishment of its axenic culture. C. burnetii infects a wide range of animals, from arthropods to humans. The genetic determinants of virulence are now better known, thanks to the achievement of determining the genome sequences of several strains of this species and comparative genomic analyses. Q fever can be found worldwide, but the epidemiological features of this disease vary according to the geographic area considered, including situations where it is endemic or hyperendemic, and the occurrence of large epidemic outbreaks. In recent years, a major breakthrough in the understanding of the natural history of human infection with C. burnetii was the breaking of the old dichotomy between "acute" and "chronic" Q fever. The clinical presentation of C. burnetii infection depends on both the virulence of the infecting C. burnetii strain and specific risks factors in the infected patient. Moreover, no persistent infection can exist without a focus of infection. This paradigm change should allow better diagnosis and management of primary infection and long-term complications in patients with C. burnetii infection.

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Section: Clinical Manifestationsmentioning

confidence: 77%

From Q Fever to Coxiella burnetii Infection: a Paradigm Change

et al. 2017

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“…The incidence of pediatric HLH due to bacterial infections in Japan was reported to be about 6% of the infectionassociated HLH cases [6]. Several studies have reported bacterial infection-associated HLH [7], including Mycoplasma pneumoniae [8,9], Listeria monocytogenes [10], Leptospira [11], Brucella [12], group B streptococcus [13], and Coxiella burnetii [14]. Secondary HLH caused by S. pneumococcus has been previously described in adults after splenectomy or with primary immunodeficiency [15,16].…”

Section: Discussionmentioning

confidence: 99%

Hemophagocytic lymphohistiocytosis complicating invasive pneumococcal disease: a pediatric case report

et al. 2020

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Background: Hemophagocytic lymphohistiocytosis (HLH) is an infrequent but life-threatening disease due to excessive immune activation. Secondary HLH can be triggered by infections, autoimmune diseases, and malignant diseases. Streptococcus pneumoniae is a pathogenic bacterium responsible for invasive pneumococcal disease (IPD) such as meningitis and bacteremia. Although the pneumococcal conjugate vaccine (PCV) has led to reductions in IPD incidence, cases of IPD caused by serotypes not included in PCV are increasing. There are few reports of secondary HLH caused by IPD in previously healthy children. We herein report a rare case of a previously healthy boy with secondary HLH complicating IPD of serotype 23A, which is not included in the pneumococcal 13-valent conjugate vaccine (PCV-13). Case presentation: An 11-month-old boy who had received three doses of PCV-13 was hospitalized with prolonged fever, bilateral otitis media, neutropenia and elevated C-reactive protein (CRP) levels. Blood culture on admission revealed S. pneumoniae, leading to a diagnosis of IPD. HLH was diagnosed based on a prolonged fever, neutropenia, anemia, hepatosplenomegaly, hemophagocytosis in the bone marrow, and elevated serum levels of triglycerides, ferritin, and soluble interleukin-2 receptor. He received broad-spectrum antibiotics and intravenous immunoglobulins for IPD and high-dose steroid pulse therapy and cyclosporine A for HLH; thereafter, his fever resolved, and laboratory findings improved. The serotype of the isolated S. pneumoniae was 23A, which is not included in PCV-13. Conclusions: It is important to consider secondary HLH as a complication of IPD cases with febrile cytopenia or hepatosplenomegaly, and appropriate treatment for HLH should be started without delay.

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“…[5] There were also published associations with hepatitis B and C viruses, [6,7] influenza viruses, [8] enteroviruses, [9] rotavirus, severe acute respiratory syndrome virus, [10] hemorrhagic fevers, [11][12][13][14][15] HIV, [15][16][17][18] and so on. Among bacterial infections associated with HLH, there were published associations with Borrelia, [19]-Babesia sp, [20] Bartonella sp, [21] Brucella sp, [22] Q fever, [23] Leptospira sp, [24] Listeria monocytogenes, [25] Mycoplasma pneumoniae, [26] and mycobacteria. [27][28][29][30][31][32] The most frequently described parasitic causes responsible for secondary HLH are: Leishmania sp, [33,34] malaria, [35][36][37] and Toxoplasma gondii.…”

Section: Discussionmentioning

confidence: 99%

Sepsis due to Streptococcus pneumoniae associated with secondary hemophagocytic lymphohistiocytosis in a splenectomized patient for spherocytosis

Bîrluțiu

Bîrluţiu

2017

Medicine

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Rationale:Hemophagocytic lymphohistiocytosis (HLH) is a syndrome that is characterized by an inappropriate hyperinflammatory immune response – primary, as a consequence of a genetic defect of NK cells and cytotoxic T lymphocytes or – secondary, in the progression of infections, rheumatic or autoimmune diseases, malignancies or metabolic diseases.Patient concerns:We present the case of a secondary HLH due to Streptococcus pneumoniae infection in a splenectomised patient for spherocytosis, a 37-year-old patient who was splenectomised in childhood for spherocytosis, without immuneprophylaxis induced by antipneumococcal vaccine.Outcomes:He developed a severe pneumococcal sepsis associated with secondary HLH, with unfavorable outcome and death.Lessons:To our knowledge, just 2 similar cases had been published in the literature, none in which the secondary HLH was the consequence of an invasive pneumococcal infection in a splenectomized patient for spherocytosis, and the association of splenectomy with HLH is surprizin.

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Coxiella burnetii causing haemophagocytic syndrome: a rare complication of an unusual pathogen

Cited by 16 publications

References 28 publications

From Q Fever to Coxiella burnetii Infection: a Paradigm Change

From Q Fever to Coxiella burnetii Infection: a Paradigm Change

Hemophagocytic lymphohistiocytosis complicating invasive pneumococcal disease: a pediatric case report

Sepsis due to Streptococcus pneumoniae associated with secondary hemophagocytic lymphohistiocytosis in a splenectomized patient for spherocytosis

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