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      A “cat”-astrophic case of Bartonella infective endocarditis causing secondary cryoglobulinemia: a case report

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          Abstract

          Background

          Culture-negative infective endocarditis (IE) constitutes approximately 10% of all cases of IE. Bartonella endocarditis is a common cause of culture-negative endocarditis and is associated with a high mortality rate. To date, no cases of Bartonella IE has been reported in association with cryoglobulinemia in the UK. We present a unique case of Bartonella IE causing secondary cryoglobulinemia in a young female.

          Case presentation

          A 17-year-old female with a background of pulmonary atresia and ventricular septal defect repaired with a cardiac conduit at the age of 4, presented with a one-year history of weight loss (from 53 to 39 kg) and poor appetite. She subsequently developed a vasculitic rash and haematoproteinuria with decline in renal function, requiring urgent hospital admission. Initial blood tests showed a near normal creatinine, but a raised cystatin C. Renal biopsy showed focal necrotizing glomerulonephritis with no acute tubular necrosis or chronic change. Subsequent blood tests supported a diagnosis of cryoglobulinaemic vasculitis (high rheumatoid factor, low complement, polyclonal gammopathy, Type 3 cryoglobulin). A weak positive PR3 meant there was some uncertainty about whether this could be a primary ANCA-associated vasculitis (AAV). Initial workup for an infectious cause, including multiple blood cultures, were negative. However, an echocardiogram showed definite vegetations on her surgical conduit. The patient did not respond to empirical antimicrobials and so was referred for surgical revision of her conduit. Tissue samples obtained intra-operatively demonstrated Bartonella species. With targeted antimicrobials post-operatively, she improved with resolution of immunologic abnormalities and at last review had a normal renal profile. On reviewing her social history, she had adopted several stray cats in the preceding year; and thus, the cause of the Bartonella infection was identified.

          Conclusion

          This is the first reported case of Bartonella endocarditis causing secondary cryoglobulinemia reported in the UK. The key learning points from this case include that Bartonella endocarditis can present as a cryoglobulinaemic vasculitis and should be considered in any differential when the cause of cryoglobulinaemia is not clear and to enquire about relevant exposures especially when culture-negative endocarditis is suspected.

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          Most cited references10

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          Biologic and clinical significance of cryoglobulins. A report of 86 cases.

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            Cryoglobulinaemia

            Cryoglobulinaemia refers to the serum presence of cryoglobulins, which are defined as immunoglobulins that precipitate at temperatures <37 °C. Type I cryoglobulinaemia consists of only one isotype or subclass of monoclonal immunoglobulin, whereas type II and type III are classified as mixed cryoglobulinaemia because they include immunoglobulin G (IgG) and IgM. Many lymphoproliferative, infectious and autoimmune disorders have been associated with mixed cryoglobulinaemia; however, hepatitis C virus (HCV) is the aetiologic agent in most patients. The underlying mechanism of the disorder is B cell lymphoproliferation and autoantibody production. Mixed cryoglobulinaemia can cause systemic vasculitis, with manifestations ranging from purpura, arthralgia and weakness to more serious lesions with skin ulcers, neurological and renal involvement. This Primer focuses on mixed cryoglobulinaemia, which has a variable course and a prognosis that is primarily influenced by vasculitis-associated multiorgan damage. In addition, the underlying associated disease in itself may cause considerable mortality and morbidity. Treatment of cryoglobulinaemic vasculitis should be modulated according to the underlying associated disease and the severity of organ involvement and relies on antiviral treatment (for HCV infection), immunosuppression and immunotherapy, particularly anti-CD20 B cell depletion therapies.
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              Diagnosis of 22 new cases of Bartonella endocarditis.

              Bartonella species are emerging pathogens that are seldom reported as a cause of blood culture-negative endocarditis. To report the occurrence of, risk factors for, and clinical features of Bartonella endocarditis and to evaluate the diagnostic tools available for this condition. Case series and comparison with past series. Multicenter international study in Halifax, Nova Scotia, Canada; Lyon, France; and Marseille, France. 22 patients from France, England, Canada, and South Africa were investigated for blood culture-negative endocarditis. Titer of antibodies to Bartonella species by microimmunofluorescence assay, blood or vegetation culture, and amplification of Bartonella DNA from valvular tissue by polymerase chain reaction. Cross-adsorption was done for patients with antibodies to Chlamydia species. 22 patients had definite endocarditis. Five were infected with B. quintana, 4 with B. henselae, and 13 with an undetermined Bartonella species. These cases were compared with the 11 previously reported cases. Of the patients with the newly reported cases, 19 had valvular surgery and 6 died. Nine were homeless, 11 were alcoholic, 4 owned cats, and 13 had preexisting valvular heart disease. Bartonella species caused 3% of the cases of endocarditis seen in the three study centers. The patients with these cases could have previously received a diagnosis of chlamydial endocarditis because of apparently high levels of cross-reacting antibodies to Chlamydia species. Bartonella species are an important cause of blood culture-negative endocarditis and can be identified by culture, serologic studies, or molecular biology techniques. Alcoholism and homelessness without previous valvular heart disease are risk factors for B. quintana infection but not for infection with other Bartonella species.
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                Author and article information

                Contributors
                arani.vivekanantham@ndorms.ox.ac.uk
                Journal
                BMC Rheumatol
                BMC Rheumatol
                BMC Rheumatology
                BioMed Central (London )
                2520-1026
                25 March 2022
                25 March 2022
                2022
                : 6
                : 16
                Affiliations
                [1 ]GRID grid.419319.7, ISNI 0000 0004 0641 2823, The Kellgren Centre of Rheumatology, , Manchester Royal Infirmary, ; Oxford Road, Manchester, UK
                [2 ]GRID grid.5379.8, ISNI 0000000121662407, Centre for Epidemiology Versus Arthritis, , University of Manchester, ; Manchester, UK
                [3 ]GRID grid.437500.5, ISNI 0000 0004 0489 5016, Liverpool Heart and Chest Hospital NHS Foundation Trust, ; Thomas Drive, Liverpool, UK
                [4 ]GRID grid.413582.9, ISNI 0000 0001 0503 2798, Alder Hey Children’s Hospital NHS Foundation Trust, ; Eaton Road, Liverpool, UK
                [5 ]GRID grid.419319.7, ISNI 0000 0004 0641 2823, Nephrology Department, , Manchester Royal Infirmary, ; Oxford Road, Manchester, UK
                [6 ]NIHR Academic Clinical Fellow and Specialist Registrar in Rheumatology, Nuffield Department of Orthopaedics, Rheumatology and Musculoskeletal Sciences, Botnar Research Centre, Windmill Road, Oxford, OX3 7HD UK
                Author information
                http://orcid.org/0000-0003-4605-6598
                Article
                248
                10.1186/s41927-022-00248-0
                8951639
                35331328
                e193648d-a0b6-4179-a1be-8f9b165137eb
                © The Author(s) 2022

                Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

                History
                : 23 September 2021
                : 27 January 2022
                Categories
                Case Report
                Custom metadata
                © The Author(s) 2022

                bartonella endocarditis,aspergillus,culture-negative endocarditis,cryoglobulinemia,case report

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