Early post-transplant cutaneous bacillary angiomatosis in a kidney recipient: Case report and review of the literature.

Bacillary angiomatosis (BA) is an uncommon systemic disease caused by Bartonella henselae (BH) or Bartonella quintana (BQ) that occurs primarily in immunocompromised hosts. Few cases of BA recipients have been reported in adult solid transplant recipients over the years, with most cases presenting years after transplant. We describe a case of a kidney transplant recipient who developed cutaneous BA very early in the post-transplant period despite not having any exposures. Retrospective testing of donor and recipient's serum was performed and raised the concern for possible donor-derived infection. A literature review encompassing 1990 to present was also performed in order to better understand the clinical presentation, diagnostics and therapeutic approach of this unusual disease. Combined serology, histopathology and molecular testing (polymerase chain reaction [PCR]) were useful in diagnosing BA in our patient as serology alone might be unreliable. Macrolides or doxycycline for at least 3 months is the recommended therapeutic strategy; however, the optimal duration of treatment is not well established in transplant recipients. In our patient, we decided to use doxycycline for 1 year due to gradual resolution of lesions and ongoing immunosuppression. Patient responded successfully without any documented relapse.

[A cutaneous case of angiolymphoid hyperplasia with eosinophilia: Presentation of a rare entity]. / Un cas cutané d'hyperplasie angiolymphoïde avec éosinophilie : présentation d'une entité rare.

We report the case of a Caucasian 73-year-old woman, without medical history, who is operated of two lesions of the forehead and the scalp measuring 0.4 and 1cm. There is no adenopathy. Histopathological examination revealed a diffuse vascular proliferation of the dermis and the hypodermis composed of small vessels bordered of plump "epthelioid" endothelial cells. There is no mitotic figure. The stroma is slightly fibrous accompanied by a polymorphic inflammatory infiltrate rich in lymphocytes, sometimes arranged in lymphoid follicles, plasma cells and eosinophils. The proposed diagnosis is angiolymphoid hyperplasia with eosinophilia. This is a rare vascular tumor, benign according with the OMS classification and preferentially observed in the skin. We report a cutaneous case, clinical and histopathological data of the reported cases and present the main differential diagnoses.

Bartonella henselae transmission by blood transfusion in mice.

BACKGROUND: Bartonella spp. are neglected fastidious Gram-negative bacilli. We isolated Bartonella henselae from 1.2% of 500 studied blood donors and demonstrated that the bacteria remain viable in red blood cell units after 35 days of experimental infection. Now, we aim to evaluate the possibility of B. henselae transmission by blood transfusion in a mouse model. STUDY DESIGN AND METHODS: Eight BALB/c mice were intraperitoneal inoculated with a 30 µL of suspension with 10(4) CFU/mL of B. henselae and a second group of eight mice were inoculated with saline solution and used as control. After 96 hours of inoculation, the animals were euthanized. We collected blood and tissue samples from skin, liver, and spleen. Thirty microliters of blood from four Bartonella-inoculated animals were transfused into a new group (n = 4). Another group received blood from the control animals. B. henselae infection was investigated by conventional and nested polymerase chain reaction (PCR). RESULTS: Blood samples from all 24 mice were negative by molecular tests though half of the tissue samples were positive by nested PCR in the intraperitoneal Bartonella-investigated animals. Tissues from two of the four mice that received blood transfusions from Bartonella-inoculated animals were also nested PCR positives. CONCLUSIONS: Transmission of B. henselae by transfusion is possible in mice even when donor animals have undetectable bloodstream infection. The impact of human Bartonella sp. transmission through blood transfusion recipients must be evaluated.

Status Epilepticus Due to Cat Scratch Disease: Recognition, Diagnosis, and Thoughts on Pathogenesis.

Despite the publication of a number of case reports since the 1950s, physician awareness of the unique relationship between cat scratch disease (CSD) and acute encephalopathy remains limited. This report alerts emergency medicine physicians to include CSD encephalopathy (CSDE) in the differential diagnosis when a previously healthy child presents with status epilepticus. Prompt recognition of this relationship impacts the selection of initial diagnostic studies and antibiotic choices and permits reliable insight into prognosis. The 2 cases are from different eras and demonstrate the significant diagnostic advances in the past 3 decades for Bartonella henselae infection. Both children were treated with antibiotics, and both had resolution of all neurological symptoms. However, the role of antibiotics in the treatment and outcome of CSDE remains speculative. Lastly, the report suggests potential areas of investigation to address immune-mediated mechanisms in the pathogenesis of CSDE.

Infection with Bartonella henselae in a Danish family.

Bartonella species constitute emerging, vector-borne, intravascular pathogens that produce long-lasting bacteremia in reservoir-adapted (natural host or passive carrier of a microorganism) and opportunistic hosts. With the advent of more sensitive and specific diagnostic tests, there is evolving microbiological evidence supporting concurrent infection with one or more Bartonella spp. in more than one family member; however, the mode(s) of transmission to or among family members remains unclear. In this study, we provide molecular microbiological evidence of Bartonella henselae genotype San Antonio 2 (SA2) infection in four of six Danish family members, including a child who died of unknown causes at 14 months of age.

Concurrent Bartonella henselae infection in a dog with panniculitis and owner with ulcerated nodular skin lesions.

BACKGROUND: Bartonella henselae, a Gram-negative, zoonotic Alphaproteobacteria that infects erythrocytes, endothelial cells and dendritic cells, has previously been implicated as a cause of panniculitis in dogs and a human. ANIMAL AND OWNER: An 8-year-old, spayed female Labrador retriever and its 78-year-old male owner living in the same household. METHODS AND RESULTS: When preliminary and advanced testing failed to identify the cause of near-simultaneous-onset dermatological lesions, Bartonella serology, Bartonella Alphaproteobacteria growth medium (BAPGM) enrichment blood culture/PCR and immunohistochemistry were used to test specimens from the dog and owner. Bartonella henselae, genotype San Antonio 2 DNA was amplified and sequenced from the man's BAPGM enrichment blood culture and the dog's panniculitis lesion. The bacterium was visualized by immunohistochemistry in the dog's panniculitis lesion; however, neither the dog nor the owner was B. henselae seroreactive. Antibiotic therapy elicited dermatological improvement in both dog and owner. CONCLUSIONS AND CLINICAL IMPORTANCE: Bartonella henselae is an emerging zoonotic pathogen that induces granulomatous inflammatory lesions in various tissues of animals, including humans. We conclude that this bacterium had a contributory or causative role in the development of the dermatological lesions in the dog and owner.

Bilateral mandibular pyogranulomatous lymphadenitis and pulmonary nodules in a dog with Bartonella henselae bacteremia.

This report describes a 2-year-old collie dog with pulmonary nodules, visualized by computed tomographic (CT) scan, with evidence of Bartonella henselae bacteremia and pyogranulomatous lymphadenitis. Clinical signs resolved with antimicrobial therapy.

Lymphadénite pyogranulomateuse mandibulaire latérale et nodules pulmonaires chez un chien atteint de bactériémie àBartonella henselae. Ce rapport décrit un chien Collie âgé de 2 ans atteint de nodules pulmonaires, visualisés par tomodensitométrie, avec des signes de bactériémie à Bartonella henselae et de lymphadénite pyogranulomateuse. Les signes cliniques se sont résorbés avec un traitement antimicrobien.(Traduit par Isabelle Vallières).

Angiolymphoid hyperplasia with eosinophilia of the infra-axillary region: report of a case.

Angiolymphoid hyperplasia with eosinophilia is an uncommon, benign hyperproliferative disorder. Papules and nodules occur predominantly in the head and neck region. Involvement of other sites such as the trunk and mucosae has been rarely reported. We herein report a case of angiolymphoid hyperplasia with eosinophilia involving the right infra-axillary region.

An autochthonous case of cutaneous bacillary angiomatosis not related to major immunosuppression: An emerging or overlooked disease?

Cutaneous bacillary angiomatosis (cBA) is a vascular proliferative disorder due to Bartonella henselae or Bartonella quintana that has been mostly described in people living with HIV. Since cBA is considered to be rare in hosts not affected by major immunosuppression, it could be underdiagnosed in this population. Moreover, antimicrobial treatment of cBA has been poorly validated, thus reporting experiences on this clinical entity is important. We reported a challenging and well-characterized case of an Italian 67-year-old gentleman without a history of major immunocompromizing conditions, although he was affected by conditions that can be associated with impaired immune function. The patient reported herein was diagnosed after a long time since the initiation of symptoms and was successfully treated with combined antibiotic therapy including macrolides and quinolones under the guidance of molecular test results. Physicians should consider cBA as a possible manifestation of Bartonella spp. Infection in patients not suffering from major immunocompromizing conditions. Until evidence-based guidelines are available, molecular tests together with severity and extension of the disease can be useful to personalize the type of treatment and its duration.

Strategy for identification & characterization of Bartonella henselae with conventional & molecular methods.

BACKGROUND & OBJECTIVES: Bartonella henselae is a fastidious gram-negative bacterium usually causing self limiting infections in immunocompetent individuals but often causes potentially life threatening infection, such as bacillary angiomatosis in immunocompromised patients. Both diagnosis of infections and research into molecular mechanisms of pathogenesis have been hindered by lack of appropriate and reliable diagnostic techniques. We undertook this study to standardize methods to characterize B. henselae in clinical samples to diagnose Bartonella infection correctly. METHODS: B. henselae ATCC 49882 strain was procured from American type culture collection, USA. This strain was revived and maintained in the laboratory, and identification and characterization of this strain was done by conventional and molecular techniques, which included culture on various media, staining by different methods including electron microscopy, biochemical analysis by conventional methods and API, polymerase chain reaction (PCR) for amplification of citrate synthase gene followed by restriction fragment length polymorphism (RFLP). RESULTS: This organism was biochemically inert due to slow growth and generated unique identification code with API. The amplification of the citrate-synthase gene with primers yielded a 381 bp product followed by specific RFLP profile for B. henselae. INTERPRETATION & CONCLUSIONS: Bartonella is fastidious and fragile organism and should be handled carefully. Extra effort and careful observation are required to isolate and characterize this organism.

Cat scratch disease and other Bartonella infections.

First described in 1931, cat scratch disease remains the most commonly identified clinical syndrome associated with Bartonella infection. Over the last 20 years, however, the discovery and use of modern diagnostic tests has greatly expanded our understanding of the pathogenesis, clinical spectrum, and treatment options for Bartonella infections of all types. Indeed, each varies substantially depending on the infecting species and the immune status of the host.

Bacillary angiomatosis presenting as a pyogenic granuloma of the hand in an otherwise apparently healthy patient.

Bacillary angiomatosis is a rare opportunistic infection caused by the gram-negative bacteria Bartonella. The infection is strongly related to human immunodeficiency virus (HIV) infection, and hence, the diagnosis is usually considered based on the fact that the patient is HIV positive. We report on a case of bacillary angiomatosis presenting as a pyogenic granuloma of the hand in an otherwise apparently healthy man. The report is aimed to increase the awareness of hand surgeons that this serious disease may be the first clinical manifestation of HIV infection. The case also demonstrates that once the correct diagnosis is made, medical treatment alone may be sufficient to cure massive recurrent lesions.

Invasive bacillary angiomatosis in a kidney transplant recipient: A challenging case on belatacept immunosuppression.

Bacillary angiomatosis is a disseminated vascular proliferative disease caused by aerobic gram-negative bacilli Bartonella henselae or Bartonella quintana. Bacillary angiomatosis is mostly described in immunosuppressed patients with HIV infection and organ transplant recipients. We describe the case of a female aged 75 years who is a kidney transplant recipient who was admitted for a 3-month history of intermittent fever, chills, vomiting, and a 12-kg weight loss. The maintenance immunosuppression was based on prednisone, mycophenolate, and monthly infusions of belatacept. Physical examination was unremarkable. Laboratory investigations revealed elevated blood acute phase proteins but all blood cultures were negative. Serological tests for Bartonella were negative. Thoracoabdominal computed tomography scan and transesophageal echocardiography were normal. A Positron Emission Tomography scan showed a hypermetabolic mass in the duodenopancreatic region, with multiple hepatic and splenic lesions. Histological findings of spleen and pancreatic biopsies were not conclusive. The histopathological examination of a celiac lymph node biopsy finally demonstrated bacillary angiomatosis. The diagnosis of bacillary angiomatosis in immunocompromised patients is most often delayed in the absence of skin involvement. A high index of clinical suspicion is needed when interpreting negative results.

Cutaneous bacillary angiomatosis in renal transplant recipients: report of three new cases and literature review.

Bacillary angiomatosis (BA) is a rare vasculoproliferative disorder due to Bartonella henselae (BH) or Bartonella quintana. It can involve many organs, including the skin, and has been mainly reported in patients with acquired immunodeficiency syndrome. In organ transplant recipients (OTR), this disorder remains misdiagnosed and therapeutic guidelines are nonexistent. We report 3 cases of BA with skin involvement in OTR and review similar cases from the literature. BA manifests on the skin with violaceous lesions mimicking Kaposi sarcoma, and is associated with fever, lymphadenopathy, and liver, spleen, or lung nodules. Bartonellosis infections in OTR are due to BH, the agent causing cat-scratch disease (CSD), but BA comprises histologically a prominent vascular proliferation, which is usually lacking in CSD. Cultures and serologic tests are poorly reliable for the diagnosis, which relies on demonstration of BH within the lesions. A history of cat exposure exists in most cases and pediatric OTR are at higher risk. Prevention consists of regular use of a flea-control product in cats and prompt cleaning of scratches. Our cases highlight several original features of this rare condition, which could potentially improve the management of BA in OTR.

Bartonella henselae-mediated disease in solid organ transplant recipients: two pediatric cases and a literature review.

Bartonella henselae, the etiologic agent of cat-scratch disease, causes a well-defined, self-limited syndrome of fever and regional lymphadenopathy in immunocompetent hosts. In immunocompromised hosts, however, B. henselae can cause severe disseminated disease and pathologic vasoproliferation known as bacillary angiomatosis (BA) or bacillary peliosis. BA was first recognized in patients infected with human immunodeficiency virus. It has become more frequently recognized in solid organ transplant (SOT) recipients, but reports of pediatric cases remain rare. Our review of the literature revealed only one previously reported case of BA in a pediatric SOT recipient. We herein present 2 pediatric cases, one of which is the first reported case of BA in a pediatric cardiac transplant recipient, to our knowledge. In addition, we review and summarize the literature pertaining to all cases of B. henselae-mediated disease in SOT recipients.

A case of multiple epithelioid angiomatous nodules.

Cutaneous epithelioid angiomatous nodule (CEAN) is a distinct type of epithelioid vascular tumor that is usually solitary. Herein we present a 31-year-old man with multiple, rapidly growing nodules on the scalp.

Detection of Bartonella vinsonii subsp. berkhoffii in an HIV patient using metagenomic Next-Generation Sequencing.

Bartonella species are fastidious, aerobic bacteria that are transmitted by blood-sucking arthropods. Bartonella spp. are responsible for cat scratch disease, Carrion's disease, bacillary angiomatosis and trench fever. On the other hand, Bartonella vinsonii is rarely reported in the literature and there exist a few reports of systemic infection caused by Bartonella vinsonii in patients with acquired immunodeficiency syndrome. A 31-year-old male (diagnosed with AIDS six years ago) had persistent fever and ulceration in the right knee. The elevated levels of inflammatory markers suggested an infectious aetiology. Despite the negative findings of blood culture, metagenomic Next-Generation Sequencing of plasma detected Bartonella vinsonii. The polymerase chain reaction of whole blood and Sanger sequencing confirmed the mNGS findings. Immunohistochemical staining had later suggested bacillary angiomatosis, which was consistent with Bartonella infection. Following antibiotic treatment, the ulcers subsided significantly, but a high fever persisted. The patient died due to sudden respiratory failure.