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.

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.

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.

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.

Cutaneous Bacillary Angiomatosis in a Renal Transplant Patient.

A 37-year-old man with a history of renal transplantation in 2013 due to focal segmental glomerulosclerosis presented to the emergency room with a 2-week history of fever, chills, anorexia, weight loss, abdominal pain, diarrhea, and a new asymptomatic lesion on the right side of the neck. The patient worked as a truck driver and frequently traveled to Wisconsin; he had not traveled internationally in the past year. He lived with his brother who had a pet cat. He was compliant with his anti-rejection medication regimen, which included mycophenolate mofetil, tacrolimus, and prednisone. Physical examination of the neck revealed an 8-mm exophytic, friable, red papule with overlying blood crusts (Figure 1). The remainder of the mucocutaneous examination was unremarkable, and there was no palpable lymphadenopathy. The patient was started on empiric intravenous cefepime and metronidazole and admitted to the hospital for further management. A punch biopsy of the lesion was performed.

Kaposi Sarcoma With Intravascular Primary Effusion Lymphoma in the Skin: A Potential Pitfall in HHV8 Immunohistochemistry Interpretation.

Primary effusion lymphoma is a rare, clinically aggressive large B-cell neoplasm universally associated with human herpesvirus 8 that occurs in the setting of immune compromise. It is classically described as a lymphomatous effusion occurring within body cavities. Recently, however, solid tumor masses, and rarely an intravascular form, have been described. We report a case of a cutaneous intravascular primary effusion lymphoma occurring within ectatic vascular spaces of a Kaposi sarcoma skin lesion in a human immunodeficiency virus-positive adult. Human herpesvirus 8 immunohistochemistry was positive in the nuclei of the Kaposi sarcoma spindled cells as well as within large intravascular plasmacytoid cells. This unusual case highlights the importance of careful assessment of the nature of human herpesvirus 8-positive staining cells in an otherwise typical Kaposi sarcoma. A careful search for dual pathology in immune-compromised patients as well as the importance of histologic assessment of skin lesions in human immunodeficiency virus-positive patients is also highlighted.

[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 in canine cavitary effusions: prevalence, identification, and clinical associations.

BACKGROUND: Previous reports suggest an association between Bartonella infection and effusions in dogs and human beings. OBJECTIVES: The aims of this study were to determine the prevalence of Bartonella infection in canine effusions and to investigate historic and clinical parameters predictive of Bartonella in dogs with effusions. METHODS: Canine cavitary effusions submitted for analysis and, if available, paired EDTA blood, were screened for Bartonella infection using the Bartonella α-proteobacteria growth medium enrichment culture/PCR diagnostic platform (Bartonella enrichment PCR or ePCR) at Galaxy Diagnostics, Inc. RESULTS: Bartonella henselaeDNA was PCR-amplified and sequenced from 15% (12/80) of sampled dogs. Enrichment culture prior to PCR testing was required for Bartonella detection in 92% (11/12) of cases. Twenty percent (4/20), 13% (8/60), and 0% (0/4) of dogs with pleural, peritoneal, and pericardial effusions, respectively, tested positive. Bartonella henselae was detected most frequently in the fall, and young and middle-aged dogs appeared to be overrepresented. Golden Retrievers and Yorkshire/Silky Terriers each comprised 25% of infected dogs (odds ratio 3.4 for Golden Retrievers). There was a weak association with hemorrhagic effusions. Fifty percent of Bartonella-positive dogs had hemorrhage as a component of their effusion compared to 37% of PCR-negative dogs (odds ratio 1.7). CONCLUSIONS: Viable B henselae organisms occur in pleural and peritoneal effusions of dogs; the clinical relevance of which remains unclear and may represent opportunistic infection. Associations found in this study included seasonal variation, age, breed, and site of effusion.

Similar presentation of cervical lymphadenitis of different etiology in two siblings.

Here, we report chronic suppurating submandibular swelling occurring in two siblings, with one case caused by nontuberculous mycobacteria and the other caused by Bartonella henselae. These two infections share a similar clinical presentation, but the treatment modalities differed.

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.

Angiomiolipoma epitelioide hepático: correlación clínico-patológica en una serie de 4 casos / Epithelioid angiomyolipoma of the liver: clinicopathological correlation in a series of 4 cases

El angiomiolipoma hepático es una neoplasia rara que puede llegar a ser difícil de diagnosticar en casos de biopsia ecoguiada. Hemos estudiado 4 casos de pacientes de sexo femenino, con una media de edad de 51 años. Ninguno de los pacientes se ha presentado con síntomas abdominales específicos, ni otras masas tumorales detectadas incidentalmente. Uno de ellos tenía antecedentes personales relevantes: carcinoma de células renales. Este mismo paciente fue diagnosticado de un angiomiolipoma renal contralateral. Ninguno de los pacientes ha mostrado evidencia de padecer esclerosis tuberosa. Tres de los tumores han sido diagnosticados por biopsia y en sólo uno de los pacientes se ha decidido la extirpación quirúrgica completa del tumor. En ninguno de los pacientes se ha observado evidencia de recidiva de la enfermedad o aumento del tamaño tumoral durante un periodo medio de 45 meses. Histológicamente los tumores se han subclasificado en angiomiolipomas epitelioides. En el 50% de los casos se ha observado hematopoyesis extramedular. La primera impresión diagnóstica mediante métodos de imagen incluía: hiperplasia nodular focal, adenoma hepático, carcinoma hepatocelular y metástasis. Mediante técnicas de inmunohistoquímica se ha demostrado que todos los tumores expresaban marcadores melanocíticos (HMB45 y Melan A) y marcadores de células musculares lisas (actina de músculo liso). El diagnóstico de estos tumores está aumentando debido a los programas de detección precoz de carcinoma hepatocelular en pacientes con cirrosis. Hay que tener en cuenta su amplio espectro de diversidad morfológica para evitar el diagnóstico incorrecto de una neoplasia maligna (AU)

Hepatic angiomyolipoma is a rare neoplasm that can be difficult to diagnose in cases of ecoguide biopsy. We studied 4 cases of female patients with a mean age of 51 years. None of the patients presented specific abdominal symptoms, or other tumour masses detected by chance. One of them had relevant personal history: Renal cell carcinoma. This same patient was diagnosed with a contralateral renal angiomyolipoma. None of the patients showed evidence of tuberous sclerosis. Three tumours have been diagnosed by biopsy and only in one patient was decided to completely remove the tumour surgically. None of the patients had evidence of recurrence of disease or an increase in tumour size over an average period of 45 months. Histologically, the tumours have been sub classified into angiomyolipomas epithelioid. In 50% of the cases, extramedullary haematopoiesis was observed. The first diagnostic impression using imaging methods included: Focal nodular hyperplasia, hepatocellular adenoma, hepatocellular carcinoma and metastasis. By immunohistochemistry, it has been demonstrated that all tumours expressing melanocytic markers (HMB45 and Melan A) and markers of smooth muscle cells (smooth muscle actin). The diagnosis of these tumours is increasing due to programmes for early detection of hepatocellular carcinoma in patients with liver cirrhosis. It must be taken into account their broad spectrum of morphological diversity to avoid incorrect diagnosis of a malignant neoplasm (AU)

[An exceptional case of tricuspid infective endocarditis due to Bartonella henseale revealed by an old pulmonary embolism]. / Un cas exceptionnel d'endocardite infectieuse tricuspide à Bartonella henselae révélée par une embolie pulmonaire « vieillie ¼.

We report a case of blood culture-negative tricuspid infective endocarditis revealed after tick bite by repeated pulmonary infection during one year due to septic pulmonary emboli in a 67-year-old farmer woman. Tricuspid vegetation and pulmonary emboli are calcified. Lyme serology is negative. Serologic test and PCR analysis are positive to Bartonella henselae. The evolution is favorable after antibiotic and anticoagulant treatment. Infective endocarditis due to B. henselae is an exceptional complication of cat scratch disease. You have to think about in case of blood culture-negative endocarditis with calcified valvular lesions even without cat bite, tick seems to be vector of the bacteria.