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.

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.

Bacillary Angiomatosis in Renal Transplant Recipient: A Case Report.

INTRODUCTION: Bacillary angiomatosis (BA) is a rare, opportunistic infectious disease caused by the aerobic Gram-negative bacilli Bartonella henselae or Bartonella quintana. The main reservoir for those microbes are cats. The disease mostly affects immunocompromised patients with human immunodeficiency virus infection, after organ transplantation, undergoing corticosteroid and methotrexate therapy or with oncological history. CASE REPORT: We represent the case of a 65-year-old man who reported to the Department of Dermatology with a high fever and numerous nodular skin lesions on the 5th month of kidney transplantation. At that time, his immunosuppressive therapy consisted of tacrolimus 6 mg/day, mycophenolate mofetil 2 g/day, and prednisone 5 mg/day. Laboratory tests revealed an increased leukocyte count and elevated values of acute-phase proteins, but blood cultures were negative. Skin biopsy was performed and BA was diagnosed. The patient was given oral doxycycline 100 mg twice a day. During antibiotic therapy, his body temperature normalized and skin lesions began to resolve. The patient continued the above treatment for the next 3 months with good tolerance, and no relapse occurred in 1 year. CONCLUSION: BA should be listed among possible opportunistic infections in organ transplant recipients.

Scalp eschar and neck lymphadenopathy after tick bite (SENLAT) caused by Bartonella henselae in Korea: a case report.

BACKGROUND: Tick-borne lymphadenopathy (TIBOLA) is an infectious disease, mainly caused by species from the spotted fever group rickettsiae and is characterized by enlarged lymph nodes following a tick bite. Among cases of TIBOLA, a case of scalp eschar and neck lymphadenopathy after tick bite (SENLAT) is diagnosed when an eschar is present on the scalp, accompanied by peripheral lymphadenopathy (LAP). Only a few cases of SENLAT caused by Bartonella henselae have been reported. CASE PRESENTATION: A 58-year-old male sought medical advice while suffering from high fever and diarrhea. Three weeks before the visit, he had been hunting a water deer, and upon bringing the deer home discovered a tick on his scalp area. Symptoms occurred one week after hunting, and a lump was palpated on the right neck area 6 days after the onset of symptoms. Physical examination upon presentation confirmed an eschar-like lesion on the right scalp area, and cervical palpation revealed that the lymph nodes on the right side were non-painful and enlarged at 2.5 × 1.5 cm. Fine needle aspiration of the enlarged lymph nodes was performed, and results of nested PCR for the Bartonella internal transcribed spacer (ITS) confirmed B. henselae as the causative agent. CONCLUSION: With an isolated case of SENLAT and a confirmation of B. henselae in Korea, it is pertinent to raise awareness to physicians in other Asian countries that B. henselae could be a causative agent for SENLAT.

Bacillary angiomatosis with bone invasion

Abstract Bacillary angiomatosis is an infection determined by Bartonella henselae and B. quintana, rare and prevalent in patients with acquired immunodeficiency syndrome. We describe a case of a patient with AIDS and TCD4+ cells equal to 9/mm3, showing reddish-violet papular and nodular lesions, disseminated over the skin, most on the back of the right hand and third finger, with osteolysis of the distal phalanx observed by radiography. The findings of vascular proliferation with presence of bacilli, on the histopathological examination of the skin and bone lesions, led to the diagnosis of bacillary angiomatosis. Corroborating the literature, in the present case the infection affected a young man (29 years old) with advanced immunosuppression and clinical and histological lesions compatible with the diagnosis.

Bacillary angiomatosis with bone invasion.

Bacillary angiomatosis is an infection determined by Bartonella henselae and B. quintana, rare and prevalent in patients with acquired immunodeficiency syndrome. We describe a case of a patient with AIDS and TCD4+ cells equal to 9/mm3, showing reddish-violet papular and nodular lesions, disseminated over the skin, most on the back of the right hand and third finger, with osteolysis of the distal phalanx observed by radiography. The findings of vascular proliferation with presence of bacilli, on the histopathological examination of the skin and bone lesions, led to the diagnosis of bacillary angiomatosis. Corroborating the literature, in the present case the infection affected a young man (29 years old) with advanced immunosuppression and clinical and histological lesions compatible with the diagnosis.

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.

The efficacy of a selamectin (Stronghold ®) spot on treatment in the prevention of Bartonella henselae transmission by Ctenocephalides felis in cats, using a new high-challenge model.

Bartonella henselae is the causative agent of cat scratch disease in humans, which is recognized as an emerging zoonotic disease. Ctenocephalides felis is the main vector, and transmission of B. henselae infection between cats and humans occurs mainly through infected flea feces. Control of feline infestation with this arthropod vector therefore provides an important strategy for the prevention of infection of both humans and cats. In the present study, a new challenge model is used to evaluate the efficacy of selamectin (Stronghold(®) spot on) in the prevention of B. henselae transmission by C. felis. In this new challenge model, domestic cats were infected by direct application of B. henselae-positive fleas. The fleas used for infestation were infected by feeding on blood that contained in vitro-cultured B. henselae. The direct application of the fleas to the animals and the use of different B. henselae strains ensured a high and consistent challenge. Two groups of six cats were randomly allocated on pre-treatment flea counts to either control (untreated cats) or the selamectin-treated group with one pipette per cat according to the label instruction. Stronghold (selamectin 6 % spot on solution) was administered on days 0 and 32. On days 3, 10, 19, 25, and 31, each cat was infested by direct application of 20 fleas that fed on blood inoculated with B. henselae. Polymerase chain reaction (PCR) on pooled fleas confirmed that the fleas were infected. Blood samples were collected from each cat on days -3 (prior to flea infestation and treatment), 9, 17, 24, 30, 37, and 44 and assayed for B. henselae antibodies using an indirect immunofluorescence (IFA), for the presence of bacteria by bacterial culture and for B. henselae DNA presence by PCR. Cats were also assessed on a daily basis for general health. There were no abnormal health observations during the study and none of the animals required concomitant treatment. None of the cats displayed any clinical signs of bartonellosis during the study. In the untreated group, all cats became bacteremic within 17 to 44 days. None of the selamectin-treated cats became positive during the study. It was concluded that Stronghold(®) spot on administered to cats was efficacious in the prevention of the transmission of B. henselae by fleas to cats in a high-challenge model.

Pathogenicity and treatment of Bartonella infections.

Bartonella spp. are responsible for emerging and re-emerging diseases around the world. The majority of human infections are caused by Bartonella henselae, Bartonella quintana and Bartonella bacilliformis, although other Bartonella spp. have also been associated with clinical manifestations in humans. The severity of Bartonella infection correlates with the patient's immune status. Clinical manifestations can range from benign and self-limited to severe and life-threatening disease. Clinical conditions associated with Bartonella spp. include local lymphadenopathy, bacteraemia, endocarditis, and tissue colonisation resulting in bacillary angiomatosis and peliosis hepatis. Without treatment, Bartonella infection can cause high mortality. To date, no single treatment is effective for all Bartonella-associated diseases. In the absence of systematic reviews, treatment decisions for Bartonella infections are based on case reports that test a limited number of patients. Antibiotics do not significantly affect the cure rate in patients with Bartonella lymphadenopathy. Patients with Bartonella spp. bacteraemia should be treated with gentamicin and doxycycline, but chloramphenicol has been proposed for the treatment of B. bacilliformis bacteraemia. Gentamicin in combination with doxycycline is considered the best treatment regimen for endocarditis, and erythromycin is the first-line antibiotic therapy for the treatment of angioproliferative lesions. Rifampicin or streptomycin can be used to treat verruga peruana. In this review, we present recent data and recommendations related to the treatment of Bartonella infections based on the pathogenicity of Bartonella spp.

Bacillary angiomatosis in a HIV-positive patient with poor adherence to antiretroviral therapy.

Bacillary angiomatosis is a systemic disease caused by Bartonella (B.) henselae and B. quintana. Today it is a rare disease that occurs predominantly in patients with poor adherence to antiretroviral therapy or with late diagnosis of human immunodeficiency virus (HIV). We report on the case of a 40-year-old Caucasian female with HIV-1 and hepatitis B virus (HBV) co-infection diagnosed 17 years ago. She presented to the emergency department with an erythematous, painless nodule located on the left naso-genian fold. In the next few weeks the disease disseminated to the oral and left tarsal mucosa and to the palm of the left hand. The histopathological findings were suggestive of bacillary angiomatosis which was confirmed by polymerase chain reaction (PCR). The patient was treated with clarithromycin 500 mg bid per os for 3 months, with complete remission of the mucocutaneous lesions. Bacillary angiomatosis is a potentially fatal disease. Early diagnosis and treatment are critical in reducing the morbidity and mortality associated with it.

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.

Angiomatosis bacilar por Bartonella quintana como primera manifestación de infección por VIH: Report of one case / Bacillary angiomatosis

Background: Bacillary angiomatosis is an unusual infectious disease, with angioproliferative lesions, typical of immunocompromised patients. It is caused by Bartonella quintana and Bartonella henselae, two infectious agents of the genus Bartonella, which trigger variable clinical manifestations, including cutaneous vascular and purpuric lesions, and regional lymphadenopathy, and even a systemic disease with visceral involvement. We report a 38-year-old HIV positive male presenting with a history of six months of cutaneous growing purple angiomatous lesions, located also in nasal fossae, rhi-nopharynx and larynx. The skin biopsy was compatible with bacillary angiomatosis. Polymerase chain reaction of a tissue sample showed homology with B. quintana strain Toulouse. The patient was treated with azithromycin and ciprofloxacin with a favorable evolution.