We present a 63-year-old man treated with alemtuzumab for chronic lymphocytic leukemia who developed multiple angiomatous papules and fever. Real-time polymerase chain reaction (RT-PCR) from a skin lesion and blood sample revealed Bartonella quintana as causative agent confirming the diagnosis of bacillary angiomatosis with bacteremia. Treatment with doxycycline, initially in combination with gentamicin, led to complete resolution of the lesions. This case shows the importance of considering bacillary angiomatosis as a rare differential diagnosis of angiomatous lesions in the immunocompromised patient, particularly in chronic lymphocytic leukemia and following lymphocyte depleting treatments as alemtuzumab. 1. Introduction Bacillary angiomatosis is a rare vasculoproliferative disorder due to Bartonella henselae or Bartonella quintana. The disease usually manifests as cutaneous angioma-like tumour. Lesions may be solitary or multiple and dissemination to visceral organs can occur. Bone lesions and subcutaneous masses are associated with B. quintana, whereas peliosis hepatis and lymph node lesions are associated with B. henselae [1]. Cases were usually described in HIV infected individuals. Although less common, the infection was reported in immunocompetent patients and in otherwise immunocompromised conditions such as solid organ transplantation and oncology patients, particularly in chronic lymphocytic leukemia [2, 3]. Because of its potentially life-threatening course, early diagnosis and adequate treatment are crucial. To our knowledge, we describe the first case of bacillary angiomatosis and bacteremia due to B. quintana in a patient with chronic lymphocytic leukemia. 2. Case Presentation A 63-year-old man was admitted for evaluation of multiple nonpruritic skin lesions that had been present for 1 month on his arms, legs, trunk, and face. His medical history was significant for chronic lymphocytic leukemia with longstanding profound neutropenia, anemia, and thrombocytopenia. He had previously been treated with chlorambucil and prednisone, as well as cladribine, rituximab, and bendamustine. At the time of presentation, he had been receiving alemtuzumab for 4 months. Current medication included prophylaxis with trimethoprim sulfamethoxazole as well as valacyclovir, and treatment with voriconazole for probable invasive pulmonary aspergillosis diagnosed during a previous febrile neutropenic episode. There had been several other episodes of neutropenic fever without specific infectious focus. Physical examination showed multiple nontender cutaneous
References
[1]
C. Foucault, P. Brouqui, and D. Raoult, “Bartonella quintana characteristics and clinical management,” Emerging Infectious Diseases, vol. 12, no. 2, pp. 217–223, 2006.
[2]
C. Moulin, J. Kanitakis, C. Ranchin, et al., “Cutaneous bacillary angiomatosis in renal transplant recipients: report of three new cases and literature review,” Transplant Infectious Disease, vol. 14, pp. 403–409, 2012.
[3]
K. Petersen, K. C. Earhart, and M. R. Wallace, “Bacillary angiomatosis in a patient with chronic lymphocytic leukemia,” Infection, vol. 36, no. 5, pp. 480–484, 2008.
[4]
A. Sander, M. Posselt, N. B？hm, M. Ruess, and M. Altwegg, “Detection of Bartonella henselae DNA by two different PCR assays and determination of the genotypes of strains involved in histologically defined cat scratch disease,” Journal of Clinical Microbiology, vol. 37, no. 4, pp. 993–997, 1999.
[5]
D. Goldenberger, R. Zbinden, I. Perschil, and M. Altwegg, “Nachweis von Bartonella (Rochalimaea) henselae/B. quintana mittels Polymerasekettenreaktion (PCR),” Schweizerische Medizinische Wochenschrift, vol. 126, no. 6, pp. 207–213, 1996.
[6]
B. La Scola and D. Raoult, “Culture of Bartonella quintana and Bartonella henselae from human samples: a 5-year experience (1993 to 1998),” Journal of Clinical Microbiology, vol. 37, no. 6, pp. 1899–1905, 1999.
[7]
J. E. Koehler, M. A. Sanchez, C. S. Garrido et al., “Molecular epidemiology of Bartonella infections in patients with bacillary angiomatosis-peliosis,” The New England Journal of Medicine, vol. 337, no. 26, pp. 1876–1883, 1997.
[8]
P. Brouqui, B. La Scola, V. Roux, and D. Raoult, “Chronic Bartonella quintana bacteremia in homeless patients,” The New England Journal of Medicine, vol. 340, no. 3, pp. 184–189, 1999.
[9]
V. Jacomo, P. J. Kelly, and D. Raoult, “Natural history of Bartonella infections (an exception to Koch's postulate),” Clinical and Diagnostic Laboratory Immunology, vol. 9, no. 1, pp. 8–18, 2002.
[10]
L. T？r？k, S. Z. Virágh, I. Borka, and M. Tápai, “Bacillary angiomatosis in a patient with lymphocytic leukaemia,” British Journal of Dermatology, vol. 130, pp. 665–668, 1994.
[11]
P. Milde, M. Brunner, F. Borchard et al., “Cutaneous bacillary angiomatosis in a patient with chronic lymphocytic leukemia,” Archives of Dermatology, vol. 131, no. 8, pp. 933–936, 1995.
[12]
D. Lange, C. Oeder, K. Waltermann et al., “Bacillary angiomatosis,” Journal of the German Society of Dermatology, vol. 7, no. 9, pp. 767–769, 2009.
[13]
N. E. Holmes, S. Opat, A. Kelman, and T. M. Korman, “Refractory Bartonella quintana bacillary angiomatosis following chemotherapy for chronic lymphocytic leukaemia,” Journal of Medical Microbiology, vol. 60, no. 1, pp. 142–146, 2011.
[14]
S. Gasquet, M. Maurin, P. Brouqui, H. Lepidi, and D. Raoult, “Bacillary angiomatosis in immunocompromised patients,” AIDS, vol. 12, no. 14, pp. 1793–1803, 1998.
[15]
J. M. Rolain, M. Maurin, M. N. Mallet, D. Parzy, and D. Raoult, “Culture and antibiotic susceptibility of Bartonella quintana in human erythrocytes,” Antimicrobial Agents and Chemotherapy, vol. 47, no. 2, pp. 614–619, 2003.
[16]
J. M. Rolain, P. Brouqui, J. E. Koehler, C. Maguina, M. J. Dolan, and D. Raoult, “Recommendations for treatment of human infections caused by Bartonella species,” Antimicrobial Agents and Chemotherapy, vol. 48, no. 6, pp. 1921–1933, 2004.
