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CASE REPORT
Year : 2010  |  Volume : 76  |  Issue : 6  |  Page : 682-685

Bacillary angiomatosis with atypical clinical presentation in an immunocompetent patient


1 Department of Dermatology, Hospitales Universitarios Virgen del Rocío, Sevilla, Spain
2 Department of Infectious Diseases, Hospitales Universitarios Virgen del Rocío, Sevilla, Spain
3 Department of Pathology, Hospitales Universitarios Virgen del Rocío, Sevilla, Spain
4 Department of AIDS and Viral Hepatitis Study Group, Hospitales Universitarios Virgen del Rocío, Sevilla, Spain

Date of Web Publication12-Nov-2010

Correspondence Address:
J Bernabeu-Wittel
Dermatology Department, Hospitales Universitarios Virgen del Rocío, Avenida Manuel Siurot s/n., 41013 Sevilla
Spain
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DOI: 10.4103/0378-6323.72469

PMID: 21079313

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  Abstract 

Bacillary angiomatosis is a recently described infectious disease that usually affects immunosupressed hosts with a previous history of contact with cats. We report a rare case of bacillary angiomatosis in an immunocompetent 59-year-old woman with no history of previous exposure to cats, and atypical clinical features (fever and subcutaneous nodules with ulceration on the left ankle). Histopathology of the lesion showed extensive ulceration and reactive tumor-like vascular proliferation of the blood vessels with swollen endothelial cells and an inflammatory infiltrate including neutrophils and lymphocytes in the dermis and subcutis. Staining with the Warthin-Starry method demonstrated the presence of clustered bacilli located in the extracellular matrix adjacent to the proliferating endothelial cells. Diagnosis was confirmed with the detection of Bartonella spp. DNA in the affected skin and in bone marrow using polymerase chain reaction.


Keywords: Bacillary angiomatosis, bartonella quintana, bartonella henselae, immunocompetent


How to cite this article:
Bernabeu-Wittel J, Luque R, Corbí R, Mantrana-Bermejo M, Navarrete M, Vallejo A, Bernabeu-Wittel M. Bacillary angiomatosis with atypical clinical presentation in an immunocompetent patient. Indian J Dermatol Venereol Leprol 2010;76:682-5

How to cite this URL:
Bernabeu-Wittel J, Luque R, Corbí R, Mantrana-Bermejo M, Navarrete M, Vallejo A, Bernabeu-Wittel M. Bacillary angiomatosis with atypical clinical presentation in an immunocompetent patient. Indian J Dermatol Venereol Leprol [serial online] 2010 [cited 2014 Sep 2];76:682-5. Available from: http://www.ijdvl.com/text.asp?2010/76/6/682/72469



  Introduction Top


The spectrum of Bartonella infections has expanded rapidly since the first HIV-infected patient with bacillary angiomatosis (BA) was described in 1983. Of the 19 species of Bartonella described until now, only 10 were acknowledged as human pathogens; B. bacilliformis, B. quintana, and B. henselae are the most frequently described. [1],[2] BA usually affects immunosuppressed hosts, especially HIV-infected patients with a decrease in the CD4 lymphocyte count. [1],[2],[3],[4],[5],[6] Hepatic peliosis is exclusively associated with B. henselae infection. BA caused by B. quintana can develop lytic bone lesions and subcutaneous masses. [2]

There are very few reported cases of BA in immunocompetent patients. [3],[4],[5],[6],[7],[8],[9],[10],[11] The skin is the most frequently affected site, presenting as papules, warts, pedunculated and subcutaneous nodules (rarely ulcerated or bleeding), or hyperkeratotic plaques. [1],[2] Most patients with BA have been exposed to cats. [1],[2],[3],[4],[5],[12],[13]

We describe an atypical case of BA in an immunocompetent woman with no previous exposure to cats.


  Case Report Top


A 59-year-old woman was admitted to our hospital with a four-week history of a painful skin lesion localized on the backside of the left ankle. The first two days she had suffered a high grade fever which remitted spontaneously. However, cutaneous lesions progressed despite empiric treatment with amoxicillin plus clavulanate. The patient denied having had direct contact with cats or wild animals, had no previous illness and received no medication. Physical examination revealed a lobulated and ulcerated plaque with an easily bleeding surface. Slight swelling of the left calf was evident [Figure 1]a. No lice infestation was detected.
Figure 1 :(a) Before treatment: subcutaneous nodules with ulceration and an easily bleeding surface on the backside of the left ankle. (b) After treatment: Healed plaque with residual hyperpigmentation

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Blood count, CD4 count, blood chemistry, urine analysis, erythrocyte sedimentation rate, X-ray and abdominal ultrasonography were normal. Doppler-ultrasonography of the left leg showed internal saphenous vein thrombosis at the level of the knee. Two sets of blood cultures, as well as a skin-biopsy culture (processed for aerobic and anaerobic bacteria, fungi, and mycobacteria), were negative. Serotesting for HIV by enzyme-linked immunoassay was negative. Serology, using an indirect immunofluorescence assay (IFA), detected an antibody titer of 1:64 (IgG) against B. henselae. IgM against B. henselae was negative. Serotesting (IFA) against B. quintana, C. burnetii, and Chlamydia were negative.

Histopathological examination of the skin lesion showed extensive ulceration, and a tumor-like vascular proliferation of the blood-vessels with swollen endothelial cells in the dermis and subcutis, predominating in the upper dermis. The dermis and subcutis showed an inflammatory infiltrate including neutrophils, and lymphocytes intermingled with the blood vessels [Figure 2]. Staining with the Warthin-Starry method demonstrated the presence of clustered bacilli located in the extracellular matrix adjacent to the proliferating endothelial cells [Figure 3]. Staining for fungi, cytomegalovirus and mycobacteria were negative.
Figure 2 :Tumor-like lobular proliferation of the blood-vessels in the dermis with swollen endothelial cells (black arrows). Inflammatory infiltrate (white arrows) including neutrophils, and lymphocytes intermingled with the blood vessels (H and E, ×40)

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Figure 3 :Solitary or clustered bacilli (arrow) located in the extracellular matrix adjacent to the proliferating endothelial cells (Warthin-Starry stain, ×100)

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Bartonella spp. DNA was detected in the skin lesion and bone marrow by polymerase chain reaction (PCR) [Figure 4]. DNA was purified from bone marrow and epihtelial cells by a DNA purification kit (Qiagen). PCR was performed using 1 μg DNA over 35 cycles consisting of 15 s at 95°C, 15 s at 50° and 45 s at 72°C in a reaction volume of 50 μl containing 0.5 units Taq polymerase, 10 mM Tris HCl pH 8.8, 50 mM KCl, 1.5 mM MgCl 2 , 0,1 % Triton X-100, 33 μM of each dNTPs, and 0.5 μM of each primer. Primers P24E 5΄-CCT CCT TCA GTT AGG CTG G-3' and P12B 5΄-GAG ATG GGT TTT GGA GAG TA-3' were used as specific primers. DNA from B. henselae and B. quintana was used as control for discrimination purposes.
Figure 4 :Detection of Bartonella spp. by polymerase chain reaction. Lanes from left to right: 1-4, patient's skin DNA; 5-8, patient's bone marrow DNA; 9, Bartonella spp. DNA; 10, negative control; 11, molecular size marker

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Oral doxycycline (100 mg/12 h) was given for two months. The lesion regressed leaving a hyperpigmented plaque [Figure 1]b. In a follow-up period of 12 months, there was no recurrence. The absence of recurrence was demonstrated with a second histopathological study of the hyperpigmented plaque.


  Discussion Top


Clinical presentation of BA as subcutaneous nodules with ulceraction in an immunocompetent host is unusual. [8] It is generally accepted that human infection occurs directly or by an arthropod vector (Ctenocephallides felis for B. henselae, and still unknown for B. quintana). However, one third of the patients with BA deny having had any previous contact with cats. [1],[2],[12],[13],[14],[15],[16] The absence of direct contact with cats and the atypical clinical presentation made the diagnosis especially difficult in this case. Clinical differential diagnosis included infections with sporotrichoid dissemination (nocardiosis, mycobacteria, and sporothrix among others) and pyogenic granulomas. Histopathological features in our patient [Figure 2] distinguished BA from Kaposi's sarcoma (lack of spindle cells, atypicallly shaped vascular channels and hyaline globules) and pyogenic granuloma (endothelial cells were large and polygonal, with a prominent inflammatory infiltrate with polymorphonuclear leukocytes scattered throughout the lesion, as opposed to classic pyogenic granuloma in which polymorphonuclear leukocytes are near the surface). Histopathological findings, serological results, the regression of the lesion with oral doxycycline, and the detection of Bartonella spp DNA in the skin lesion and bone marrow all confirmed the diagnosis of BA in our patient.

The culture of the skin lesion processed for aerobic and anaerobic bacteria, fungi, and mycobacteria was negative. Culture in blood agar (5% defibrinated rabbit blood) incubated at 37C in 5% CO 2 could not be done due to lack of laboratory facility. However, the vast majority of BA cases are diagnosed histologically, with identification of the causative bacteria by Warthin-Starry staining. Bartonella spp. is an extremely fastidious Gram negative rod, and requires special culture conditions for isolation; hence culture is not routinely performed for diagnosis. [1],[2] Serological tests, using an indirect immunofluorescence assay (IFA), show relatively high specificity to the genus level against Bartonella spp., but are not species specific. It is well known that Bartonella spp. cross-react with other genera and species, such as C. burnetii and Chlamydia. However, in our patient, serological (IFA) tests against B. quintana, C. burnetii, and Chlamydia were negative. [17],[18] PCR can be very useful in demonstrating Bartonella spp. DNA in clinical samples, enabling an early therapeutic approach. [1],[2]

Endocarditis and asymptomatic bacteremia caused by B. henselae and B. quintana in the absence of BA, and other evidence of primary inoculation site have been previously described in both immunosuppressed and immunocompetent patients. [1],[2],[18],[19],[20],[21] In this sense, Bartonella spp. DNA found in our patients' bone marrow and the presence of low specific IgG titers against B. henselae could easily have been a sign of a past infection with hematogenous dissemination. We do not know whether the skin lesion was the primary inoculation site in this case, or on the contrary, a secondary manifestation of a previous asymptomatic bacteremia.

 
  References Top

1.Spach DH, Koehler JE. Bartonella-Associated Infections. Infect Dis Clin North Am 1998;12:137-55.  Back to cited text no. 1
    
2.Maguina C, Gotuzzo E. Bartonellosis: New and Old. Infect Dis Clin North Am 2000;14:1-22.  Back to cited text no. 2
    
3.Gangopadhyay AK, Sharma PK. Bacillary angiomatosis in an immune-competent patient. Indian J Dermatol Venereol Leprol 2001;67:37-8  Back to cited text no. 3
    
4.Turgut M, Alabaz D, KarakaΊ M. Bacillary angiomatosis in an immunocompetent child with a grafted traumatic wound. J Dermatol 2004;31: 44-7.  Back to cited text no. 4
    
5.Karakaώ M, Baba M, Aksungur VL, Homan S, Memisoπlu HR, Uπuz A. Bacillary angiomatosis on a region of burned skin in a immunocompetent patient. Br J Dermatol 2000;143:609-11.  Back to cited text no. 5
    
6.Schwartz RA, Gallardo MA, Kapila R, Gascσn P, Herscu J, Siegel I, et al. Bacillary angiomatosis in an HIV seronegative patient on systemic steroid therapy. Br J Dermatol 1996;135:982-7.  Back to cited text no. 6
    
7.Kayaselηuk F, Ceken I, Bircan S, Tuncer I. Bacillary angiomatosis of the scalp in a human immunodeficiency virus-negative patient. J Eur Acad Dermatol Venereol 2002;16:612-14.  Back to cited text no. 7
    
8.Karakaώ M, Baba M, Homan S, Akman A, Acar MA, Memiώoπlu HR, et al . A case of bacillary angiomatosis presenting as leg ulcers. J Eur Acad Dermatol Venereol 2003;17:65-7.  Back to cited text no. 8
    
9.Cockerell CJ, Bergstresser PR, Myrie-Williams C, Tierno PM. Bacillary epithelioid angiomatosis occurring in an immunocompetent individual. Arch Dermatol 1990;126:787-90.  Back to cited text no. 9
    
10.Schwartz RA, Nychay SG, Janniger CK, Lambert WC. Bacillary angiomatosis: Presentation of six patients, some with unusual features. Br J Dermatol 1997;136:60-5.  Back to cited text no. 10
    
11.Paul MA, Fleischer AB Jr, Wieselthier JS, White WL. Bacillary angiomatosis in an immunocompetent child: the first reported case. Pediatr Dermatol 1994;11:338-41.  Back to cited text no. 11
    
12.Breitschwerdt EB, Kordick DL. Bartonella Infection in Animals: Carriership, Reservoir Potential, Pathogenicity, and Zoonotic Potential for Human Infection. Clin Microbiol Rev 2000;13:428-38.   Back to cited text no. 12
    
13.Kravetz JD, Federman DG. Cat-Associated Zoonoses. Arch Intern Med 2002;162:1945-52.  Back to cited text no. 13
    
14.Koehler JE, Sanchez MA, Garrido CS, Whitfeld MJ, Chen FM, Berger TG, et al. Molecular epidemiology of Bartonella infections in patients with bacillary angiomatosis-peliosis. N Engl J Med 1997;337:1916-7.  Back to cited text no. 14
    
15.Drancourt M, Mainardi JL, Brouqui P, Vandenesch F, Carta A, Lehnert F, et al. Bartonella (rochalimaea) quintana endocarditis in three homeless men. N Engl J Med 1995;332:419-23.  Back to cited text no. 15
    
16.Spach DH, Kanter AS, Dougherty MJ, Larson AM, Coyle MB, Brenner DJ, et al. Bartonella (Rochalimaea) quintana bacteremia in inner-city patients with chronic alcoholism. N Engl J Med 1995;332:424-8.  Back to cited text no. 16
    
17.Blanco JR, Oteo JA, Martνnez V, Ramalle E, Garcνa A, Ibarra V, et al. Seroepidemiologνa de la infecciσn por Bartonella henselae en un colectivo de riesgo. Rev Clin Esp 1998;198:805-9.   Back to cited text no. 17
    
18.Blanco JR, Oteo JA, Martνnez V, Ramalle E, Garcνa A, Ibarra V, et al. Seroepidemiologνa de la infecciσn por Bartonella henselae en pacientes infectados por el VIH. Enferm Infecc Microbiol Clin 1999;17:434-8.   Back to cited text no. 18
    
19.Chomel BB, Kasten RW, Sykes JE, Boulouis HJ, Breitschwerdt EB. Clinical impact of persistent Bartonella bacteremia in humans and animals. Ann N Y Acad Sci 2003;990:267-78.  Back to cited text no. 19
    
20.Lesprit P, Noλl V, Chazouillθres P, Brun-Buisson C, Deforges L. Cure of Bartonella endocarditis of a prosthetic aortic valve without surgery: Value of serologic follow-up. Clin Microbiol Infect 2003;9:239-41.  Back to cited text no. 20
    
21.Tappero JW, Mohle-Boetani J, Koehler JE, Swaminathan B, Berger TG, LeBoit PE, et al. The epidemiology of bacillary angiomatosis and bacillary peliosis. JAMA 1993;296:770-5.  Back to cited text no. 21
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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