Abstract
We present an interesting case report of two sarcoid-like lesions on a port-wine stain (PWS) birthmark in a Brazilian patient which on investigation proved to be cutaneous leishmaniasis.
Hemangioma, capillary; Leishmaniasis, cutaneous; Port-Wine Stain; Tropism
A 30-year-old housewife who was born and lived in the state of Maranhão in the northeast of
Brazil was admitted to our dermatology department with a complaint of two painless
erythematous infiltrative papules on her forehead that had started two months previously.
Her stepdaughter had been treated for cutaneous leishmaniasis on her leg. The patient
presented with two nonulcerated erythematous infiltrative papules which measured 8 and 10
mm in diameter on a Port-wine stain (nevus flammeus) on the left side of her forehead
(Figure 1). An intradermal test for leishmaniasis
(Montenegro test) was positive (10mm). A cutaneous biopsy was taken and processed for
conventional optical microscopy using H&E, Fite-Faraco, PAS and Grocott-Gomori
stains and for immunohistochemistry to detect
Leishmania.11. Penna GO, Domingues CM, Siqueira Jr JB, Elkhoury AN, Cechinel MP,
Grossi MA, et al, Dermatological diseases of compulsory notification in Brazil. An
Bras Dermatol. 2011;86:865-77.
2. Stebut EV. Immunology of cutaneous leishmaniasis: the role of mast
cells, phagocytes and dendritic cells for protective immunity. Eur J Dermatol.
2007;17:115-22.-33. Basu MK, Ray M. Macrophage and Leishmania: an unacceptable
coexistence. Crit Rev Microbiol. 2005;31:145-54. Histopathology revealed chronic diffuse dermatitis
with a granulomatous tissue reaction and plasmacytosis (Figure 2A). Immunohistochemistry staining to detect Leishmania
antigens performed as previously described using antibodies against
Leishmania chagasi was positive (Figure
2B).44. Amato VS, Tuon FF, de Andrade HF Jr, Bacha H, Pagliari C, Fernandes
ER, et al. Immunohistochemistry and polymerase chain reaction on paraffin-embedded
material improve the diagnosis of cutaneous leishmaniasis in the Amazon region. Int J
Dermatol. 2009;48:1091-5.
5. Carrasco L, Pastor A, Fariña C, Martín L, Manzarbeitia F, Requena L.
Acral arteriovenous tumor developed within a nevus flammeus in a patient with
Sturge-Weber syndrome. Am J Dermatopathol. 2003;25:341-5.-66. Lefèvre T, Koella JC, Renaud F, Hurd H, Biron DG, Thomas F. New
prospects for research on manipulation of insect vectors by pathogens. PLoS Pathog.
2006;2:e72. Histological staining and cultures for acid-fast bacilli and fungi
were negative.
A. Port-wine stain birthmark in the left frontotemporal area. Note the two sarcoid-like lesions on the hemangioma; B and C. Histopathological examination of hematoxylin- eosin (H&E)-stained tissue revealed dermal granulomatous inflammation with a predominantly lymphoid infiltrate, several plasma cells and dilated blood vessels with thin walls (H&E; OM 200x)
A. Detail of the histopathological examination showing dermal granulomatous inflammation with a with numerous lymphoid and plasma cells. (H&E; OM 400x); B. Immunohistochemistry with anti-Leishmania antibody (ABC peroxidase) showing a few amastigote structures (arrow) inside the macrophage cytoplasm
This is the first report of cutaneous leishmaniasis on a hemangioma in the literature. We believe a sand fly was attracted to the lesion to feed probably because of the increased vascularization in this area. Mann et al. suggested that the attraction of Lutzomyia spp. to colors and odors was species-specific and demonstrated that certain colors, such as red and red-blue, exert a greater attraction on these species.77. Mann RS, Kaufman PE, Butler JF. Lutzomyia spp. (Diptera: Psychodidae) response to olfactory attractant- and light emitting diode-modified Mosquito Magnet X (MM-X) traps. J Med Entomol. 2009;46:1052-61.
REFERENCES
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1Penna GO, Domingues CM, Siqueira Jr JB, Elkhoury AN, Cechinel MP, Grossi MA, et al, Dermatological diseases of compulsory notification in Brazil. An Bras Dermatol. 2011;86:865-77.
-
2Stebut EV. Immunology of cutaneous leishmaniasis: the role of mast cells, phagocytes and dendritic cells for protective immunity. Eur J Dermatol. 2007;17:115-22.
-
3Basu MK, Ray M. Macrophage and Leishmania: an unacceptable coexistence. Crit Rev Microbiol. 2005;31:145-54.
-
4Amato VS, Tuon FF, de Andrade HF Jr, Bacha H, Pagliari C, Fernandes ER, et al. Immunohistochemistry and polymerase chain reaction on paraffin-embedded material improve the diagnosis of cutaneous leishmaniasis in the Amazon region. Int J Dermatol. 2009;48:1091-5.
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5Carrasco L, Pastor A, Fariña C, Martín L, Manzarbeitia F, Requena L. Acral arteriovenous tumor developed within a nevus flammeus in a patient with Sturge-Weber syndrome. Am J Dermatopathol. 2003;25:341-5.
-
6Lefèvre T, Koella JC, Renaud F, Hurd H, Biron DG, Thomas F. New prospects for research on manipulation of insect vectors by pathogens. PLoS Pathog. 2006;2:e72.
-
7Mann RS, Kaufman PE, Butler JF. Lutzomyia spp. (Diptera: Psychodidae) response to olfactory attractant- and light emitting diode-modified Mosquito Magnet X (MM-X) traps. J Med Entomol. 2009;46:1052-61.
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Financial funding: None
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*
Study carried out at the Mycology and Leishmaniasis Outpatient Unit, Department of Dermatology, Hospital das Clínicas, School of Medicine, University of São Paulo (HC-FMUSP), São Paulo, SP, Brazil.
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How to cite this article: Criado PR, Valente NS, Noda A, Belda W Jr. Cutaneous New World Leishmaniasis on a Port-Wine Stain Birthmark. An Bras Dermatol. 2014;89(4):669-70.
Publication Dates
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Publication in this collection
Jul-Aug 2014
History
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Received
12 Feb 2013 -
Accepted
18 June 2013