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Cutaneous leishmaniasis: An unusual case with atypical recurrence
Journ al of the Am erican Academ y of Dermatology Volume 28, Number 3
7. Goldberg HS. Basal cell epitheliomas developing in a localized linear epidermal nevus. Cutis 1980;25:295-9. 8. Rosenblum GA. Large basal cell carcinoma in a congenital nevus. J DermatoJ Surg Oncol 1986;12:166-8. 9. Mehta VR. Basal cell epithelioma in a junctional nevus: a case report with ulceration and comedonous changes. Indi an J Med Sci 1980;34:8-10. 10. Lever WF, Sch au mberg-Lever G . Histopathology of .the skin. 7th ed. Philad elphia: JB Lippincott , 1990:622-34.
Brief communications 495 II. Wick MR. Malignant tumors of the epidermis. In: Farmer ER, Hood AF, eds. Pathology of the skin. E Norwalk, Conn: Appleton & Lange, 1991:568-95 . 12. Mehregan HA . P inkus' Guide to dermatohistopatbology. 4th ed . Norw alk, Conn: Appleton-Century-Crofts, 1986: 509-20, 539. 13. PattersonJW. Basal cellcarcinoma. Clio Dermatol1988:126.
Cutaneous leishmaniasis: An unusual case with atypical recurrence Lina F. Kanj, MD, Abdul-Ghani Kibbi, MD, and Shukrallah Zaynoun, MD Beirut, Lebanon Recently there has been a renewed interest in leishmaniasis, revealing new information as to the role of the vector in the transmission of the disease, as well as factors in the host that playa role in the fate of the parasite. New avenues of therapy are being explored, and hitherto unrecognized clinical presentations are being defined. We report a case of cutaneous leishmaniasis with an atypical recurrence not conforming to any of the known presentations.
extending into the subcutaneous fa t composed of epithelioid cells, lymphocytes, and plasma cells . Histiocytes with a bundant cytopl asm contained nonencapsulated round to oval amastigotes, approximately 3 Jlm in diameter ( Fig. 2). The patient was given several courses of intramuscular pentavalent antimonials (Glucantime). Significant improvement was achieved.
CASE REPORT
Although there is no proof that the initial skin lesions were leishmaniasis, the circumstantial evidence isstrong: the highly endemic a rea in which the patient lived, the location of the lesions on the face, eventual healing after I to 2 years, and the characteristic scars.l" The appearance of scaly, erythematous papules at the periphery of the scars of healed leishmaniasis is a typical presentation of leishmania recidivans or recurrent leishmaniasis.1-4 In this condition , it is postulated that "dormant" organisms remain within the skin and, for unknown reasons, reactivate. The period of dormancy usually varies from 1 year to 10 to 15 years.s 4-6 In leishmaniasis recidivans the active disease is limited to the periphery of the scar and the zone immediately contiguous to it. 1-5 In our patient, not only was the period between initial healing and the appearance of new papules prolonged, but also the recurrence was not restricted to the periphery of the scars. Histologically, leishmaniasis recidivans typically shows epithelioid cell tubercles and histiocytic infiltrates with few organisms.v 4, 7,8 In our patient, the histologic features were those of acute leishmaniasis (numerous histiocytes laden with organisms) .2, 7, 8 Atypical presentations of infectious diseases oc-
A 47-year-old white man had painful lesions on the face and right ear of 2 years' duration. During his childhood "Aleppo boils" developed on both cheeks, his right ear, and his lower lip. The lesions healed 1 to 2 years later with residual scarring. Two years ago multiple small, scaly, erythematous papules on his left cheek, lips, and right ear developed around the scars of previously healed lesions. He was treated continuously with prednisone, 40 mg daily, and a topical corticosteroid cream. The lesions progressively enlarged. Physical examination revealed that the left half of the chin, left half of the upper and lower lips, adjacent half of the face, and the entire right ear were erythematous and indurated (Fig. I) . There was no associated lymphadenopathy. Hematoxylin-and-eosin- and Giemsa-stained sections of a biopsy specimen from the patient's left cheek showed a diffuse noncaseating granulomatous dermal infiltrate From the Departmen t of Dermatology, Am erican University of Beiru t-M edical Ce nter. Reprint requ ests: Shu krallah Zaynoun , MD, Department of Dermatology, American Uni versity of Beirut-Medical Center, 850 T hird Ave., New York, NY 10022-
J
AM A CAD D ERMATOL
1993;28:495-6.
Copyright @ 1993 by the American Academy of Dermatology, Inc. 0190-9622 /93 $1.00
+ .10
16/54/41031
DISCUSSION
496
Brief communications
Journal of the American Academy of Dermatology March 1993
Fig. 1. A, Active lesions and scars on left side of face. B, Active lesions and scars on right pinna.
ciency syndrome (AIDS).9 Our patient had a negative test for the human immunodeficiency virus but had been treated with systemic steroids for more than 2 years. Corticosteroids are known immunosuppressants 1aand, when given for a prolonged time, can reduce the size and intensity of the Leishmania skin test and promote proliferation of Leishmania organisms. 11 REFERENCES
Fig. 2. Dermal infiltrate of epithelioid cells, lymphocytes, and histiocytes filled with Leishman bodies. (Hematoxylin-eosin stain; X200.)
cur in immunocompromised states. The extent ofthe disease depends on the degree of immunosuppression and the virulence of the organism. Dissemination of leishmaniasis can occur when immunity is deeply impaired, as in the acquired immunodefi-
1. Farah FS, Malak JA. Cutaneous leishmaniasis. Arch Dermatol1971;103:467-74. 2. Lever WF, Schaumburg-Lever G. Histopathology of the skin. Philadelphia: JB Lippincott, 1990:394-5. 3. Farah FS. Protozoan and helminth infections. In: Fitzpatrick TB, Eisen AZ, Wolff K, et aI, eds. Dermatology in general medicine. New York: McGraw-Hill, 1986:247793. 4. Strick RA, Borok M, Gasiorowski He. Recurrent cutaneous leishmaniasis. J AM ACAD DERMATOL 1983;9:437-43. 5. Berlin C. Leishmania recidiva cutis; Leishmanid. Arch Dermatol SyphiloI1940;41:874-86. 6. Hart M, Livingood CS, Goltz RW, et al. Late cutaneous leishmaniasis. Arch Dermatol 1969;99:455-8. 7. Kurban AK, Malak JA, Farah FS, et al. Histopathology of cutaneous leishmaniasis. Arch Dermatoll966;93:396-401. 8. Kurban AK, Kibbi AG. Diseases caused by protozoa. In: Farmer ER, Hood AF, eds. Pathology of the skin. Norwalk, Conn: Appleton & Lange, 1990:366-71. 9. Yebra M, Segovia J, Manzano L, et al. Disseminated-toskin kala azar and the acquired immunodeficiency syndrome. Ann Intern Med 1988;108:490-1. 10. Parrillo JE, Fauci AS. Meehan ismsof glucocorticoid action on immune processes. Ann Rev Pharmacol Toxicol 1979;19:179-201. I 1. Dostrovsky A. Steroids as a supplementary treatment in late leishmaniasis. Br J Dermatol 1958;70:288-92.
7. Goldberg HS. Basal cell epitheliomas developing in a localized linear epidermal nevus. Cutis 1980;25:295-9. 8. Rosenblum GA. Large basal cell carcinoma in a congenital nevus. J DermatoJ Surg Oncol 1986;12:166-8. 9. Mehta VR. Basal cell epithelioma in a junctional nevus: a case report with ulceration and comedonous changes. Indi an J Med Sci 1980;34:8-10. 10. Lever WF, Sch au mberg-Lever G . Histopathology of .the skin. 7th ed. Philad elphia: JB Lippincott , 1990:622-34.
Brief communications 495 II. Wick MR. Malignant tumors of the epidermis. In: Farmer ER, Hood AF, eds. Pathology of the skin. E Norwalk, Conn: Appleton & Lange, 1991:568-95 . 12. Mehregan HA . P inkus' Guide to dermatohistopatbology. 4th ed . Norw alk, Conn: Appleton-Century-Crofts, 1986: 509-20, 539. 13. PattersonJW. Basal cellcarcinoma. Clio Dermatol1988:126.
Cutaneous leishmaniasis: An unusual case with atypical recurrence Lina F. Kanj, MD, Abdul-Ghani Kibbi, MD, and Shukrallah Zaynoun, MD Beirut, Lebanon Recently there has been a renewed interest in leishmaniasis, revealing new information as to the role of the vector in the transmission of the disease, as well as factors in the host that playa role in the fate of the parasite. New avenues of therapy are being explored, and hitherto unrecognized clinical presentations are being defined. We report a case of cutaneous leishmaniasis with an atypical recurrence not conforming to any of the known presentations.
extending into the subcutaneous fa t composed of epithelioid cells, lymphocytes, and plasma cells . Histiocytes with a bundant cytopl asm contained nonencapsulated round to oval amastigotes, approximately 3 Jlm in diameter ( Fig. 2). The patient was given several courses of intramuscular pentavalent antimonials (Glucantime). Significant improvement was achieved.
CASE REPORT
Although there is no proof that the initial skin lesions were leishmaniasis, the circumstantial evidence isstrong: the highly endemic a rea in which the patient lived, the location of the lesions on the face, eventual healing after I to 2 years, and the characteristic scars.l" The appearance of scaly, erythematous papules at the periphery of the scars of healed leishmaniasis is a typical presentation of leishmania recidivans or recurrent leishmaniasis.1-4 In this condition , it is postulated that "dormant" organisms remain within the skin and, for unknown reasons, reactivate. The period of dormancy usually varies from 1 year to 10 to 15 years.s 4-6 In leishmaniasis recidivans the active disease is limited to the periphery of the scar and the zone immediately contiguous to it. 1-5 In our patient, not only was the period between initial healing and the appearance of new papules prolonged, but also the recurrence was not restricted to the periphery of the scars. Histologically, leishmaniasis recidivans typically shows epithelioid cell tubercles and histiocytic infiltrates with few organisms.v 4, 7,8 In our patient, the histologic features were those of acute leishmaniasis (numerous histiocytes laden with organisms) .2, 7, 8 Atypical presentations of infectious diseases oc-
A 47-year-old white man had painful lesions on the face and right ear of 2 years' duration. During his childhood "Aleppo boils" developed on both cheeks, his right ear, and his lower lip. The lesions healed 1 to 2 years later with residual scarring. Two years ago multiple small, scaly, erythematous papules on his left cheek, lips, and right ear developed around the scars of previously healed lesions. He was treated continuously with prednisone, 40 mg daily, and a topical corticosteroid cream. The lesions progressively enlarged. Physical examination revealed that the left half of the chin, left half of the upper and lower lips, adjacent half of the face, and the entire right ear were erythematous and indurated (Fig. I) . There was no associated lymphadenopathy. Hematoxylin-and-eosin- and Giemsa-stained sections of a biopsy specimen from the patient's left cheek showed a diffuse noncaseating granulomatous dermal infiltrate From the Departmen t of Dermatology, Am erican University of Beiru t-M edical Ce nter. Reprint requ ests: Shu krallah Zaynoun , MD, Department of Dermatology, American Uni versity of Beirut-Medical Center, 850 T hird Ave., New York, NY 10022-
J
AM A CAD D ERMATOL
1993;28:495-6.
Copyright @ 1993 by the American Academy of Dermatology, Inc. 0190-9622 /93 $1.00
+ .10
16/54/41031
DISCUSSION
496
Brief communications
Journal of the American Academy of Dermatology March 1993
Fig. 1. A, Active lesions and scars on left side of face. B, Active lesions and scars on right pinna.
ciency syndrome (AIDS).9 Our patient had a negative test for the human immunodeficiency virus but had been treated with systemic steroids for more than 2 years. Corticosteroids are known immunosuppressants 1aand, when given for a prolonged time, can reduce the size and intensity of the Leishmania skin test and promote proliferation of Leishmania organisms. 11 REFERENCES
Fig. 2. Dermal infiltrate of epithelioid cells, lymphocytes, and histiocytes filled with Leishman bodies. (Hematoxylin-eosin stain; X200.)
cur in immunocompromised states. The extent ofthe disease depends on the degree of immunosuppression and the virulence of the organism. Dissemination of leishmaniasis can occur when immunity is deeply impaired, as in the acquired immunodefi-
1. Farah FS, Malak JA. Cutaneous leishmaniasis. Arch Dermatol1971;103:467-74. 2. Lever WF, Schaumburg-Lever G. Histopathology of the skin. Philadelphia: JB Lippincott, 1990:394-5. 3. Farah FS. Protozoan and helminth infections. In: Fitzpatrick TB, Eisen AZ, Wolff K, et aI, eds. Dermatology in general medicine. New York: McGraw-Hill, 1986:247793. 4. Strick RA, Borok M, Gasiorowski He. Recurrent cutaneous leishmaniasis. J AM ACAD DERMATOL 1983;9:437-43. 5. Berlin C. Leishmania recidiva cutis; Leishmanid. Arch Dermatol SyphiloI1940;41:874-86. 6. Hart M, Livingood CS, Goltz RW, et al. Late cutaneous leishmaniasis. Arch Dermatol 1969;99:455-8. 7. Kurban AK, Malak JA, Farah FS, et al. Histopathology of cutaneous leishmaniasis. Arch Dermatoll966;93:396-401. 8. Kurban AK, Kibbi AG. Diseases caused by protozoa. In: Farmer ER, Hood AF, eds. Pathology of the skin. Norwalk, Conn: Appleton & Lange, 1990:366-71. 9. Yebra M, Segovia J, Manzano L, et al. Disseminated-toskin kala azar and the acquired immunodeficiency syndrome. Ann Intern Med 1988;108:490-1. 10. Parrillo JE, Fauci AS. Meehan ismsof glucocorticoid action on immune processes. Ann Rev Pharmacol Toxicol 1979;19:179-201. I 1. Dostrovsky A. Steroids as a supplementary treatment in late leishmaniasis. Br J Dermatol 1958;70:288-92.