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Interstitial Keratitis Caused By American (Mucocutaneous) Leishmaniasis
INTERSTITIAL KERATITIS CAUSED BY AMERICAN (MUCOCUTANEOUS) LEISHMANIASIS JAIME R O I Z E N B L A T T , Los Angeles,
American leishmaniasis is a disease caused by the flagellate protozoa, Leishmania braziliensis, which was observed in 1909 by Lindenberg 1 and Carini and Paranhos 2 in smears of patients with Bauru ulcer in Sao Paulo, Brazil. In 1911, Vianna 3 studied this type of leishmania, considered it a new species and named it L. braziliensis. The disease occurs in the areas from southern Mexico to northern Argentina and is extensive in Brazil and Peru. 4 The Leishmania (L. brasiliensis, pro ducing American leishmaniasis; L. donovani, producing kala-azar; and L. tropica, producing Oriental sore) has two stages of development: the aflagellate stage occur ring in man and other vertebrates and the flagellate stage occurring in insect vectors and culture media. The vector, the mosquitoes of the genus Phlebotomus, are small hairy mosquitoes that acquire the infection by ingesting the parasite when taking a blood meal from an infected host. The parasite is transmit ted to a healthy human or other vertebrate host by the bite of the infected female mosquito. The extra-human reservoirs are wild rodents, including rats, mice, agoutis, and pacas, which are common in the neotropics. The incubation period varies from two weeks to two months," after which an
From the Department of Ophthalmology, Jules Stein Eye Institute, UCLA School of Medicine, Los Angeles. Dr. Roizenblatt is a special fellow in retinal diseases from Clinica Oftalmologica, Hospital das Cliniuas, Faculdade de Medicine da Universidade de Sao Paulo, Brazil. This study was supported by Alcon Laboratories and the LaSalle Foundation. Reprint requests to Jaime Roizenblatt, M.D., Jules Stein Eve Institute, UCLA School of Medicine, Los Angeles, CA 90024.
M.D.
California
erythematous papule develops at the site of inoculation and gradually increases in size by peripheral extension. The papule may ulcerate, vesiculate, or take on a mulberry aspect. Regional adenopathy may be noted. Generally, the lesions are secondarily infected. The cutaneous lesions have an infiltrat ed raised border and the interior of the sore is often covered by a serosanguinous exudate or yellow membrane. A lymphat ic form may be seen simulating sporotrichosis extending along the lymph chan nels, manifested by thickening of the lymph vessels, nodules lymphangitis, and adenitis. Mucous membrane lesions differenti ate American leishmaniasis from Oriental sore. The mucous membrane lesions al most always begin in the nasal mucosa, which becomes inflamed, edematous, and finally ulcerative. The nasal cartilage usu ally is invaded and destroyed, resulting in a deformity known as "tapir's nose," "camel's nose," or "parrot's beak." The upper lip becomes thickened and pro trudes forward, accentuating the defor mity. The involvement of nasal fossae, pharynx, soft palate, floor of the mouth, tonsils, and tonsilar fauces, as well as the respiratory tract, pharynx, and larynx may cause difficulty in breathing, feeding, and deglutition. As a result of acute respira tory infection, malnutrition and death may occur. Disseminated anergic leishmaniasis as manifested by specific anergy to leismanin antigen (Montenegro's antigen), absence of visceral involvement but widespread involvement of the skin, probably from hematogenous dissemina tion, may occur. Ocular complications caused by Ameri can leishmaniasis have been reported, 5 - 2 5
AMERICAN JOURNAL O F OPHTHALMOLOGY 87:175-179, 1979
175
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but reports of interstitial keratitis as a result of American leishmaniasis are uncommon, 5 ' 6,9,13 ' 18,26 and I am unaware of any report in the past 25 years. I report herein a case seen at the Eye Clinic of the Hospital das Clinicas Medical Faculty, Sao Paulo University. CASE REPORT A 58-year-old man came to the eye clinic com plaining of decreased vision in both eyes, and sores in the nose of 20 years' duration (Fig. 1). The patient had been bitten by a mosquito behind the right ear 35 years previously. Treatment with antimonial drugs apparently resulted in cicatrization of the ulcer, but ten years later, new sores appeared that involved his nose, and over the past two years the sores became much worse. The patient noted photo phobia, tearing, hyperemia, and decreased vision in both eyes for over 20 years. Examination revealed visual acuity was reduced to finger counting at 2 m in the right eye and 20 cm in the left eye, which did not improve with lenses or pinhole. The limbal conjunctiva was hyperemic. A diffuse leukoma and cellular infiltration
FEBRUARY, 1979
involved the entire cornea of both eyes, together with diffuse vascularization of the superficial stroma. The epithelium was intact (Fig. 2). The lens, iris, and fundus were poorly visible but appeared to be grossly normal. The general examination revealed diffuse enlarge ment of the nose with ulcers in various stages of development and purulent discharge. The nasal septum was absent. Ulcers occurred in the patient's gums, tonsillar pillae, and the left malar region. There was a cicatrix behind the right ear in the spot where the patient said the mosquito had bitten him. Laboratory studies included Montenegro test (positive at 48 hours); indirect immunofluorescence for anti-Leishmania antibodies (positive at 1:10); VDRL and FTA-ABS (negative); Mantoux test (neg ative at 1:1,000 dilution); and chest x-ray film (normal). Cultures of the conjunctiva and cornea for fungi, bacteria and parasites, including cultures on Nicole, Novy, McNeal medium, were negative.
Fig. 1 (Roizenblatt). Front view (left) and side view (right) of facial aspects of patient before treatment.
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Fig. 2 (Roizenblatt). Corneal (left) and conjunctival (right) aspects of the right and left eyes.
Scrapings of the bulbar conjunctiva and cornea including studies with the Leishman dye and acidfast stain were negative. A biopsy specimen of the nasal lesions showed hyperkeratosis, corneal pearls of the epidermis, and atrophy and hydropic degeneration of the basal cell layer and pigmentary drop off into the dermis. Perivascular round cell infiltration was noted in the dermis, together with vacuolation of the histiocytes (Fig. 3). Biopsy of the conjunctiva showed exfoliation of the epithelial layer, round cell infiltration in the stroma, and tuberculoid granulomas without caseation (Fig. 4). Treatment with amphotericin B (total dose of 1 g) was given and the ulcerative areas showed marked improvement (Fig. 5). DISCUSSION
Although we were unable to identify the parasite in this patient, the clinical, laboratory, and pathologic findings and the therapeutic response suggest that the patient was suffering from American
leishmaniasis. Many authors believe that the lack of finding a parasite in the lesion does not rule out the diagnosis. 7,16 ~ 18,27 Epidemiologically, the patient was born in an endemic area for leishmaniasis (Bahia), and at the time of the original ulcer formation, he was working as a farmer in another area endemic for leish maniasis (Marilia, Sao Paulo). The Montenegro test, positive in this patient, is positive in more than 90% of patients with the disease. 4 ' 7,18,20 It has certain limitations in endemic zones, as patients may have a primary infection with Leishmania, which evolves sponta neously and thus gives a positive re sponse. Differential diagnosis—The dermatologic aspects of this patient suggest various diagnoses, but we are unable to
">Jtl Fig. 3 (Roizenblatt). Biopsy specimen of the nasal lesion showing hyperkeratosis and corneal pearls of the epidermis together with atrophy and hydropic degeneration of the basal cell layer and pigmentary drop off into the dermis. Perivascular round cell infiltration is present in the dermis, together with vacuolation of the histiocytes (xlOO and x400).
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Fig. 4 (Roizenblatt). Biopsy specimen of the con junctiva showing exfoliation of the epithelial layer, round cell infiltration in the stroma, and tuberculoid granulomas without caseation.
establish evidence of fungal infections such as paracoccidioidomycosis, sporotrichosis, histoplasmosis, and chromoblastomycosis, or bacterial infections such as rhinoscleroma, tuberculosis, or leprosy on culture or smear. The ocular aspects of the patient sug gest various other diseases, including tra choma, syphilis, tuberculosis, leprosy, trypanosomiasis, and onchocerciasis. Trachoma was not considered a likely cause, as the vascularization occurred in the superficial stroma and did not re-
Fig. 5 (Roizenblatt). Facial aspect of patient after treatment.
FEBRUARY, 1979
semble the pannus seen with trachoma. Additionally, there was no supporting ev idence of trachoma, such as follicjes, conjunctival scarring, trichiasis, or Herbert's pits. The vascularization of the cornea and the inflammatory reaction associated with syphilis is predominate in deep stroma, in contrast to the superficial inflammation in this case. Additionally, there were no other signs of congenital syphilis, such as Hutchinson teeth, raspberry molars, sad dle nose, or deafness, and the serologic tests virtually ruled out the probability of syphilis. Tuberculosis must be considered in the differential diagnosis of interstitial keratitis; however, the patient was anergic to tuberculin, the chest x-ray film was nega tive, and the biopsy specimen showed no caseation. Lepromatous leprosy may cause a su perficial vascularization of the cornea to gether with skin changes, somewhat simi lar to that noted in this patient, but smears and bjopsy usually show the specimen teeming with organisms. Trypanosimiasis and onchocerciasis may cause corneal vascularization, but these diseases are not seen in the areas in which the patient lived, and the organ isms were not seen on biopsy. Other causes of interstitial keratitis in cluding herpes simplex keratitis (herpes simplex is the great imitator), lymphogranuloma venereum (which may cause a segmental sclerokeratitis), and mumps (which do not cause vascularization of the corneal stroma) have not been ruled out as the cause of the ocular lesions, but their presence is unlikely, and they would not cause the skin lesions. American leishmaniasis has various forms, including cutaneous American leishmaniasis, mucocutaneous American leishmaniasis, disseminated anergic leish maniasis, and Mexican leishmaniasis. The present case falls into the category of mucocutaneous American leishmaniasis.
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AMERICAN LEISHMANIASIS
Ocular findings associated with mucocutaneous American leishmaniasis include involvement of the eyelids 6 , 8 , 9 , 1 1 , 1 7 " 1 9 , 2 6 , 2 8 and conjunctiva, 10,13,15,18,23,25-27 described as eyelid edema; scarring or even destruc tion of the tarsus; and nodular granulomas of the tarsal or bulbar conjunctiva. Ulceration of the conjunctiva and cornea and ulcerative 5 , 6 , 1 8 , 2 5 - 2 7 or granulomatous 5,10,16,18,24 "" 26 lesions of the cornea, as well as interstitial keratitis 5,6,9,13,18,26 also have been described. The eye may be involved by contiguous spread to the eyelid and conjunctiva, by hematogenous spread, or by inoculation of the conjunctiva by the patient's own fingers. 5,6,13,15,17,18,20,26 In our patient, the most likely spread was by way of the blood stream, as there were no signs of tarsal or conjunctival involvement. The disfigurement and lack of satisfac tory treatment have often hampered case findings of patients with leishmaniasis, and when found, little attention has been paid to the ocular findings. Case findings and ophthalmologic examination would reveal many more cases than the reports suggest. SUMMARY
A 58-year-old man with American (mucocutaneous) leishmaniasis had the un usual ocular complication of interstitial keratitis. Although the parasite was not identified, clinical laboratory and histologic findings were compatible with leishmaniasis, and tests ruled out other causes. Ulcerative areas improved after treatment with 1 g of amphotericin B. REFERENCES 1. Lindenberg, A.: A ulcera de Bauru e o sen microbio. Communicacao preventiva. Rev. Med. Sao Paulo 12:116, 1909. 2. Carini, A., and Paranhos, U.: Identifieacao das ulceras de Bauru ao botao do Oriente. Communicac ao preliminar. Rev. Med. Sao Paulo 12:111, 1909. 3. Vianna, G.: Sobre uma nova especie de Leishmania (Nota preliminar). Brasil-Med. 25:411, 1911. 4. Forattini, O. P.: Leishmanioses tegumentares
179
americanas. In Entomologia Medica, vol. 4. Sao Paulo, Edgar Blticher/Editora da Universidade Sao Paulo, 1973, pp. 570-610. 5. Andrade, C. de: Fatores climaticos na patologia ocular. An. Fac. Med. Bahia 1942-1943, pp. 124-127. 6. Andrade, C. de: Interstitial and ulcerous kerati tis in leishmaniasis. Arch. Ophthalmol. 27:1193, 1942. 7. Azulay, R. D.: Leishmaniose tegumentar, the sis. Rio de Janeiro, 1952. 8. Burnier, F. F.: Leishmaniose primitiva da palpebra. Arq. Inst. P. Burnier (Campinas) 4:225, 1939. 9. Bussaca, A.: Keratitis na leishmania. Congr. Bras. Oftalmol. 10:261, 1936. 10. Cecilia, J. S.: Leishmaniose ocular com localizacao na conjunctiva bulbar. Arq. Mineir DermatoSiphilograf 1:21, 1919. 11. Cerqueira, A. G. C : Da leishmaniose te gumentar, thesis. Bahia, 1914, p. 77. 12. Chagas, C.: Notas sobre a epidemiologia do Amazonas. Brasil Med. 27:450, 1913. 13. Costa, P. R., Jr.: Da Leishmaniose ulcerosa e suas lesoes oculares, thesis. Bahia, 1916. 14. Duke-Elder, S., and Leigh, A. G.: Diseases of the Outer Eye. Cornea and Sclera. In Duke-Elder, S. (ed.): System of Ophthalmology, vol. 8, pt. 2. Lon don, Ki'mpton, 1965, p p . 811-856. 15. Dusseldorp, M.: Leishmaniosis cutaneaamericana com complicationes oculares. Bol. Inst. Clin. Quir. 3:452, 1927. 16. Duke-Elder, S.: Diseases of the Outer Eye. Conjunctiva. In Svstem of Ophthalmology, vol. 8, pt. 1. London, Kimpton, 1965, pp. 398-401. 17. Machado, N. R., Machado, J. G. C , and Moura, P. A.: Sobre urn caso de leishmaniose ocular. Rev. Bras. Oftalmol. 17:279, 1958. 18. Marbak, H.: Lesoes oculares da leishmaniose tegumentar americana, thesis. Bahia, 1953. 19. Monteiro, S.: Leishmaniose primitiva da palpebra. Arq. Inst. P. Burnier (Campinas) 8:180, 1949. 20. Pessoa, S. B., and Barreto, M. P.: Leishman iose tegumentar americana. Rio de Janieiro, Minist. de Educacao e Cultura/Imprensa Nacional, 1948, pp. 244-245. 21. Pina, M. C : Breves consideracoes sobre a leishmaniose ulcerosa, thesis. Bahia, 1912. 22. Rabello, E.: Contribuicao ao estudo da leish maniose tegumentar no Brasil. An. Bras. Derm. Sif. 1:3031, 1925. 23. Silva, F.: Leishmaniose tegumentar (lesoes multiplas, inclusive de conjuntiva palpebral). An. Fac. Med. Bahia 1942-1943, pp. 117-123. 24. Simoes, E.: Tracoma e Leishmaniose. Arq. Clin. Oftalmol Oto-rino-laryngol. 5:174, 1938. 25. Spinola, C : Kerato conjuncitite a leishmania. In 1st Congresso Argentino de Oftalmologica. Bue nos Aires, 1937, pp. 372-378. 26. Andrade, C.: Oftalmologia Tropical. Rio de Janeiro, Rodriques and Cia, 1940, pp. 43-64. 27. Campos, E.: Lichimaniose ocular. In Almanak Laemmert (ed.): Consultas Oftalmologieas. Rio de Janeiro, 1935, pp. 65-67. 28. Bussaca, A., and Maia, J.: Oedema chronique des paupieres du a la leismania. Folia Ophthalmol. Orient. 13:372, 1934.
American leishmaniasis is a disease caused by the flagellate protozoa, Leishmania braziliensis, which was observed in 1909 by Lindenberg 1 and Carini and Paranhos 2 in smears of patients with Bauru ulcer in Sao Paulo, Brazil. In 1911, Vianna 3 studied this type of leishmania, considered it a new species and named it L. braziliensis. The disease occurs in the areas from southern Mexico to northern Argentina and is extensive in Brazil and Peru. 4 The Leishmania (L. brasiliensis, pro ducing American leishmaniasis; L. donovani, producing kala-azar; and L. tropica, producing Oriental sore) has two stages of development: the aflagellate stage occur ring in man and other vertebrates and the flagellate stage occurring in insect vectors and culture media. The vector, the mosquitoes of the genus Phlebotomus, are small hairy mosquitoes that acquire the infection by ingesting the parasite when taking a blood meal from an infected host. The parasite is transmit ted to a healthy human or other vertebrate host by the bite of the infected female mosquito. The extra-human reservoirs are wild rodents, including rats, mice, agoutis, and pacas, which are common in the neotropics. The incubation period varies from two weeks to two months," after which an
From the Department of Ophthalmology, Jules Stein Eye Institute, UCLA School of Medicine, Los Angeles. Dr. Roizenblatt is a special fellow in retinal diseases from Clinica Oftalmologica, Hospital das Cliniuas, Faculdade de Medicine da Universidade de Sao Paulo, Brazil. This study was supported by Alcon Laboratories and the LaSalle Foundation. Reprint requests to Jaime Roizenblatt, M.D., Jules Stein Eve Institute, UCLA School of Medicine, Los Angeles, CA 90024.
M.D.
California
erythematous papule develops at the site of inoculation and gradually increases in size by peripheral extension. The papule may ulcerate, vesiculate, or take on a mulberry aspect. Regional adenopathy may be noted. Generally, the lesions are secondarily infected. The cutaneous lesions have an infiltrat ed raised border and the interior of the sore is often covered by a serosanguinous exudate or yellow membrane. A lymphat ic form may be seen simulating sporotrichosis extending along the lymph chan nels, manifested by thickening of the lymph vessels, nodules lymphangitis, and adenitis. Mucous membrane lesions differenti ate American leishmaniasis from Oriental sore. The mucous membrane lesions al most always begin in the nasal mucosa, which becomes inflamed, edematous, and finally ulcerative. The nasal cartilage usu ally is invaded and destroyed, resulting in a deformity known as "tapir's nose," "camel's nose," or "parrot's beak." The upper lip becomes thickened and pro trudes forward, accentuating the defor mity. The involvement of nasal fossae, pharynx, soft palate, floor of the mouth, tonsils, and tonsilar fauces, as well as the respiratory tract, pharynx, and larynx may cause difficulty in breathing, feeding, and deglutition. As a result of acute respira tory infection, malnutrition and death may occur. Disseminated anergic leishmaniasis as manifested by specific anergy to leismanin antigen (Montenegro's antigen), absence of visceral involvement but widespread involvement of the skin, probably from hematogenous dissemina tion, may occur. Ocular complications caused by Ameri can leishmaniasis have been reported, 5 - 2 5
AMERICAN JOURNAL O F OPHTHALMOLOGY 87:175-179, 1979
175
176
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but reports of interstitial keratitis as a result of American leishmaniasis are uncommon, 5 ' 6,9,13 ' 18,26 and I am unaware of any report in the past 25 years. I report herein a case seen at the Eye Clinic of the Hospital das Clinicas Medical Faculty, Sao Paulo University. CASE REPORT A 58-year-old man came to the eye clinic com plaining of decreased vision in both eyes, and sores in the nose of 20 years' duration (Fig. 1). The patient had been bitten by a mosquito behind the right ear 35 years previously. Treatment with antimonial drugs apparently resulted in cicatrization of the ulcer, but ten years later, new sores appeared that involved his nose, and over the past two years the sores became much worse. The patient noted photo phobia, tearing, hyperemia, and decreased vision in both eyes for over 20 years. Examination revealed visual acuity was reduced to finger counting at 2 m in the right eye and 20 cm in the left eye, which did not improve with lenses or pinhole. The limbal conjunctiva was hyperemic. A diffuse leukoma and cellular infiltration
FEBRUARY, 1979
involved the entire cornea of both eyes, together with diffuse vascularization of the superficial stroma. The epithelium was intact (Fig. 2). The lens, iris, and fundus were poorly visible but appeared to be grossly normal. The general examination revealed diffuse enlarge ment of the nose with ulcers in various stages of development and purulent discharge. The nasal septum was absent. Ulcers occurred in the patient's gums, tonsillar pillae, and the left malar region. There was a cicatrix behind the right ear in the spot where the patient said the mosquito had bitten him. Laboratory studies included Montenegro test (positive at 48 hours); indirect immunofluorescence for anti-Leishmania antibodies (positive at 1:10); VDRL and FTA-ABS (negative); Mantoux test (neg ative at 1:1,000 dilution); and chest x-ray film (normal). Cultures of the conjunctiva and cornea for fungi, bacteria and parasites, including cultures on Nicole, Novy, McNeal medium, were negative.
Fig. 1 (Roizenblatt). Front view (left) and side view (right) of facial aspects of patient before treatment.
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Fig. 2 (Roizenblatt). Corneal (left) and conjunctival (right) aspects of the right and left eyes.
Scrapings of the bulbar conjunctiva and cornea including studies with the Leishman dye and acidfast stain were negative. A biopsy specimen of the nasal lesions showed hyperkeratosis, corneal pearls of the epidermis, and atrophy and hydropic degeneration of the basal cell layer and pigmentary drop off into the dermis. Perivascular round cell infiltration was noted in the dermis, together with vacuolation of the histiocytes (Fig. 3). Biopsy of the conjunctiva showed exfoliation of the epithelial layer, round cell infiltration in the stroma, and tuberculoid granulomas without caseation (Fig. 4). Treatment with amphotericin B (total dose of 1 g) was given and the ulcerative areas showed marked improvement (Fig. 5). DISCUSSION
Although we were unable to identify the parasite in this patient, the clinical, laboratory, and pathologic findings and the therapeutic response suggest that the patient was suffering from American
leishmaniasis. Many authors believe that the lack of finding a parasite in the lesion does not rule out the diagnosis. 7,16 ~ 18,27 Epidemiologically, the patient was born in an endemic area for leishmaniasis (Bahia), and at the time of the original ulcer formation, he was working as a farmer in another area endemic for leish maniasis (Marilia, Sao Paulo). The Montenegro test, positive in this patient, is positive in more than 90% of patients with the disease. 4 ' 7,18,20 It has certain limitations in endemic zones, as patients may have a primary infection with Leishmania, which evolves sponta neously and thus gives a positive re sponse. Differential diagnosis—The dermatologic aspects of this patient suggest various diagnoses, but we are unable to
">Jtl Fig. 3 (Roizenblatt). Biopsy specimen of the nasal lesion showing hyperkeratosis and corneal pearls of the epidermis together with atrophy and hydropic degeneration of the basal cell layer and pigmentary drop off into the dermis. Perivascular round cell infiltration is present in the dermis, together with vacuolation of the histiocytes (xlOO and x400).
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Fig. 4 (Roizenblatt). Biopsy specimen of the con junctiva showing exfoliation of the epithelial layer, round cell infiltration in the stroma, and tuberculoid granulomas without caseation.
establish evidence of fungal infections such as paracoccidioidomycosis, sporotrichosis, histoplasmosis, and chromoblastomycosis, or bacterial infections such as rhinoscleroma, tuberculosis, or leprosy on culture or smear. The ocular aspects of the patient sug gest various other diseases, including tra choma, syphilis, tuberculosis, leprosy, trypanosomiasis, and onchocerciasis. Trachoma was not considered a likely cause, as the vascularization occurred in the superficial stroma and did not re-
Fig. 5 (Roizenblatt). Facial aspect of patient after treatment.
FEBRUARY, 1979
semble the pannus seen with trachoma. Additionally, there was no supporting ev idence of trachoma, such as follicjes, conjunctival scarring, trichiasis, or Herbert's pits. The vascularization of the cornea and the inflammatory reaction associated with syphilis is predominate in deep stroma, in contrast to the superficial inflammation in this case. Additionally, there were no other signs of congenital syphilis, such as Hutchinson teeth, raspberry molars, sad dle nose, or deafness, and the serologic tests virtually ruled out the probability of syphilis. Tuberculosis must be considered in the differential diagnosis of interstitial keratitis; however, the patient was anergic to tuberculin, the chest x-ray film was nega tive, and the biopsy specimen showed no caseation. Lepromatous leprosy may cause a su perficial vascularization of the cornea to gether with skin changes, somewhat simi lar to that noted in this patient, but smears and bjopsy usually show the specimen teeming with organisms. Trypanosimiasis and onchocerciasis may cause corneal vascularization, but these diseases are not seen in the areas in which the patient lived, and the organ isms were not seen on biopsy. Other causes of interstitial keratitis in cluding herpes simplex keratitis (herpes simplex is the great imitator), lymphogranuloma venereum (which may cause a segmental sclerokeratitis), and mumps (which do not cause vascularization of the corneal stroma) have not been ruled out as the cause of the ocular lesions, but their presence is unlikely, and they would not cause the skin lesions. American leishmaniasis has various forms, including cutaneous American leishmaniasis, mucocutaneous American leishmaniasis, disseminated anergic leish maniasis, and Mexican leishmaniasis. The present case falls into the category of mucocutaneous American leishmaniasis.
VOL. 87, NO. 2
AMERICAN LEISHMANIASIS
Ocular findings associated with mucocutaneous American leishmaniasis include involvement of the eyelids 6 , 8 , 9 , 1 1 , 1 7 " 1 9 , 2 6 , 2 8 and conjunctiva, 10,13,15,18,23,25-27 described as eyelid edema; scarring or even destruc tion of the tarsus; and nodular granulomas of the tarsal or bulbar conjunctiva. Ulceration of the conjunctiva and cornea and ulcerative 5 , 6 , 1 8 , 2 5 - 2 7 or granulomatous 5,10,16,18,24 "" 26 lesions of the cornea, as well as interstitial keratitis 5,6,9,13,18,26 also have been described. The eye may be involved by contiguous spread to the eyelid and conjunctiva, by hematogenous spread, or by inoculation of the conjunctiva by the patient's own fingers. 5,6,13,15,17,18,20,26 In our patient, the most likely spread was by way of the blood stream, as there were no signs of tarsal or conjunctival involvement. The disfigurement and lack of satisfac tory treatment have often hampered case findings of patients with leishmaniasis, and when found, little attention has been paid to the ocular findings. Case findings and ophthalmologic examination would reveal many more cases than the reports suggest. SUMMARY
A 58-year-old man with American (mucocutaneous) leishmaniasis had the un usual ocular complication of interstitial keratitis. Although the parasite was not identified, clinical laboratory and histologic findings were compatible with leishmaniasis, and tests ruled out other causes. Ulcerative areas improved after treatment with 1 g of amphotericin B. REFERENCES 1. Lindenberg, A.: A ulcera de Bauru e o sen microbio. Communicacao preventiva. Rev. Med. Sao Paulo 12:116, 1909. 2. Carini, A., and Paranhos, U.: Identifieacao das ulceras de Bauru ao botao do Oriente. Communicac ao preliminar. Rev. Med. Sao Paulo 12:111, 1909. 3. Vianna, G.: Sobre uma nova especie de Leishmania (Nota preliminar). Brasil-Med. 25:411, 1911. 4. Forattini, O. P.: Leishmanioses tegumentares
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