Bilateral Ptosis and Lower Eyelid Ectropion Secondary to Cutaneous Leishmaniasis

CLINICOPATHOLOGIC REPORT Bilateral Ptosis and Lower Eyelid Ectropion Secondary to Cutaneous Leishmaniasis

Imtiaz A. Chaudhry, MD, PhD; Camille Hylton, MS; Beatrice DesMarchais, MD

73-year-old white woman had a 14-month history of an extensive, disfiguring facial lesion involving the cheeks, nose, and eyelids, resulting in exposure keratopathy. A biopsy of the facial lesion established the diagnosis of cutaneous leishmania, and the lesion responded to treatment with itraconazole. A Arch Ophthalmol. 1998;116:1244-1245

Leishmaniasis, a parasitic infection caused pion and ptosis with weakened levator pal- by a hemoflagellate protozoan of the ge- pebral superior function, requiring use of nus Leishmania, is rarely seen in the United her frontalis muscles to assist in lid eleva- States. It is endemic, however, in the Medi- tion. Visual acuity was correctable to 20/25 terranean littoral, the Middle East, Af- OD and 20/30 OS. Slitlamp examination re- rica, and Central Asia. With the current vealed bilateral inferior superficial punc- regularity of worldwide travel, physi- tate keratopathy and palpebral conjuncti- cians around the world must have an in- val injection and hypertrophy with dex of suspicion for this diagnosis. We de- granulation and diffuse papillae. The re- scribe a patient from Italy who had facial sults of the remainder of the ocular exami- cutaneous leishmaniasis and associated bi- nation were normal. Systemic examina- lateral ptosis, ectropion, and exposure tion did not reveal any lymphadenopathy keratopathy. or hepatosplenomegaly. A biopsy specimen was obtained from REPORT OF A CASE the edge of the lesion on the cheek. He- matoxylin-eosin staining of the formalin- A 73-year-old woman who had immi- fixed specimen showed chronic inflam- grated 4 months previously from Italy, matory infiltration with focal areas of where she worked on a farm, was first seen granulomatous inflammation (Figure 2, with a chief ocular complaint of facial top).The inflammatory cells consisted pri- sores, associated with itching, burning, marily of histiocytes, plasma cells, and lym- photophobia, and mucoid discharge. Four- phocytes without evidence of necrosis. teen months previously, she noted a small Multiple Leishman-Donovan bodies were pimple on her nose, which gradually de- present within the cytoplasm of histio- veloped into an extensive facial lesion that cytes as well as in free form in the der- did not respond to treatment with a vari- mis, confirming the diagnosis of leish- ety of antibiotics. The medical and surgi- maniasis (Figure 2, bottom). After 2 cal histories were unremarkable. months of treatment with itraconazole, 100 External examination revealed an ex- mg orally daily, lubricating drops, and oint- tensive disfiguring, verrucous facial lesion ment, a significant improvement was noted with multiple areas of crusting, lichnifica- in the facial lesions, ptosis, ectropion, and tion, and vegetation primarily involving the keratopathy. nose and cheeks with extension to the eyelids (Figure 1). There was bilateral ectro- COMMENT

From the Department of Ophthalmology and Visual Sciences, Yale University School of Cutaneous leishmaniasis, which is caused Medicine, New Haven, Conn. Dr Chaudhry is now with the Department of by Leishmania major and Leishmania tropica Ophthalmology, Baylor College of Medicine, Houston, Tex. and transmitted by sand flies, typically be-

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©1998 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/24/2021 In most immunocompetent in- dividuals, the infection and cutaneous manifestations spontaneously re- solve. Otherwise treatment with pentavalent antimonial, sodium sti- bogluconate (pentostam), is typically curative. However, serious systemic adverse effects, such as fatal cardiac arrhythmias, hepatotoxic- ity, nephropathy, and gastrointesti- nal systemic effects, such as nausea and vomiting, limit its use,3 and more tolerable antifungal agents (ketaconazole, itraconazole, and am- photericin B), as well as pentami- Figure 1. External appearance of cutaneous leishmaniasis demonstrating extensive dine and allopurinol, may be equally disfiguring, verrucous facial lesion with multiple effective. Systemic therapy with in- areas of crusting, lichenification, vegetation, terferon gamma has also been shown bilateral ptosis, and ectropion. to be effective in the treatment of cu- 5 gins as a small erythematous papule taneous leishmaniasis. Residual on the face or extremity at the site of scarring and deformity may re- inoculation. It progresses slowly to quire surgical correction. an indurated, verrucous plaquelike nodule, reaching 1 to 2 cm in diam- Accepted for publication April 17, eter, and after several weeks or 1998. Corresponding author: Imtiaz A. months develops into a shallow Figure 2. Photomicrographs of biopsy specimen ulcer containing a central crust.1 that show dermis containing massive infiltrate of Chaudhry, MD, PhD, Baylor College Small satellite nodules are char- inflammatory cells (top) (hematoxylin-eosin, of Medicine, 6560 Fannin St, Suite original magnification ϫ100) and 902, Houston, TX 77030. acteristically present at the edge of the granulomatous reaction (prominent histiocytic lesion. Involvement of the lid and infiltrate) with numerous extracellular and ocular adnexa may simulate dac- intracellular organisms (Leishman-Donovan REFERENCES bodies) (bottom) (hematoxylin-eosin, original ryocystitis, a chalazion, or a tumor magnification ϫ400). 2 of the lid. As in the present case, 1. Font RL. Eyelids and lacrimal drainage system. In: ocular complications of cutaneous Spencer WH, ed. Ophthalmic Pathology: An At- leishmaniasis may include cicatri- microscopic examination, the cuta- las and Textbook. 4th ed. Philadelphia, Pa: WB Saunders Co; 1997:2364-2367. cial ectropion and ptosis. Other neous lesion discloses a moderate 2. Ferry AP. Cutaneous leishmaniasis (Oriental infectious conditions that can dermal infiltrate of lymphocytes and sore) of the eyelid. Am J Ophthalmol. 1977;84: mimic the clinical picture typical plasma cells intermixed with a 349-354. of leishmanias include leproma- granulomatous inflammatory reac- 3. Glover AT. Eyelid infections. In: Albert DM, Jako- tous leprosy, sporotrichosis, herpes tion composed of histiocytes, epi- biec FA, eds. Principles and Practice of Ophthal- mology. Philadelphia, Pa: WB Saunders Co; 1994: zoster ophthalmicus, herpes sim- theloid cells, and multinucleated gi- 1702-1712. 4 plex, vaccina, anthrax, toxoplas- ant cells. The Leishmania organism 4. Chu FC, Rodriques MM, Cogan DG, Franklin AN. mosis, histoplasmosis, blastomyco- within the cytoplasm of the histio- Leishmaniasis affecting the eyelids. Arch Oph- sis, and candidiasis.3 cytes appears as a round to oval thalmol. 1983;43:91-93. 5. Kolde G, Luger T, Sorg C, Sunderkotter C. Suc- The diagnosis of the infection body, measuring 2 to 3 µm in diam- cessful treatment of cutaneous leishmaniasis us- is established based on clinical sus- eter, containing a nucleus and a ing systemic interferon-gamma. Dermatology. picion and confirmed by biopsy. On small kinetoplast (paranucleus).1 1996;192:56-60.

100 Years Ago in the ARCHIVES

A look at the past. . .

SRAELSOHN (Smolensk) gave an account of 42 cases of Distichiasis and trichiasis cured by transplantation of a piece of mucous membrane from the lip. Kuhnt proceeded in the same manner, only without the use of sutures, I and Matkovec had had success by the same method. Reference: Arch Ophthalmol. 1898;27:105.

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