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In recent years, exceptions to these classic forms of the diseases have been reported. Mild visceral disease (low-grade fever, asthenia, and moderate hepatomegaly) without cutaneous involvement was caused by a cutaneous species in Iraq.2 Disseminated polymorphic cutaneous disease (10–300 cutaneous lesions that are a mixture of acneiform, papular, nodular, and ulcerated types) often with concomitant mucosal involvement was observed in northeastern Brazil.3 Erysipiloid cutaneous disease was recently the subject of a case report.4
We report a patient with an unusual case of cutaneous leishmaniasis. Symptoms included cutaneous lesions that were boggy indurations as well as, papules, and ulcers; mucosal disease; and systemic involvement in the sense of asthenia and hepatocellular abnormalities.
By the end of December 2006, cutaneous lesions had appeared on her forehead, right wrist, left arm, chest, abdomen, and back. The lesions on her wrist and arm consisted of large boggy swellings with and without central ulcerations. She had painful crusts in her right ear, and throat pain that limited swallowing. In addition, she had low-grade fevers and chills. At this point, the patient was taking 2,000–2,500 mg of aspirin per day for the throat pain.
In early January 2007, she was referred to a dermatology unit. Biopsy specimens of the original "chigger bite" lesion on her right arm, a back lesion, and the right wrist lesion were sent to the local dermatopathology laboratory, where Leishmania were visualized, and then to the Centers for Disease Control and Prevention (Atlanta, GA). Leishmania organisms were visualized by staining with Giemsa in all biopsy specimens, and Leishmania braziliensis was cultured from one specimen and identified by isoenzyme electrophoresis. Flexible laryngoscopy on January 19, 2007, showed an erythematous crusty nasal cavity with hypopharynx findings of intense erythema with boggy mucosa and scattered petechea. Laboratory tests showed a hematocrit of 40% and a leukocyte count of 5,800 cells/mm3 (neutrophils = 69%). Results of liver function tests were abnormal (aspartate aminotransferase [AST] = 108 U/L, alanine aminotransferase [ALT] = 169 U/L), and results of a test for HIV were negative.
By February 2007, the cutaneous lesions continued to appear and spread. Particularly notable lesions were 1) a crusted exudative lesion covering approximately two-thirds of interior of the right ear (Figure 1A
) accompanied by a prominent cervical lymph node, 2) a swollen nodule (approximately 2 cm in diameter) on the left elbow, 3) four ulcers surrounded by boggy swellings on the left wrist, 4) a large boggy swelling plus several ulcers on the right wrist (Figure 1B), 5) many small ulcers on the back, and 6) many ulcers on the left leg and the right leg (Figure 1C). Throat pain had increased such that even saliva was hard to swallow. Although the patient had no difficulty in breathing, she was coughing up bloody exudates, and her weight had decreased to 130 lbs. Chills had increased and she was almost prostrate with no energy.
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The standard treatment for leishmaniases, pentavalent antimony (used under an investigational new drug application in the United States), frequently causes hepatocellular dysfunction and might exacerbate the unknown hepatic process. The patient was treated with a new drug (miltefosine) because it rarely causes hepatocellular dysfunction, is orally administered (antimony is administered by injection), and was recently shown to be effective for mucosal leishmaniasis in Bolivia.5 Treatment with miltefosine was initiated on February 8, 2007, at the standard dose of 2.5 mg/kg/day, three times a day, with meals for 28 days. Other than vomiting when oxycodone was coadministered for throat and ear pain, miltefosine was well tolerated with side effects limited to nausea 30–120 minutes after each dose. After a gastroenterology consultation, the patient stopped taking aspirin because the high doses she was using may have caused or contributed to her hepatitis.
Two weeks after beginning treatment with miltefosine and stopping treatment with aspirin, liver function test results had improved (AST = 96 U/L, ALT = 140 U/L). By the end of the 28-day treatment period, liver function test results had essentially normalized (AST = 48 U/L, ALT = 40 U/L). Because the cutaneous lesions had improved somewhat but had not resolved completely, three additional courses of miltefosine (2.5 mg/kg/day for 28 days) were administered until June 2007. By April–June 2007, the ear lesion had only a small amount of ulcer and discharge (Figure 2A). The boggy lesions on the arm had improved considerably (Figure 2B), as had many of the leg lesions (Figure 2C). However, new papules and ulcers had appeared on the legs during therapy with miltefosine. There was mild pain with swallowing but no exudate. The patient’s energy level improved and chills were absent, although her weight had decreased to 112 lbs.
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Despite substantial improvement in her upper extremity lesions, throat symptoms, ear disease, and elevated levels of liver enzymes, by the end of four courses of miltefosine, some throat symptoms and extremity lesions remained and new leg lesions had appeared. Although she received 280 mg of miltefosine/kg of body weight, probably more than any other U.S. citizen to date, the drug was well tolerated with only minor gastrointestinal difficulties. One course of antimony was then given with no discernable improvement in the short period of 28 days during which it was administered. Complete cure was then rapidly accomplished by two 20-mg/kg courses of Ambisome.
The boggy characteristics of some of her lesions, symptoms in multiple organs, and the slow overall response to a drug to which some of her lesions were susceptible suggests that the patient has a specific immunologic defect in her ability to control infection with Leishmania. We did not have the opportunity to investigate this hypothesis by evaluating the interaction of the parasite with her immune cells in vitro.
As travel continues to facilitate contact of a wide variety of human hosts with parasitic microorganisms, both classic and unusual presentations such as this one are likely to be seen more frequently for leishmaniasis.
Received April 23, 2008. Accepted for publication July 29, 2008.
Acknowledgment: We thank the Division of Parasitic Diseases at the Centers for Disease Control and Prevention (Atlanta, GA) for advice and assistance.
* Address correspondence to Jonathan Berman, 6205 Poindexter Lane, Rockville, MD 20852. E-mail: Jbe9320457{at}aol.com 
Authors’ addresses: Steven Mings, 100 Warm Springs Avenue, Boise, ID 83712, E-mail: Dermn8r{at}hotmail.com. Jill C. Beck, Division of Otolaryngology, Department of Surgery, St. Luke’s Regional Medical Center, 900 North Liberty, Suite 400, Boise, ID 83704, E-mail: Jillbeck{at}swient.org. Christopher Davidson, Botanical Research Foundation of Idaho, 637 Warm Springs Avenue, Boise, ID 83712, E-mail: cdavidson3{at}juno.com. Andrew L. Ondo, 4351 East Lohman Avenue, Suite 208, Las Cruces, NM 88011, E-mail: andyondo{at}yahoo.com. Stuart D. Shanler, 100 Winston Drive, Cliffside Park, NJ 07010, E-mail: sdsmd{at}yahoo.com. Jonathan Berman, 6205 Poindexter Lane, Rockville, MD 20852, E-mail: Jbe9320457{at}aol.com.
Reprints requests: Steven Mings, 100 Warm Springs Avenue, Boise, ID 83712.
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