|
CASE REPORT |
|
| Year : 2009 | Volume
: 54
| Issue : 5 | Page : 37-40 |
|
| Leishmaniasis of the face: Report of a case |
|
|
Mohammad Hosein Kalantar Motamedi1, Parvin Alami Harandi2, Taghi Azizi3
1 Trauma Research Center, Baqiyatallah Medical Sciences University, Attending Faculty, Azad University of Medical Sciences, Tehran, Iran
2 Clinic of Dermatology, Baqiyatallah Medical Center, BMSU, Tehran, Iran
3 Department of Pathology, Baqiyatallah Medical Center, BMSU, Tehran, IR, Iran
Correspondence Address:
Mohammad Hosein Kalantar Motamedi
Africa Expressway, Golestan St., Giti Blvd. No. 11 Tehran, 19667, IR
Iran
 Source of Support: None, Conflict of Interest: None  | Check |
|
|
 Abstract | | |
Leishmaniasis is a parasitic disease prevalent throughout the world. According to the geographic region in which different Leishmania species are found it can present variably. Because of the increase in both international travel and immigrant populations, the numbers of reported cases of leishmaniasis is on the increase. It is important that clinicians are able to recognize the cutaneous lesions and unusual presentations of leishmaniasis (CL). Our case was an incidental finding during treatment of a gunshot wound. The lesions were mistaken for facial pimples because they were numerous and the patient was a teenager. The patient was treated with sodium stibogluconate.
Keywords: Leishmaniasis, cutaneous leishmaniasis, acne like lesions
How to cite this article:
Motamedi MK, Harandi PA, Azizi T. Leishmaniasis of the face: Report of a case. Indian J Dermatol 2009;54, Suppl S1:37-40 |
| Introduction | |  |
Leishmaniasis is a parasitic disease caused by the hemoflagellate, Leishmania spp. The parasite is transmitted by the bite of an infected female phlebotomine sandfly. The disease is prevalent throughout the world and in at least 88 countries. [1] It occurs throughout Africa, Asia, South America, the Middle East, and the Mediterranean regions. Leishmaniasis is a disease with diverse clinical manifestations, and is a public health problem in endemic countries such as Brazil. [2]
In humans, the disease can affect the skin, viscera, or mucocutaneous areas, varying according to the geographic region in which different Leishmania species are found as well as the host response. Infections with Leishmania are increasing worldwide because of tourism and job-related travel. [3] In patients with a weak immune response, the reticuloendothelial system is laden with infected macrophages, whereas in subjects with an increased immune response, granulomas form in the lymph nodes, liver, and spleen, with few clinical symptoms. [4] A localized lesion may form at the site of the insect bite. In endemic areas, permanent immunity develops to a particular species of Leishmania , with inapparent infection. Because of the increase in both international travel and immigrant populations, the numbers of reported cases of leishmaniasis is likely to increase. It is therefore important that clinicians are able to recognize the cutaneous lesions and unusual presentations of leishmaniasis. [4],[5] Clinicians in rural and remote areas may be the first to confront such patients presenting with cutaneous or mucocutaneous leishmaniasis. They can readily diagnose such patients presenting with nonhealing papular leasions by obtaining smear samples from the depth of the lesions and staining with Giemsa or hematoxylin-eosin and recognition of the parasite or Donovani bodies via light microscopy. Our case was an incidental finding during treatment of a patient suffering from a gunshot wound.
| Case History | | |
A 20-year-old male war refugee from Afghanistan presented to our clinic for treatment of a defect of the right body of the mandible following a gunshot wound in Afghanistan. Clinically, the skin and mucosa of the entrance and exit wounds of the bullet had healed. Several small, painless, papular lesions with indurated, erythematous borders and central ulcerations were evident on the forehead and cheek [Figure 1],[Figure 2]. There was no nerve deficit or adenopathy in the head and neck. The medical history was not significant and the patient was in general health. The patient underwent mandibular reconstruction of the defect using an autogenous corticocancellous bone graft. The patient received prophylactic antibiotics (cephalothin, gentamicin, and metronidazole) during the first postoperative week to prevent graft infection. Then, he was prescribed cephalexin which he took for the next three weeks. After two months the patient referred for removal of the hardware. It was noticed that the acne-appearing lesions were still present on the face and had enlarged slightly despite the extensive antibiotic therapy (which often cures ordinary pimples and acne) during this time period. Thus, smears and biopsies were indicated and done for the lesions under local anesthesia and submitted for pathologic examination. Light microscopy showed skin with hyperkeratosis, parakeratosis, and acanthosis. The dermis was filled with aggregates of large, pink, histiocytes, and mixed chronic inflammatory cells. The histiocytes contained dot-like organisms typical of cutaneous leishmaniasis [Figure 3],[Figure 4]. Treatment was started with sodium stibogluconate for three weeks. The lesions disappeared a month later and there has been no recurrence to date.
| Discussion | | |
Features
Leishmaniasis is an intracellular infection caused by the protozoa leishmania, which is transmitted by the sandfly. It occurs widely throughout Africa, Asia, South America, the Middle East, and the Meditteranean region. [5] The WHO reported a morbidity of nearly 12 million people in 88 countries around the world. [6] In the U.S. malaria and leishmaniasis continue to be the most common deployment-related illnesses. [7] Cutaneous leishmaniasis (CL) caused by L. tropica and L. major is endemic in Pakistan. [4] Leishmania donovani identified as the agent of CL in Sri Lanka is closely related to that causing visceral leishmaniasis in the Indian subcontinent. [8]
Course
Leishmania can affect the skin, viscera, or mucocutaneous areas, varying according to the geographic region in which different Leishmania species are found and the host response. [5] Infections become apparent after return from an endemic region. Depending on the Leishmania species and the host immune status, different forms of cutaneous (CL), mucocutaneous (MCL) ( L. brasiliensis complex) or visceral leishmaniasis ( L. donovani and L. infantum ) may develop. CL may heal spontaneously with scarring or evolve into diffuse CL (with reduced immune response to L. amazonensis , L. guyanensis , L. mexicana or L. aethiopica ) or into recurrent CL. [3]
Life cycle
The life cycle of the parasite involves two stages: The amastigote (Leishman-Donovan body) is present in hosts such as humans, dogs, and rodents. The parasite invades and multiplies in macrophages and other reticuloendothelial (RE) cells, which then rupture and release the organisms into the bloodstream. [5] When the female sandfly bites an infected host, the amastigotes enter the sandfly and develop into the infective promastigotes. The cycle is completed when the sandfly bites another host.
Prognosis
In patients with a weak immune response, the reticuloendothelial system is laden with infected macrophages, whereas in subjects with an increased immune response, granulomas form in the lymph nodes, liver, and spleen, with few clinical symptoms. A localized lesion may form at the site of the insect bite. In endemic areas, permanent immunity develops to a particular species of leishmania, with inapparent infection being common. CL (oriental sore) characterized by painless nodules that occur on exposed areas of the skin after the sandfly bite enlarge and ulcerate, and have a characteristic erythematous raised border. An overlying crust may develop. The lesions heal slowly over months or years, sometimes leaving a disfiguring scar. [5]
Diagnosis
Diagnostic criteria for CL include travel from an endemic area, ulcerated plaques or nodules on an exposed site which show no tendency towards healing over three to four weeks. Differential diagnostic considerations include ecthyma, other infectious ulcers, and neoplasms. Clinicians in rural and remote areas can readily diagnose patients with CL or MCL noninvasively by finding Leishmania (and characteristic Donovan bodies) in a smear or tissue biopsy stained using Giemsa or hematoxylin-eosin. Donovan bodies are found in smears of the blood or bone marrow, as well as in aspirates of lymph node, liver, or spleen. Apositive Leishmanin skin test (negative in the early stage); a culture of the parasite (in the Nicolle-Novy-McNeal culture medium), and isoenzyme or DNA studies are among the other diagnostic tests. [3] Anemia, neutropenia and thrombocytopenia may also be seen. Leishman-Serologic tests also are available. [5] Thepolymerase chain reaction (PCR) assay has also been reported for the diagnosis of cutaneous leishmaniasis (CL). [9]
Treatment
The causative species of CL determines the clinical features, courses, and treatment. Intralesional or systemic antimonials are the gold standard for the treatment of this disease. Paromomycin ointments are effective in Leishmania major, L. tropica, L. mexicana , and L. panamensis lesions. In L. braziliensis localized leishmaniasis, both paromomycin and imiquimod may be topically applied. Oral fluconazole and zinc sulfate are useful in L. major . Intramuscular pentamidine is required for L. guyanensis CL, for which systemic antimony is not effective. [10] Large lesions, or those in prominent areas of the body, can also be treated locally with surgical excision, curettage, or cryotherapy. Mucosal lesions usually involve the nose and larynx. Oral involvement is unusual and often becomes evident several years after resolution of the original cutaneous lesions. Oral lesions classically appear as mucosal ulcerations in the hard or soft palate. [2]
Visceral leishmaniasis (Kala-azar) is caused by L.Donovani , and usually affects young people, often with an incubation period of months or even years. Clinical symptoms include a characteristic biphasic fever, cough, and diarrhea. Splenic enlargement occurs, which may be massive, and hypersplenism is chiefly responsible for the pancytopenia seen. If left untreated, death usually occurs within 2 to 3 years because of pulmonary or gastrointestinal superinfection. Low red meat intake and poor zinc and retinol status may characterize a group at higher risk of symptomatic disease. [11],[12]
Medical management involves the parenteral use of pentavalent antimony compounds (e.g., sodium stibogluconate) given for up to 21 days. Patients with a poor response to the initial therapy are treated with intravenous amphotericin or pentamadine. The pentavalent antimony compounds have remained mainstay for nearly 75 years. However, emergence of resistance has led to the use of other compounds - amphotericin B, pentamidine, paromomycin, allopurinol etc. Amphotericin B, an antifungal macrolide polyene is is the only drug with highest cure rate. Recently, anticancer alkylphosphocholines have been found to be the most effective oral compounds. The most promising of these are miltefosine (hexadecylphosphocholine), edelfosine (ET-18-OCH(3)) and ilmofosine (BM 41.440). However, the recent focus has been on identifying newer therapeutic targets in the parasite such as DNA topoisomerases. [1]
Another important issue is co-infection of VL and HIV in recent years. [13] Many are refractory to treatment. [14] Diffuse cutaneous leishmaniasis in patients with AIDS has also been reported. The leishmaniasis-AIDS co-infection enhances invasive capacity for both causal microorganisms. [15] Atypical disseminated dermal leishmaniasis is also seen in HIV-infected patients. [16]
| Conclusion | | |
In our patient who presented for treatment of a maxillofacial gunshot wound attention was directed primarily to treatment of this injury. However, the facial lesions although noticed were thought to be acne. Suspicion occurred when the lesions did not regress and failed to heal after two months of antibiotic therapy. Because of the increase in Afghan immigrants from developing countries in neighboring states, the numbers of reported cases of leishmaniasis may be likely to increase. It is therefore prudent that clinicians especially in rural and remote areas be able to recognize, diagnose and differentiate the cutaneous lesions caused by leishmaniasis.
| References | | |
| 1. | Mishra J, Saxena A, Singh S. Chemotherapy of leishmaniasis: Past, present and future. Curr Med Chem 2007;14:1153-69.  [PUBMED] [FULLTEXT] |
| 2. | Motta AC, Lopes MA, Ito FA, Carlos-Bregni R, de Almeida OP, Roselino AM. Oral leishmaniasis: A clinicopathological study of 11 cases. Oral Dis 2007;13:335-40. [PUBMED] [FULLTEXT] |
| 3. | von Stebut E, Sunderkφtter C. Cutaneous leishmaniasis. Hautarzt 2007;58:445-58. |
| 4. | Ejaz A, Raza N, Iftikhar N. Recurrent cutaneous leishmaniasis presenting as sporotrichoid abscesses: A rare presentation near Afghanistan border. Dermatol Online J 2007;13:15. |
| 5. | Amin M, Manialli M. Cutaneous leishmaniasis of the face: Report of a case. J Oral Maxillofac Surg 2000;56:1066-9. |
| 6. | Wysluch A, Sommerer F, Ramadan H, Loeffelbein D, Wolff KD, Hφlzle F. The Leishmaniasis: A parasitel infection as differential diagnosis of malignant tumours of oral mucosa, a case report and review of literature. Mund Kiefer Gesichtschir 2007;11:167-73. |
| 7. | Nguyen DR. Illness in a redeployed soldier. Mil Med 2007; 172:541-3. [PUBMED] |
| 8. | Siriwardana HV, Noyes HA, Beeching NJ, Chance ML, Karunaweera ND, Bates PA. Leishmania donovani and cutaneous leishmaniasis, Sri Lanka. Emerg Infect Dis 2007;13:476-8. [PUBMED] [FULLTEXT] |
| 9. | Wortmann G, Hochberg LP, Arana BA, Rizzo NR, Arana F, RyanJR. Diagnosis of cutaneous leishmaniasis in Guatemala using a real-time polymerase chain reaction assay and the Smartcycler. Am J Trop Med Hyg 2007;76:906-8. |
| 10. | Minodier P, Parola P. Cutaneous leishmaniasis treatment. Travel Med Infect Dis 2007;5:150-8. [PUBMED] |
| 11. | Kumar R, Bumb RA, Ansari NA, Mehta RD, Salotra P. Cutaneous leishmaniasis caused by Leishmania tropica in Bikaner, India: Parasite identification and characterization using molecular and immunologic tools. Am J Trop Med Hyg 2007;76:896-901. [PUBMED] [FULLTEXT] |
| 12. | Bern C, Haque R, Chowdhury R, Ali M, Kurkjian KM, Vaz L, et al. The epidemiology of visceral leishmaniasis and asymptomatic leishmanial infection in a highly endemic Bangladeshi village. Am J Trop Med Hyg 2007;76:909-14. [PUBMED] [FULLTEXT] |
| 13. | Kallel K, Ammari L, Kaouech E, Belhadj S, Anane S, KilaniB, et al . Asymptomatic bearing of Leishmania infantum among tunisian HIV infected patients. Pathol Biol (Paris) 2007;55:521-4. |
| 14. | Troya J, Casquero A, Refoyo E, Fernαndez-Guerrero ML, Górgolas M. Long term failure of miltefosine in the treatment of refractory visceral leishmaniasis in AIDS patients. Scand J Infect Dis 2008;40:78-80. |
| 15. | Pιrez C, Solνas Y, Rodrνguez G: Diffuse cutaneous leishmaniasis in a patient with AIDS. Biomedica 2006;26:485-97. |
| 16. | Boumis E, Chinello P, Della Rocca C, Paglia MG, Proietti MF, Petrosillo N. Atypical disseminated leishmaniasis resembling post-kala-azar dermal leishmaniasis in an HIV-infected patient. Int J STD AIDS 2006;17:351-3. [PUBMED] [FULLTEXT] |
[Figure 1], [Figure 2], [Figure 3], [Figure 4] |
|
| This article has been cited by | | 1 |
Visceral leishmaniasis in a patient with cutaneous lesions, negative Leishman-Donovan bodies and immunological test: A case report |
|
| Khorvash, F., Naeini, A.E., Behjati, M., Abdi, F. | | Journal of Research in Medical Sciences. 2011; 16(11): 1507-1510 | | [Pubmed] | |
|
|
|
|
|
|
|
|
|
|
|
| Article Access Statistics | | | Viewed | 3348 | | | Printed | 96 | | | Emailed | 1 | | | PDF Downloaded | 161 | | | Comments | [Add] | | | Cited by others | 1 | | |
|
|
