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Year : 2021  |  Volume : 10  |  Issue : 1  |  Page : 50-53

Isolated leishmaniasis at lower lip

Department of Otorhinolaryngology, Institute of Medical Sciences (IMS) and SUM Hospital, Siksha ‘O’ Anusandhan (Deemed to be University), Bhubaneswar, Odisha, India

Date of Submission27-Mar-2020
Date of Decision26-May-2020
Date of Acceptance01-Jun-2020
Date of Web Publication4-Mar-2021

Correspondence Address:
Santosh Kumar Swain
Professor, Department of Otorhinolaryngology, IMS and SUM Hospital, Bhubaneswar 751 003, Odisha
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/JCSR.JCSR_31_20

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Leishmaniasis caused by Leishmania spp. is a vector-borne disease and transmitted by the Phlebotomus sand fly. Lip is an extremely rare site for this lesion. The lesion often begins as erythematous papules, and slowly enlarges and ulcerates. The diagnosis is usually based on clinical features and laboratory tests. Here, we reported a case of an isolated lesion of the leishmaniasis at the lower lip. The patient presented with an ulcerative lesion and initially not healed by conservative treatment such as antibiotics. Histopathological and smear examination confirmed the diagnosis. The isolated lesion of leishmaniasis at the lower lip is rare and often confusing, which challenges diagnosis. Clinicians should keep in mind about this isolated localised lesion as a differential diagnosis of chronic pathology of the lower lip so that delayed diagnosis and treatment can be avoided.

Keywords: Leishmaniasis, lower lip, mucocutaneous leishmaniasis, zoonotic disease

How to cite this article:
Swain SK, Anand N. Isolated leishmaniasis at lower lip. J Clin Sci Res 2021;10:50-3

How to cite this URL:
Swain SK, Anand N. Isolated leishmaniasis at lower lip. J Clin Sci Res [serial online] 2021 [cited 2021 Aug 3];10:50-3. Available from: https://www.jcsr.co.in/text.asp?2021/10/1/50/310764

  Introduction Top

Leishmaniasis is a protozoan infectious disease caused by the Leishmania parasite, which occurs through the bite of an infected sand fly. Leishmaniasis is a group of infective diseases, occurs by a protozoan parasite, which belongs to the genus Leishmania (order Kinetoplastida). This zoonotic disease is transmitted by sand flies of the species, such as Phlebotomus and Lutzomyia. The reservoirs of the Leishmaniasis are a wide range of mammals and rarely human beings.[1] Leishmaniasis is classified into three types, varying in severity from spontaneous healing skin ulcers in cutaneous leishmaniasis (CL) to destructive type of mucocutaneous leishmaniasis (MCL) to more fatal variety of visceral leishmaniasis (VL). CL is found throughout South America, Africa, Asia, Middle East and Mediterranean regions.[1] Leishmaniasis has diverse clinical presentations, which lead to public health problems in endemic areas.[2] This disease can affect the skin, mucocutaneous area and viscera. Often, the leishmaniasis is mistaken for certain skin disorders such as fungal infections, eczema, bacterial infections and chronic ulcers, hence the delay in the diagnosis and treatment of this disease. In many cases, patients consult many clinicians before confirming the diagnosis of CL. Here, we present a case of isolated lower lip leishmaniasis, which confused us as a chronic ulcer and not healed by local treatment.

  Case Report Top

A 55-year-old male attended the outpatient department of otorhinolaryngology with complaints of a painless wound at the lower lip. The ulcerative wound started 4 months back at the centre of the lower lip as a small nodule and slowly extending to the whole of the lower lip and then spontaneously ulcerated. The ulcerative lesion of the lower was covered by a dried crust [Figure 1]. He consulted with a local physician, but the condition did not improve with a course of antibiotics. He had no past history of trauma to the lower lip. On examination, the lesion measured approximately a 3 cm × 5 cm and appeared as painless, indurated, necrotic ulcer on the lower lip. There was no palpable lymphadenopathy in the body of the patient. The patient had no cutaneous lesion anywhere of the body. He had no fever, hepatosplenomegaly and weight loss. Systemic examination showed no abnormality. General examinations and routine blood investigations were within normal limit. Human virus immunodeficiency test was also negative, and slit-skin smears from the lesions stained with Ziehl–Neelsen stain revealed negative for acid-fast bacilli. Then, tissue from the chronic ulcerative lesion was excised and sent for the histopathological examination, and also smear was extracted from the ulcerative margin to rule out any malignancy and chronic granulomatous lesions. This was done under local anaesthesia. The smear from the ulcerative lesion of the lower lip revealed intracellular and extracellular Leishmania amastigotes in Giemsa stain [Figure 2]. The patient was treated with sodium stibogluconate for 3 wk (intramuscular 20 mg/kg daily for 3 wk). The lesions from the lower lip disappeared, and there has been no evidence of the recurrence after 1 year.
Figure 1: Clinical photograph showing erythematous crusted ulceration on the lower lip

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Figure 2: Photomicrograph of the smear from ulcer showing numerous intracellular and extracellular Leishmania amastigotes (Giemsa, ×1000)

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  Discussion Top

Human leishmaniasis is a vector-borne protozoan-induced disease transmitted by sand fly. Leishmaniasis is caused by unicellular, flagellate, intracellular protozoa belongs to the genus Leishmania transmitted from animals to human beings through phlebotomine sand fly vectors. Leishmaniasis is classified into cutaneous, mucocutaneous and visceral. The CL is a protozoan-induced disorder, which affects people in around 98 tropical, subtropical and Mediterranean countries.[1] Leishmaniasis affects people in areas of South America, Southern Europe, Africa and Asia.[1] It has been a marked increase in non-endemic zones because of increased travel both for tourism and returning of military personnel from endemic areas such as Iran and Afghanistan, where the incidence is high.[3] The Leishmania species responsible for visceral disease may also cause the cutaneous variety.[4] Leishmania major, Leishmania tropica, Leishmania aethiopica and Leishmania infantum are the aetiological agents for leishmaniasis in the old world, whereas Leishmania donovani and Leishmania braziliensis are the aetiological agents for MCL at South and Central America and cause VL all over the world, respectively.[5] The CL is the most common type and is characterised by the presence of ulcerations with a well-defined erythematous margin, and a central crust which is usually haemorrhagic and found in the exposed area of the body.[6]

The earliest lesion is cutaneous nodules in the location where sand fly inoculate, and then, the parasites attack the reticuloendothelial system of the body. The clinical presentations include intermittent fever, hepatosplenomegaly, anaemia, agranulocytosis and thrombocytopenia. Patients are usually susceptible to secondary infection, which may produce gastrointestinal and pulmonary infections, which may lead to death in case of untreated cases. Mucosal disease often becomes evident several years after healing of the cutaneous lesions. The CL is characterised by single or multiple erythematous papules, which may be seen just after bite of the sand fly. The papule slowly grows to around 2 cm over a period of several weeks and assumes a dusky violaceous hue. Eventually, this lesion changed to crusted at the centre with a shallow ulcer and raised indurated border (volcano sign). There are small satellite papules found at the periphery of the lesion, and sometimes, subcutaneous nodules along the course of proximal lymphatic may develop. Ulcerations often persist for 3–6 months or longer period. Then, these ulcerative lesions heal with a slightly depressed scar. The isolated lesion of the lower lip leishmaniasis is very uncommon. Lip leishmaniasis is clinically characterised by gradual involvement of the one lip to both lips and finally leads to macrocheilitis.[7] The lesion at the lip is started with nodule or plaque and undergoes an ulceration, which may be covered with scaling and crusts. The consistency of the whole lesion is characterised as parenchymatous hard. The lip involvement may occur from direct spread from the nearby skin lesions or from haematogenous or lymphatic dissemination of the leishmaniasis amastigotes from the skin.[8] Patients of isolated lip leishmaniasis are usually in good health condition. The incubation period of CL varies from 2 weeks to several months along with a varied spectrum of clinical features such as cutaneous ulceration to mucosal involvement.[9] The ulcerations have a characteristic erythematous border, and a central crust which is usually haemorrhagic and seen in exposed parts of the body.[9] The differential diagnosis of this clinical entity includes herpes labialis, syphilitic chancre, Melkersson–Rosenthal syndrome, cheilitis granulomatosa, orofacial granulomatosis, Wegener granulomatosis, oral Crohn's disease, sarcoidosis, discoid erythematosus, skin tuberculosis, discoid lupus erythematosus, leprosy, mycotic infections, neuroma, basal cell carcinoma and squamous cell carcinoma.[10],[11]

Common methods used for the diagnosis of leishmaniasis are the demonstration of the parasites in the tissue with the help of light microscopic examination of the stained specimen in vivo or vitro. Immunodiagnosis can be done by detection of the parasite antigen in the blood, tissue or urine sample by detecting the antileishmanial antibodies or assessment of Leishmania-specific cell-mediated immunity. The detection of parasites DNA in tissue samples can be done by polymerase chain reaction (PCR).[12] Isolation of Leishmania parasite in dermal macrophages with the help of skin biopsy or dermal scraping can confirm the diagnosis of disease. When doing slit smear, it is important for scraping the edges of the lesion. However, in chronic illness, the parasites may be rare. Hence, failure to see amastigote in histopathology does not exclude the diagnosis of MCL. PCR, culture and immunofluorescent-antibody can be considered as significant diagnostic techniques.[6]

Once the diagnosis confirmed, the treatment often used is antimony medications. The single therapy may not be helpful always. The combination treatment includes allopurinol and antimony drugs, which usually shorten the duration of treatment period. Combination therapy is usually successful in resistant cases of VL.[12] Local treatment is done instead of systemic treatment if there is no risk of MCL. Local treatment acts as an adjuvant to systemic treatment and helps to accelerate the healing process of the lesion. Topical ointment consisting of 15% paromomycin and 12% methylbenzethonium chloride is applied for two times daily for 20 days and it also helps to heal the lesion.[2] Paromomycin ointment is helpful in L. tropica, L. major, Leishmania mexicana and Leishmania panamensis. Both paromomycin and imiquimod may be useful for local application in L. braziliensis. Oral preparation of zinc sulphate and fluconazole is helpful in L. major. Intramuscular pentamidine is needed for Leishmania guyanen in CL where systemic antimony is not much effective.[13] After the use of pentavalent antimonials, it remains the choice of drug for both cutaneous and VL in most of the world. Pentamidine and amphotericin B are the second line of choice antileishmanial drugs, although they need a long course of parenteral administration.[14] Imidazoles are useful therapeutic agents in case of CL. It acts by blocking the synthesis of ergosterol, which is present in the cell wall of Leishmania spp. The efficacy of the ketaconazole is usually variable in CL.[15] Majority of the drugs used leishmaniasis are toxic and do not eliminate the parasites from the infected patients. Failure to treat this disease is usually due to increased chemoresistance of the parasites. On the basis of the causative Leishmania species, the choice of the drug is decided by the clinicians. Although spontaneous healing is the rule, the rate of recovery varies depends on the basis of the species of etiological agents and may need months or years to heal completely.

Leishmaniasis is a tropical dermatose with diverse clinical presentations. It is caused by a protozoa Leishmania transmitted by the sand fly. It may present in an unusual shape or unusual location, which causes a diagnostic challenge to the clinicians. The leishmaniasis lesion at the lower lip is often mistaken for leprosy, fungal dermatitis and malignancy. Histopathological and smear examinations should be done early for any ulcerative lesions more than 2 wk under local anaesthesia and ensure the diagnosis. Our observation suggests that when an unusual ulceration is noticed on the lip leishmaniasis should also be considered in the differential diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

  References Top

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Ameen M. Cutaneous leishmaniasis: Advances in disease pathogenesis, diagnostics and therapeutics. Clin Exp Dermatol 2010;35:699-705.  Back to cited text no. 3
Bryceson AD, Hay RJ. Parasitic worms and protozoa. In: Rook A, Wilkinson DS, Ebling FJG, Champion RH, editors. Rook/Wilkinson/Ebling Textbook of Dermatology. 6th ed., Vol. 5. Oxford, UK: Blackwell Science Ltd.; 1998. p. 1411-4.  Back to cited text no. 4
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Goto H, Lindoso JA. Current diagnosis and treatment of cutaneous and mucocutaneous leishmaniasis. Expert Rev Anti Infect Ther 2010;8:419-33.  Back to cited text no. 6
Roundy S, Almony J, Zislis T. Cutaneous leishmaniasis of the lower lip in a United States soldier. J Oral Maxillofac Surg 2008;66:1513-5.  Back to cited text no. 7
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.  Back to cited text no. 8
David CV, Craft N. Cutaneous and mucocutaneous leishmaniasis. Dermatol Ther 2009;22:491-502.  Back to cited text no. 9
Boukhris I, Azzabi S, Cherif E, Kechaou I. Orofacial granulomatosis: Do not forget leishmaniasis. BMJ Case Rep 2015;2015:bcr2015211919.  Back to cited text no. 10
Saab J, Fedda F, Khattab R, Yahya L, Loya A, Satti M, et al. Cutaneous leishmaniasis mimicking inflammatory and neoplastic processes: A clinical, histopathological and molecular study of 57 cases. J Cutan Pathol 2012;39:251-62.  Back to cited text no. 11
Kirigi G, Mbuchi MW, Mbui JK, Rashid JR, Kinoti DM, Njoroge SN, et al. A successful treatment of a Kenyan case of unresponsive cutaneous leishmaniasis with a combination of pentostam and oral allopurinol: Case report. East Afr Med J 2010;87:521-4.  Back to cited text no. 12
Wortmann G, Hochberg LP, Arana BA, Rizzo NR, Arana F, Ryan JR. 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.  Back to cited text no. 13
Amato VS, Tuon FF, Bacha HA, Neto VA, Nicodemo AC. Mucosal leishmaniasis . Current scenario and prospects for treatment. Acta Trop 2008;105:1-9.  Back to cited text no. 14
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