Abstract

Background: Muco-cutaneous leishmaniasis (MCL) is a disease of new world and characterized by involvement of oral, nasal, pharyngeal, laryngeal and lip mucosae. Mucosal involvement is rarely seen with old world leishmaniasis. Recently, cases of lip mucosal leishmaniasis (LML) have been reported from old world and they are considered to be a part of cutaneous leishmaniasis (CL) rather than of MCL.

Methods: All newly diagnosed cases of CL were registered and detailed clinico-epidemiological variables were recorded. Data of patients having lesions over lips was extracted out and analysed separately. Imprint smears for Leishmania bodies and biopsies for histopathology were done only in atypical cases to confirm clinical diagnosis.

Results: Out of total 337 registered cases, 73 patients had lesions over lips either as sole presentation or in association with cutaneous lesions. Amongst affected cases of LML no predilection was found for specific gender or age group. Duration of disease varied from 20 days to one year but most [60 (67.3%)] cases presented with in six month of lesion onset. Although multiple lesions were also present but 42(57.5%) cases presented with single lesion. Nodules and plaques were the most frequent morphologies found in 28(38.4%) and 26(35.6%) cases respectively. Upper lip was most commonly affected site in 43(58.9%) cases followed by lower lip in 15(20.54%) and oral commissure in 9 (12.33%) cases. Out 73 cases 21(28.76%) cases had associated skin lesions and 3(4.1%) cases had extension of lesions to either oral or nasal mucosa (MCL). Biopsies and imprint smear examination were done in 11(15.06%) atypical cases. Out of these 11 cases, 8(72.7%) cases showed features suggestive of leishmaniasis on HPE and only 2(18.18%)  cases showed imprint smear positivity while in rest of  the cases histopathology and imprint smears were non-conclusive (excluded out from study). Most cases were treated effectively with intralesional injection of sodium stibogluconate (I/L SSG). But in 3 cases with MCL, intravenous SSG was given together with oral itraconazole to achieve complete cure. Most 69(94.5%) cases were cured completely after 3rd cycle of I/L SSG, only 3(4.1%) patients took 4 cycles and 1(1.7%) patient took 5 cycles for complete resolution of lesion.  Immediate side effects like pain, swelling, bleeding and ulceration were observed almost in every patient. Scarring, milia formation and reactive sub-mandibular lymphadenopathy were other less commonly observed side effects.

Conclusion: This is the first study on lip leishmaniasis from Himachal Pradesh. Lip leishmaniasis is not uncommon but very rarely reported entity from our region. Unusual presentation poses diagnostic challenge and we recommend facilities of culture, PCR and molecular studies in order to know about the exact characteristics of parasite and vector in this region. Sodium stibogluconate is the treatment of choice.

Keywords: Lip, Leishmaniasis, Epidemiology, Muco-cutaneous.