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Mucosal Leishmaniasis with Primary Oral Involvement: a Case Series and a Review of the Literature

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Mucosal Leishmaniasis with Primary Oral Involvement: a Case Series and a Review of the Literature Oral Diseases (2014) doi:10.1111/odi.12268 © 2014 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd All rights reserved www.wiley.com REVIEW ARTICLE Mucosal leishmaniasis with primary oral involvement: a case series and a review of the literature MD Mignogna1, A Celentano1, S Leuci1, M Cascone1, D Adamo1, E Ruoppo1, G Favia2 1Department of Neurosciences, Reproductive and Odontostomatological Sciences, University Federico II of Naples, Naples; 2Head Oral Surgery Unit, Interdisciplinary Department of Medicine, University of Bari, Bari, Italy OBJECTIVE: To analyze retrospectively a case series of Introduction primary oral leishmaniasis and to review the literature on head–neck primary mucosal leishmaniasis (ML) in Leishmaniasis is a parasitic disease caused by several pro- immunocompetent patients. tozoan species of the genus Leishmania, belonging to the SUBJECTS AND METHODS: A PUBMED search was family Trypanosomatidae. After malaria and African try- ‘ ’ carried out from 1950 to 2013. Clinical records of panosomiasis ( sleeping sickness ), the leishmaniases are patients with primary head–neck mucosal manifesta- the third most important group of vectorborne diseases tions of leishmaniasis were analyzed. In addition, clinical and are ranked ninth in terms of the global burden of dis- records between 2001 and 2012 of patients with pri- ease of all infectious and parasitic diseases (Prabhu et al, mary oral manifestations were collected in two indepen- 1992; Stockdale and Newton, 2013). dent hospitals. The leishmaniases are widely dispersed, with transmis- fi RESULTS: Our multicenter case series revealed seven sion to humans on ve continents, and are endemic in 98 patients with oral leishmaniasis. The most commonly countries. However, the human disease burden is concen- affected site was the tongue (four patients, 57%), and trated mainly in a few major foci. The epidemiological the most common clinical presentation was an exophy- data on leishmaniasis are very complex, with intra- and fi tic lesion (six patients, 85%). The literature review interspeci c variations in transmission cycles, reservoir fl showed 11 reports published between 2005 and 2013, hosts, sand y vectors, and clinical forms (World Health describing 13 patients (100% male) affected by head– Organization, 2010). neck primary ML (54% laryngeal, 31% oral, 23% pharyn- Travelers and soldiers who have to stay in endemic geal, and 15% endonasal). The most common clinical areas, foreign citizens, migrant workers, asylum seekers, presentation was an exophytic lesion (69%). refugees from endemic areas, migrants, and immunosup- CONCLUSIONS: The literature analysis revealed that pressed patients are considered as risk groups (Grimaldi in immunocompetent patients, the oral mucosa is the and Schottelius, 2001). second most frequently affected site of the head and In humans, the clinical forms of the leishmaniases are neck region. In the oral cavity, the tongue is the most broadly categorized into three groups: visceral leishmania- affected site. Diagnosis of oral leishmaniasis represents sis (VL), cutaneous leishmaniasis (CL), and mucosal leish- fi a challenge but must be considered in any differential maniasis (ML). However, a more complete classi cation diagnosis of exophytic lesions of oral mucosa. encompasses 11 different clinical forms (World Health Oral Diseases Organization, 2010). Among these, mucocutaneous leish- (2014) doi: 10.1111/odi.12268 maniasis (MCL) or ML pure forms can occur with a latency of months, even years, after exposure in endemic Keywords: primary oral leishmaniasis; mucosal; immunocompetent; – areas and because of this huge latency, the diagnosis is head neck leishmaniasis often seriously delayed. All the clinical forms of leishman- iasis can start with primary mucosal lesions in the head– neck region, sometimes affecting the oral cavity, or certain significant symptoms detectable by the dental practitioner. Such symptoms include swallowing difficulties, dyspho- Correspondence: Mignogna Michele Davide, MD, DMD, Department of nia, and dyspnea. Therefore, dentists play an important Neurosciences, Reproductive and Odontostomatological Sciences, Univer- role in the early diagnosis of oral leishmaniasis, to avoid sity Federico II of Naples, Naples, Italy. Tel: +39 0817462182, the systemic spread of the disease (Pellicioli et al, 2012). Fax: +39 0817462181, E-mail: [email protected] All persons and only persons who meet authorship criteria and partici- In any such systemic spread, the sites most commonly pated in the preparation of the manuscript are listed as authors. involved are the liver, spleen, abdominal lymphatic sys- Received 11 April 2014; revised 6 June 2014; accepted 9 June 2014 tem, lymph nodes, and bone marrow. Oral leishmaniasis and literature review MD Mignogna et al 2 Management of leishmaniasis still represents a big challenge. Presently, the main target of any drugs used is to fight the parasite in its different physiological and biochemical aspects and to promote the immune response of the host. The therapeutic strategies currently available include pentavalent antimony derivatives, systemically or intralesionally administered, pentamidine, metronidazole, amphotericin B, azoles, and miltefosine (Masmoudi et al, 2013). The primary objective of this study is to present a retro- spective multicenter case series describing seven cases of primary oral leishmaniasis. The secondary objective is to review the literature on head and neck primary ML in immunocompetent/otherwise healthy patients, as no simi- lar data are currently available in the literature, and to update the current literature with our seven new cases. Patients and methods Multicenter case series assessment We retrospectively selected and analyzed the clinical data Figure 1 Flow chart of leishmaniasis patients from the archives of two inde- pendent hospitals, the outpatient clinic of the Oral Medi- cine Unit, Department of Head and Neck Diseases, year of publication, age, sex, underlying or past diseases, Federico II University of Naples and the Department of predisposing risk factors, local conditions, involved sites, Dental Sciences and Surgery, University of Bari, reporting symptoms, lesion description, diagnostic procedures, treat- seven new cases with a history of ML with exclusive and ment, follow-up period, and outcomes. primary oral involvement, diagnosed and treated between 2001 and 2012. Results The study was approved by the Ethics Committee of University of Naples ‘Federico II’. The case series from our database shows seven patients, All patients, after providing their written informed con- three of whom were treated at our institution. sent, were hospitalized and examined by routine hemato- All the patients underwent incisional or excisional logical testing, HIV, HBV, HCV, and oral biopsies with biopsy (cases 2, 4, and 7), and in all cases, the biopsy histopathological evaluation. Abdominal CT and bone showed inflammatory lesions, with the presence of para- marrow biopsies were made in some cases to exclude any sites belonging to Leishmania spp., allowing the diagnosis suspected visceral forms. The study was performed in of leishmaniasis. Subsequently, all patients underwent rou- accordance with the Declaration of Helsinki. tine laboratory examinations, CT scanning, and bone mar- row biopsy, revealing no significant findings or evidence Literature review of leishmaniasis elsewhere except for splenomegaly in Subsequently, we retrospectively reviewed all articles from case 5 (Figures 2–6). 1950 to 2013 focused on cases of ML in immunocompe- tent/otherwise healthy patients, by searching in the Case reports PubMed database on each of the following keywords: Case 1 – A 55-year-old Caucasian man with a history of a ‘mucosal’, ‘oral’, ‘buccal’, ‘nose’, ‘nasal’, ‘pharynx’, ‘pha- 4-week painful swelling affecting the left side of the ton- ryngeal’, ‘esophagus’, ‘esophageal’, ‘larynx’, ‘laryngeal’ gue. The patient had lived in Senegal for 20 years and in association with ‘leishmaniasis’, and alternatively with was a smoker (25 cigarettes daily for 30 years). ‘healthy’ and ‘immunocompetent’. Physical examination showed a nodular, whitish, and The inclusion criteria of this review encompass (i) arti- ulcerated lesion on the left edge of the tongue, 1.7 cm of cles published in the English language reporting primary maximum diameter, with some mild perilesional fissura- ML for all patients described; (ii) healthy or immunocom- tions. petent patients who contracted an infection of leishmania- The patient was treated with 28 intramuscular injections À sis between 1950 and 2013; (iii) mucosal lesions that of meglumine antimoniate at a daily dose of 5 mg kg 1 presented in the regions of the nasal mucosa, oral mucosa, body weight. nasopharynx, oropharynx, hypopharynx, esophageal, and After 1 month, the patient had a complete remission of laryngeal mucosa; (iv) the absence of any visceral or cuta- the tongue lesion, but after 8 months, the disease recurred neous manifestations; and 5) documentation on the leish- with the onset of a cutaneous form of leishmaniasis. The mania infection. patient was referred to the Infectious Diseases Department A flow chart is shown in Figure 1. for treatment. In relation to the reviewed articles, we collected, tabu- Case 2 – A 48-year-old Caucasian man with a history lated, and depicted the following information: country and of a 7-week painful lesion of the tongue. The patient was Oral Diseases Oral leishmaniasis and literature review MD Mignogna et al
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