Case Report

Oral leishmaniasis: Report of two cases

Rennan Luiz Oliveira Dos Santos1, Jefferson Rocha Tenório1, Lígia Gonzaga Fernandes1, Ana Isabel Moreira Ribeiro2,
Sabrina Araújo Pinho Costa1, Marília Trierveiler1, Celso Augusto Lemos1, Norberto Nobou Sugaya1
1
Department of Stomatology, School of Dentistry, University of São Paulo, São Paulo, Brazil, 2Department of Stomatology,
Faculty of Dental Medicine, University of Porto, Porto, Portugal

Abstract Leishmaniasis is a chronic inflammatory disease caused by several species of the parasite Leishmania that is
transmitted by insects of the genus Phlebotomus spp. or Lutzomyia spp. This disease can affect skin, mucous
membranes and viscera being classified as cutaneous, mucocutaneous and visceral leishmaniasis, depending
on the spectrum of clinical manifestations. Diagnosis can be achieved through biopsy, microscopical analysis,
Montenegro intradermoreaction and/or ELISA. The dentist plays an important role in the diagnosis of this
disease due to frequent involvement of oral mucosa. This article reports two clinical cases of leishmaniasis
with oral mucosa involvement, their diagnosis workup and treatment.

Keywords: Leishmaniasis, mucocutaneous leishmaniasis, oral medicine

Address for correspondence: Dr. Rennan Luiz Oliveira Dos Santos, School of Dentistry, University of São Paulo, São Paulo, Brazil.
E-mail: rennan_475@hotmail.com
Submitted: 05-Dec-2018, Revised: 11-May-2020, Accepted: 18-May-2020, Published: 09-Sep-2020

INTRODUCTION involved. The fundamental lesions are similar, consisting

of ulcerated and papulonodular lesions that, regarding the
Leishmaniasis is a chronic inflammatory disease transmitted extension and involved organs, produce symptoms with
by an insect vector and caused by the flagellate protozoan systemic or local repercussions.[5-8] Leishmaniasis clinical
of the genus Leishmania. This parasite has a dimorphic classification is determined by the topographical lesions
life cycle, characterized by the promastigote form in the distribution: cutaneous, mucocutaneous and visceral
insect, where it lives and develops extracellularly, and the leishmaniasis.[6,7]
amastigote form that multiplies intracellularly in the host
macrophages.[1-4] The disease is transmitted by insects Mucosal involvement is relatively rare and results from
belonging to the genus Phlebotomus spp. or Lutzomyia spp. the hematogenous or lymphatic dissemination of the
and is considered by the World Health Organization to be amastigotes from the skin to the nasal, oropharyngeal,
one of the most prominent infectious diseases worldwide laryngeal or tracheal mucosa. [4] When it affects the
due to its high detection coefficient and the high level oral region, the involvement of the posterior portion
of morbidity that it causes due to its capacity to produce of the palate and the tongue is more frequent, but
extensive tissue loss.[1,4-6] lip involvement has been described either. It is also
known to affect middle-aged patients with male
The clinical presentation differs depending on the predominance.[1,4,5,9]
immune response of the host and the protozoan species

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DOI: How to cite this article: Dos Santos RL, Tenório JR, Fernandes LG,
10.4103/jomfp.JOMFP_306_18 Moreira Ribeiro AI, Pinho Costa SA, Trierveiler M, et al. Oral leishmaniasis:
Report of two cases. J Oral Maxillofac Pathol 2020;24:402.

© 2020 Journal of Oral and Maxillofacial Pathology | Published by Wolters Kluwer - Medknow
 Dos Santos, et al.: Oral leishmaniasis

The diagnosis of leishmaniasis can be made through The microscopical study evidenced areas of intense
a series of tests such as anatomopathological study of diffuse inflammatory infiltrate, with organized areas in
biopsy specimens, Montenegro intradermoreaction the form of granulomas. In addition, the presence of
(IDRM) and/or ELISA and PCR to identify Leishmania multinucleated giant cells was observed, and a descriptive
species. IDRM is a skin test of high sensitivity, simple to diagnosis of nonspecific chronic inflammatory process
perform and of great diagnostic value.[4,5,9,10] The differential was provided [Figure 2a and b]. Regarding the clinical
diagnosis of Leishmaniasis-like mucosal lesions is leprosy, and histopathological information, the hypothesis of
lupus vulgaris, squamous cell carcinoma (SCC), Langerhans mucocutaneous leishmaniasis was raised, and IDRM
cell histiocytosis and other granulomatous infections.[4,11] test was requested, which confirmed the suspicion. The
In addition, skin lesions may bear some resemblance to patient was referred to an infectious diseases specialist to
fungal infections such as blastomycosis, histoplasmosis or start treatment with Glucantime® (N-methylglucamine
antimoniate). The dosage used was 20 mg Sb5 (pentavalent
coccidioidomycosis.[4]
antimonial) +/kg/day intravenously for 30 days. Complete
The objective of this article is to report two cases of remission of the lesions was observed post treatment and
atypical leishmaniasis with oral involvement, highlighting after a follow-up of 12 months the patient remained with
no signs of the disease and just a minor cicatricial sequelae.
the importance of the role of dentist in the diagnosis and
treatment of this disease. Patient 2
A 62-year-old male patient, rural worker, smoker, and
CASE REPORT
chronic alcohol user, HIV negative, was referred to the
Patient 1 stomatology clinic with upper lip and upper alveolar ridge
An 80-year-old male patient, living in a rural area, ex- lesions present for 2 months. During anamnesis, sudden
smoker and social drinker, HIV negative, was referred weight loss was reported. Medical history recorded no other
to the stomatology clinic complaining of lesions on the important data. Extraoral physical examination showed a
cutaneous lesion in the left thigh region [Figure 3a] and
upper lip, soft and hard palate that had been presented for
along with ulcerated lesions of the nasal mucosa. Cervical
9 months, producing severe pain in the affected regions.
lymphadenopathy was undetected on palpation.
The patient underwent two biopsies in other clinical
Physical intraoral examination revealed ulcerated and
settings with inconclusive results. On physical examination,
irregular lesions in the mucosa of upper alveolar ridge and
the right side of the upper lip presented an erythematous palate [Figure 3b and c]. An incisional biopsy was performed
swelling associated with a granulomatous ulceration of under local anesthesia considering a differential diagnosis
labial mucosa that extended to the hard palate and seemed with paracoccidioidomycosis, tuberculosis and leukemia.
to infiltrate to the nasal region [Figure 1a-c]. No skin
involvement was observed. Cervical lymphadenopathy Serologies for histoplasmosis and toxoplasmosis were
with inflammatory features as swelling, pain and firm subsequently requested, both resulting in non-reactive
consistency was noticed during palpation. antibodies. Therefore, mucocutaneous leishmaniasis was
suspected. Histopathologic data showed a non-specific
The clinical hypothesis of tuberculosis, histoplasmosis chronic inflammatory process. Immunohistochemistry
and SCC were investigated, and an incisional biopsy was procedures revealed cells positive for CD68 and Leishmania
performed under local anesthesia. The patient’s medical antigen [Figure 4a and b]. In addition, the positivity of the
history recorded no relevant data. IDRM test proved the hypothesis. Treatment, therapeutic

a b c
Figure 1: (a) Extraoral aspect showing asymmetry due to an indurated swelling of the upper lip on the right, accompanied by inflammatory
features of edema and erythema. (b) Granulomatous lesion with ulceration involving the upper lip mucosa up to the posterior region of hard
palate. (c) Granulomatous ulcer on upper lip mucosa extending to vestibule, alveolar ridge and hard palate

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 Dos Santos, et al.: Oral leishmaniasis

response and follow-up of this patient were similar to those and center-west of the country.[2] The patients reported in
cited for the previous patient. this study were diagnosed in a large urban center in the
southern region, where the level of suspicion for the disease
DISCUSSION by health teams is much lower than in those places where
the occurrence is higher. The same situation shall occur in
Leishmaniasis is an infectious disease of worldwide
those countries where the disease incidence is lower or in
distribution, with cases reported in Asia, Africa, Europe places that receive imported or nonautochthonous cases.
and the Americas. Brazil is home to most of the cases
of leishmaniasis that affect mankind, as all forms of the As was described in the cases reported here diagnosis can
disease have high incidence in this country, besides the be difficult if leishmaniasis is not included in the differential
fact that dogs, rodents and other wild animals constitute diagnosis. The IDRM test is extremely valuable in the
natural reservoirs of the parasites.[2] Leishmania braziliensis diagnosis of Leishmaniasis, since the histopathology of
is the most common etiological agent in leishmaniasis with biopsied lesions can hardly disclose the definitive diagnosis
mucosal involvement, capable of producing ulcerated and due to the scarcity of parasites in these specimens.[1,4] This
papulonodular lesions, affecting oral mucosa, nasal mucosa information corroborates with the first case reported, but
and other sites besides the skin.[1,5,6,9] the professional should not rule out the biopsy, which allows
to eliminate other diagnostic possibilities and increase the
It is estimated that near 2 million people develop level of suspicion regarding leishmaniasis. The IDRM
leishmaniasis each year with about 50,000 deaths due to indicates contact with the parasite but not necessarily
complications of the disease and lack of proper treatment. active disease; however, the symptomatology allied to the
The disease occurs worldwide but mainly in the tropics: positivity of the test enable the diagnosis. The safest way
Africa, Asia, Southern Europe, Central and South America. to conclude the diagnosis of leishmaniasis is through the
Brazil, Ethiopia, Sudan, India and Bangladesh encompass detection of protozoan DNA in immunological tests such
90% of potentially fatal cases of leishmaniasis.[12] as ELISA, immunofluorescence and other techniques.[2]
Although leishmaniasis cases occur all over the country in The cases reported here presented mucous lesions
Brazil, the highest incidence occurs in the north, northeast very similar to each other, involving nasal mucosa and
palate, sites characteristically affected by mucocutaneous
leishmaniasis. Distinctive features, which may be considered
uncommon in the exposed cases, refer to the involvement
of the alveolar ridge mucosa, which both cases have
demonstrated, and the coexistence of cutaneous lesions
in limbs and mucosal lesions as in case 2, as cutaneous
lesion is usually an initial manifestation that heals even with
a b
no treatment, followed by late expression of secondary
Figure 2: (a) Lining epithelium showing thin and elongated projections,
with intense spongiosis and lymphocyte exocytosis. The lamina propria
mucosal lesions. Another fact to be highlighted was
is a dense collagenated connective tissue with intense, diffuse and deep the absence of previous skin lesion in case 1, or even a
inflammatory infiltrate (H&E, ×100). (b) Higher magnification showing reference to it in the anamnesis conducted with the patient.
the nature of the inflammatory infiltrate that is lymphoplasmocytic and
rich in macrophages (H&E, ×400). (b) It is also possible to observe
areas with marked presence of eosinophils with granular cytoplasm The process of diagnosis can be challenging according
(H&E, ×400) to clinical presentation, health team experience, and

a b c
Figure 3: (a) Ulcer covered by a darkened crust, with indurated borders, discrete erythema and regular circular shape on forearm skin. (b and c)
Extensive ulcerated irregular lesions, covered by yellowish fibrinous exudate, associated with edema and erythema, involving almost all upper
vestibule, anterior alveolar ridge, central region of hard palate and the left side of soft palate

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 Dos Santos, et al.: Oral leishmaniasis

towards the development of preventive programs, vectors

control or researches on more effective treatment protocols.

CONCLUSION

The manifestations of mucocutaneous leishmaniasis, even

in Brazil, which leads the world statistics in number of
a b
occurrences, can bring difficulties and delay in diagnosis,
Figure 4: (a) Immunohistochemistry for anti-CD68 antibody revealing
positive and intense cytoplasmic positivity in macrophages (IHC, ×400).
due to the variability of clinical presentation, and the need
(b) Immunohistochemistry for the anti-Leishmania antibody revealing of a combination of test results or more sophisticated
positive labeling of amastigotes within the cytoplasm of macrophages and expensive technology, such as protozoan DNA
(IHC, ×400)
identification. Clinicians should develop a higher level
of suspicion regarding the disease in the presence of
occurrence in areas of low or unexpected incidence. ulcerogranulomatous lesions located in the oral and nasal
Such scenario may cause a delayed diagnosis and mucosa, with or without cutaneous involvement, in order
mistreating, leading to sequelae and poor prognosis. to adopt objective guidelines towards the diagnosis and
The differential diagnosis encompasses other infectious subsequently to enable early treatment and better prognosis
diseases, and noninfectious diseases such as pemphigus for the patients.
vulgaris, pemphigoid, plasma cell gingivitis, anemia and
even malignancies as leukemia and SCC.[9] In the case 1 Declaration of patient consent
described before, the clinical aspect of a deep ulcer with The authors certify that they have obtained all appropriate
elevated borders lead to inclusion of SCC in differential patient consent forms. In the form the patient(s) has/have
diagnosis, despite the 9 months history of evolution given his/her/their consent for his/her/their images and
and the lack of associated necrotic tissue. In case 2, the other clinical information to be reported in the journal.
possibility of malignancy was also discussed, among other The patients understand that their names and initials will
infectious diseases, but leukemia instead of SCC because not be published and due efforts will be made to conceal
of the bulging aspect of gingiva and palate mucosa along their identity, but anonymity cannot be guaranteed.
with multiple fibrinous ulcers.
Financial support and sponsorship
The treatment is based on the clinical presentation of Nil.
the disease and on the medical history of each patient.
There are two pentavalent antimonial drugs considered Conflicts of interest
as first choice: N-methylglucamine antimoniate and There are no conflicts of interest.
sodium stibogluconate. Without success with these
REFERENCES
drugs, pentamidines and amphotericin B are alternatives
available.[13] The effectiveness of the treatment depends on 1. Almeida TF, da Silveira EM, Dos Santos CR, León JE, Mesquita AT.
the form and extent of the disease. Therapy with single Exclusive primary lesion of oral leishmaniasis with immunohistochemical
drug present recurrence rates around 20%, which justifies diagnosis. Head Neck Pathol 2016;10:533-7.
2. Gontijo CMF, Melo MN. Leishmaniose visceral no Brasil: Quadro atual,
and requires prolonged follow-up of the patients, as it has desafios e perspectivas. Rev Bras Epid 2004;7:338-49.
been conducted in the cases presented.[7] 3. Handler MZ, Patel PA, Kapila R, Al-Qubati Y, Schwartz RA. Cutaneous
and mucocutaneous leishmaniasis: Differential diagnosis, diagnosis,
Early detection of Leishmaniasis reduces the risk of histopathology, and management. J Am Acad Dermatol 2015;73:911-26.
4. Miranda Lessa M, Andrade Lessa H, Castro TW, Oliveira A, Scherifer A,
mucocutaneous complications such as disfigurement and
Machado P, et al. Mucosal leishmaniasis: Epidemiological and clinical
recurrence of infection. The oral cavity is a frequent site aspects. Braz J Otorhinolaryngol 2007;73:843-7.
of manifestation of this severe form of the disease and 5. Mohammadpour I, Motazedian MH, Handjani F, Hatam GR. Lip
therefore, the dentist represents a crucial health agent in leishmaniasis: A case series with molecular identification and literature
review. BMC Infect Dis 2017;17:96.
the detection of initial lesions preventing the progression 6. Nadler C, Enk CD, Leon GT, Samuni Y, Maly A, Czerninski R. Diagnosis
of the disease to stages of greater tissue loss and favoring and management of oral leishmaniasis-case series and literature review.
more effective therapeutic results. J Oral Maxillofac Surg 2014;72:927-34.
7. Copeland NK, Aronson NE. Leishmaniasis: Treatment updates and
clinical practice guidelines review. Curr Opin Infect Dis 2015;28:426-37.
Unfortunately, leishmaniasis is included in the group of 8. Paniz-Mondolfi AE, Talhari C, García Bustos MF, Rosales T,
tropical diseases that affect mainly poor countries and the Villamil-Gomez WE, Marquez M, et al. American cutaneous
poor people of these countries, with very low investments leishmaniasis in infancy and childhood. Int J Dermatol 2017;56:1328-41.

Journal of Oral and Maxillofacial Pathology | Volume 24 | Issue 2 | May-August 2020

 Dos Santos, et al.: Oral leishmaniasis

9. Mignogna MD, Celentano A, Leuci S, Cascone M, Adamo D, Ruoppo E, Of Disease. 9nd ed. Philadelphia (PA): Saunders Ipswich; 2014.
et al. Mucosal leishmaniasis with primary oral involvement: A case series 12. Pigott DM, Bhatt S, Golding N, Duda KA, Battle KE, Brady OJ, et al.
and a review of the literature. Oral Dis 2015;21:e70-8. Global distribution maps of the leishmaniases. Elife 2014;3:1-21.
10. Garg S, Tripathi R, Tripathi K. Oral mucosal involvement in visceral 13. Handlers JP. Diagnosis and management of oral soft-tissue lesions: The
leishmaniasis. Asian Pac J Trop Med 2013;6:249-50. use of biopsy, toluidine blue staining, and brush biopsy. J Calif Dent
11. Kumar V, Abbas AK, Aster JC. Robbins and Cotran Pathologic Basis Assoc 2001;29:602-6.

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