|Year : 2018 | Volume
| Issue : 2 | Page : 89-91
A case of hyperkeratotic cutaneous leishmaniasis on the hallux: an unusual presentation of American tegumentary leishmaniasis
Marcus H. de S.B Xavier1, Sabha Mushtaq2, Luiz A.B Pôrto1, Ana C.R Fonseca1, Moisés S Pedrosa1
1 Hospital Foundation of the State of Minas Gerais, FHEMIG, Belo Horizonte, MG, Brazil
2 Department of Dermatology, Jawaharlal Nehru Medical College (JNMC), Aligarh, Uttar Pradesh, India
|Date of Submission||30-Mar-2018|
|Date of Acceptance||02-Jun-2018|
|Date of Web Publication||17-Aug-2018|
Department of Dermatology, Jawaharlal Nehru Medical College (JNMC), Aligarh, Uttar Pradesh 202001
Source of Support: None, Conflict of Interest: None
Cutaneous leishmaniasis (CL) is endemic in various parts of Brazil and is known for its clinical pleiomorphism in humans. Beyond the classical picture, CL may appear at unusual sites or with atypical morphologies which may elude the diagnosis at first instance. Herein, we report the case of a 48-year-old Brazilian male, who presented with a 2-year history of persistent hyperkeratotic plaque on the left hallux, which turned out to be cutaneous leishmaniasis. The wide variety of cutaneous findings and unusual presentations of CL often pose a diagnostic challenge especially for inexperienced physicians. The objective of this case report is to emphasize on the unpredictability of clinical expressions of CL and to apprise the clinicians of its masquerading potential.
Keywords: cutaneous leishmaniasis, hyperkeratotic, meglumine antimoniate, mucocutaneous leishmaniasis, new world leishmaniasis
|How to cite this article:|
Xavier MS, Mushtaq S, Pôrto LA, Fonseca AC, Pedrosa MS. A case of hyperkeratotic cutaneous leishmaniasis on the hallux: an unusual presentation of American tegumentary leishmaniasis. Egypt J Dermatol Venerol 2018;38:89-91
|How to cite this URL:|
Xavier MS, Mushtaq S, Pôrto LA, Fonseca AC, Pedrosa MS. A case of hyperkeratotic cutaneous leishmaniasis on the hallux: an unusual presentation of American tegumentary leishmaniasis. Egypt J Dermatol Venerol [serial online] 2018 [cited 2019 Jul 21];38:89-91. Available from: http://www.ejdv.eg.net/text.asp?2018/38/2/89/237842
| Introduction|| |
Cutaneous leishmaniasis (CL) is considered an important public health issue, causing considerable morbidity in many parts of the world including Brazil ,. According to the WHO’s estimates, nearly 1.5 million cases of CL occur every year in 98 countries and territories on five continents . CL is widely distributed, with approximately one-third of cases occurring in each of the three epidemiological regions: the Americas, the Mediterranean basin, and Western Asia . A recent rise in cases of CL in the United States has been noted, and it is attributable to international travel to and from endemic regions, whether by immigrants, refugees, tourists, or soldiers ,.
Although the typical cases of CL are easy to diagnose in endemic regions, the unusual forms may dodge even seasoned physicians. In the recent past, there has been an increase in reporting of atypical/unusual clinical presentations of CL, both in Old and the New World . Dermatologists, therefore, should be aware of the clinical manifestations of CL, as its clinicopathological picture is variable, often making diagnosis difficult besides being a condition sometimes forgotten by physicians.
| Case report|| |
A 48-year-old male from a rural settlement to the North of Minas Gerais state of Brazil presented with a 2-year history of an asymptomatic plaque over his left big toe. It began as an erythematous papule which gradually developed into a plaque over a period of 6 months. There was no history of obvious trauma at the site. Since 1 month, the patient had noticed thick crust covering the plaque and a small ulceration at one place. The patient had consulted several family physicians and dermatologists without a definitive diagnosis. He had taken multiple courses of antibiotics without any improvement. His medical history was otherwise unremarkable.
Cutaneous examination revealed a well-defined plaque measuring 3 cm×2.5 cm over dorsum of left hallux. The margin of the plaque had a violaceous hue and the surface was hyperkeratotic, covered with thick adherent yellowish-gray crust. A small ulcer was seen at the anterior end of the plaque ([Figure 1]). However, there was no active ooze or discharge. There was no regional lymphadenopathy. The differential diagnosis included tuberculosis verrucosa cutis, cutaneous leishmaniasis and fixed cutaneous sporotrichosis.
Biopsy specimens were obtained for histological examination and culture (mycobacterial and fungal). Histopathology revealed pseudoepitheliomatous hyperplasia with the dermis showing nodular infiltrate of histiocytes, lymphocytes, and tuberculoid-type histiocytic granulomas. Leishman-Donovan bodies in the cytoplasm of histiocytes could be demonstrated on Giemsa stain, thus confirming the diagnosis of CL ([Figure 2]a–[Figure 2]c). The mycobacterial and fungal culture was negative. The results of chest radiograph, blood chemistry, blood count, and urinalysis were within normal limits.
|Figure 2 (a) Pseudoepitheliomatous hyperplasia with dense lymphohistiocytic infiltrate in the dermis (hematoxylin and eosin, ×40). (b) Higher power showing histiocytes and multiple Leishman-Donovan bodies (hematoxylin and eosin, ×100). (c) Few histiocytes with multiple Leishman-Donovan bodies (Giemsa, ×400).|
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The patient was given intralesional meglumine antimoniate administered fortnightly for 8 weeks. At a 3-month follow-up, the lesion had completely healed.
| Discussion|| |
Leishmaniasis is a diverse vector borne disease caused by different species of protozoa Leishmania. It is transmitted to humans by the bite of a tiny (2–3 mm long) infected sand-fly (Phlebotomus and Lutzomyia) . As per geographic categorization, leishmaniasis is divided into (a) Old World leishmaniasis (transmitted by vector Phlebotomus and caused by Leishmania species found in Africa, Asia, the Middle East, the Mediterranean, and India), which produces cutaneous or visceral disease and (b) New world leishmaniasis (transmitted by vector Lutzomyia and caused by Leishmania species found in Central and South America), which produces cutaneous, mucocutaneous, and visceral disease ,.
The main Leishmania species causing New World CL include Leishmania mexicana and Leishmania (Viannia) braziliensis species complex. In Brazil, the disease is also known as American tegumentary leishmaniasis (ATL) caused by L. (V.) braziliensis, Leishmania (V.) guyanensis, and Leishmania (Leishmania) amazonensis and is widely distributed in endemic regions of northeast Brazil ,,.
CL is a major public health problem in many countries with 90% of all cases occurring in seven countries including Brazil, where every year ∼28 000 cases of CL are reported . The disease is endemic in whole of Brazil, and the major clinical forms include localized or disseminated cutaneous and mucocutaneous disease . Localized CL is most frequently caused by L. guyanensis and L. braziliensis in Brazil. The disseminated cutaneous and mucocutaneous disease are most frequently caused by L. braziliensis . Our patient belonged to Minas Gerais state of Brazil where ATL is endemic, and most cases are of local CL as per the official record .
A ‘classical’ lesion of ATL originates as a macule over the exposed area at the site of inoculation followed by a papule that ulcerates and expands to a typical round-to-oval crateriform lesion or evolves as a nodular lesion . The clinical manifestations of CL are variable and depend not only on the causative Leishmania species but also on endemic region, host factors and immuno-inflammatory responses .
A myriad of atypical presentations of localized CL have been reported over the years: lupoid, eczematous, erysipeloid, verrucous, zosteriform, paronychial, sporotrichoid, chancriform, psoriasiform, and annular . However, there are only two reports of hyperkeratotic form of CL in the literature, and both are from Turkey. Yesilova et al.  and Gülüm et al.  reported CL presenting as hyperkeratotic plaque on the nose and penis, respectively. Both these cases had a long standing history similar to the present case. The hyperkeratotic form of CL is reported in Old World CL though sporadically ,, but to the best of our literature search, we could not find any report of the same in the New World.
The most common diagnostic method for CL is microscopic examination, and the causative Leishmania species can be identified by PCR . In the present case, species identification could not be performed owing to limitation of resources.
The diagnosis of CL is quite easy in typical cases found in the endemic areas. However, the lesions may appear at unusual sites or with atypical morphologies imitating various infectious, inflammatory, and neoplastic conditions, which may lead to delay in diagnosis and mismanagement . A high index of suspicion in such cases is crucial in arriving at the diagnosis. CL is a self-limiting disease, but treatment may be sought for esthetic reasons, delay in spontaneous healing and disfiguring scar. A battery of treatment options are available, but pentavalent antimonials remain the first line of treatment ,.
In our patient, the relatively uncommon site, the atypical clinical presentation and absence of medical suspicion eluded the diagnosis for 2 years. As our patient belonged to an endemic area and owing to the painless nature of the lesion, CL was kept as one of the differential diagnosis, which was confirmed by histopathology.
| Conclusion|| |
CL with its protean manifestations may mimic a variety of diseases posing a diagnostic challenge. The delay in diagnosis may not only lead to mismanagement but also financial burden on the patient. The present case report highlights the unusual morphological presentations of CL and emphasizes keeping a high index of suspicion for timely diagnosis and appropriate treatment.
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Conflicts of interest
There are no conflicts of interest.
| References|| |
Al-Natour SH. Update in the treatment of cutaneous leishmaniasis. J Family Community Med 2009; 16:41–47.
Romero GA, Vinitius de-Farias-Guerra MV, Gomes Paes MG, de Oliveria Macêdo V. Comparison of cutaneous leishmaniasis due to Leishmania (Viannia) braziliensis
and L.(V.) guyanensis
in Brazil: clinical findings and diagnostic approach. Clin Infect Dis 2001; 32:1304–1312.
World Health Organization Expert Committee. Control of the Leishmaniases. WHO technical report series 949. Geneva, Switzerland: World Health Organization 2010. 1–186
Alvar J, Vélez ID, Bern C, Desjeux P, Cano J, Jannin J, den Boer M, WHO Leishmaniasis Control Team. WHO Leishmaniasis Control Team. Leishmaniasis worldwide and global estimates of its incidence. PLoS One 2012; 7:e35671.
Handler MZ, Patel PA, Kapila R, Al-Qubati Y, Schwartz RA. Cutaneous and mucocutaneous leishmaniasis: clinical perspectives. J Am Acad Dermatol 2015; 73:897–908.
Meireles CB, Maia LC, Soares GC, Teodoro IPP, Gadelha MDSV, da Silva CGL, de Lima MAP. Atypical presentations of cutaneous leishmaniasis: a systematic review. Acta Trop 2017; 172:240–254.
Alves CF, Alves CF, Figueiredo MM, Souza CC, Machado-Coelho GL, Melo MN et al.
American tegumentary leishmaniasis: effectiveness of an immunohistochemical protocol for the detection of leishmania in skin. PLoS ONE 2013; 8:e 63343.
Yesilova Y, Turan E, Sürücü HA, Bozkurt F, Zeyrek FY, Tanrikulu O. Hyperkeratotic cutaneous leishmaniasis: a rare presentation. Balkan Med J 2013; 30:129–130.
Gülüm M, Yeşilova Y, Savaş M, Çiftçi H, Yeni E, Zeyrek FY. A case of giant hyperkeratotic cutaneous leishmaniasis in the penis. Turkiye Parazitol Derg 2013; 37:53–54.
Mushtaq S, Dogra D, Dogra N. Clinical response with intralesional Amphotericin B in the treatment of old world cutaneous leishmaniasis: a preliminary report. Dermatol Ther 2016; 29:398–405.
[Figure 1], [Figure 2]