Mucocutaneous leishmaniasis: an imported infection among travellers to central and South America
http://www.100md.com
《英国医生杂志》
1 Royal National Throat, Nose and Ear Hospital, London, 2 Hospital for Tropical Diseases, London
Correspondence to: S D Lawn, Clinical Research Unit, Department of Infectious and Tropical Diseases, London School of Hygiene and Tropical Medicine, London WC1E 7HT stevelawn@yahoo.co.uk
Introduction
Mucocutaneous leishmaniasis is reported infrequently among travellers returning from Latin America to countries where the disease is not endemic.4-9 The cases described here were all young healthy travellers who had spent time in tropical forest areas of Latin America; none had any risk factors for HIV infection. MCL is endemic in such areas between southern Mexico and the northern tip of Argentina.2 The trend towards "adventure travel" to Latin America may lead to MCL being more often imported to the United Kingdom. Patients with MCL may present to a wide variety of clinicians, including general practitioners, all of whom should be aware of the potential significance of a history of rural travel in Latin America. Familiarity with the manifestations of cutaneous leishmaniasis and MCL may speed diagnosis and limit disease progression.
Recognition of cutaneous leishmaniasis lesions may help to prevent development of MCL or facilitate diagnosis of established MCL. The disease can be prevented by treating L viannia cutaneous leishmaniasis before mucosal involvement. Alternatively, in patients with established mucosal disease, the presence of cutaneous leishmaniasis (or a history suggestive of a previous self healing cutaneous leishmaniasis lesion) may provide a strong clue to a diagnosis of mucocutaneous disease.
Cutaneous leishmaniasis lesions often develop on exposed parts of the body within a few weeks of exposure to infected sandfly bites. Single or multiple ulcers typically have a raised, indurated margin and a sloughy base (fig 2). There may be satellite lesions (sporotrichoid spread) and cord-like infiltration of proximal lymphatic vessels and local lymphadenopathy.
Fig 2 A 4 cm diameter cutaneous leishmaniasis ulcer on lower leg caused by L viannia subgenus species showing the typical raised, indurated margin
Box 1: Differential diagnosis for nasal congestion
Rhinitis (allergy)
Anatomical cause (septal deviation, hypertrophic turbinates)
Hormonal disorder (puberty, pregnancy, hypothyroidism, acromegaly)
Granulomatous disease (sarcoidosis, Wegener's granulomatosis)
Drug induced cause (rhinitis medicamentosa, prazosin, cocaine)
Impaction (crusts, foreign body)
Mass (adenoids, nasal polyps, nasopharyngeal carcinoma)
Infection (leishmaniasis, tuberculosis, leprosy, syphilis, rhinoscleroma, coccidioidomycosis, histoplasmosis, blastomycosis)
The differential diagnosis for cutaneous leishmaniasis includes secondary infected insect bites, sporotrichosis (a fungal infection implanted into the skin by a thorn or other penetrating injury), or more rarely cutaneous tuberculosis. Appropriate drug treatment promotes rapid healing of cutaneous leishmaniasis lesions, but the great majority will heal spontaneously over several months; such healing, however, does not preclude the later development of MCL, as illustrated by case 1.
The most common symptom of MCL is persistent nasal congestion, for which the differential diagnosis is broad (box 1). Anterior nasal septal granulomas may be visible with a light source and Thudicum's speculum, whereas posteriorly located granulomas require nasendoscopy to be seen. Progressive MCL lesions destroy upper respiratory tract mucosa over months and years. Common sites are the turbinates and nasal septum,7 9 where erosion of underlying tissue and cartilage may result in perforation. Progressive tissue destruction at the nasal mucocutaneous junction in advanced disease may cause marked disfigurement, requiring reconstructive surgery. Lesions may also affect the palate, pharynx, and larynx, causing palatal dysfunction, dysphagia, dysphonia, and aspiration.1 2 Bony structures are not involved.
Diagnoses of cutaneous leishmaniasis and MCL are established by demonstrating the presence of Leishmania parasites in infected tissues. Serology is rarely helpful except in advanced MCL. Punch biopsies should be taken from the raised, indurated edge of cutaneous leishmaniasis lesions and sent fresh for microbiological and parasitological examination and in formalin for histopathological assessment. Examination of Giemsa stained impression smears for the presence of the intracellular form of the parasites (amastigotes) may quickly yield a diagnosis in some patients. Amastigotes may also be seen in histopathological sections. The flagellate form of the parasite (promastigote) may be cultured from biopsies on modified Novy-McNeal-Nicolle medium incubated for up to three weeks.
Polymerase chain reaction is the most sensitive diagnostic test, however, and is also used to differentiate L viannia subgenus infections, which are associated with the greatest risk of MCL.10 11 Similarly, in patients with suspected MCL, biopsies of mucosal lesions are required for diagnosis, and additional biopsies may be taken from the turbinates even if these are not overtly involved. Histopathology of mucosal biopsies shows granulomatous changes for which the differential diagnosis is broad (box 2). Compared with cutaneous leishmaniasis, Leishmania parasites are less readily visualised or cultured from MCL lesions because the vigorous host response limits the tissue parasite burden. In recent years, however, polymerase chain reaction has proved to be a sensitive tool.11
Box 2: Differential diagnosis for nasal granulomas
Bacterial infection (tuberculosis, leprosy, syphilis, rhinoscleroma)
Fungal infection (coccidioidomycosis, histoplasmosis, blastomycosis, rhinosporidiosis)
Parasitic infection (leishmaniasis)
Autoimmune (Wegener's granulomatosis, systemic lupus erythematosis)
Sarcoidosis
Lymphoma
Foreign body
Heavy metals (beryllium, nickel)
Idiopathic cause
MCL requires prolonged parenteral treatment with pentavalent antimonials (treatment of choice) or amphotericin preparations. Such treatment is associated with toxicity and risk of relapse.1 2 Patients require joint management from infectious or tropical diseases physicians plus otorhinolaryngologists who have clinical experience of MCL and access to the necessary specialist laboratory investigations. Expeditious and appropriate care, however, can be given only after the diagnosis has been suspected.
Mucocutaneous leishmaniasis may be acquired by travellers to central and South America
Contributors: SA helped to collect patients' data and to write the manuscript. SDL provided medical care for the patients, collected patients' data, and wrote the manuscript. JK and HG provided otorhinolaryngology assessments and contributed to the manuscript. DNJL was responsible for the care of the patients and contributed to the manuscript. DNJL is the guarantor.
Funding: None.
Competing interests: None declared.
References
Herwaldt BL. Leishmaniasis. Lancet 1999;354: 1191-9.
Dedet JP, Pratlong F. Leishmaniasis. In: Cook GC, Zumla A, eds. Manson's tropical diseases. 21st ed. London: Saunders, 2003: 1339-64.
David C, Dimier-David L, Vargas F, Torrez M, Dedet JP. Fifteen years of cutaneous and mucocutaneous leishmaniasis in Bolivia: a retrospective study. Trans R Soc Trop Med Hyg 1993;87: 7-9.
Rosbotham JL, Corbett EL, Grant HR, Hay RJ, Bryceson AD. Imported mucocutaneous leishmaniasis. Clin Exp Dermatol 1996;21: 288-90.
Scope A, Trau H, Bakon M, Yarom N, Nasereddin A, Schwartz E. Imported mucosal leishmaniasis in a traveler. Clin Infect Dis 2003;37: e83-7.
Costa JW Jr, Milner DA Jr, Maguire JH. Mucocutaneous leishmaniasis in a US citizen. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;96: 573-7.
Lohuis PJ, Lipovsky MM, Hoepelman AI, Hordijk GJ, Huizing EH. Leishmania braziliensis presenting as a granulomatous lesion of the nasal septum mucosa. J Laryngol Otol 1997;111: 973-5.
Singer C, Armstrong D, Jones TC, Spiro RH. Imported mucocutaneous leishmaniasis in New York city. Report of a patient treated with amphotericin B. Am J Med 1975;59: 444-7.
Galioto P, Fornaro V. A case of mucocutaneous leishmaniasis. Ear Nose Throat J 2002;81: 46-8.
De Bruijin MHL, Barker DC. Diagnosis of New World leishmaniasis: specific detection of species of the Leishmania braziliensis complex by amplification of kinetoplast DNA. Acta Tropica 1992;52: 45-58.
Pirmez C, da Silva Trajano V, Paes-Oliveira Neto M, da-Cruz AM, Goncalves-da-Costa SC, Catanho M, et al. Use of PCR in diagnosis of human American tegumentary leishmaniasis in Rio de Janeiro, Brazil. J Clin Microbiol 1999;37: 1819-23.(Sukhbir Ahluwalia, senior)
Correspondence to: S D Lawn, Clinical Research Unit, Department of Infectious and Tropical Diseases, London School of Hygiene and Tropical Medicine, London WC1E 7HT stevelawn@yahoo.co.uk
Introduction
Mucocutaneous leishmaniasis is reported infrequently among travellers returning from Latin America to countries where the disease is not endemic.4-9 The cases described here were all young healthy travellers who had spent time in tropical forest areas of Latin America; none had any risk factors for HIV infection. MCL is endemic in such areas between southern Mexico and the northern tip of Argentina.2 The trend towards "adventure travel" to Latin America may lead to MCL being more often imported to the United Kingdom. Patients with MCL may present to a wide variety of clinicians, including general practitioners, all of whom should be aware of the potential significance of a history of rural travel in Latin America. Familiarity with the manifestations of cutaneous leishmaniasis and MCL may speed diagnosis and limit disease progression.
Recognition of cutaneous leishmaniasis lesions may help to prevent development of MCL or facilitate diagnosis of established MCL. The disease can be prevented by treating L viannia cutaneous leishmaniasis before mucosal involvement. Alternatively, in patients with established mucosal disease, the presence of cutaneous leishmaniasis (or a history suggestive of a previous self healing cutaneous leishmaniasis lesion) may provide a strong clue to a diagnosis of mucocutaneous disease.
Cutaneous leishmaniasis lesions often develop on exposed parts of the body within a few weeks of exposure to infected sandfly bites. Single or multiple ulcers typically have a raised, indurated margin and a sloughy base (fig 2). There may be satellite lesions (sporotrichoid spread) and cord-like infiltration of proximal lymphatic vessels and local lymphadenopathy.
Fig 2 A 4 cm diameter cutaneous leishmaniasis ulcer on lower leg caused by L viannia subgenus species showing the typical raised, indurated margin
Box 1: Differential diagnosis for nasal congestion
Rhinitis (allergy)
Anatomical cause (septal deviation, hypertrophic turbinates)
Hormonal disorder (puberty, pregnancy, hypothyroidism, acromegaly)
Granulomatous disease (sarcoidosis, Wegener's granulomatosis)
Drug induced cause (rhinitis medicamentosa, prazosin, cocaine)
Impaction (crusts, foreign body)
Mass (adenoids, nasal polyps, nasopharyngeal carcinoma)
Infection (leishmaniasis, tuberculosis, leprosy, syphilis, rhinoscleroma, coccidioidomycosis, histoplasmosis, blastomycosis)
The differential diagnosis for cutaneous leishmaniasis includes secondary infected insect bites, sporotrichosis (a fungal infection implanted into the skin by a thorn or other penetrating injury), or more rarely cutaneous tuberculosis. Appropriate drug treatment promotes rapid healing of cutaneous leishmaniasis lesions, but the great majority will heal spontaneously over several months; such healing, however, does not preclude the later development of MCL, as illustrated by case 1.
The most common symptom of MCL is persistent nasal congestion, for which the differential diagnosis is broad (box 1). Anterior nasal septal granulomas may be visible with a light source and Thudicum's speculum, whereas posteriorly located granulomas require nasendoscopy to be seen. Progressive MCL lesions destroy upper respiratory tract mucosa over months and years. Common sites are the turbinates and nasal septum,7 9 where erosion of underlying tissue and cartilage may result in perforation. Progressive tissue destruction at the nasal mucocutaneous junction in advanced disease may cause marked disfigurement, requiring reconstructive surgery. Lesions may also affect the palate, pharynx, and larynx, causing palatal dysfunction, dysphagia, dysphonia, and aspiration.1 2 Bony structures are not involved.
Diagnoses of cutaneous leishmaniasis and MCL are established by demonstrating the presence of Leishmania parasites in infected tissues. Serology is rarely helpful except in advanced MCL. Punch biopsies should be taken from the raised, indurated edge of cutaneous leishmaniasis lesions and sent fresh for microbiological and parasitological examination and in formalin for histopathological assessment. Examination of Giemsa stained impression smears for the presence of the intracellular form of the parasites (amastigotes) may quickly yield a diagnosis in some patients. Amastigotes may also be seen in histopathological sections. The flagellate form of the parasite (promastigote) may be cultured from biopsies on modified Novy-McNeal-Nicolle medium incubated for up to three weeks.
Polymerase chain reaction is the most sensitive diagnostic test, however, and is also used to differentiate L viannia subgenus infections, which are associated with the greatest risk of MCL.10 11 Similarly, in patients with suspected MCL, biopsies of mucosal lesions are required for diagnosis, and additional biopsies may be taken from the turbinates even if these are not overtly involved. Histopathology of mucosal biopsies shows granulomatous changes for which the differential diagnosis is broad (box 2). Compared with cutaneous leishmaniasis, Leishmania parasites are less readily visualised or cultured from MCL lesions because the vigorous host response limits the tissue parasite burden. In recent years, however, polymerase chain reaction has proved to be a sensitive tool.11
Box 2: Differential diagnosis for nasal granulomas
Bacterial infection (tuberculosis, leprosy, syphilis, rhinoscleroma)
Fungal infection (coccidioidomycosis, histoplasmosis, blastomycosis, rhinosporidiosis)
Parasitic infection (leishmaniasis)
Autoimmune (Wegener's granulomatosis, systemic lupus erythematosis)
Sarcoidosis
Lymphoma
Foreign body
Heavy metals (beryllium, nickel)
Idiopathic cause
MCL requires prolonged parenteral treatment with pentavalent antimonials (treatment of choice) or amphotericin preparations. Such treatment is associated with toxicity and risk of relapse.1 2 Patients require joint management from infectious or tropical diseases physicians plus otorhinolaryngologists who have clinical experience of MCL and access to the necessary specialist laboratory investigations. Expeditious and appropriate care, however, can be given only after the diagnosis has been suspected.
Mucocutaneous leishmaniasis may be acquired by travellers to central and South America
Contributors: SA helped to collect patients' data and to write the manuscript. SDL provided medical care for the patients, collected patients' data, and wrote the manuscript. JK and HG provided otorhinolaryngology assessments and contributed to the manuscript. DNJL was responsible for the care of the patients and contributed to the manuscript. DNJL is the guarantor.
Funding: None.
Competing interests: None declared.
References
Herwaldt BL. Leishmaniasis. Lancet 1999;354: 1191-9.
Dedet JP, Pratlong F. Leishmaniasis. In: Cook GC, Zumla A, eds. Manson's tropical diseases. 21st ed. London: Saunders, 2003: 1339-64.
David C, Dimier-David L, Vargas F, Torrez M, Dedet JP. Fifteen years of cutaneous and mucocutaneous leishmaniasis in Bolivia: a retrospective study. Trans R Soc Trop Med Hyg 1993;87: 7-9.
Rosbotham JL, Corbett EL, Grant HR, Hay RJ, Bryceson AD. Imported mucocutaneous leishmaniasis. Clin Exp Dermatol 1996;21: 288-90.
Scope A, Trau H, Bakon M, Yarom N, Nasereddin A, Schwartz E. Imported mucosal leishmaniasis in a traveler. Clin Infect Dis 2003;37: e83-7.
Costa JW Jr, Milner DA Jr, Maguire JH. Mucocutaneous leishmaniasis in a US citizen. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;96: 573-7.
Lohuis PJ, Lipovsky MM, Hoepelman AI, Hordijk GJ, Huizing EH. Leishmania braziliensis presenting as a granulomatous lesion of the nasal septum mucosa. J Laryngol Otol 1997;111: 973-5.
Singer C, Armstrong D, Jones TC, Spiro RH. Imported mucocutaneous leishmaniasis in New York city. Report of a patient treated with amphotericin B. Am J Med 1975;59: 444-7.
Galioto P, Fornaro V. A case of mucocutaneous leishmaniasis. Ear Nose Throat J 2002;81: 46-8.
De Bruijin MHL, Barker DC. Diagnosis of New World leishmaniasis: specific detection of species of the Leishmania braziliensis complex by amplification of kinetoplast DNA. Acta Tropica 1992;52: 45-58.
Pirmez C, da Silva Trajano V, Paes-Oliveira Neto M, da-Cruz AM, Goncalves-da-Costa SC, Catanho M, et al. Use of PCR in diagnosis of human American tegumentary leishmaniasis in Rio de Janeiro, Brazil. J Clin Microbiol 1999;37: 1819-23.(Sukhbir Ahluwalia, senior)