Case Report: Old World Mucosal Leishmaniasis: Report of Five Imported Cases to the Hospital for Tropical Diseases, London, United Kingdom

Trupti A. Patel Hospital for Tropical Diseases, University College London Hospitals NHS Foundation Trust, London, United Kingdom;

Search for other papers by Trupti A. Patel in
Current site
Google Scholar
PubMed
Close
,
Glenis K. Scadding Royal National Throat, Nose and Ear Hospital, University College London Hospitals NHS Foundation Trust, London, United Kingdom;

Search for other papers by Glenis K. Scadding in
Current site
Google Scholar
PubMed
Close
,
David E. Phillips Department of Ear, Nose and Throat Surgery, Warwick Hospital, Warwick, United Kingdom

Search for other papers by David E. Phillips in
Current site
Google Scholar
PubMed
Close
, and
Diana N. Lockwood Hospital for Tropical Diseases, University College London Hospitals NHS Foundation Trust, London, United Kingdom;

Search for other papers by Diana N. Lockwood in
Current site
Google Scholar
PubMed
Close
Restricted access

Old World species of Leishmania typically cause visceral and cutaneous leishmaniasis. Mucosal involvement is typically seen with infection by Leishmania species found in South America, usually after the healing of cutaneous leishmaniasis. We present five imported cases of mucosal leishmaniasis caused by Old World Mediterranean Leishmania infantum exclusively affecting the nasal mucosa or vocal cord. In only one case was there a recollection of a preceding cutaneous lesion compatible with cutaneous Leishmaniasis. Of significance was that four out of five cases were receiving local corticosteroids for chronic lung disorders and four were systemically immunosuppressed. This report highlights the importance of considering mucosal leishmaniasis in the differential diagnosis in those presenting with upper respiratory tract mucosal lesions with a relevant travel history to the Mediterranean and in whom malignancy has been excluded.

Author Notes

Address correspondence to Trupti A. Patel, Royal Free London NHS Foundation Trust, Pond Street, London NW3 2QG, UK. E-mail: truptipatel1@nhs.net

Authors’ addresses: Trupti A. Patel and Diana N. Lockwood, Hospital for Tropical Diseases, University College London Hospitals NHS Foundation Trust, London, UK, E-mails: truptipatel1@nhs.net and diana.lockwood@lshtm.ac.uk. Glenis K. Scadding, Royal National Throat, Nose and Ear Hospital, University College London Hospitals NHS Foundation Trust, London, UK, E-mail: gscadding@gmail.com. David E. Phillips, Department of Ear, Nose and Throat Surgery, Warwick Hospital, Warwickshire, UK, E-mail: david.phillips@swft.nhs.uk.

  • 1.

    World Health Organization, 2013. Sustaining the Drive to Overcome the Global Impact of Neglected Tropical Diseases. Second WHO Report on Neglected Tropical Diseases. , Geneva, Switzerland: World Health Organization. Available at: http://www.who.int/neglected_diseases/9789241564540/en/. Accessed December 1, 2016.

  • 2.

    Alborzi A, Rasouli M, Shamsizadeh A, 2006. Leishmania tropica-isolated patient with visceral leishmaniasis in southern Iran. Am J Trop Med Hyg 74: 306307.

    • Search Google Scholar
    • Export Citation
  • 3.

    World Health Organization, 2010. Technical Report Series 949. Control of the Leishmaniases. Available at: http://whqlibdoc.who.int/trs/WHO_TRS_949_eng.pdf. Accessed January 11, 2017.

  • 4.

    Sacks DL, Kenney RT, Kreutzer RD, Jaffe CL, Gupta AK, Sharma MC, Sinha SP, Neva FA, Saran R, 1995. Indian kala-azar caused by Leishmania tropica. Lancet 345: 959961.

    • Search Google Scholar
    • Export Citation
  • 5.

    Magill AJ, 2009. Leishmania species: Visceral (kala-azar), cutaneous, and mucosal leishmaniasis. Mandell GL, Bennett JE, Dolin R, eds. Mandell, Douglas and Bennett’s Principles and Practice of Infectious Diseases, Vol. 2, 7th edition. Philadelphia, PA: Churchill Livingstone, 3481.

    • Search Google Scholar
    • Export Citation
  • 6.

    Jones TC et al.., 1987. Epidemiology of American cutaneous leishmaniasis due to Leishmania braziliensis braziliensis. J Infect Dis 156: 7383.

    • Search Google Scholar
    • Export Citation
  • 7.

    Lessa MM, Lessa HA, Castro TW, Oliveira A, Scherifer A, Machado P, Carvalho EM, 2007. Mucosal leishmaniasis: epidemiological and clinical aspects. Rev Bras Otorrinolaringol (Engl Ed) 73: 843847.

    • Search Google Scholar
    • Export Citation
  • 8.

    Aliaga L, Cobo F, Mediavilla JD, Bravo J, Osuna A, Amador JM, Martín-Sánchez J, Cordero E, Navarro JM, 2003. Localized mucosal leishmaniasis due to Leishmania (Leishmania) infantum: clinical and microbiologic findings in 31 patients. Medicine (Baltimore) 82: 147158.

    • Search Google Scholar
    • Export Citation
  • 9.

    Cocuzza S, Strazzulla A, Pinzone MR, Cosentino S, Serra A, Caltabiano R, Lanzafame S, Cacopardo B, Nunnari G, 2013. Isolated laryngeal leishmaniasis in immunocompetent patients: an underdiagnosed disease. Case Rep Infect Dis 2013: 165409.

    • Search Google Scholar
    • Export Citation
  • 10.

    Richter J, Hanus I, Haussinger D, Löscher T, Harms G, 2011. Mucosal Leishmania infantum infection. Parasitol Res 109: 959962.

  • 11.

    Tiseo D, Tosone G, Conte MC, Scordino F, Mansueto G, Mesolella M, Parrella G, Pennone R, Orlando R, 2008. Isolated laryngeal leishmaniasis in an immunocompetent patient: a case report. Infez Med 16: 233235.

    • Search Google Scholar
    • Export Citation
  • 12.

    Alvar J, Ballesteros JA, Soler R, Benito A, van Eys GJ, Schooner GJ, Cabrera B, 1990. Mucocutaneous leishmaniasis due to Leishmania (Leishmania) infantum: biochemical characterization. Am J Trop Med Hyg 43: 614618.

    • Search Google Scholar
    • Export Citation
  • 13.

    Tomson N, Symonds RP, Moir AA, Kendall CH, Wiselka MJ, 2002. New World leishmaniasis from Spain. Postgrad Med J 78: 757758.

  • 14.

    Garcia de Marcos JA, Dean FA, Alamillos GF, Ruiz Masera JJ, Cortés Rodríguez B, Vidal Jiménez A, García Lainez A, Lozano Rodríguez-Mancheno A, 2007. Localized Leishmaniasis of the oral mucosa. A report of three cases. Med Oral Patol Oral Cir Bucal 12: E281E286.

    • Search Google Scholar
    • Export Citation
  • 15.

    Borzoni F, Gradoni L, Gramiccia M, Maccioni A, Valdes E, Loddo S, 1991. A case of lingual and palatine localization of a viscerotropic Leishmania infantum zymodeme in Sardinia, Italy. Trop Med Parasitol 42: 193194.

    • Search Google Scholar
    • Export Citation
  • 16.

    Casolari C, Guaraldi G, Pecorari M, Tamassia G, Cappi C, Fabio G, Cesinaro AM, Piolini R, Rumpianesi F, Presutti L, 2005. A rare case of localized mucosal leishmaniasis due to Leishmania infantum in an immunocompetent Italian host. Eur J Epidemiol 20: 559561.

    • Search Google Scholar
    • Export Citation
  • 17.

    Cobo F, Aliaga L, Talavera P, Concha A, 2007. The histological spectrum of non-granulomatous localized mucosal leishmaniasis caused by Leishmania infantum. Ann Trop Med Parasitol 101: 689694.

    • Search Google Scholar
    • Export Citation
  • 18.

    Pau M, Atzori L, Aste N, Aste N, 2009. Two cases of primary endonasal leishmaniasis in Sardinia (Italy). Dermatol Online J 15: 5.

  • 19.

    Maroli M, Rossi L, Baldelli R, Capelli G, Ferroglio E, Genchi C, Gramiccia M, Mortarino M, Pietrobelli M, Gradoni L, 2008. The northward spread of leishmaniasis in Italy: evidence from retrospective and ongoing studies on the canine reservoir and phlebotomine vectors. Trop Med Int Health 13: 256264.

    • Search Google Scholar
    • Export Citation
  • 20.

    Noyes HA, Reyburn H, Bailey JW, Smith D, 1998. A nested-PCR-based schizodeme method for identifying Leishmania kinetoplast minicircle classes directly from clinical samples and its application to the study of the epidemiology of Leishmania tropica in Pakistan. J Clin Microbiol 36: 28772881.

    • Search Google Scholar
    • Export Citation
  • 21.

    Adams ER, Jacquet D, Schooner G, Gidwani K, Boelaert M, Cunningham J, 2012. Leishmaniasis direct agglutination test: using pictorials as training materials to reduce inter-reader variability and improve accuracy. PLoS Negl Trop Dis 6: e1946.

    • Search Google Scholar
    • Export Citation
  • 22.

    Sundar S, Pai K, Sahu M, Kumar V, Murray HW, 2002. Immunochromatographic strip-test detection of anti-K39 antibody in Indian visceral leishmaniasis. Ann Trop Med Parasitol 96: 1923.

    • Search Google Scholar
    • Export Citation
  • 23.

    Fsadni C, Fsadni P, Piscopo T, Mallia AC, 2007. Laryngeal leishmaniasis in Malta. J Infect 54: e61e63.

  • 24.

    Faucher B et al.., 2011. Mucosal Leishmania infantum leishmaniasis: specific pattern in a multicentre survey and historical cases. J Infect 63: 7682.

    • Search Google Scholar
    • Export Citation
  • 25.

    Habibzadeh F, Sajedianfard J, Yadollahie M, 2005. Isolated lingual leishmaniasis. J Postgrad Med 51: 218219.

  • 26.

    Michel G, Pomares C, Ferrua B, Marty P, 2011. Importance of worldwide asymptomatic carriers of Leishmania infantum (L. chagasi) in human. Acta Trop 119: 6975.

    • Search Google Scholar
    • Export Citation
  • 27.

    Guerra JA, Coelho LI, Pereira FR, Siqueira AM, Ribeiro RL, Almeida TM, Lacerda MV, Barbosa Md, Talhari S, 2011. American tegumentary leishmaniasis and HIV-AIDS association in a tertiary care center in the Brazilian Amazon. Am J Trop Med Hyg 85: 524527.

    • Search Google Scholar
    • Export Citation
  • 28.

    World Health Organization, 2013. Leishmaniasis in the Americas. Recommendations for Treatment. Geneva, Switzerland: World Health Organization. Available at: http://www.who.int/leishmaniasis/research/en/. Accessed December 1, 2016.

  • 29.

    Aronson N et al.., 2016. Diagnosis and treatment of leishmaniasis: clinical practice guidelines by the (IDSA) and the American Society of Tropical Medicine and Hygiene (ASTMH). Clin Infect Dis 63: e202e264.

    • Search Google Scholar
    • Export Citation
  • 30.

    van Thiel PP et al.., 2010. Miltefosine treatment of Leishmania major infection: an observational study involving Dutch military personnel returning from northern Afghanistan. Clin Infect Dis 50: 8083.

    • Search Google Scholar
    • Export Citation
  • 31.

    Mohebali M, Fotouhi A, Hooshmand B, Zarei Z, Akhoundi B, Rahnema A, Razaghian AR, Kabir MJ, Nadim A, 2007. Comparison of miltefosine and meglumine antimoniate for the treatment of zoonotic cutaneous leishmaniasis (ZCL) by a randomized clinical trial in Iran. Acta Trop 103: 3340.

    • Search Google Scholar
    • Export Citation
  • 32.

    Rahman SB, ul Bari A, Mumtaz N, 2007. Miltefosine in cutaneous leishmaniasis. J Coll Physicians Surg Pak 17: 132135.

  • 33.

    Mosimann V, Blazek C, Grob H, Chaney M, Neumayr A, Blum J, 2016. Miltefosine for mucosal and complicated cutaneous old world leishmaniasis: a case series and review of the literature. Open Forum Infect Dis 3: ofw008.

    • Search Google Scholar
    • Export Citation
  • 34.

    Sundar S, Jha TK, Thakur CP, Bhattacharya SK, Rai M, 2006. Oral miltefosine for the treatment of Indian visceral leishmaniasis. Trans R Soc Trop Med Hyg 100: S26S33.

    • Search Google Scholar
    • Export Citation
  • 35.

    Sundar S, Jha TK, Thakur CP, Bhattacharya SK, Rai M, 2002. Oral miltefosine for Indian visceral leishmaniasis. N Engl J Med 347: 17391746.

  • 36.

    Bhattacharya SK et al.., 2007. Phase 4 trial of miltefosine for the treatment of Indian visceral leishmaniasis. J Infect Dis 196: 591598.

  • 37.

    Sindermann H, Engel KR, Fischer C, Bommer W, 2004. Miltefosine compassionate use program: oral miltefosine for leishmaniasis in immunocompromised patients: compassionate use in 39 patients with HIV infection. Clin Infect Dis 39: 15201523.

    • Search Google Scholar
    • Export Citation
  • 38.

    Soto J et al.., 2007. Treatment of Bolivian mucosal leishmaniasis with miltefosine. Clin Infect Dis 44: 350356.

  • 39.

    Soto J, Rea J, Valderrama M, Toledo J, Valda L, Ardiles J, Berman J, 2009. Efficacy of extended (six weeks) treatment with miltefosine for mucosal leishmaniasis in Bolivia. Am J Trop Med Hyg 81: 387389.

    • Search Google Scholar
    • Export Citation
Past two years Past Year Past 30 Days
Abstract Views 51 51 8
Full Text Views 505 107 0
PDF Downloads 200 45 0
 
Membership Banner
 
 
 
Affiliate Membership Banner
 
 
Research for Health Information Banner
 
 
CLOCKSS
 
 
 
Society Publishers Coalition Banner
Save