|
|
| Year : 2009 | Volume
: 52
| Issue : 1 | Page : 62-64 |
|
| Primary laryngeal leishmaniasis: A rare case report |
|
|
Bipin Kumar, Anand Ghimire, Smriti Karki, Paricha Upadhyaya
Department of Pathology, B. P. Koirala Institute of Health Sciences, Dharan, Nepal
Click here for correspondence address and email
|
|
 |
|
 Abstract | | |
Laryngeal leishmaniasis is extremely rare. We report a case of primary laryngeal leishmaniasis in a 70-year-old male who was admitted with complaints of gradual progressive hoarseness of the voice, dyspnea, cough for the past 3 months and noisy breathing for the past 5 days. An X-ray of the soft tissue of the neck showed a prevertebral soft tissue mass causing narrowing of the airway at the C6-C7 vertebral level. A computerized tomography (CT) scan showed a soft tissue mass in the subglottic region causing significant narrowing of the airway. A direct laryngoscopy showed a pinkish-white, friable mass involving the subglottic region and the anterior half of the vocal cords. With the clinical suspicion of malignancy, an endoscopic biopsy was done. A histopathological examination showed diffuse mixed inflammatory cell infiltrate in subepithelium with numerous Leishmania donovani bodies in the cytoplasm of histiocytes. Keywords: Laryngeal leishmaniasis, leishmania donovani, sub-glottic region
How to cite this article:
Kumar B, Ghimire A, Karki S, Upadhyaya P. Primary laryngeal leishmaniasis: A rare case report. Indian J Pathol Microbiol 2009;52:62-4 |
| Introduction | |  |
Leishmaniasis is a zoonotic disease caused by the genus Leishmania. Humans are affected by this disease during the sucking of blood by a vector known as Phlebotomus.[1],[2],[3] Visceral, mucosal and cutaneous are the three clinical forms of the disease. The visceral form is the most common and the mucosal form of the disease is extremely rare in India and Nepal. We report a rare case of primary laryngeal leishmaniasis diagnosed by histopathological examination of the subglottic endoscopic biopsy.
| Case History | | |
A 70-year-old male was admitted with complaints of hoarseness of voice, dyspnea, cough for the past 3 months and noisy breathing for the past 5 days. Upon examination, an enlarged right lobe of the thyroid with nodules of 6 cm and 4 cm in diameter was found. An X-ray of the soft tissues of the neck showed a prevertebral soft tissue mass causing narrowing of the airway at the C6 and C7 vertebral level. A computerized axial tomography revealed a soft tissue mass in the subglottic region causing significant narrowing of the airway. A direct laryngoscopy showed a pinkish-white mass involving the subglottis and the anterior half of the vocal cords. A laryngoscopic biopsy with right sided hemithyroidectomy and neck exploration with pretracheal and paratracheal lymph node dissection was done and sent for a histopathological examination.
Routine hematological, biochemical and urine examinations did not reveal any abnormality.
A histopathological examination of the right hemithyroidectomy specimen showed features of multinodular goiter; pretracheal and paratracheal lymphnodes showed features of sinus histiocytosis. A laryngoscopic biopsy revealed chronic nonspecific inflammation. No evidence of malignancy was seen even on repeat biopsy, which was done due to the strong clinical suspicion of malignancy.
The patient's condition improved after treatment with antibiotics and corticosteroids. Thyroid function tests were normal and an indirect laryngoscopy showed bilateral mobile vocal cords. Six months after being discharged from the hospital, the patient was re-admitted with similar symptoms. Upon examination, a biphasic stridor was found. An immediate tracheostomy was done. A flexible nasolaryngoendoscopy showed whitish-pink growth in the anterior two-thirds of sub-glottic region [Figure 1]. A biopsy was done and the tissue was sent for a histopathological examination.
The tissue consisted of three bits of tissue together measuring 3x3x1 cms. A microscopic examination showed diffuse mixed inflammatory cell infiltrates comprising of lymphocytes, plasma cells and histiocytes with numerous intra-cytoplasmic Leishmania donovani bodies (LD bodies) in the subepithelium [Figure 2] and [Figure 3].
An ultrasonography of the abdomen did not reveal any organomegaly and the bone marrow examination did not show any parasites. Clinical features indicating cutaneous leishmaniasis were not seen. A serological test done for HIV was negative. Based on these findings, a diagnosis of primary laryngeal leishmaniasis was made. The patient was treated with intravenous liposomal amphoterecin B daily for 14 days. The patient's condition improved. The tracheostomy tube was removed and he was discharged from the hospital. After 8 months of follow-up, the patient was free from any symptoms.
| Discussion | | |
Mucosal leishmaniasis is rare and only a few cases involving the oral mucosa and/or upper respiratory tract have been reported. [1-10] The nasal septum was found to be the most common localization. The lesions may be single or multiple along with the involvement of the turbinate and larynx. [6] These lesions were usually associated with visceral or cutaneous leishmaniasis, HIV, or other immunosuppressive conditions like malnutrition, solid organ transplantation, steroid treatment, chemotherapy, or chronic alcoholism. [1],[2],[3],[4],[5],[6],[7],[10] Localization of cutaneous lesions on the upper half of the body has been reported as a risk factor for mucosal leishmaniasis. [6] Exogenous factors such as trauma or a concomitant unrelated illness may act as precipitating factors for the expression of a latent infection. [6] Age range at presentation of mucosal leishmaniasis was 7-75 years and the disease is seen more frequently in males. [3],[5],[6] The time from the onset of symptoms to diagnosis ranged from 3 weeks to 4.5 years. [5] However, long incubation periods of even 16 years have also been reported for laryngeal leishmaniasis. [10] The course of the disease may be very long ranging - up to 23 years. [9] Mucosal leishmaniasis as a complication of cutaneous leishmaniasis is caused either by direct extension or through the bloodstream and lymphatics. [3],[6] However, cases without an underlying disease or immunodeficiency have also been reported. [5],[7],[8]
A case of primary laryngeal leishmaniasis is extremely rare. [1],[2],[10] We report a case of primary laryngeal leishmaniasis that had strong clinical and radiological suspicion of malignancy. The patient was treated with antibiotics for 2 weeks and with corticosteroids for 6 months. Leishmania donovani (LD) bodies were seen on the histopathological section of the third biopsy. The use of corticosteroids in our case caused immunosuppression, which caused increased proliferation of the parasite that could be detected only on the third biopsy. Cases of isolated laryngeal leishmaniasis having similar types of clinical features with clinical suspicion of malignancy were also reported. [1],[7]
Many times, LD bodies fail to demonstrate in the histological sections because of the presence of a smaller number of parasites and features of only nonspecific chronic inflammation may be seen. [3],[8]
| Acknowledgement | | |
We acknowledge Dr. Asis Kumar Karak (Professor, Department of Pathology AIIMS, New Delhi; deputed at the Department of Pathology, B.P. Koirala Institute of Health Sciences, Dharan, Nepal) for his guidance during the preparation of this manuscript.
| References | | |
| 1. | Fsadni C, Fsadni P, Piscopo T, Mallia Azzopardi C. Laryngeal leishmaniasis in Malta. J Infect 2007;54:e61-3.  [PUBMED] [FULLTEXT] |
| 2. | Diaz Sastre MA, Padilla Parrado M, Morales Puebla JM, Jimenez Antolin JA, Caro Garcia MA, Chacon Martinez J, et al . Laryngeal leishmaniasis. An Otorrinolaringol Ibero Am 2007;34:17-25. |
| 3. | Di Lella F, Vincenti V, Zennaro D, Afeltra A, Baldi A, Giordano D, et al . Mucocutaneous leishmaniasis: Report of a case with massive involvement of nasal, pharyngeal and laryngeal mucosa. Int J Oral Maxillofac Surg 2006;35:870-2. [PUBMED] [FULLTEXT] |
| 4. | Gonzalez-Anglada MI, Pena JM, Barbado FJ, Gonzalez JJ, Redondo C, Galera C, et al . Two cases of laryngeal leishmaniasis in patients infected with HIV. Eur J Clin Microbiol Infect Dis 1994;13:509-11. |
| 5. | Aliaga L, Cobo F, Mediavilla JD, Bravo J, Osuna A, Amador JM, et al . Localized mucosal leishmaniasis due to Leishmania infantum: Clinical and microbiologic findings in 31 patients. Medicine (Baltimore) 2003;82:147-58. [PUBMED] [FULLTEXT] |
| 6. | Osorio LE, Castillo CM, Ochoa MT. Mucosal leishmaniasis due to Leishmania (Viannia) panamensis in Colombia: Clinical characteristics. Am J Trop Med Hyg 1998;59:49-52. [PUBMED] [FULLTEXT] |
| 7. | Guddo F, Gallo E, Cillari E, La Rocca AM, Moceo P, Leslie K, et al . Detection of Leishmania infantum kinetoplast DNA in laryngeal tissue from an immunocompetent patient. Hum Pathol 2005;36:1140-2. [PUBMED] [FULLTEXT] |
| 8. | Casolari C, Guaraldi G, Pecorari M, Tamassia G, Cappi C, Fabio G. A rare case of localized mucosal leishmaniasis due to Leishmania infantum in an immunocompetent Italian host. Eur J Epidemiol 2005;20:559-61. |
| 9. | Ravisse P, Bensimon P, Lapicorey G. A case of laryngeal leishmaniasis with a long course. Bull Soc Pathol Exot Filiales 1984;77:305-11. [PUBMED] |
| 10. | Grant A, Spraggs PD, Grant HR, Bryceson AD. Laryngeal leishmaniasis. J Laryngol Otol 1994;108:1086-8. |
Correspondence Address:
Bipin Kumar
Department of Pathology, B.P. Koirala Institute of Health Sciences, Dharan
Nepal
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0377-4929.44967
[Figure 1], [Figure 2], [Figure 3] |
|
| This article has been cited by | | 1 |
Isolated Laryngeal Leishmaniasis: A Diagnostic Dilemma |
|
| Karan Aggarwal,Rajeev Kumar,Nishu Bhardwaj,Bhinyaram Jat,Rakesh Kumar | | Indian Journal of Otolaryngology and Head & Neck Surgery. 2019; | | [Pubmed] | [DOI] | | | 2 |
Generalized Leishmanides in an Immunodeprived Man |
|
| Angel Fernandez-Flores,Aram Boada,Maria T. Fernández-Figueras | | The American Journal of Dermatopathology. 2017; 39(2): e29 | | [Pubmed] | [DOI] | | | 3 |
Isolated Laryngeal Leishmaniasis in Immunocompetent Patients: An Underdiagnosed Disease |
|
| Salvatore Cocuzza,Alessio Strazzulla,Marilia Rita Pinzone,Stefano Cosentino,Agostino Serra,Rosario Caltabiano,Salvatore Lanzafame,Bruno Cacopardo,Giuseppe Nunnari | | Case Reports in Infectious Diseases. 2013; 2013: 1 | | [Pubmed] | [DOI] | | | 4 |
Laryngeal leishmaniasis: Case report of a rare infection |
|
| Trevor A. Teemul,Montserrat Giles-Lima,John Williams,Shane E. Lester,Peter Andersen | | Head & Neck. 2013; 35(9): E277 | | [Pubmed] | [DOI] | | | 5 |
Leishmaniasis laríngea, modificación quirúrgica tras estudio intraoperatorio. Comunicación de un caso |
|
| Roberto Vera-Berón,Rubén Darío García-Ángel,Maria Luisa García-Mauriño Guelbenzu,Blanca M. Martínez-Duro,María García-Sánchez,Juan Pablo García-de la Torre | | Revista Española de Patología. 2013; 46(2): 122 | | [Pubmed] | [DOI] | | | 6 |
Endemic tegumentary leishmaniasis in Brazil: Correlation between level of endemicity and number of cases of mucosal disease |
|
| Bedoya-Pacheco, S.J., Araujo-Melo, M.H., Valete-Rosalino, C.M., Pimentel, M.I.F., Conceição-Silva, F., Schubach, A.O., Marzochi, M.C.A. | | American Journal of Tropical Medicine and Hygiene. 2011; 84(6): 901-905 | | [Pubmed] | | | 7 |
Endemic Tegumentary Leishmaniasis in Brazil: Correlation between Level of Endemicity and Number of Cases of Mucosal Disease |
|
| Sandro J. Bedoya-Pacheco,Maria Inês F. Pimentel,Fátima Conceição-Silva,Maria H. Araujo-Melo,Mauro C. A. Marzochi,Claudia M. Valete-Rosalino,Armando O. Schubach | | The American Journal of Tropical Medicine and Hygiene. 2011; 84(6): 901 | | [Pubmed] | [DOI] | | | 8 |
Recidivant laryngeal leishmaniosis: An unusual case in an immunocompetent patient treated with corticosteroids | [Leishmaniosis laríngea recidivante: Un caso inusual en un paciente inmunocompetente tratado con corticoides] |
|
| Casero, R., Laconte, L., Fraenza, L., Iglesias, N., Quinteros Greco, C., Villablanca, M.L. | | Revista Argentina de Microbiologia. 2010; 42(2): 118-121 | | [Pubmed] | |
|
|
|
|
|
|
|
|
|
|
|
|
| Article Access Statistics | | | Viewed | 4995 | | | Printed | 151 | | | Emailed | 2 | | | PDF Downloaded | 225 | | | Comments | [Add] | | | Cited by others | 8 | | |
|
|
