Giulia Querzoli, Margherita Ortalli, Stefania Varani, Matteo Errani, Andrea Ambrosini-Spaltro, Marina Del Vecchio, Anna Rita Lombardi, Paolo Rinaldi, Carlo Biagetti, Riccardo Albertini, Luca Amorosa, Alessandro Rosti, Marco Trebbi, Paolo Farneti, Ernesto Pasquini, Antonio Mastroianni, Maria Pia Foschini, Valeria Gaspari
{"title":"Tonsillar Leishmaniasis: A Rare Clinical Entity Mimicking Malignancy in the Oropharynx - A Case Series from Northeastern Italy.","authors":"Giulia Querzoli, Margherita Ortalli, Stefania Varani, Matteo Errani, Andrea Ambrosini-Spaltro, Marina Del Vecchio, Anna Rita Lombardi, Paolo Rinaldi, Carlo Biagetti, Riccardo Albertini, Luca Amorosa, Alessandro Rosti, Marco Trebbi, Paolo Farneti, Ernesto Pasquini, Antonio Mastroianni, Maria Pia Foschini, Valeria Gaspari","doi":"10.1007/s12105-025-01773-3","DOIUrl":null,"url":null,"abstract":"<p><strong>Purpose: </strong>Tegumentary leishmaniasis (TL) is a neglected disease in Europe, often underdiagnosed or misdiagnosed due to its variable clinical presentation. Mucosal leishmaniasis (ML) is a rare manifestation of TL, and isolated tonsillar leishmaniasis is an even rarer finding, with very few reported cases. This study aims to expand knowledge on this unusual clinical entity by describing five cases of isolated tonsillar leishmaniasis diagnosed in the Emilia-Romagna region (ERR), northeastern Italy, emphasizing diagnostic challenges and treatment outcomes.</p><p><strong>Methods: </strong>Between January 2014 and December 2024, all consecutive patients presenting with unilateral tonsillar swelling and pharyngodynia were evaluated at otolaryngology units in ERR hospitals. Histopathological analysis, special stains (H&E, Giemsa, PAS, Ziehl-Neelsen), and immunostaining (CD1a) were performed at the referring hospital. Microbiological confirmation was obtained through real-time PCR targeting Leishmania kDNA and rRNA. Treatment was administered according to current TL guidelines.</p><p><strong>Results: </strong>We included five patients who presented with unilateral tonsillar swelling mimicking malignancy and with a histological diagnosis of non-necrotizing granulomas. Histology revealed amastigotes in four cases. PCR confirmed Leishmania infection in all cases. Treatment with liposomal amphotericin B or pentamidine led to complete clinical remission.</p><p><strong>Conclusion: </strong>Isolated tonsillar leishmaniasis should be considered in the differential diagnosis of head and neck tumors, especially in endemic regions. Histological and molecular tools are essential for accurate diagnosis. Increased awareness among clinicians and pathologists is necessary to improve recognition and management of this rare presentation.</p>","PeriodicalId":47972,"journal":{"name":"Head & Neck Pathology","volume":"19 1","pages":"39"},"PeriodicalIF":3.2000,"publicationDate":"2025-03-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11947358/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Head & Neck Pathology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1007/s12105-025-01773-3","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"PATHOLOGY","Score":null,"Total":0}
引用次数: 0
Abstract
Purpose: Tegumentary leishmaniasis (TL) is a neglected disease in Europe, often underdiagnosed or misdiagnosed due to its variable clinical presentation. Mucosal leishmaniasis (ML) is a rare manifestation of TL, and isolated tonsillar leishmaniasis is an even rarer finding, with very few reported cases. This study aims to expand knowledge on this unusual clinical entity by describing five cases of isolated tonsillar leishmaniasis diagnosed in the Emilia-Romagna region (ERR), northeastern Italy, emphasizing diagnostic challenges and treatment outcomes.
Methods: Between January 2014 and December 2024, all consecutive patients presenting with unilateral tonsillar swelling and pharyngodynia were evaluated at otolaryngology units in ERR hospitals. Histopathological analysis, special stains (H&E, Giemsa, PAS, Ziehl-Neelsen), and immunostaining (CD1a) were performed at the referring hospital. Microbiological confirmation was obtained through real-time PCR targeting Leishmania kDNA and rRNA. Treatment was administered according to current TL guidelines.
Results: We included five patients who presented with unilateral tonsillar swelling mimicking malignancy and with a histological diagnosis of non-necrotizing granulomas. Histology revealed amastigotes in four cases. PCR confirmed Leishmania infection in all cases. Treatment with liposomal amphotericin B or pentamidine led to complete clinical remission.
Conclusion: Isolated tonsillar leishmaniasis should be considered in the differential diagnosis of head and neck tumors, especially in endemic regions. Histological and molecular tools are essential for accurate diagnosis. Increased awareness among clinicians and pathologists is necessary to improve recognition and management of this rare presentation.
期刊介绍:
Head & Neck Pathology presents scholarly papers, reviews and symposia that cover the spectrum of human surgical pathology within the anatomic zones of the oral cavity, sinonasal tract, larynx, hypopharynx, salivary gland, ear and temporal bone, and neck.
The journal publishes rapid developments in new diagnostic criteria, intraoperative consultation, immunohistochemical studies, molecular techniques, genetic analyses, diagnostic aids, experimental pathology, cytology, radiographic imaging, and application of uniform terminology to allow practitioners to continue to maintain and expand their knowledge in the subspecialty of head and neck pathology. Coverage of practical application to daily clinical practice is supported with proceedings and symposia from international societies and academies devoted to this field.
Single-blind peer review
The journal follows a single-blind review procedure, where the reviewers are aware of the names and affiliations of the authors, but the reviewer reports provided to authors are anonymous. Single-blind peer review is the traditional model of peer review that many reviewers are comfortable with, and it facilitates a dispassionate critique of a manuscript.