Adult onset Xanthogranuloma presenting as a solitary laryngeal localization: case report and review of literature

Gianluca Velletrani, Beatrice Francavilla, Valentina Rosati, Belen Padial, Lucia Anemona, Stefano Di Girolamo

Abstract


Juvenile Xanthogranuloma (XG) is a rare disorder that belongs to the heterogeneous group of histiocytic neoplasms, characterized by a clonal expansion of non-Langerhans cell histiocytes that share a dermal macrophage phenotype. Although the head and neck region is the most common reported site of involvement by the Juvenile Xanthogranuloma family, laryngeal localization is extremely rare. We report a unique case of Adult Onset Xanthogranuloma with subglottic localization, presenting as a solitary laryngeal mass without other systemic or cutaneous lesions. A review of the previously described cases of laryngeal Xanthogranuloma has been performed, highlighting 7 cases of Juvenile Xanthogranuloma and only 3 cases of Adult Onset Xanthogranuloma. Despite the extreme rarity of laryngeal localization of XG, this histiocytic neoplasm should be considered as a differential diagnosis for laryngeal masses causing airway obstruction, even in the absence of other concomitant manifestations.


Keywords


Xanthogranuloma; Larynx; Subglottis; Histiocytic Neoplasm; Non-Langerhans; Histiocytosis

Full Text:

PDF

References


Ceppi F, Abla O. Juvenile Xanthogranuloma and Related Non-LCH Disorders. Springer International Publishing. 2018;293–311.

Khoury JD, Solary E, Abla O, Akkari Y, Alaggio R, Apperley JF, et al. The 5th edition of the World Health Organization Classification of Haematolymphoid Tumours: Myeloid and Histiocytic/Dendritic Neoplasms. Leukemia. 2022;36(7):1703–19.

Emile JF, Abla O, Fraitag S, Horne A, Haroche J, Donadieu J, et al. Revised classification of histiocytoses and neoplasms of the macrophage-dendritic cell lineages. Blood. 2016;127(22):2672–81.

Sharma, A., McAfee, J., Wang, L., Cook, E., Ababneh, E. and Bergfeld, W. Utility of Cyclin D1 Immunostaining in Cutaneous Xanthogranuloma. The American Journal of Dermatopathology. 2021;43(12):141-145.

Sampedro‐Ruiz R, Martos‐Cabrera L, Fraga‐Fernández J, Aragüés M, Chicharro P. Multiple adult xanthogranuloma with upper airway involvement. Int J Dermatol. 2021;60(8).

Li S, Weidenbecher M. Adult onset xanthogranuloma presenting as laryngeal mass. Am J Otolaryngol. 2016;37(2):112–5.

Yoon D, Jang D, Byun HJ, Park CS, Park JH, Lee JH, et al. A case of generalized xanthogranuloma with systemic involvement. JAAD Case Rep. 2019;5(12):1097–100.

Verma C, Taneja K, Mahajan A. Juvenile Xanthogranuloma of Subglottis: Rare, Recurrent and Refractory. Indian Pediatr. 2022;59(6):501–501.

Kawamoto A, Katori Y, Honkura Y, Ishii K, Ogura M, Naganuma H, et al. Isolated Juvenile Xanthogranuloma in the Larynx of a Three-Year-Old Child. Tohoku J Exp Med. 2013;230(2):123–7.

Wang LM, Chen QH, Zhang YX, Fang YQ, Wang ZC, Zhu Q. Laryngeal juvenile xanthogranuloma: Imaging finding. Int J Pediatr Otorhinolaryngol. 2010;74(8):949–51.

Somorai M, Goldstein NA, Alexis R, Giusti RJ. Managing isolated subglottic juvenile xanthogranuloma without tracheostomy: Case report and review of literature. Pediatr Pulmonol. 2007;42(2):181–5.

Sahhar HS, Marra S, Shahid R, Akhter J. Juvenile Xanthogranuloma: A Rare Cause of Subglottic Cyst and Stenosis. Ear Nose Throat J. 2003;82(9):725–6.

Thevasagayam MS, Ghosh S, O’Neill D, Panarese A, Bull PD. Isolated juvenile xanthogranuloma of the subglottis: Case report. Head Neck. 2001;23(5):426–9.

Benjamin B, Motbey J, Ivers C, Kan A. Benign juvenile xanthogranuloma of the larynx. Int J Pediatr Otorhinolaryngol. 1995;32(1):77–81.




DOI: http://dx.doi.org/10.22551/2022.37.0904.10221

Copyright (c) 2022 Gianluca Velletrani, Beatrice Francavilla, Valentina Rosati, Belen Padial, Lucia Anemona, Stefano Di Girolamo

Creative Commons License
Archive of Clinical Cases is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.

 

ISSN: 2360-6975