Abstract
This report describes two cases of oral localized amyloidosis (LA). In case 1, a 52-year-old man appeared with painful slightly, yellowish multiple nodules located on the dorsum of the tongue, of unknown duration. Incisional biopsy was performed, and the histopathologic analysis revealed a homogeneous, eosinophilic, and extracellular material. Congo red stain showed salmon pink coloration at light microscopy and apple-green birefringence at polarized light. In case 2, a 74-year-old man presented asymptomatic nodular lesions on the labial commissures with duration of several months. An excisional biopsy was performed in both lesions, and microscopically the specimen demonstrated the same histopathologic features of the case 1. Furthermore, amyloidosis with systemic involvement was excluded after investigations for both patients. Thus, the final diagnosis for both cases was LA. The patient 1 refused the surgical excision of the residual lesion, and in both cases, no signs of clinical and systemic progression were observed after 24 and 84 months of follow up. Although it is rare, LA should be considered in the differential diagnosis of multiple or single yellowish nodules on the oral cavity.
Similar content being viewed by others
Data Availability
Not applicable.
Code Availability
Not applicable.
References
Fahrner KS, Black CC, Gosselin BJ. Localized amyloidosis of the tongue: a review. Am J Otolaryngol. 2004;25(3):186–9. doi:https://doi.org/10.1016/j.amjoto.2004.01.007.
Cengiz MI, Wang HL, Yıldız L. Oral involvement in a case of AA amyloidosis: a case report. J Med Case Rep. 2010;4:200. doi:https://doi.org/10.1186/1752-1947-4-200.
Cohen AS, Calkins E. Electron microscopic observations on a fibrous component in amyloid of diverse origins. Nature. 1959;183(4669):1202–3. doi:https://doi.org/10.1038/1831202a0.
Falk RH, Comenzo RL, Skinner M. The systemic amyloidoses. N Engl J Med. 1997;337(13):898–909. doi:https://doi.org/10.1056/NEJM199709253371306.
Pentenero M, Davico Bonino L, Tomasini C, Conrotto D, Gandolfo S. Localized oral amyloidosis of the palate. Amyloid. 2006;13(1):42–6. https://doi.org/10.1080/13506120500537343.
Balatsouras DG, Eliopoulos P, Assimakopoulos D, Korres S. Primary local amyloidosis of the palate. Otolaryngol Head Neck Surg. 2007;137(2):348–9. doi:https://doi.org/10.1016/j.otohns.2007.03.039.
Aono J, Yamagata K, Yoshida H. Local amyloidosis in the hard palate: a case report. Oral Maxillofac Surg. 2009;13(2):119–22. doi:https://doi.org/10.1007/s10006-009-0158-4.
Pang KP, Chee LWJ, Busmanis I. Amyloidoma of the nose in a pediatric Patient: a case report. Am J Otolaryngol. 2001;22(2):138–41. doi:https://doi.org/10.1053/ajot.2001.22576.
Babburi S, B R, Rv S, Srivastava VA G. Amyloidosis of the tongue – report of a rare case. J Clin Diagn Res. 2013;7(12):3094-5. Doi: https://doi.org/10.7860/JCDR/2013/7028.3865.
Stoor P, Suuronen R, Lindqvist C, Hietanen J, Laine P. Local primary (AL) amyloidosis in the palate. A case report. Int J Oral Maxillofac Surg. 2004;33(4):402–3. doi:https://doi.org/10.1016/j.ijom.2003.08.010.
Yamagata K, Bukawa H. Oral localized amyloidosis. In: Güvenç IA ed. Amyloidosis-an insight to disease of systems and novel therapies. London: IntechOpen; 2011. pp. 141–52. doi:https://doi.org/10.5772/1785.
Haraguchi H, Ohashi K, Yamada M, Hasegawa M, Maeda S, Komatsuzaki A. Primary localized nodular tongue amyloidosis associated with Sjögren’s syndrome. J Otorhinolaryngol Relat Spec. 1997;59(1):60–3. doi:https://doi.org/10.1159/00027690.
Koren R, Veltman V, Halpern M, Szabo R, Gal R. Localized amyloid tumor of the tongue. A case report and review of the literature. Rom J Morphol Embryol. 1998;44(1–4):179–82.
Asaumi J, Yanagi Y, Hisatomi M, Konouchi H, Kishi K. CT and MR imaging of localized amyloidosis. Eur J Radiol. 2001;39(2):83–7. doi:https://doi.org/10.1016/s0720-048x(01)00300-x.
Singh A, Haq M, Gautam P, Gautam D, Handa AC, Handa KK. Clinical Profile of Patients with Head and Neck Amyloidosis: A Single-Institution Retrospective Chart Review. Int Arch Otorhinolaryngol. 2020;24(4):e450–6. doi:https://doi.org/10.1055/s-0039-3402494.
O’Reilly A, D’Souza A, Lust J, Price D. Localized tongue amyloidosis: a single institutional case series. Otolaryngol Head Neck Surg. 2013;149(2):240–4. doi:https://doi.org/10.1177/0194599813490896.
McAlpine JC, Fuller AP. Localized laryngeal amyloidosis, a report of a case with a review of the literature. J Laryngol Otol. 1964;78:296–314. doi:https://doi.org/10.1017/s0022215100062113.
Yamoaka Y, Suzuki A, Hatakeyama S, Noda M, Hiraga M, Sekiyama S. Median rhomboid glossitis associated with amyloid deposition. Act Pathol Jpn. 1978;23:319–23. doi:https://doi.org/10.1111/j.1440-1827.1978.tb00544.x.
Liu X, Zhou P, Hua H. Characteristics of Orofacial Amyloidosis: A Case Series. Ann Clin Case Rep. 2016;1:1133.
Noguchi T, Jinbu, Sakurai S. Kusama. Primary Localized Amyloidosis of the Angle of Mouth: Report of a case. Oral Med Pathol. 2003;8(1):27–30. doi:https://doi.org/10.3353/omp.8.27.
Silva WPP, Wastner B, Bohn JC, Jung JE, Schussel JL, Sassi LM. Unusual presentation of oral amyloidosis. Contemp Clin Dent. 2015;6(1):282-4. doi:https://doi.org/10.4103/0976-237X.166814.
Timoşca G, Gavriliţă L. Primary localized amyloidosis of the palate. Oral Surg. 1977;44(1):76–83. doi:https://doi.org/10.1016/0030-4220(77)90247-x.
Rennie JS, Critchlow HA. Solitary oral amyloid. A report of two cases. Int J Oral Surg. 1982;11(1):73–6. doi:https://doi.org/10.1016/s0300-9785(82)80053-7.
Takeda Y, Sekiyama S, Suzuki A, Hirose H. Localized oral amyloidosis: ultrastructural and immunohistochemical study. J Oral Pathology. 1987;16(5):278–81. doi:https://doi.org/10.1111/j.1600-0714.1987.tb01493.x.
Madani M, Harwick RD, Chen SY, Miller AS. Amyloidosis of the oral cavity: report of five cases. Compendium. 1991;12(5):336. 338 – 42.
Kerner MM, Wand MD, Angier G, Calcaterra TC, Ward PH. Amyloidosis of the head and neck—a clinicopathologic study of the UCLA experience. Arch Otolaryngol Head Neck Surg. 1995;121(7):778–82. doi:https://doi.org/10.1001/archotol.1995.01890070064014.
Alvi A, Goldstein MN. Amyloidosis of the palate. Otolaryngol Head Neck Surg. 1999;120(2):287. doi:https://doi.org/10.1016/S0194-5998(99)70424-9.
Penner CR, Muller S. Head and neck amyloidosis: a clinicopathologic study of 15 cases. Oral Oncol. 2006;42(4):421–9. doi:https://doi.org/10.1016/j.oraloncology.2005.09.010.
Henley E, Houghton N, Bucknall R, Triantafyllou A, Field EA. Localized amyloidosis of the palate. Clin Exp Dermatol. 2008;33(1):100–1. doi:https://doi.org/10.1111/j.1365-2230.2007.02528.x.
Akyildiz S, Doganavsargil B, Göde S, Veral A. Solitary amyloid tumor of the tongue base. Int J Otolaryngol. 2009;2009:515068. doi:https://doi.org/10.1155/2009/515068.
Angiero F, Seramondi R, Magistro S, Crippa R, Benedicenti S, Rizzardi C, Cattoretti G. Amyloid deposition in the tongue: clinical and histopathological profile. Anticancer Res. 2010;30(7):3009–14.
Andreadis D, Poulopoulos A, Papadopoulos P, Epivatianos A. Localized tongue amyloidosis in a patient with neurofibromatosis type II. Head Neck Pathol. 2011;5(3):302–5. doi:https://doi.org/10.1007/s12105-011-0251-5.
Gouvêa AF, Ribeiro ACP, Léon JE, Carlos R, De Almeida OP, Lopes MA. Head and neck amyloidosis: clinicopathological features and immunohistochemical analysis of 14 cases. J Oral Pathol Med. 2012;41(2):178–85. doi:https://doi.org/10.1111/j.1600-0714.2011.01073.x.
Bucci T, Bucci E, Rullan AMP, Bucci P, Nuzzolo. Localized amyloidosis of the upper gingiva: a case report. J Med Case Rep. 2014;8:198. doi:https://doi.org/10.1186/1752-1947-8-198.
Folkard SS, Gibbs SD, Shah KA, Dhariwak DK. A rare case of localized oral amyloid of the labial mucosa. Br J Oral Maxillofac Surg. 2014;52(4):e24–5. doi:https://doi.org/10.1016/j.bjoms.2014.01.014.
Matsuo FS, De Paulo LFB, Servato JPS, De Faria PR, Cardoso SV, Loyola AM. Involvement of oral tissue by AL amyloidosis: a literature review and report of eight new cases. Clin Oral Invest. 2016;20(8):1913–20. doi:https://doi.org/10.1007/s00784-015-1649-3.
Adamo D, Gasparro R, Marenzi G, Mascolo M, Cervasio M, Cerciello G, De Novellis D, Mignogna MD. Amyloidoma of the tongue: case report, surgical management, and review of the literature. J Oral Maxillofac Surg. 2020;78(9):1572–82. doi:https://doi.org/10.1016/j.joms.2020.04.022.
Morelato L, Smojver I, Seiwerth S, Gabric D. Extremely rare intraoral presentation of localized amyloidosis. Case Rep Dent. 2021;2021:5541320. doi: https://doi.org/10.1155/2021/5541320.
Thompson LDR, Deringer GA, Wenig BM. Amyloidosis of the larynx: a clinicopathologic study of 11 cases. Mod Pathol. 2000;13(5):528–35. doi:https://doi.org/10.1038/modpathol.3880092.
Deng J, Chen Q, Ji P, Zeng X, Jin X. Oral amyloidosis: A strategy to differentiate systemic amyloidosis involving the oral cavity and localized amyloidosis. Oral Dis. 2019;25(3):670–5. doi:https://doi.org/10.1111/odi.12870.
Funding
Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq), Brazil.
Author information
Authors and Affiliations
Contributions
Conceptualization: Danyel Elias da Cruz Perez; José Divaldo Prado; Rafael Segura Saint-Gerons. Supervision: Danyel Elias da Cruz Perez; José Divaldo Prado. Visualization: Elaine Judite de Amorim Carvalho. Writing – original draft: Hélen Kaline Farias Bezerra; Talita Ribeiro Tenório de França. Writing – review & editing: Danyel Elias da Cruz Perez; Elaine Judite de Amorim Carvalho; Rafael Segura Saint-Gerons.
Corresponding author
Ethics declarations
Conflict of interest
The authors declare no conflict of interest.
Ethical Approval
The study was performed following the 1964 Helsinki Declaration standards.
Consent to participate
Not applicable.
Consent for publication
Not applicable.
Additional information
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Bezerra, H.K.F., de França, T.R.T., Prado, J.D. et al. Oral localized amyloidosis. Head and Neck Pathol 16, 818–822 (2022). https://doi.org/10.1007/s12105-022-01436-7
Received:
Revised:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12105-022-01436-7