Abstract
Amyloidoma is a solitary mass of amyloid protein that arises in patients with or without evidence of systemic amyloidosis, and can be found in a variety of different organ systems. Herein, we describe three cases of localized biopsy-positive amyloidomas with no evidence of systemic involvement—primary amyloidoma. Our cases include a patient with a paraspinal soft tissue amyloidoma, a patient with multiple primary amyloidomas involving the thoracic cavity and flank, and a patient with insulin-injection induced amyloidoma of the left shoulder. We present these cases to provide further insights into the clinical presentation of this uncommon clinical entity. We review the pathophysiology of amyloidosis and discuss our cases in the context of previous reports of amyloidoma.
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References
Kumar V, Abbas AK, Aster JC, Perkins JA. Robbins basic pathology. Tenth ed. Philadelphia: Elsevier; 2018. xiv, 935 pages
Esplin BL, Gertz MA. Current trends in diagnosis and management of cardiac amyloidosis. Curr Probl Cardiol. 2013;38(2):53–96.
Goldsbury C, Baxa U, Simon MN, Steven AC, Engel A, Wall JS, et al. Amyloid structure and assembly: insights from scanning transmission electron microscopy. J Struct Biol. 2011;173(1):1–13.
Sipe JD, Cohen AS. Review: history of the amyloid fibril. J Struct Biol. 2000;130(2–3):88–98.
Pettersson T, Konttinen YT. Amyloidosis-recent developments. Semin Arthritis Rheum. 2010;39(5):356–68.
Bely M, Kapp P, Szabo TS, Lakatos T, Apathy A. Electron microscopic characteristics of beta2-microglobulin amyloid deposits in long-term haemodialysis. Ultrastruct Pathol. 2005;29(6):483–91.
Wechalekar AD, Gillmore JD, Hawkins PN. Systemic amyloidosis. Lancet. 2016;387(10038):2641–54.
Yanagisawa A, Ueda M, Sueyoshi T, Nakamura E, Tasaki M, Suenaga G, et al. Knee osteoarthritis associated with different kinds of amyloid deposits and the impact of aging on type of amyloid. Amyloid. 2016;23(1):26–32.
Chang YS, Ku MC, Lee TS. Primary amyloidoma of the spine. Zhonghua Yi Xue Za Zhi (Taipei). 1993;51(1):85–90.
Fierens J, Mees U, Vanbockrijck M, Hendrikx M. Amyloidoma of the chest wall: a rare entity. Interact Cardiovasc Thorac Surg. 2008;7(6):1194–5.
Haridas A, Basu S, King A, Pollock J. Primary isolated amyloidoma of the lumbar spine causing neurological compromise: case report and literature review. Neurosurgery. 2005;57(1):E196 discussion E196.
Hwang SS, Park YH, Kim JY, Jung SL, Ahn MI, Park CK, et al. Primary amyloidoma of the cervical spine. AJNR Am J Neuroradiol. 2000;21(3):601–3.
Landau D, Avgeropoulos N, Ma J. Cerebral amyloidoma mimicking intracranial tumor: a case report. J Med Case Rep. 2010;4:308.
Mayhew JM, Alan T, Kalidindi V, Gandamihardja TAK. Isolated insulin-derived amyloidoma of the breast. BMJ Case Rep. 2017;2017(1):e219491.
Mizuno J, Nakagawa H, Tsuji Y, Yamada T. Primary amyloidoma of the thoracic spine presenting with acute paraplegia. Surg Neurol. 2001;55(6):378–82.
Reitboeck JG, Feldmann R, Loader D, Breier F, Steiner A. Primary cutaneous amyloidoma: a case report. Case Rep Dermatol. 2014;6(3):264–7.
Savant D, Adler M, Kahn L, Cocker R. Amyloidoma of stomach: a case report. Acta Cytol. 2018;62(3):231–3.
Suri VS, Tatke M, Kumar S, Gupta V. Amyloidoma of the thoracic spine. Case report. J Neurosurg. 2001;94(2 Suppl):299–301.
Walsh NM, Lano IM, Green P, Gallant C, Pasternak S, Ly TY, et al. AL Amyloidoma of the skin/subcutis: cutaneous amyloidosis, plasma cell dyscrasia or a manifestation of primary cutaneous marginal zone lymphoma? Am J Surg Pathol. 2017;41(8):1069–76.
Kapoor A, Manje Gowda A, Kaur S, Estifan E, Maroules M. A rare case of cerebral amyloidoma mimicking a hemorrhagic malignant central nervous system neoplasm. Cureus. 2020;12(3):e7245.
Dalal BS, Sarkar P, Maity N, Nadeem SM. Soft-tissue amyloidoma with associated plasmacytoma. Indian J Pathol Microbiol. 2016;59(4):535–7.
Kaltoft B, Schmidt G, Lauritzen AF, Gimsing P. Primary localised cutaneous amyloidosis--a systematic review. Dan Med J. 2013;60(11):A4727.
Leeson MC, Rechtine GR, Makley JT, Carter JR. Primary amyloidoma of the spine. A case report and review of the literature. Spine (Phila Pa 1976). 1985;10(4):303–6.
Verhoeven F, Prati C, Wendling D. Amyloidoma, an unusual cause of fracture. Case Rep Rheumatol. 2014;2014:424056.
Lai KN, Chan KW, Siu DL, Wong CC, Yeung D. Pathologic hip fractures secondary to amyloidoma. Case report and review of the literature. Am J Med. 1984;77(5):937–43.
Hashmi H, Dhanoa J, Manapuram S. Trigeminal amyloidoma: case report and review of literature. Cureus. 2018;10(12):e3795.
Bornemann A, Bohl J, Hey O, Storkel S, Gamm H, Muller-Forell W, et al. Amyloidoma of the gasserian ganglion as a cause of symptomatic neuralgia of the trigeminal nerve: report of three cases. J Neurol. 1993;241(1):10–4.
Menetti F, Bartolomei I, Ambrosini-Spaltro A, Salvi F, Agati R, Leonardi M. Amyloidoma involving the orbit, Meckel’s cave and infratemporal fossa: 3T MRI findings. Neuroradiol J. 2009;22(1):41–7.
Matsumoto T, Tani E, Maeda Y, Natsume S. Amyloidomas in the cerebellopontine angle and jugular foramen. Case report J Neurosurg. 1985;62(4):592–6.
Consales A, Roncaroli F, Salvi F, Poppi M. Amyloidoma of the brachial plexus. Surg Neurol. 2003;59(5):418–23 discussion 423.
Bookland MJ, Bagley CA, Schwarz J, Burger PC, Brem H. Intracavernous trigeminal ganglion amyloidoma: case report. Neurosurgery. 2007;60(3):E574 discussion E574.
D’Souza A, Theis JD, Vrana JA, Dogan A. Pharmaceutical amyloidosis associated with subcutaneous insulin and enfuvirtide administration. Amyloid. 2014;21(2):71–5.
Nakamura M, Misumi Y, Nomura T, Oka W, Isoguchi A, Kanenawa K, et al. Extreme adhesion activity of amyloid fibrils induces subcutaneous insulin resistance. Diabetes. 2019;68(3):609–16.
Amdursky N, Gazit E, Rosenman G. Formation of low-dimensional crystalline nucleus region during insulin amyloidogenesis process. Biochem Biophys Res Commun. 2012;419(2):232–7.
Landreh M, Stukenborg JB, Willander H, Soder O, Johansson J, Jornvall H. Proinsulin C-peptide interferes with insulin fibril formation. Biochem Biophys Res Commun. 2012;418(3):489–93.
Dickman CA, Sonntag VK, Johnson P, Medina M. Amyloidoma of the cervical spine: a case report. Neurosurgery. 1988;22(2):419–22.
Meyers SP, Mullins KJ, Kazee AM. Unifocal primary amyloidoma of the spine causing compression of the cervical spinal cord: MR findings. J Comput Assist Tomogr. 1996;20(4):592–3.
Hegarty JL, Rao VM. Amyloidoma of the nasopharynx: CT and MR findings. AJNR Am J Neuroradiol. 1993;14(1):215–8.
Urban BA, Fishman EK, Humphrey RL. Primary amyloidosis of the sternum. Imaging findings. Clin Imaging. 1994;18(1):75–8.
Lee J, Krol G, Rosenblum M. Primary amyloidoma of the brain: CT and MR presentation. AJNR Am J Neuroradiol. 1995;16(4):712–4.
Hamidi Asl K, Liepnieks JJ, Nakamura M, Benson MD. Organ-specific (localized) synthesis of Ig light chain amyloid. J Immunol. 1999;162(9):5556–60.
Sari I, Birlik M, Kasifoglu T. Familial Mediterranean fever: an updated review. Eur J Rheumatol. 2014;1(1):21–33.
Qin Y, Smyrk TC, Topazian MD. Biliary amyloidoma as a cause for pancreatitis. Clin Gastroenterol Hepatol. 2019;17(11):A25–6.
Schneider JR, Kwan K, Kulason KO, Faltings LJ, Colantonio S, Safir S, et al. Primary solitary retro-clival amyloidoma. Surg Neurol Int. 2018;9:100.
Pasternak S, Wright BA, Walsh N. Soft tissue amyloidoma of the extremities: report of a case and review of the literature. Am J Dermatopathol. 2007;29(2):152–5.
Koczka CP, Goodman AJ. Gastric amyloidoma in patient after remission of non-Hodgkin’s lymphoma. World J Gastrointest Oncol. 2009;1(1):93–6.
Sin AH, Gonzalez-Toledo E, Fowler M, Minagar A, Nanda A. Amyloidoma presenting as a butterfly glioma on positron emission tomography scan and magnetic resonance-spectroscopy: a case report and review of the literature. J La State Med Soc. 2008;160(1):44–7 49-50.
Umeda Y, Demura Y, Takeda N, Morikawa M, Uesaka D, Nakanishi M, et al. FDG-PET findings of nodular pulmonary amyloidosis with a long-term observation. Nihon Kokyuki Gakkai Zasshi. 2007;45(5):424–9.
Xu L, Frazier A, Burke A. Isolated pulmonary amyloidomas: report of 3 cases with histologic and imaging findings. Pathol Res Pract. 2013;209(1):62–6.
Kiss E, Keusch G, Zanetti M, Jung T, Schwarz A, Schocke M, et al. Dialysis-related amyloidosis revisited. AJR Am J Roentgenol. 2005;185(6):1460–7.
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Desai, S.S., Rizzo, M.G., Rush, A.J. et al. Amyloidoma: a review and case report. Skeletal Radiol 50, 437–444 (2021). https://doi.org/10.1007/s00256-020-03560-3
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DOI: https://doi.org/10.1007/s00256-020-03560-3