Laryngeal manifestations of IgG4-related disease. Systematic review
Abstract Introduction and objectives: IgG4-Related disease (IgG4-RD) is a systemic autoimmune disorder characterized by tissue infiltration with IgG4-secreting plasma cells, fibrosis and organic disfunction. It can involve multiple sites. The objectives in this article are to identify the laryngeal involvement of this disease. Methods: A systematic review was conducted by collecting the literature published between 2003 and February 2021, seeking for case reports of IgG4-RD with laryngeal involvement. The bibliographic databases utilized were Pubmed, Web of Science and Scopus. 22 cases were selected, all of them including histological findings in the larynx. Results: Most of the patients were male, between 41 and 60 years old. The most common laryngeal manifestations were pseudotumoral neoformations. The supraglottis was the area were most of these lesions appeared. In this disease the standard treatment involves glucocorticoids, often with an optimal clinical response. However, a great part of the patients included in the review got a delayed diagnosis, some of them leading to aggressive surgical procedures. Conclusions: Laryngeal manifestations in the IgG4-Related disease are unusual. That´s why it should be considered when considering the differential diagnosis of malignant tumors. This is an illness that needs to be suspected in some cases, to ensure a prompt diagnosis and a proper treatment, thus avoiding long-term sequelae.
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Takano K, Yamamoto M, Takahashi H, Himi T. Recent advances in knowledge regarding the head and neck manifestations of IgG4-related disease. Auris Nasus Larynx. 2017;44(1):7-17. doi:10.1016/j.anl.2016.10.011
Hamano H, Kawa S, Horiuchi A, et al. High Serum IgG4 Concentrations in Patients with Sclerosing Pancreatitis. N Engl J Med. 2001; 344(10). doi:10.1056/nejm200103083441005
Kamisawa T, Funata N, Hayashi Y, et al. A new clinicopathological entity of IgG4-related autoimmune disease. J Gastroenterol. 2003;38(10):982-984. doi:10.1007/s00535-003-1175-y
Beyer G, Schwaiger T, Lerch MM, Mayerle J. IgG4-related disease: A new kid on the block or an old aquaintance? United Eur Gastroenterol J. 2014;2(3). doi:10.1177/2050640614532457
Fujita A, Sakai O, Chapman MN, Sugimoto H. IgG4-related disease of the head and neck: CT and MR imaging manifestations. Radiographics. 2012;32(7):1945-1958. doi:10.1148/rg.327125032
Reder L, Della-Torre E, Stone JH, Mori M, Song P. Clinical Manifestations of IgG4-Related Disease in the Pharynx: Case Series and Review of the Literature. Ann Otol Rhinol Laryngol. 2015;124(3). doi:10.1177/0003489414549574.
Kamisawa T, Zen Y, Pillai S, Stone JH. IgG4-related disease. Lancet. 2015;385(9976):1460-1471. doi:10.1016/S0140-6736(14)60720-0
Maritati F, Peyronel F, Vaglio A. IgG4-related disease: A clinical perspective. Rheumatol (United Kingdom). 2020;59:iii123-iii131. doi:10.1093/rheumatology/kez667
Deshpande V, Zen Y, Chan JKC, et al. Consensus statement on the pathology of IgG4-related disease. Mod Pathol. 2012;25(9): 1181-1192. doi:10.1038/modpathol.2012.72
Sánchez-Oro R, Alonso-Muñoz EM, Martí Romero L. Revisión de la enfermedad relacionada con la IgG4. Gastroenterol Hepatol. 2019;42(10). doi:10.1016/j.gastrohep.2019.08.009
Baltar-martín JM, García-monteavaro C, Manzanos TV, Parejo-garcía L. Afectación renal de las enfermedades relacionadas con IgG4. NefroPlus. 2015;7(1):1-10. doi:10.3265/NefroPlus.pre2015.Jan.12960
Martínez-Valle F, Orozco-Gálvez O, Fernández-Codina A. Update in ethiopathogeny, diagnosis and treatment of the IgG4 related disease. Med Clin (Barc). 2018;151(1):18-25. doi:10.1016/j.medcli.2017.10.034
Karim F, Paridaens D, Westenberg LEH, et al. Infliximab for IgG4-Related Orbital Disease. Ophthal Plast Reconstr Surg. 2017; 33(3S). doi:10.1097/IOP.0000000000000625
Khan ML, Colby T V., Viggiano RW, Fonseca R. Treatment with bortezomib of a patient having hyper IgG4 disease. Clin Lymphoma, Myeloma Leuk. 2010;10(3). doi:10.3816/CLML.2010.n.034
Idoyaga P, Fielli M, González A, Ferreyra Mufarregue LR, Fernández Casares M, Crevena G. [IgG4-related interstitial lung disease]. Medicina (B Aires). 2018;78(6):449-452.
Urrútia G, Bonfilll X. La declaración prisma: Un paso adelante en la mejora de las publicaciones de la revista Española de salud pública. Rev Esp Salud Publica. 2013;87(2):99-102. doi:10.4321/S1135-57272013000200001
Völker HU, Scheich M, Zettl A, Hagen R, Müller-Hermelink HK, Gattenlöhner S. Laryngeal inflammatory myofibroblastic tumors: Different clinical appearance and histomorphologic presentation of one entity. Head Neck. 2010;32(11). doi:10.1002/hed.21232
Virk JS, Stamatoglou C, Kwame I, Salama A, Sandison A, Sandhu G. IgG4-Sclerosing Pseudotumor of the Trachea. Arch Otolaryngol Neck Surg. 2012;138(9):864. doi:10.1001/archoto.2012.1821
Khoo JF, Batt M, Stimpson P, Safdar A. Supraglottic immunoglobulin-G4 related plasma cell granuloma: Case report and literature review. Head Neck. 2014;36(6). doi:10.1002/hed.23519
Shaib Y, Ton E, Goldschmeding R, Tekstra J. IgG4-related disease with atypical laryngeal presentation and Behçet/granulomatous polyangiitis mimicking features. BMJ Case Rep. Published online 2013. doi:10.1136/bcr-2013-009158
Morita M, Kitamura M, Hirano S, et al. A case of igg4-related disease with pseudotumor of the Larynx. Pract Otol Suppl. 2015;141. doi:10.5631/jibirinsuppl.141.86
Sabato V, Vanderveken OM, Van den Wyngaert T, Van Laer C, Ebo D. A patient with a severe glottic stenosis and saddle nose. Acta Clin Belgica Int J Clin Lab Med. 2017;72(2):130-132. doi:10.1080/17843286.2015.1111657
Comeche Casanova L, Ortega de Victoria L, Moradiellos J. Tracheal stenosis and IgG4-related disease. Arch Bronconeumol. 2016;52(3): 175. doi:10.1016/j.arbr.2016.01.011
Mustafaev DM. IgG4-related sclerosing disease of the larynx. Vestn Otorinolaringol. 2017;82(2). doi:10.17116/otorino201782277-79
Ferrante SS, Wang L, Kurant D, Daniero JJ. Laryngeal immunoglobulin G4-related disease resulting in severe airway obstruction. JAMA Otolaryngol - Head Neck Surg. 2017;143(4):426-427. doi:10.1001/jamaoto.2016.3931
Suárez-Díaz S, Núñez-Batalla F, Fernández-García MS, Fernández-Llana MB, Yllera-Gutiérrez C, Caminal-Montero L. Aphthous Stomatitis and Laryngitis, Another Form of Presentation of an IgG4-Related Disease? Reumatol Clin. 2020;16(5):416-418. doi:10.1016/j.reuma.2018.08.011
Hamadani S, Wang B, Gupta S. IgG4-related disease presenting as hoarseness and postcricoid ulcer. Ann Allergy, Asthma Immunol. 2018;120(2):211-212. doi:10.1016/j.anai.2017.10.035
Jordan VA, Herrera Hernandez LP, Cofer SA, Roby BB. Pediatric Laryngeal Expression and Surgical Treatment of IgG4-Related Disease. JAMA Otolaryngol - Head Neck Surg. 2018;144(12):1183-1184. doi:10.1001/jamaoto.2018.2258
Matsushima K, Ohira S, Matsui H, et al. IgG4-related disease with pseudotumor formation in the larynx. Auris Nasus Larynx. 2020;47(2):305-308. doi:10.1016/j.anl.2019.05.008
Gradoni P, Frausini G, Pandolfini M, Bedetta S, Migliori G. Laryngeal involvement of immunoglobulin G4-related disease : case report and literature review. Published online 2019:51-54.
Maughan EF, Michaels J, Miller B, et al. Primary Immunoglobulin G4-Related Laryngeal Disease: A Case Series and Review of Literature. Clin Med Insights Case Reports. 2020;13. doi:10.1177/1179547620960197
Poo SX, Pepper RJ, Onwordi L, Ghufoor K, Sandhu G, Salama AD. Sirolimus use in patients with subglottic stenosis in the context of granulomatosis with polyangiitis (GPA), suspected GPA, and immunoglobulin G4-related disease. Scand J Rheumatol. 2021;50(1):52-57. doi:10.1080/03009742.2020.1777324
Atienza-Mateo B, Díaz de Terán-López T, Gómez-Román J, et al. Atypical presentation of immunoglobulin G4-related disease as subglottic stenosis: a case-based review. Rheumatol Int. Published online 2021. doi:10.1007/s00296-021-04816-4
Bledsoe JR, Della-Torre E, Rovati L, Deshpande V. IgG4-related disease: review of the histopathologic features, differential diagnosis, and therapeutic approach. Apmis. 2018;126(6):459-476. doi:10.1111/apm.12845
Lara Martínez MF, Díaz Silván A, Cabello García D, López Acosta M, Allende Riera AJ. 18F-FDG PET/CT in the assessment of a patient with Immunoglobulin G4-related disease. Rev Española Med Nucl e Imagen Mol (English Ed). Published online 2020. doi:10.1016/j.remnie.2020.08.001
Hamano H, Kawa S, Horiuchi A, et al. High Serum IgG4 Concentrations in Patients with Sclerosing Pancreatitis. N Engl J Med. 2001; 344(10). doi:10.1056/nejm200103083441005
Kamisawa T, Funata N, Hayashi Y, et al. A new clinicopathological entity of IgG4-related autoimmune disease. J Gastroenterol. 2003;38(10):982-984. doi:10.1007/s00535-003-1175-y
Beyer G, Schwaiger T, Lerch MM, Mayerle J. IgG4-related disease: A new kid on the block or an old aquaintance? United Eur Gastroenterol J. 2014;2(3). doi:10.1177/2050640614532457
Fujita A, Sakai O, Chapman MN, Sugimoto H. IgG4-related disease of the head and neck: CT and MR imaging manifestations. Radiographics. 2012;32(7):1945-1958. doi:10.1148/rg.327125032
Reder L, Della-Torre E, Stone JH, Mori M, Song P. Clinical Manifestations of IgG4-Related Disease in the Pharynx: Case Series and Review of the Literature. Ann Otol Rhinol Laryngol. 2015;124(3). doi:10.1177/0003489414549574.
Kamisawa T, Zen Y, Pillai S, Stone JH. IgG4-related disease. Lancet. 2015;385(9976):1460-1471. doi:10.1016/S0140-6736(14)60720-0
Maritati F, Peyronel F, Vaglio A. IgG4-related disease: A clinical perspective. Rheumatol (United Kingdom). 2020;59:iii123-iii131. doi:10.1093/rheumatology/kez667
Deshpande V, Zen Y, Chan JKC, et al. Consensus statement on the pathology of IgG4-related disease. Mod Pathol. 2012;25(9): 1181-1192. doi:10.1038/modpathol.2012.72
Sánchez-Oro R, Alonso-Muñoz EM, Martí Romero L. Revisión de la enfermedad relacionada con la IgG4. Gastroenterol Hepatol. 2019;42(10). doi:10.1016/j.gastrohep.2019.08.009
Baltar-martín JM, García-monteavaro C, Manzanos TV, Parejo-garcía L. Afectación renal de las enfermedades relacionadas con IgG4. NefroPlus. 2015;7(1):1-10. doi:10.3265/NefroPlus.pre2015.Jan.12960
Martínez-Valle F, Orozco-Gálvez O, Fernández-Codina A. Update in ethiopathogeny, diagnosis and treatment of the IgG4 related disease. Med Clin (Barc). 2018;151(1):18-25. doi:10.1016/j.medcli.2017.10.034
Karim F, Paridaens D, Westenberg LEH, et al. Infliximab for IgG4-Related Orbital Disease. Ophthal Plast Reconstr Surg. 2017; 33(3S). doi:10.1097/IOP.0000000000000625
Khan ML, Colby T V., Viggiano RW, Fonseca R. Treatment with bortezomib of a patient having hyper IgG4 disease. Clin Lymphoma, Myeloma Leuk. 2010;10(3). doi:10.3816/CLML.2010.n.034
Idoyaga P, Fielli M, González A, Ferreyra Mufarregue LR, Fernández Casares M, Crevena G. [IgG4-related interstitial lung disease]. Medicina (B Aires). 2018;78(6):449-452.
Urrútia G, Bonfilll X. La declaración prisma: Un paso adelante en la mejora de las publicaciones de la revista Española de salud pública. Rev Esp Salud Publica. 2013;87(2):99-102. doi:10.4321/S1135-57272013000200001
Völker HU, Scheich M, Zettl A, Hagen R, Müller-Hermelink HK, Gattenlöhner S. Laryngeal inflammatory myofibroblastic tumors: Different clinical appearance and histomorphologic presentation of one entity. Head Neck. 2010;32(11). doi:10.1002/hed.21232
Virk JS, Stamatoglou C, Kwame I, Salama A, Sandison A, Sandhu G. IgG4-Sclerosing Pseudotumor of the Trachea. Arch Otolaryngol Neck Surg. 2012;138(9):864. doi:10.1001/archoto.2012.1821
Khoo JF, Batt M, Stimpson P, Safdar A. Supraglottic immunoglobulin-G4 related plasma cell granuloma: Case report and literature review. Head Neck. 2014;36(6). doi:10.1002/hed.23519
Shaib Y, Ton E, Goldschmeding R, Tekstra J. IgG4-related disease with atypical laryngeal presentation and Behçet/granulomatous polyangiitis mimicking features. BMJ Case Rep. Published online 2013. doi:10.1136/bcr-2013-009158
Morita M, Kitamura M, Hirano S, et al. A case of igg4-related disease with pseudotumor of the Larynx. Pract Otol Suppl. 2015;141. doi:10.5631/jibirinsuppl.141.86
Sabato V, Vanderveken OM, Van den Wyngaert T, Van Laer C, Ebo D. A patient with a severe glottic stenosis and saddle nose. Acta Clin Belgica Int J Clin Lab Med. 2017;72(2):130-132. doi:10.1080/17843286.2015.1111657
Comeche Casanova L, Ortega de Victoria L, Moradiellos J. Tracheal stenosis and IgG4-related disease. Arch Bronconeumol. 2016;52(3): 175. doi:10.1016/j.arbr.2016.01.011
Mustafaev DM. IgG4-related sclerosing disease of the larynx. Vestn Otorinolaringol. 2017;82(2). doi:10.17116/otorino201782277-79
Ferrante SS, Wang L, Kurant D, Daniero JJ. Laryngeal immunoglobulin G4-related disease resulting in severe airway obstruction. JAMA Otolaryngol - Head Neck Surg. 2017;143(4):426-427. doi:10.1001/jamaoto.2016.3931
Suárez-Díaz S, Núñez-Batalla F, Fernández-García MS, Fernández-Llana MB, Yllera-Gutiérrez C, Caminal-Montero L. Aphthous Stomatitis and Laryngitis, Another Form of Presentation of an IgG4-Related Disease? Reumatol Clin. 2020;16(5):416-418. doi:10.1016/j.reuma.2018.08.011
Hamadani S, Wang B, Gupta S. IgG4-related disease presenting as hoarseness and postcricoid ulcer. Ann Allergy, Asthma Immunol. 2018;120(2):211-212. doi:10.1016/j.anai.2017.10.035
Jordan VA, Herrera Hernandez LP, Cofer SA, Roby BB. Pediatric Laryngeal Expression and Surgical Treatment of IgG4-Related Disease. JAMA Otolaryngol - Head Neck Surg. 2018;144(12):1183-1184. doi:10.1001/jamaoto.2018.2258
Matsushima K, Ohira S, Matsui H, et al. IgG4-related disease with pseudotumor formation in the larynx. Auris Nasus Larynx. 2020;47(2):305-308. doi:10.1016/j.anl.2019.05.008
Gradoni P, Frausini G, Pandolfini M, Bedetta S, Migliori G. Laryngeal involvement of immunoglobulin G4-related disease : case report and literature review. Published online 2019:51-54.
Maughan EF, Michaels J, Miller B, et al. Primary Immunoglobulin G4-Related Laryngeal Disease: A Case Series and Review of Literature. Clin Med Insights Case Reports. 2020;13. doi:10.1177/1179547620960197
Poo SX, Pepper RJ, Onwordi L, Ghufoor K, Sandhu G, Salama AD. Sirolimus use in patients with subglottic stenosis in the context of granulomatosis with polyangiitis (GPA), suspected GPA, and immunoglobulin G4-related disease. Scand J Rheumatol. 2021;50(1):52-57. doi:10.1080/03009742.2020.1777324
Atienza-Mateo B, Díaz de Terán-López T, Gómez-Román J, et al. Atypical presentation of immunoglobulin G4-related disease as subglottic stenosis: a case-based review. Rheumatol Int. Published online 2021. doi:10.1007/s00296-021-04816-4
Bledsoe JR, Della-Torre E, Rovati L, Deshpande V. IgG4-related disease: review of the histopathologic features, differential diagnosis, and therapeutic approach. Apmis. 2018;126(6):459-476. doi:10.1111/apm.12845
Lara Martínez MF, Díaz Silván A, Cabello García D, López Acosta M, Allende Riera AJ. 18F-FDG PET/CT in the assessment of a patient with Immunoglobulin G4-related disease. Rev Española Med Nucl e Imagen Mol (English Ed). Published online 2020. doi:10.1016/j.remnie.2020.08.001
Bedia-Cadelo, J. ., & Morales-Angulo, C. (2022). Laryngeal manifestations of IgG4-related disease. Systematic review. Revista ORL, 13(1), e27445. https://doi.org/10.14201/orl.27445
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