Abstract
Thymomas are the most common mediastinal neoplasms in adults. Patients with thymoma are frequently diagnosed with paraneoplastic syndromes and are generally associated with several autoimmune conditions. Sjögren's syndrome (SS) is an autoimmune inflammatory disorder, characterized by the lymphocyte-mediated destruction of the exocrine glands, which leads to the absence of glandular secretion, particularly salivary and lacrimal; hyposalivation can vary from a slight reduction in the flow of saliva with transient discomfort, to a serious deterioration of oral health and psychological distress. Mucous membrane pemphigoid (MMP) is a chronic, autoimmune disease that shows predominant mucosal involvement, most commonly affecting the oral cavity, followed by the conjunctiva, the nasal cavity and the esophagus; intraorally includes scaly gingivitis, vesicles, erosions covered by pseudomembranes and ulcers. The objective of this work is to present the first case of a patient with a history of thymoma, which, jointly presented SS and MMP; also highlight the importance of multidisciplinary management for an accurate and early diagnosis, as this increases the efficiency and efficacy of the treatment strategy.
References
Kelly R. J., Petrini I., Rajan A., Wang Y., Giaccone G. Thymic malignancies: from clinical management to targeted therapies. J Clin Oncol. 2011; 29, 4820-7.
Scorsetti M., Leo F., Trama A., D ’angelillo R., et al. Thymoma and thymic carcinomas. Crit Rev Oncol Hematol. 2016; 99: 332-50. 10.1016/j.critrevonc.2016.01.012
Evoli A., Minicuci G. M., Vitaliani R., Battaglia A., Della M. G., Lauriola L., Fattorossi A. Paraneoplastic diseases associated with thymoma. J Neurol. 2007 Jun; 254 (6): 756-62.
Kondo K., Monden Y. Thymoma and myasthenia gravis: a clinical study of 1,089 patients from Japan. Ann Thorac Surg. 2005; 79 (1): 219-24.
Souadjian, J.V., Enriquez, P., Silverstein, M.N., Pepin, J.M. The spectrum of diseases associated with thymoma. Arch. Intern. Med.1974; 134 (2), 374-379.
Rosenow 3rd, E.C., Hurley, B.T. Disorders of the thymus. Arch. Intern. Med.1984; 144 (4), 763-770.
Regnard, J.-F., Magdeleinat, P., Dromer, C., Dulmet, E., de Montpreville, V., Levi, J.F., et al. Prognostic factors and long-term results after thymoma resection: a series of 307 patients. J. Thorac. Cardiovasc. Surg. 1996; 112 (2), 376-384.
Venuta, F., Rendina, E.A., Pescarmona, E.O., De Giacomo, T., Vegna, M.L., Fazi, P., et al. Multimodality treatment of thymoma: a prospective study. Ann. Thorac. Surg. 1997; 64 (6), 1585-1592.
Napeñas JJ, Brennan MT, Fox PC. Diagnosis and treatment of xerostomia (dry mouth). Odontology. 2009; 97 (2): 76-83. doi: 10.1007/s10266-008-0099-7
Scully C, Felix DH. Oral medicine -- update for the dental practitioner: dry mouth and disorders of salivation. Br Dent J 2005; 199: 423-7.
Saccucci M., Di Carlo G., Bossù M., Giovarruscio F., Salucci A., Polimeni A. Autoimmune Diseases and Their Manifestations on Oral Cavity: Diagnosis and Clinical Management. Journal of Immunology Research. 2018; 2018:6061825. doi:10.1155/2018/6061825
Malladi A. S., Sack K. E., Shiboski S. C., et al. Primary Sjogren's syndrome as a systemic disease: a study of participants enrolled in an international Sjogren's syndrome registry. Arthritis Care Res (Hoboken) 2012; 64: 911-918.
Bascones-Martínez A., García-García V., Meurman J. H., Requena-Caballero L. Immune-mediated diseases: what can be found in the oral cavity? International Journal of Dermatology. 2015; 54 (3): 258-270. doi: 10.1111/ijd.12681
Margaix-Muñoz M., Bagán J. V., Poveda R., Jiménez Y., Sarrión G. Sjögren’s syndrome of the oral cavity. Review and update. Medicina Oral, Patología Oral y Cirugía Bucal. 2009; 14 (7): E325-E330.
Shiboski S. C., Shiboski C. H., Criswell L., et al. American College of Rheumatology classification criteria for Sjögren's syndrome: a data-driven, expert consensus approach in the Sjögren's International Collaborative Clinical Alliance cohort. Arthritis Care Res (Hoboken). 2012; 64: 475-487.
Schmidt E., Zillikens D. Pemphigoid diseases. Lancet. 2013; 381: 320-32.10.1016/S0140-6736 (12) 61140-4.
Xu H. H., Werth V. P., Parisi E., Sollecito T. P. Mucous membrane pemphigoid. Dent Clin North Am. 2013; 57:611-30.
Scully C., Carrozzo M., Gandolfo S., Puiatti P., Monteil R. Update on mucous membrane pemphigoid: A heterogeneous immune-mediated subepithelial blistering entity. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999; 88: 56-68.
Scully C., Muzio Lo L. Oral mucosal diseases: mucous membrane pemphigoid. Br J Oral Maxillofac Surg. 2008; 46: 358-66.10.1016/j.bjoms.2007.07.205
Chan L. S., Ahmed A. R., Anhalt G. J., Bernauer W., Cooper K. D., Elder M. J., et al. The first international consensus on mucous membrane pemphigoid: definition, diagnostic criteria, pathogenic factors, medical treatment, and prognostic indicators. Arch Dermatol. 2002; 138:370-9.10.1001/archderm.138.3.370.
Evoli A., Minisci C., Di Schino C., Marsili F., Punzi C., Batocchi A. P., Tonali P. A., Doglietto G. B., Granone P., Trodella L., Cassano A., Lauriola L. Thymoma in patients with MG: characteristics and long-term outcome. Neurología. 2002 Dec 24; 59 (12): 1844-50.
Pescio J. J. Clinical epidemiological study of xerostomia in elderly totally edentulous. Rev Fac Cien Med Univ Nac Cordoba. 2006; 63 (2 Suppl): 6-12.
Löfgren C. D., Wickström C., Sonesson M., Lagunas P. T., Christersson C. A systematic review of methods to diagnose oral dryness and salivary gland function. BMC Oral Health. 2012; 12:29. doi:10.1186/1472-6831-12-29
Wolff A., Joshi R. K., Ekström J., et al. A Guide to Medications Inducing Salivary Gland Dysfunction, Xerostomia, and Subjective Sialorrhea: A Systematic Review Sponsored by the World Workshop on Oral Medicine VI. Drugs R D. 2016; 17 (1): 1-28.
Shimizu C., Kuriyama T., Williams D. W. Association of oral yeast carriage with specific host factors and altered mouth sensation. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008; 105: 445-451.
Minato H., Kinoshita E., Nakada S., et al. Thymic lymphoid hyperplasia with multilocular thymic cysts diagnosed before the Sjögren syndrome diagnosis. Diagnostic Pathology. 2015; 10:103. doi:10.1186/s13000-015-0332-y
Xin Y., Cai H., Li Y., Cui Y. Thymic hyperplasia associated with primary Sjogren’s syndrome cured by thymectomy. Journal of Thoracic Disease. 2017; 9 (2): E130-E132. doi:10.21037/jtd.2017.02.24
Daniels T. E. Labial salivary gland biopsy in Sjögren's syndrome: assessment as a diagnostic criterion in 362 suspected cases. Arthritis Rheum. 1984; 27: 147-56.
Chisholm D. M., Mason D. K. Labial salivary gland biopsy in Sjögren's disease. J Clin Pathol. 1968; 21 (5): 656-60.
Vitali C., Bombardieri S., Jonsson R., et al. Classification criteria for Sjögren's syndrome: a revised version of the European criteria proposed by the American-European Consensus Group. Ann Rheum Dis. 2002; 61: 554558.
Daniels T. E., Cox D., Shiboski C. H., et al. Associations between salivary gland histopathologic diagnoses and phenotypic features of Sjögren's syndrome among 1,726 registry participants. Arthritis & Rheumatism. 2011; 63: 2021-2030.
Silvestre F. J., Miralles L., Martínez V. Tratamiento de la boca seca: puesta al día. Med Oral. 2004; 9: 273-279.
Guggenheimier J., Moore P. Xerostomia: etiology, recognition and treatment. J Am Dent Assoc. 2003; 134:61-9.
Van der Putten G. J., Brand H. S., Schols JMGA, de Baat C. The diagnostic suitability of a xerostomia questionnaire and the association between xerostomia, hyposalivation and medication use in a group of nursing home residents. Clin Oral Invest. 2011; 15: 185-192.
López-Jornet M. P., García-Teresa G., Viñas M., Vinuesa T. Clinical and antimicrobial evaluation of a mouthwash and toothpaste for xerostomia: A randomized, double-blind, crossover study. Journal of Dentistry. 2011; 39: 757-63. doi: 10.1016/j.jdent.2011.08.007
Cassolato S. F., Turnbull R. S. Xerostomia: clinical aspects and treatment. Gerodontology. 2003; 20: 64-77.
Kamaguchi M., Iwata H., Ujiie I., Ujiie H., Sato J., Kitagawa Y and Shimizu H. Direct Immunofluorescence Using Non-Lesional Buccal Mucosa in Mucous Membrane Pemphigoid. Front. Med. 2018; 5:20. doi: 10.3389/fmed.2018.00020
McCluskey P., Chang J. H., Singh R., Wakefield D. Methotrexate therapy for ocular cicatricial pemphigoid. Ophthalmology. 2004;111 (4): 796-801. doi.org/10.1016/j.ophtha.2003.07.010
Theander E., Vasaitis L., Baecklund E., et al. Lymphoid organisation in labial salivary gland biopsies is a possible predictor for the development of malignant lymphoma in primary Sjogren's syndrome. Annals of the Rheumatic Diseases. 2011; 70:1363-1368.