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Extranodal marginal zone lymphomas of mucosa-associated lymphoid tissue (MALT) are low-grade B-cell neoplasms, which arise in mucosal sites with prolonged lymphoid proliferation. Primary tracheal MALT lymphoma is an exceedingly rare entity for which the optimal treatment approach has not been determined. Here, we report a case of MALT lymphoma involving the trachea in a 64-year-old smoking woman who received desobstructive endoscopy and was thereafter successfully treated with (anti-CD20) immunotherapy and radiotherapy.