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Endobronchial ultrasound-guided transbronchial needle aspiration (EBUS-TBNA) is highly accurate in diagnosing mediastinal lymphadenopathies of lung cancer and benign disorders, with the advantage that it is a minimally invasive technique, unlike open surgery and mediastinoscopy. However, the diagnostic accuracy of EBUS-TBNA for the diagnosis of lymphoma in patients with mediastinal lymphadenopathy is not well defined. The lack of tissue architecture obtained by cytological needles decreases the diagnostic accuracy for diagnosis and subtyping of de novo and relapsed mediastinal lymphomas. We present the first described case in the literature of an anaplastic large cell lymphoma relapsed, diagnosed on tissue fragments obtained by EBUS-TBNA with the particularity of using a histological needle.