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Introduction Respiratory Epithelial Adenomatoid Hamartoma (REAH) is a benign disease that can resemble other malignant entities. Thus, it is essential to diagnose it accurately as the treatment approach differs, from radical surgeries in malignant cases, to a simple excision in hamartoma. We present an unusual case of bilateral REAH that was misdiagnosed, and hence it was treated aggressively. Case report A 57-year-old male patient presented with anosmia, 2-years history of bilateral nasal obstruction, and was accompanied with a moderate headache. An impression of olfactory neuroblastoma was made after history taking physical examination, and imaging studies. The patient underwent Functional Endoscopic Sinus Surgery (FESS), excisional biopsy of the cribriform plate mass bilaterally, and superior septectomy. Histopathologic examination of the bilateral masses showed sinonasal polyposis with crypting of surface mucosa and pseudoglandular formation. A diagnosis of sinonasal polyps with REAH was established. The patient's nasal obstruction improved, with no recurrence of sinusitis ± polyposis. However, he still complains of anosmia after 2-years follow-up. Conclusion Although REAH is a benign disease, it is critical to reach the correct diagnosis, in order to avoid aggressive treatment. Unfortunately, the preoperative investigations were not consistent with REAH, thus it was misdiagnosed and treated aggressively., Highlights • Respiratory Epitheliel Adenomatoid Hamartoma (REAH) is a benign disease that can resemble other malignant diseases. • The disease is commoner in males and in the third to ninth decades of life. • The significance of this article is to share our experience with this disease as we did not diagnose the case accurately. Thus the patient was treated aggressively for a malignant case, instead of a simple excision. Hence, one should keep REAH in mind when encountered a similar case to ours.