Introduction: Uterine arteriovenous malformations (AVMs) are one of the causes of potential severe genital hemorrhages. Acquired AVMs are considered iatrogenic and mainly diagnosed after pregnancy termination and/ or uterine surgery. The diagnosis is based on ultrasound, computed tomography, magnetic resonance imaging and angiography. Uterine artery embolization and hysterectomy represent the current treatments of choice. Uterine curettage is not recommended for AVMs treatment due to high risk of hemorrhage. Although hysteroscopy is the reference method for studying intra-uterine pathologies, few reports described hysteroscopy features of AVMs. We report on a patient with an AVM occasionally diagnosed and managed by hysteroscopy, a treatment never reported in literature. Case Report: A 52-year-old patient complaining of abnormal uterine bleeding, submitted to a cesarean section 29 years before, was scheduled to hysteroscopy resection of an Giancarlo Garuti1, Attilio Di Spiezio Sardo3, Maurizio Mirra2, Stefania Calabrese3, Cristina Gonfiantini2, Marco Di Mario1 Affiliations: 1Obstetric and Gynecology Department, Public Hospital of Lodi, via Savoia 1, 26900-Lodi, Italy; 2Pathology Department, Public Hospital of Lodi, via Savoia 1, 26900Lodi, Italy; 3Department of Gynecology and Obstetrics and Pathology of Human Reproduction, University of Naples “Federico II°”, via Pansini 5, Naples, Italy. Corresponding Author: Giancarlo Garuti (MD), Obstetrics and Gynecology Department, Lodi Hospital, via Savoia n° 1, 26900-Lodi, Italy. The corresponding Author is the Guarantor of Submission; Ph: 39-371-372349, 39-338-2702675; Email: giancarlo.garuti@tiscali.it Received: 13 June 2015 Accepted: 06 Augsut 2015 Published: 12 December 2015 intrauterine lesion suggestive of submucosal myoma at saline infusion ultrasonography. Rather than a myoma, hysteroscopy imaging was consistent with an endometrial polyp with no abnormal vascularization. The slicing of the mass was hampered by bleeding from a crowding of arterial and venous vessels; near the pedicle, a worsening of bleeding precluded an adequate visualization causing the premature interruption of the surgery. Bleeding control was obtained by the placement of a balloon. The pathologic examination confirmed an AVM. One month later, ultrasound, computed tomography and hysteroscopy showed no residual mass. Twelve months after intervention no vaginal bleeding was recorded and normal findings were found at physical and ultrasound examination. Conclusion: AVMs can be misdiagnosed as a submucous myoma or an endometrial polyp at ultrasounds and hysteroscopy, respectively. Although theoretically effective, hysteroscopy resection of AVMs can lead to hemorrhagic complications.