Salter Wm Zachary, Nagy Kolos K, Smith Drew H, Dauod Arif, Nguyen Tam Q
School of Medicine, Texas Tech University Health Sciences Center, Lubbock, Texas, USA.
Department of Otolaryngology-Head and Neck Surgery, Texas Tech University Health Sciences Center, Lubbock, Texas, USA.
Case Rep Otolaryngol. 2025 Jun 15;2025:1538233. doi: 10.1155/crot/1538233. eCollection 2025.
Angioleiomyoma (ALM) is a subtype of leiomyoma characterized by vascular involvement alongside the proliferation of smooth muscle cells. These tumors are generally found in the lower limb and rarely occur in the head and neck region. Herein, we present a rare case of ALM of the external auditory canal (EAC) in a 12-year-old female. To our knowledge, this is the sixth case and the second youngest patient reported with ALM occurring in the EAC. Initially, this patient was diagnosed with otitis externa with polypoid change and prescribed a course of Ciprodex. Follow-up CT demonstrated a soft tissue density in the right EAC, consistent with medial canal fibrosis, and an exam under anesthesia with local excision of the mass was scheduled. Surgical findings showed a cartilaginous, firm mass originating from the fissure of Santorini that was carefully excised completely along with an EAC cholesteatoma seen lateral to the tympanic membrane. The foramen of Huschke was uninvolved and the tympanic membrane was intact without perforation. Final pathology confirmed benign ALM. The EAC was packed with floxin-soaked gelfoam and left packed for 2 weeks with instructions for daily floxin drop placement. Once the gelfoam was removed in the clinic, the EAC was found to be healing well and the patient noted improved subjective hearing. There has not been any subsequent recurrence over a period of 5 months. This case documents a rare presentation of an ALM in an extremely rare anatomical position which was managed successfully.
血管平滑肌瘤(ALM)是平滑肌瘤的一种亚型,其特征是伴有平滑肌细胞增殖的血管受累。这些肿瘤通常发生在下肢,很少出现在头颈部区域。在此,我们报告一例12岁女性外耳道(EAC)血管平滑肌瘤的罕见病例。据我们所知,这是第六例报告的发生在EAC的血管平滑肌瘤病例,也是第二年轻的患者。最初,该患者被诊断为伴有息肉样改变的外耳道炎,并接受了一个疗程的环丙沙星治疗。后续CT显示右侧EAC有软组织密度影,与内侧管纤维化一致,遂安排在麻醉下进行肿物局部切除检查。手术发现一个起源于Santorini裂的软骨样坚实肿物,连同鼓膜外侧可见的EAC胆脂瘤一起被仔细完整切除。Huschke孔未受累,鼓膜完整无穿孔。最终病理证实为良性血管平滑肌瘤。EAC用浸有氟氧沙星的明胶海绵填充,并留置2周,同时告知患者每日滴入氟氧沙星。在诊所取出明胶海绵后,发现EAC愈合良好,患者主观听力有所改善。在5个月的时间里没有出现任何后续复发。本病例记录了血管平滑肌瘤在极其罕见的解剖位置的罕见表现,并成功得到处理。