Nasal Angioleiomyoma: A Unique Case Report and Literature Review

Nasal Angioleiomyoma: A Unique Case Report and Literature Review


Camille Duggal MD(c) 1, Kelti Munroe MD 1, Mark Taylor MD, FRCS(C)*

 

  1. Department of Otolaryngology– Head and Neck Surgery (K.M., M.T.), Dalhousie University, Queen Elizabeth II Health Sciences Centre, Halifax, NS, Schulich School of Medicine and Dentistry (C.D.), Western University, London, Ontario.

 

*Correspondence to: Mark Taylor, MD, FRCS(c), Department of Otolaryngology–Head and Neck Surgery, Dickson Building Queen Elizabeth II Health Sciences Centre, Rm. 3044, 5820 University Ave, Halifax, NS B3H 1V7, Canada.


Copyright.

© 2026 Mark Taylor, This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Received: 22 July 2026

Published: 01 August 2026

DOI: https://doi.org/10.5281/zenodo.21715252

 

Abstract

Background: Angioleiomyoma is a rare benign tumor originating from vascular smooth muscle. This paper aims to describe the clinicopathologic features and management of nasal angioleiomyoma, emphasizing its rarity and diagnostic challenges.

Methods: A 73-year-old male presented to the Otolaryngology-Head and Neck Surgery with chronic right nasal obstruction and facial pressure. Physical examination revealed a cystic mass in the nasal vestibule, confirmed by flexible nasopharyngoscopy as originating from the right anterior lateral wall. CT scan showed interval growth of the mass to 2.0 x 2.3 x 1.8 cm.

Results: A right lateral rhinotomy was performed, and the mass was excised with no postoperative complications. Histological examination confirmed angioleiomyoma, characterized by spindle cells with strong desmin positivity.

Conclusion: A review of the literature in this article outlines the rarity of angioleiomyomas in the sinonasal tract.  This case underscores the importance of accurate diagnosis and complete surgical excision as treatment of choice.

Key words

Head and neck, angioleiomyoma, vascular leiomyoma, Sinonasal angiomyoma, sinonasal tumor.

Nasal Angioleiomyoma: A Unique Case Report and Literature Review

Introduction

Angioleiomyoma is a benign tumour arising from vascular smooth muscle, which is rarely found in the nose. Although angioleiomyomas were initially considered a leiomyoma subtype, they have recently been classified as a separate entity by the World Health Organization.[1] Angioleiomyomas are very rare in the head and neck region, especially in the sinonasal cavity, constituting < 1% of all angioleiomyomas.[2,3] The first case of an angioleiomyoma in the nasal cavity was reported in 1966 with only five cases reported in the next 12 consecutive years.[4] A recent systematic review reported that only 87 prior cases of sinonasal-tract angioleiomyomas had been described.[5]

Given the paucity of published cases regarding angioleiomyoma’s in the nasal cavity, an updated review of patient etiology, diagnostic indicators, localization of tumor site and excisional technique is essential. Our review aims to increase awareness of this rare tumor, highlighting the clinicopathologic characteristics of nasal angioleiomyoma.

 

Case Presentation

Clinical Course

Informed consent for this case report was provided by the patient.  A 73-year-old male with a 20-pack year smoking history presented to the Otolaryngology-Head and Neck Surgery clinic in 2019 with the chief complaint of longstanding right nasal obstruction. The patient reported progressive pressure and pain on the right side of his face. No epistaxis, nasal drainage or constitutional symptoms were noted.

Physical examination with anterior rhinoscopy revealed a cystic mass with a bluish hue. Flexible nasopharyngoscopy confirmed that the cystic mass was confined to the nasal vestibule and originating deep to the right anterior lateral wall. The mass measured 1.4 x 1.2 cm at the base of the ala, abutting the piriform aperture. It did not appear to be attached to the inferior turbinate and ended superiorly at the upper lateral cartilage. The mass did not increase in size with a valsalva. Examination of the oropharynx, hypopharynx, larynx and left nasal cavity was unremarkable.

Imaging (CT sinuses) redemonstrated the 1.2 x 1.4 cm soft tissue mass, with benign imaging characteristics.  A FNA was performed and demonstrated atypical cells, with a clinicopathological differential that included venous malformation, Klestadt (nasolabial) cyst or a minor salivary gland tumor.
The patient was offered surgery for definitive management but was delayed due to the Covid pandemic and the need for anticoagulation for a transient ischemic attack (TIA).  Two years following the initial assessment, a repeat CT scan demonstrated an interval increase in size (2.0 x 2.3 x 1.8 cm) of the mass arising from the posterolateral aspect of the right nostril with no osseous involvement (see Figure 1). There was no bony indentation, erosion, expansion into the gingiva on the oral side or sinogenic obstruction.

The patient underwent a right lateral rhinotomy with excision of the masomaxillary cystic mass.  (Figure 2). The nasal cartilage and mucosa overlying the mass was carefully preserved. Blood loss was minimal with no complications. The patient’s postoperative recovery period was uncomplicated. No septal perforation, bleeding or scarring was noted.  The patient has remained disease free and well at 2-year follow-up.

 

Pathology:
The mass was circumferentially dissected and sent as a gross total resection to pathology. The specimen consisted of a pink-red, rubbery tissue (Figure 3).

Histological examination demonstrated a circumscribed, spindle cell nodule with intersecting fascicles containing bland ovoid nuclei with paranuclear vacuoles, surrounding flattened slit like vascular spaces. No atypia, mitotic activity or necrosis was identified. Immunohistochemical stain shows the proliferation to be strongly positive for antibodies to desmin, and negative for S100 and EMA. The morphologic and immunohistochemical features supported the diagnosis of angioleiomyoma.

 

Discussion

Leiomyomas are benign smooth muscle, soft tissue tumors most commonly in the uterus (95%), skin (3%), and gastrointestinal tract (1.5%). Interestingly, MED12 mutations have been frequently observed with uterine leiomyomas, but not demonstrated in extrauterine masses.[6] Angioleiomyomas are vascular leiomyomas, with unique histological characteristics and clinical presentation that influence surgical treatment.  Our case is the unique because of the 2-year interval gap between first presentation and subsequent surgery, highlighting the natural history.  Although during this lengthy time interval, there was a significant increase in tumor size, the tumor, however, had a benign histology.

Angioleiomyoma’s are described as circumscribed proliferation of spindle cells around a central endothelium-lined vascular channel.[7] Angioleiomyomas can be stratified into three histologic types. The most common being a solid type composed of mostly muscle with collapsed vascular lumina, generally found in the limbs.[7] The cavernous type involves a large dilated vascular lumina, while the venous type involves thick muscle-walled veins.[7] The etiology of angioleiomyomas remain unclear. However, literature suggests three hypotheses to explain the origin of the sinonasal leiomyoma. One theory proposes that the tumor arises directly from the smooth muscle in the nasal cavity. A second theory suggests it originates from the smooth muscle of a blood vessel, while the other posits that it is derived from aberrant undifferentiated mesenchyme.[3,8]

Due to the rarity of sinonasal angioleiomyomas, a review of the literature was completed including case reports and retrospective reviews. No time setting was employed to capture as many studies as possible. This literature review conducted on Pubmed with the following key words: Angioleiomyoma, Vascular leiomyoma, Angiomyoma and sinonasal, nasal cavity, sinonasal, nose, nasal mucosa, yielded 87 patients (see Table 1). Two articles were excluded as no full text was found. There were 6 retrospective reviews with the remaining entries being case reports. In total 87 patients were identified with 43% being male. The youngest patient identified in this review was 12, while the oldest was 82. Nasal obstruction, facial pain and epistaxis were the most common symptoms experienced. Surgical excision was the treatment in all cases except on two occasions.

 

Treatment for these tumors is local resection, and there are no reports of recurrence after total excision.[2] There is a single reported case of tumor recurrence after incomplete resection.[9] This highlights the importance of a complete surgical excision, if possible, to prevent local recurrence.[9] The choice of the surgical approach, whether by nasal endoscopic surgery or by lateral rhinotomy, is dependent on tumor location and extension, as well as the concern for improved hemostasis.  The literature suggests that an open surgical approach is the preferred technique for complete excision of mass with negative margins and is associated with a lower recurrence rate in comparison to endoscopic resection.[10] Given its minimally invasive nature, endoscopic resection has been associated with decreased hospital stay and surgical complications.[10] Our preference for an open surgical approach in this case was via a lateral rhinotomy given the size of the mass and the lateral extension demonstrated on preoperative imaging.

 

Conclusion

This case underscores the rarity of sinonasal angioleiomyomas and the importance of considering them in the differential diagnosis for a nasal mass. The natural history of angioleiomyomas is progressive enlargement.  Complete surgical excision as treatment of choice to prevent local recurrence.

 

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