Bleomycin Electrosclerotherapy (BEST) for Slow-Flow Malformations of the Upper Aerodigestive Tract

Abstract

Background/Objectives: Bleomycin electrosclerotherapy (BEST), which combines intralesional bleomycin administration with electroporation, enhances drug uptake and has shown efficacy in treating vascular malformations resistant to conventional therapies. While BEST is increasingly used in various anatomical sites, its application in the upper aerodigestive tract remains underexplored. This study evaluates the safety and effectiveness of BEST in managing slow-flow vascular malformations of the oral cavity, tongue, larynx, and hypopharynx. Methods: In this retrospective, multicenter study, 20 patients with symptomatic slow-flow vascular malformations of the upper aerodigestive tract were treated with BEST. Clinical and radiological assessments were used to evaluate the treatment response, categorized as "significantly reduced", "reduced", "stable disease", or "lesion growth". Postprocedural complications and functional outcomes were systematically recorded. Results: A total of 29 BEST sessions were performed. Lesions of the tongue (n = 8) and combined oral cavity and tongue (n = 6) showed the highest response rates, with significant symptom reduction in five out of eight and five out of six patients, respectively. Among isolated oral cavity lesions (n = 4), one out of four demonstrated a significant reduction. In contrast, laryngeal and hypopharyngeal lesions (n = 2) had limited response, with one case showing partial reduction and the other remaining stable. Severe complications, including bleeding and dyspnea requiring tracheostomy, limited further treatment in these locations. No systemic adverse events, such as pulmonary toxicity, were observed. Conclusions: BEST is effective for treating vascular malformations of the upper aerodigestive tract, particularly in the tongue and oral cavity, but presents significant risks in laryngeal and hypopharyngeal lesions. A multidisciplinary approach is required to optimize treatment protocols for these challenging locations.

Keywords: BEST; bleomycin; electrosclerotherapy; lymphatic malformation; mucosal malformation; sclerotherapy; slow-flow malformation; vascular anomalies; vascular malformations; venous malformation.

Figure 1

A 49-year-old female patient with an extensive venous malformation (VM, indicated by white arrows) of the oral cavity and tongue undergoing 3 sequential bleomycin electrosclerotherapy (BEST) sessions with intralesional drug administration. (A) T2-weighted (T2w) axial and coronal magnetic resonance (MR) images obtained prior to BEST treatment, with indications including swelling, pain, dysphagia, and impaired speech. (B) T2w axial and coronal MR images acquired 3 months following the second BEST session, demonstrating partial regression of the malformation. (C) T2w axial and coronal MR images 6 months after the third BEST session, showing near-complete resolution of the malformation. Following the third session, no further swelling or pain was reported, and lesion regression led to the restoration of unimpaired food intake and speech.

Figure 2

Patients with lesions in the oral cavity and tongue demonstrated the highest rates of lesion reduction following bleomycin electrosclerotherapy (BEST). (A) A 41-year-old male patient with a venous malformation (VM) of the right palatal arch presented with swelling, dysphagia, and dyspnea. Three months post-treatment, the lesion was completely resolved, and the patient was symptom-free. (B) A 10-year-old male patient with a lymphatic malformation (LM) of the tongue presented with swelling, pain, and dysphagia. Seven months post-treatment, the lesion was completely resolved, and the patient was symptom-free. (C) A 6-year-old female patient with a VM of the lower lip mucosa presented with swelling, pain, and dysphagia. One year post-treatment, the lesion was significantly reduced, and the patient was symptom-free. (D) A 26-year-old female patient with a VM involving the floor of the mouth, tongue, mucosa of the right cheek, and lower lip presented with swelling and pain. Three months post-treatment, the lesion was significantly reduced, and the patient was symptom-free.

Figure 3

A 64-year-old female patient with an extensive venous malformation (VM, indicated by arrows) of the laryngeal and hypopharyngeal mucosa with severe bleeding, swelling, and dyspnea following bleomycin electrosclerotherapy (BEST). (A) T2-weighted (T2w) coronal magnetic resonance (MR) image and sagittal photographic documentation of the lesion prior to treatment, showing the extent of the VM. The patient reported symptoms of dysphagia and dyspnea. (B) T2w coronal MR image and sagittal photographic documentation taken 2 months after the first BEST session demonstrated regression of the lesion. However, postinterventional swelling and bleeding necessitated a tracheostomy, precluding a second BEST procedure.