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Patients with unresectable extracranial head and neck arteriovenous malformations (HNAVMs) appeared to benefit from taking sirolimus and embolization in combination, authors of a small case series said.

Favorable outcomes were seen in six patients receiving endovascular embolization and up to 3 years of sirolimus treatment, reported Joyce Teng, MD, PhD, of Stanford University in California, and colleagues in .

However, they noted, "the optimal perioperative dosing and duration of sirolimus treatment still need to be investigated."

The six patients (mean age 24.5) had extracranial head and neck arteriovenous malformations.

Patients began adjuvant sirolimus therapy, at trough level ranging from 10 to 15 ng/mL over the course of treatment, at least one month prior to their first embolic procedure. The average adjuvant sirolimus therapy duration was 2.67 months. All had at least one endovascular embolization and three patients had three embolizations each over the course of 1 to 2 months.

Outcomes were as follows:

• Patient one suffered from the following adverse effects: localization ulceration requiring debridement, skin infection treated with mupirocin, and oral erosion. The outcomes for patient one were: near-complete resolution of bleeding events and a decline in volume by 75%, and improvement in pain scale score, the study authors noted.

• Patient two had an oral erosion which eventually led to 90% volume decrease of arteriovenous malformations (AVMs), resolution of vascular prominence and headaches each day.

• Patient three also suffered from oral erosion and eventually a 67% volume decline of AVM, total resolution of reoccurring headaches and of facial pain and pressure.

• Patient four had pulmonary embolism resulting from cyanoacrylate glue and oral erosion. The outcomes were as follows: substantial volume reduction of AVM, "improvement in pain scale, interval growth on follow-up MRI 2 years after treatment, reinitiation of sirolimus therapy stabilized growth for over 15 months."
• Patient five had oral erosion and later total resolution of AVM and bleeding.

• Patient six had the adverse effect of pruritus and an outcome of "80% volume reduction of AVM and improvement in the pain scale score," the investigators wrote.

"Clinical follow-up demonstrated significant improvement in all patients; follow-up magnetic resonance imaging confirmed significant or complete resolution of malformations in all patients as well," the investigators wrote.

"Our results therefore suggest that sirolimus can be used as a safe and effective adjuvant therapy with endovascular embolization in the management of complicated, unresectable HNAVMs." However, Teng and colleagues said "well controlled, larger-scale multicenter studies" are needed to confirm this method.