We report a case of amyloidosis localized to the medium-sized arteries of the face and presenting as prominent perioral swelling. The condition was identified as primary AL-type amyloidosis on the basis of immunohistochemical reactivity, predominantly for anti-lambda light chain antibody within amyloid deposits. Subsequent radiographic and laboratory evaluation of the patient disclosed multiple myeloma. Although amyloid deposits were subsequently detected in the temporal arteries, evidence of widespread amyloidosis has not been observed after 1 year of follow-up. To the best of our knowledge, this is the first report of myeloma-associated amyloidosis mainly localized to the medium-sized arteries of the face.