Cricoarytenoid joint arthritis is most frequently reported in Rheumatoid Arthritis and in other systemic diseases such as Sjogren's syndrome, Systemic Lupus Erythematosus, Ankylosing Arthritis, Juvenile Chronic Arthritis, and autoimmune hepatitis but it has not been reported in dermatomyositis. In this paper, we report the case of a 43 years-old woman treated for dermatomyositis who presented with hoarseness and severe odynophagia. The laryngoscopy revealed the presence of an extensive white swelling of the left cricoarytenoid joint with reduced mobility of the left vocal cord, consistent with left cricoarytenoid joint arthritis, which has not previously been described in dermatomyositis to our knowledge. Treatment with high doses of prednisone produced a complete resolution of the laryngeal symptoms.