Madelung disease, also known as benign symmetrical lipomatosis, is a rare condition characterized by symmetrical diffuse adipose tissue in the neck, shoulders, and arms. The present report described the case of a 51-year-old man diagnosed with Madelung disease who presented with masses primarily in the neck. He had previously shown partial improvement after injection lipolysis and shoulder surgery. However, 4 years later, following corticosteroid administration for the treatment of acute deafness, cervical lipomatosis progressed to the extent that he was unable to fasten his shirt. The initial treatment plan involved bilateral surgical excision of the lipomatous masses of the neck and liposuction for those in the submental area. However, the lipomas were adherent to the surrounding tissue and were partially fibrosed, presumably due to the previous injection lipolysis; thus, liposuction was not possible, and all the masses around the neck were carefully excised using cervical lymph node dissection technique. Thirty-two months later, the patient showed good cosmetic results, with no recurrence of cervical lipomatosis. Radical resection of the lipomas using a cervical dissection technique is useful in the treatment of Madelung disease; however, clinicians must consider the potential for adhesions and plan a meticulous dissection in those who have undergone injection lipolysis before the surgery.