Linear scleroderma “en coup de sabre” is characterized by atrophy and furrowing of the skin of the front parietal region above the level of the eyebrow. In most cases, it occurs as a single paramedian line that may be associated with hypoplasia of underlying structures and hemiatrophy of the face. The affected region is a depression that may be associated with hypoplasia of the underlying soft tissues and bone that results in facial hemiatrophy. If the lesion is narrow, it can be resected and directly sutured; in the case of a wide lesion, many different reconstructive techniques, directed at augmentation of deficient soft tissue volume, have been proposed such as autologous tissue grafts, biomaterials, pedicled flaps, and free flaps. Adipose-derived regenerative cells (ADRCs) can be easily processed from lipoaspirated fat and can provide a significant quantity of multipotent cells for a variety of therapeutic regenerative medicine therapies. There is an increasing interest in a possible therapeutic role of ADRCs from processed lipoaspirate for many applications, including their use as soft-tissue fillers. We introduce the application of a successful ADRC therapy for a linear scleroderma en coup de sabre deformity.