Anorexia nervosa (AN) is a highly disabling mental disorder with high rates of morbidity and mortality. Few psychological treatments and pharmacotherapy are proven to be effective for adult AN. Two invasive stereotactic neurosurgical interventions, deep brain stimulation (DBS) and anterior capsulotomy, are now commonly used as investigational approaches for the treatment of AN. Here, we report the long-term safety and efficacy of rescue bilateral anterior capsulotomy after the failure of bilateral nucleus accumbens (NAcc)-DBS in an 18-year-old female patient with life-threatening and treatment-resistant restricting subtype AN. Improvements in the neuropsychiatric assessment were not documented 6 months after the NAcc-DBS. Rescue bilateral anterior capsulotomy was proposed and performed, resulting in a long-lasting restoration of body weight and a significant and sustained remission in AN core symptoms. The DBS pulse generator was exhausted 2 years after capsulotomy and removed 3 years postoperatively. No relapse was reported at the last follow-up (7 years after the first intervention). From this case, we suggest that capsulotomy could be a rescue treatment for patients with treatment-resistant AN after NAcc-DBS failure. Further well-controlled studies are warranted to validate our findings.