Rationale: Epidermolysis bullosa (EB) is an inherited disease characterized by fragile skin with painful blistering, which requires lifelong skin and wound care. This case report describes the use of inhaled nitrous oxide (N 2 O) for procedural pain control at home during wound care in a young man with severe dystrophic EB. To our knowledge, only 1 case was reported by Ingelmo et al in 2017 regarding the use of N 2 O at home in a 4-year-old-child. To date, no such attempt has been made in adult patients. Patient concerns: Our patient was a 28-year-old man. Frequent blisters appear spontaneously, and are often preceded by erythema and itching. Patient required daily treatment daily consisting of lancing blisters with a needle and emptying them by compression. Diagnoses: Severe recessive dystrophic EB diagnosed at the time of delivery. Interventions: Procedural pain control was managed by the auto-administration of an inhaled N 2 O and air gas mixture. Outcomes: Conscious sedation with N 2 O leads to beneficial effects, such as reduction in dressing duration, acute procedural pain, local antibiotic needing, medication memory, anxiety, anticipatory pain, and fatigue after the dressing session. Lessons: N 2 O analgesia is safe and effective, resulting in a significant reduction in procedural pain and an improvement in the quality of life of patients and their caregivers.