Rationale:The traditional Japanese Kampo medicine Yokukansan (TSUMURA Yokukansan extract granules) was originally used to treat neurosis, insomnia, night crying, and irritability and/or agitation in infants and recently it has also been used for neuropsychiatric symptoms in Alzheimer's disease or other dementia in Japan. Furthermore, several recent studies have reported the efficacy of Kampo medicines for various types of headache. Here, we report a case of severe chronic migraine refractory to prophylactic therapy using various western medicines and Japanese Kampo medicines that had resulted in a leave of absence from work, but for which the frequency and severity were markedly decreased by Yokukansan (2.5 g 3 times/d), enabling the patient to return to work fully.Patient concerns:The patient was a 39-year-old woman with a diagnosis of migraine without aura, which started around the age of 17 years and had been well managed with oral triptan preparations. However, due to lifestyle changes after childbirth, the frequency and severity of migraine increased at 38 years of age, prompting her to visit our hospital.Diagnoses:Our initial examination found no neurological abnormality, and our diagnosis was also migraine without aura based on the International Classification of Headache Disorders version 3.Interventions:Her migraine had become refractory to several western medicines (lomerizine hydrochloride, propranolol, sodium valproate, amitriptyline, and duloxetine) and 2 Japanese Kampo medicines (Goshuyuto and Chotosan). The migraine episodes worsened, and consequently she took a leave of absence from work.Outcomes:Yokukansan was then tried, and this markedly improved the chronic migraine, enabling her full return to work.Lessons:Yokukansan might have exerted a prophylactic effect on chronic migraine via its action on the glutamatergic and serotonergic systems, inhibitory action on orexin A secretion, and anti-inflammatory action. Yokukansan might be useful as a prophylactic for migraine worldwide, and a future large-scale clinical study is warranted.