INTRODUCTION: Kartagener's syndrome (KS) is a rare disease characterized by a triad of situs inversus totalis, chronic sinusitis, and bronchiectasis. The disorder is caused by a hereditary genetic abnormality that impairs ciliary movement. Although aberrant pass course of the inferior laryngeal nerves due to visceral inversion should be considered during thyroid surgery in patients with KS, no report of surgical treatment for Graves' disease (GD) in patients with KS has been found to date. CASE PRESENTATION: A Japanese male in his 40s was referred to our hospital for surgical treatment for drug-refractory GD. He was diagnosed to have KS by genetic alteration of the CONCLUSION: We reported the first case of surgical treatment for GD in a patient with KS and discussed the preoperative diagnosis of NRLN.