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Male patients with hypogonadism have an increased risk of developing rheumatic diseases. Most causes of hypogonadism related with rheumatic disease are karyotype abnormality such as Klinefelter's syndrome or Turner's syndrome and gonadal dysgenesis. A 24-year-old year male was admitted for pain of both hip joints that had worsened over 2 months. He had hip joint involvement from ankylosing spondylitis and did not show secondary sex characteristics. His sex hormones and gonadotropins levels indicated hypergonadotropic hypogonadism. The karyotype was 46 XY, and there was no obvious cause of hypogonadism. Here we report on clinical features of this first Korean case of primary hypogonadism accompanying ankylosing spondylitis.