Two Cases of Monostotic Paget's Disease: Effects of Bisphosphonate

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  • HOSHIRO Madoka
    Department of Endocrinology, Metabolism and Diabetes Mellitus, Kinki University School of Medicine
  • HARADA Takeshi
    Department of Endocrinology, Metabolism and Diabetes Mellitus, Kinki University School of Medicine
  • IWAI Hiroshi
    Department of Endocrinology, Metabolism and Diabetes Mellitus, Kinki University School of Medicine
  • MIYATAKE Toshiyuki
    Department of Endocrinology, Metabolism and Diabetes Mellitus, Kinki University School of Medicine
  • NISHIMURA Akiyoshi
    Department of Endocrinology, Metabolism and Diabetes Mellitus, Kinki University School of Medicine
  • OHNO Yasuhiro
    Department of Endocrinology, Metabolism and Diabetes Mellitus, Kinki University School of Medicine
  • AOKI Norihiko
    Department of Endocrinology, Metabolism and Diabetes Mellitus, Kinki University School of Medicine

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We report two cases of monostotic Paget's disease which were effectively treated with bisphosphonate. Case 1 was a 60-year-old female. Medical examination revealed high alkaline phosphatase (ALP) levels making her visit our clinic. Hematological examination showed high levels of ALP isozyme 3 and bone metabolism markers, and bone scintigraphy demonstrated strong accumulation of 99mTc on the skull. With the diagnosis of monostotic Paget's disease of the skull, treatment with bisphosphonate (etidronate) was started. The response to etidronate was good and after 12 weeks of treatment, the ALP levels decreased to about 26% of the levels before treatment, without the appearance of any symptoms or lesion development. One year and three months later, ALP increased again, and etidronate administration was resumed. However, four years after the diagnosis of the disease, etidronate became ineffective and oral administration of alendronate, a stronger bisphosphonate, was started at 5 mg/day. The patient responded favorably to the bisphosphonate and is still under observation. Case 2 was a 71-year-old female. High ALP levels were found during the follow-up of type 2 diabetes, and the case was diagnosed as monostotic Paget's disease of the pelvis based on bone metabolism markers and bone scintigraphy. Etidronate treatment at 200 mg/day resulted in the improvement of bone metabolism markers and bone scintigraphy findings. When she died of colon cancer twelve months later, with no marked progress of the Paget's disease of bone observed clinically.<br>

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  • Endocrine Journal

    Endocrine Journal 50 (4), 385-392, 2003

    一般社団法人 日本内分泌学会

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