Anderson D C, O'Driscoll J B, Buckler H M, Cantrill J, Brown J D
University of Manchester Department of Medicine, Hope Hospital, Salford.
Br J Radiol. 1988 Nov;61(731):996-1001. doi: 10.1259/0007-1285-61-731-996.
Two patients with osteoporosis circumscripta of the skull are presented who have each been treated with a 3-month course of intravenous infusions of (3-amino-1-hydroxypropylidene)-1,1-bisphosphonate (APD) (total dose of 185 and 375 mg), which in both cases was associated with rapid and dramatic remineralization of the porotic bone and marked clinical and biochemical remission. However, osteolytic disease reappeared (after 9 and 18 months, respectively) in both cases as an osteolytic ring showing high uptake of radiolabelled bisphosphonate on bone scan, in formerly unaffected bone just distal to the original leading edge of osteolysis. Further treatment was associated with healing of these new lesions. These cases suggest that the diseased osteoclasts entering previously normal bone are protected against the drug probably because normal bone matrix takes up relatively little APD, and that APD taken up by diseased bone behind this front confers long-term resistance to further resorption. In assessing treatment strategies with bisphosphonates for Paget's disease, the response at the leading edge of osteoporosis circumscripta may be particularly informative.
本文报告了两名颅骨局限性骨质疏松症患者，他们均接受了为期3个月的静脉输注（3-氨基-1-羟基亚丙基）-1,1-二膦酸盐（APD）治疗（总剂量分别为185毫克和375毫克），在这两例患者中，治疗均与多孔性骨的快速显著再矿化以及明显的临床和生化缓解相关。然而，两例患者均在治疗后（分别为9个月和18个月）出现溶骨性疾病复发，表现为溶骨环，在骨扫描中显示放射性标记的双膦酸盐在先前未受影响的骨中摄取增加，位于原溶骨前缘远端。进一步治疗使这些新病变愈合。这些病例表明，进入先前正常骨的病变破骨细胞可能受到药物保护，这可能是因为正常骨基质摄取的APD相对较少，而在这一前沿之后病变骨摄取的APD赋予了对进一步吸收的长期抵抗力。在评估双膦酸盐治疗佩吉特病的策略时，局限性骨质疏松症前缘的反应可能特别具有参考价值。
