Eulry F, Poirier J M, Perard D, Bergamasco P, Lechevalier D, Magnin J
Rheumatology Department, Bégin Army Instruction Hospital, Saint-Mandé, France.
Rev Rhum Engl Ed. 1997 Jul-Sep;64(7-9):495-9.
A patient with an osteolytic L2-L3 pagetic block and pagetic lesions of L1 and the sacrum seen only as increased radionuclide activity became resistant to etidronate after the fifth course (5 mg/kg/d six months per year) and developed severe cauda equina syndrome (reduction in walking distance to 30 m and sphincter dysfunction) due primarily to vertebral hypertrophy. Five months after a ten-day course of intravenous pamidronate (22.5 mg/d), the clinical symptoms were unchanged, although the alkaline phosphatase level was down 50%. Oral clodronate (1,600 mg/day for six months per year) in combination with calcium and vitamin D supplementation dramatically improved the walking distance and sphincter disorders. Resolution of the neurological manifestations was complete after the second clodronate course. At last follow-up nine months after the fourth clodronate course, there was no evidence of a relapse and the alkaline phosphatase level was normal. The time course of events in this patient does not allow to affirm that pamidronate was ineffective and suggests that calcium and vitamin D supplementation improved mineralization of the pagetic block and enhanced the effect of bisphosphonate therapy.
一名患有溶骨性L2-L3骨炎样骨块以及L1和骶骨骨炎样病变(仅表现为放射性核素活性增加)的患者,在接受第五个疗程(每年6个月,5毫克/千克/天)依替膦酸钠治疗后产生耐药性,并因椎体肥大主要发展为严重的马尾综合征(步行距离缩短至30米且出现括约肌功能障碍)。在接受为期10天的静脉注射帕米膦酸钠(22.5毫克/天)疗程五个月后,尽管碱性磷酸酶水平下降了50%,但临床症状未改变。口服氯屈膦酸(每年6个月,1600毫克/天)并补充钙和维生素D显著改善了步行距离和括约肌功能障碍。在第二个氯屈膦酸疗程后,神经症状完全消失。在第四个氯屈膦酸疗程九个月后的最后一次随访中,没有复发迹象,碱性磷酸酶水平正常。该患者的病程发展情况无法确定帕米膦酸钠无效,提示补充钙和维生素D改善了骨炎样骨块的矿化并增强了双膦酸盐治疗的效果。