Morlock G, Sanvy J, Serre H
Rev Rhum Mal Osteoartic. 1975 Nov;42(11):669-79.
The authors report 12 cases of Paget's disease with sarcomatous degeneration that were observed among 311 patients with Paget's disease hospitalized in the Montpellier Rheumatology Clinic. They compare their experience with information they were able to extract from the literature in French and English. The frequency of such degeneration in cases of Paget's disease is difficult to determine but did not appear to exceed 1%. Degeneration rarely occurred before the age of 50 years, affected men twice as frequently as women, and occurred particularly in cases of diffuse Paget's disease, mainly in the femur or the humerus; the rachis was rarely affected. Pain was the main symptom, was practically constant, was remarkable because of its permanence and its intensity. Tumefaction was frequently seen. Pathological fractures were seen in almost a third of the patients with sarcomas of the long bones. Radiculo-medullary compression characterized the rare cases with involvement of the vertebral column or the sacrum. Radiography showed rupture of cortical layers with invasion of the soft parts without any periostal reaction. Histological investigation confirmed the diagnosis by demonstrating one of the three types of osteogenic sarcoma: osteosarcomas were the most frequent. Less frequently giant-cell sarcomas or reticulosarcomas were found. The affected patients nearly always died, survival at five years being rare. Treatment, amputation or radiotherapy, was disappointing.
作者报告了在蒙彼利埃风湿病诊所住院的311例佩吉特病患者中观察到的12例伴有肉瘤样变性的佩吉特病病例。他们将自己的经验与从法语和英语文献中获取的信息进行了比较。佩吉特病病例中这种变性的发生率难以确定,但似乎不超过1%。变性很少发生在50岁之前,男性受影响的频率是女性的两倍,尤其发生在弥漫性佩吉特病病例中,主要累及股骨或肱骨;脊柱很少受累。疼痛是主要症状,几乎持续存在,因其持续性和强度而显著。经常可见肿胀。几乎三分之一的长骨肉瘤患者出现病理性骨折。神经根脊髓受压是脊柱或骶骨受累的罕见病例的特征。X线检查显示皮质层破裂,软组织受侵,无任何骨膜反应。组织学检查通过证实三种类型的骨肉瘤之一确诊:骨肉瘤最为常见。较少见巨细胞肉瘤或网状细胞肉瘤。受影响的患者几乎总是死亡,五年生存率罕见。治疗方法,截肢或放疗,效果令人失望。