Longato Lorena
Polyambulatory A.S.L. of Biella (BI), Italy.
Clin Cases Miner Bone Metab. 2014 Jan;11(1):49-58.
The Ambulatory of Biella is the only service of Rheumatology of the Biella area. The Paget's disease is the 0.5% and appears to be representative of the territory of Biella. We evaluated the pagetics locations, disease activity and quality of life, the presence of gene mutation and frequency of comorbidities and therapeutic efficacy. Patients with active disease were treated with intravenous bisphosphonate therapy and investigated in relation to the side effects: flu-like syndrome and therapeutic efficacy by ALP and scintigraphy. 15 out of 20 patients analyzed resulted in the active disease with pain in the upper-middle pagetic location. Treatment with bisphosphonates has led to remission of disease: normalization of ALP values in 90%, documented by the scintigraphy of control. In the 86% of cases it was not necessary to make a second infusion, even 3 years after the first administration. In only two cases was carried out another infusion for the persistence of pain, despite levels of ALP prove the rule, in such cases, the scintigraphy can showed uptake of the sites involved. Bone scintigraphy is more sensitive and specific of ALP in determining disease activity and should be used in cases of doubt of retreatment. 53% of cases showed as side effect a flu-like syndrome; 6 months after infusion secondary hyperparathyroidism in 46% of cases. SQSTM1 mutation was positive in two patients originating from Veneto and Sardinia, but in no patients in Biella. The presence of two aggregations family suggesting other mutations in the Biella population.
比耶拉门诊是比耶拉地区唯一的风湿病科服务机构。佩吉特病占比0.5%,似乎代表了比耶拉地区的情况。我们评估了佩吉特病的发病部位、疾病活动度和生活质量、基因突变的存在情况、合并症的发生率以及治疗效果。活动性疾病患者接受静脉双膦酸盐治疗,并就副作用(类流感综合征)以及通过碱性磷酸酶(ALP)和骨闪烁显像评估的治疗效果进行调查。在分析的20例患者中,有15例患有活动性疾病,病变部位在上中部,伴有疼痛。双膦酸盐治疗使疾病缓解:90%的患者ALP值恢复正常,通过对照骨闪烁显像得以证实。在86%的病例中,即使在首次给药3年后也无需进行第二次输注。仅在两例患者中,尽管ALP水平表明病情缓解,但因疼痛持续仍进行了另一次输注,在这种情况下,骨闪烁显像可显示受累部位有摄取。在确定疾病活动度方面,骨闪烁显像比ALP更敏感、更具特异性,在对再次治疗有疑问的情况下应使用。53%的病例出现类流感综合征副作用;输注6个月后,46%的病例出现继发性甲状旁腺功能亢进。两名分别来自威尼托和撒丁岛的患者SQSTM1基因突变呈阳性,但比耶拉地区的患者均未出现。两个家族聚集现象表明比耶拉人群中存在其他突变。