Tsilchorozidou Tasoula, Yovos John G
Division of Endocrinology, Diabetes and Metabolism, AHEPA University Hospital, Thessaloniki, Greece.
Hormones (Athens). 2005 Jul-Sep;4(3):171-6. doi: 10.14310/horm.2002.11156.
Osteomalacia associated with adult onset Fanconi syndrome is thought to result from hypophosphataemia due to renal phosphate loss and relative 1,25-dihydroxyvitamin D3 deficiency. In this disorder, the impaired renal phosphate uptake occurs as part of a generalized tubular defect in association with other features such as bicarbonuria, glycosuria and aminoaciduria. Fanconi syndrome is either hereditary--juvenile form--or is associated with various acquired or heritable diseases. In adults, the disease is similar to the juvenile form, but osteomalacia is a prominent feature. We report a sporadic, adult onset, hypophosphataemia in a 19-year old female patient who presented after puberty complaining of bone and joint pain and difficulty in walking following a minor fall. Radiological examination revealed numerous bilateral fractures of the ribs and pelvis while biochemical investigations showed combination of high phosphate clearance, low serum bicarbonate, glycosuria and glycinuria. Known causes of acquired renal tubular dysfunction were ruled out. The patient was diagnosed as having idiopathic Fanconi syndrome and started on vitamin D3 (Alfacalcidol 1 mg/day) and oral phosphorus (Joulie Solution, 1.5 g/day), which led to resolution of symptoms and an increase in serum phosphate (from 0,54 to 0,71 mmol/l) within few months following the initiation of therapy. However, radiological re-examination showed no signs of fracture healing.
成人起病的范科尼综合征相关的骨软化症被认为是由于肾磷丢失和相对的1,25 - 二羟维生素D3缺乏导致的低磷血症所致。在这种疾病中,肾磷摄取受损是作为与其他特征(如重碳酸盐尿、糖尿和氨基酸尿)相关的全身性肾小管缺陷的一部分出现的。范科尼综合征要么是遗传性的——青少年型——要么与各种获得性或遗传性疾病相关。在成年人中,该病与青少年型相似,但骨软化症是一个突出特征。我们报告了一名19岁女性患者,青春期后发病,表现为散发性、成人起病的低磷血症,患者在轻微跌倒后出现骨和关节疼痛及行走困难。放射学检查发现双侧肋骨和骨盆有多处骨折,而生化检查显示高磷清除率、低血清重碳酸盐、糖尿和甘氨酸尿并存。排除了已知的获得性肾小管功能障碍的病因。该患者被诊断为特发性范科尼综合征,并开始服用维生素D3(阿法骨化醇1毫克/天)和口服磷(朱利溶液,1.5克/天),治疗开始后几个月内症状缓解,血清磷升高(从0.54升至0.71毫摩尔/升)。然而,放射学复查未显示骨折愈合迹象。