Sumida K, Ubara Y, Hoshino J, Hayami N, Suwabe T, Hiramatsu R, Hasegawa E, Yamanouchi M, Sawa N, Fujii T, Takaichi K
Nephrology Center, Toranomon Hospital, 2-2-2, Toranomon, Minato-ku, Tokyo, 105-8470, Japan,
Osteoporos Int. 2015 Apr;26(4):1435-41. doi: 10.1007/s00198-014-2987-8. Epub 2014 Dec 13.
A bone biopsy specimen in a long-term hemodialysis patient with sarcoidosis coexisting with severe hypoparathyroidism has demonstrated that a persistent near physiological level of 1,25-dihydroxyvitamin D3 contributes to the preservation of bone remodeling and has the potential to retard the development of vascular calcification and atherosclerosis. Sarcoidosis-related hypercalcemia and hypoparathyroidism, which is characterized by 1,25-dihydroxyvitamin D3 (1,25(OH)2D3) overproduction, is rarely seen in hemodialysis patients. Herein, we describe a 60-year-old Japanese woman on hemodialysis for 35 years who presented with malaise and hypercalcemia. Severe hypoparathyroidism without parathyroidectomy and a preserved 1,25(OH)2D3 level were detected. Computed tomography showed bilateral axillary lymphadenopathy and minimal aortic and soft tissue calcification. The axillary node biopsy led to a definite diagnosis of sarcoidosis. A bone biopsy specimen obtained from the right iliac crest showed remodeling of normal lamellar bone with scalloped cement lines and clear double labeling by tetracycline on fluorescence microscopy. Histomorphometric analysis revealed that the bone formation rate was preserved (30.0 %/year), together with a decrease of osteoid volume (5.75 %) and fibrous volume (0 %), indicating that the patient did not have adynamic bone disease and only showed mild disease. This is the first documented case of sarcoidosis-related hypercalcemia associated with severe hypoparathyroidism in a long-term hemodialysis patient who underwent bone histomorphometry. Our findings suggest that, in hemodialysis patients with sarcoidosis coexisting with severe hypoparathyroidism, a persistent near physiological level of 1,25(OH)2D3 contributes to the preservation of bone remodeling and has the potential to retard the development of vascular calcification and atherosclerosis.
一名长期接受血液透析的结节病患者，同时合并严重甲状旁腺功能减退，其骨活检标本显示，持续接近生理水平的1,25 - 二羟维生素D3有助于维持骨重塑，并有可能延缓血管钙化和动脉粥样硬化的发展。结节病相关的高钙血症和以1,25 - 二羟维生素D3（1,25(OH)2D3）过度产生为特征的甲状旁腺功能减退在血液透析患者中很少见。在此，我们描述了一名60岁的日本女性，她接受血液透析35年，出现不适和高钙血症。检测到严重甲状旁腺功能减退且未进行甲状旁腺切除术，同时1,25(OH)2D3水平保持正常。计算机断层扫描显示双侧腋窝淋巴结肿大，主动脉和软组织有轻微钙化。腋窝淋巴结活检确诊为结节病。从右髂嵴获取的骨活检标本显示正常板层骨重塑，骨水泥线呈扇贝状，荧光显微镜下四环素双标记清晰。组织形态计量学分析显示骨形成率保持正常（每年30.0%），同时类骨质体积减少（5.75%），纤维体积减少（0%），表明该患者没有骨再生障碍性骨病，仅表现为轻度疾病。这是首例记录在案的长期血液透析患者中与严重甲状旁腺功能减退相关的结节病性高钙血症病例，并进行了骨组织形态计量学分析。我们的研究结果表明，在合并严重甲状旁腺功能减退的结节病血液透析患者中，持续接近生理水平的1,25(OH)2D3有助于维持骨重塑，并有可能延缓血管钙化和动脉粥样硬化的发展。
