Vuorimies Ilkka, Mäyränpää Mervi K, Valta Helena, Kröger Heikki, Toiviainen-Salo Sanna, Mäkitie Outi
Children's Hospital and.
Folkhälsan Institute of Genetics, 00029 Helsinki, Finland.
J Clin Endocrinol Metab. 2017 Apr 1;102(4):1333-1339. doi: 10.1210/jc.2016-3745.
The short-term benefits of bisphosphonates (BPs) are evident in the treatment of children with osteogenesis imperfecta (OI), but some concerns related to long-term effects remain.
To elucidate the effect of BPs on characteristics of femoral fractures in children with OI.
Retrospective cohort study at a university hospital.
The study included 93 patients with OI. We recorded fracture histories and analyzed all femoral fractures for location and fracture type using radiographs obtained at fracture diagnosis. Effects of BPs were evaluated by comparing fracture characteristics in three groups: patients (1) naive to BPs, (2) receiving ongoing BP treatment, and (3) whose treatment was discontinued.
In total, 127 femoral fractures occurred in 24 patients. Of the fractures, 63 (50%) occurred in patients naive to BPs, 44 (35%) during BP treatment, and 20 (16%) after treatment discontinuation. Mid or distal shaft fractures were most common (41%), followed by subtrochanteric (33%) and distal (20%) fractures. Almost all fractures were transverse (65%) or oblique (28%). The pattern of femoral fractures was similar in all three BP treatment groups (P = 0.78 for location; P = 0.35 for fracture type) and was not related to cumulative BP dose. Instead, OI type correlated with fracture characteristics, and distal location and transverse configuration were more common in the more severe types III and IV compared with type I OI.
Characteristics of femoral fractures in children with OI are affected by OI type but not by BP exposure.
双膦酸盐(BPs)在治疗成骨不全(OI)儿童中的短期益处是明显的,但仍存在一些与长期影响相关的担忧。
阐明BPs对OI儿童股骨骨折特征的影响。
在一家大学医院进行的回顾性队列研究。
该研究纳入了93例OI患者。我们记录了骨折病史,并使用骨折诊断时获得的X线片分析了所有股骨骨折的位置和骨折类型。通过比较三组患者的骨折特征来评估BPs的效果:(1)未使用过BPs的患者;(2)正在接受BPs治疗的患者;(3)已停止治疗的患者。
共有24例患者发生了127例股骨骨折。其中,63例(50%)骨折发生在未使用过BPs的患者中,44例(35%)发生在BPs治疗期间,20例(16%)发生在治疗停止后。股骨干中、远端骨折最为常见(41%),其次是转子下骨折(33%)和远端骨折(20%)。几乎所有骨折均为横行(65%)或斜行(28%)。在所有三个BPs治疗组中,股骨骨折的模式相似(位置P = 0.78;骨折类型P = 0.35),且与BPs累积剂量无关。相反,OI类型与骨折特征相关,与I型OI相比,更严重的III型和IV型OI中,远端骨折位置和横行骨折形态更为常见。
OI儿童股骨骨折的特征受OI类型影响,而非BPs暴露情况的影响。