Jeleč Željko, Primorac Dragan, Antičević Darko
St Catherine Specialty Hospital.
University of Osijek, School of Medicine, Osijek, Croatia.
J Pediatr Orthop B. 2019 Sep;28(5):505-508. doi: 10.1097/BPB.0000000000000598.
Osteogenesis imperfecta (OI) is a genetic disorder characterized by fragile bones. It is our aim to illustrate variability in clinical presentation of severe form of OI. As an example of personalized surgery approach we present an 11-year-old girl with OI type III. Prior to referral to our hospital, she was treated with 18 cycles of bisphosphonates as well as with several different surgical procedures. Due to no improvement in her mobility status she underwent two additional surgeries at our hospital with a 5-month interval between them. Prior to the surgery, molecular genetic analysis was performed and the clinical diagnosis of OI was confirmed. Using the Fassier-Duval intramedullary telescoping nail, we performed correction osteotomies of both femurs and lower legs in two separate settings, with a very good final result. According to our experience, the Fassier-Duval nailing system is good option, but one should pay attention to many details while performing surgery. Thus, making treatment of OI patients very personalized. In this paper we present a unique personalized approach in OI: firstly, diagnosing COL1A1 gene mutations and secondly, performing a complex two-part surgery.
成骨不全症(OI)是一种以骨骼脆弱为特征的遗传性疾病。我们的目的是阐述重度成骨不全症临床表现的变异性。作为个性化手术方法的一个例子,我们介绍一位11岁的III型成骨不全症女孩。在转诊到我院之前,她接受了18个周期的双膦酸盐治疗以及几种不同的外科手术。由于她的活动能力没有改善,她在我院又接受了两次手术,两次手术间隔5个月。手术前,进行了分子遗传学分析,确诊为成骨不全症。我们使用法西耶 - 杜瓦尔髓内伸缩钉,在两个不同阶段分别对双侧股骨和小腿进行了截骨矫正手术,最终效果非常好。根据我们的经验,法西耶 - 杜瓦尔钉系统是个不错的选择,但手术时应注意诸多细节。因此,成骨不全症患者的治疗极具个性化。在本文中,我们介绍了一种针对成骨不全症独特的个性化方法:首先,诊断COL1A1基因突变;其次,实施复杂的两阶段手术。