Al-Senaidi Khalfan S, Ullah Irfan, Javad Hashim, Al-Khabori Murtadha, Al-Yaarubi Saif
Departments of Child Health, Sultan Qaboos University Hospital, Muscat, Oman.
Haematology, Sultan Qaboos University Hospital, Muscat, Oman.
Sultan Qaboos Univ Med J. 2015 Nov;15(4):e456-62. doi: 10.18295/squmj.2015.15.04.003. Epub 2015 Nov 23.
Structural and functional cardiovascular abnormalities have been reported in adults with osteogenesis imperfecta (OI); however, there is a lack of paediatric literature on this topic. This study aimed to investigate cardiovascular abnormalities in children with OI in comparison to a control group.
This case-control study was conducted at the Sultan Qaboos University Hospital in Muscat, Oman, between May 2013 and August 2014. Data from eight patients with OI and 24 healthy controls were compared using conventional and tissue Doppler echocardiography (TDE).
The OI group had significantly lower peak early mitral valve flow velocity (P = 0.027), peak a-wave reversal in the pulmonary vein (P = 0.030) and peak early diastolic velocity of the mitral valve and upper septum (P = 0.001 each). The peak late diastolic velocities of the mitral valve (P = 0.002) and the upper septum (P = 0.037) were significantly higher in the OI group; however, the peak early/late diastolic velocity ratios of the mitral valve (P = 0.002) and upper septum (P = 0.001) were significantly lower. Left ventricular dimensions and aortic and pulmonary artery diameters were larger in the OI group when indexed for body surface area. Both groups had normal systolic cardiac function.
Children with OI had normal systolic cardiac function. However, changes in myocardial tissue Doppler velocities were suggestive of early diastolic cardiac dysfunction. They also had increased left ventricular dimensions and greater vessel diameters. These findings indicate the need for early and detailed structural and functional echocardiographic assessment and follow-up of young patients with OI.
已有报道称成骨不全(OI)成人患者存在心血管结构和功能异常;然而,关于这一主题的儿科文献却很匮乏。本研究旨在对比OI患儿与对照组,调查OI患儿的心血管异常情况。
本病例对照研究于2013年5月至2014年8月在阿曼马斯喀特的苏丹卡布斯大学医院开展。使用传统及组织多普勒超声心动图(TDE)对8例OI患者和24例健康对照的数据进行比较。
OI组二尖瓣早期血流峰值速度显著降低(P = 0.027),肺静脉a波反向峰值降低(P = 0.030),二尖瓣及上间隔舒张早期峰值速度均显著降低(均为P = 0.001)。OI组二尖瓣(P = 0.002)及上间隔(P = 0.037)舒张晚期峰值速度显著更高;然而,二尖瓣(P = 0.002)及上间隔(P = 0.001)舒张早期/晚期峰值速度比值显著更低。以体表面积计算时,OI组左心室尺寸、主动脉及肺动脉直径更大。两组的心脏收缩功能均正常。
OI患儿的心脏收缩功能正常。然而,心肌组织多普勒速度的变化提示存在舒张早期心脏功能障碍。他们的左心室尺寸也增大,血管直径更宽。这些发现表明,需要对OI患儿进行早期、详细的结构和功能超声心动图评估及随访。