Balci Tansel Ansal, Ciftci Ismail, Kabakus Nimet, Aydin Mustafa
Department of Nuclear Medicine, Firat University Medical Faculty, Elazig, Turkey.
Tohoku J Exp Med. 2006 Oct;210(2):169-73. doi: 10.1620/tjem.210.169.
Fragile X syndrome (FXS) is known as the most common form of inherited mental retardation. In our study, brain perfusion single photon emission computed tomography (SPECT) was performed in a 6 year-old boy diagnosed with FXS. Diffuse bilateral uptake of Technetium-99m hexamethyl propylene amine oxime (99mTc-HMPAO) was noted in his orbits, as well as cortical perfusion defects (hypoperfusion in the right parietal and the left temporal lobe). Ophthalmologic examination showed no pathological findings. Magnetic resonance imaging (MRI) revealed no abnormality in the orbital structures, although hypoplasia of cerebellum and vermis was visualized. Since the patient was crying during the injection, the increased blood flow or the increased metabolism of the eyes and/or ocular muscles may be responsible for this orbital finding. Alternatively, the enhanced uptake of HMPAO in the orbits may reflect the pathology associated with FXS, because patients with FXS might have visual-motor abnormalities. To the best of our knowledge, there has been no report documenting such an orbital uptake of HMPAO. Moreover, the visualization of decreased cerebral perfusion, with the normal findings of MRI, indicates that brain SPECT imaging with HMPAO is helpful for detecting brain abnormalities in children with FXS.
脆性X综合征(FXS)是已知最常见的遗传性智力障碍形式。在我们的研究中,对一名被诊断为FXS的6岁男孩进行了脑灌注单光子发射计算机断层扫描(SPECT)。在他的眼眶中发现了双侧弥漫性的锝-99m六甲基丙烯胺肟(99mTc-HMPAO)摄取,以及皮质灌注缺损(右侧顶叶和左侧颞叶灌注不足)。眼科检查未发现病理结果。磁共振成像(MRI)显示眼眶结构无异常,尽管可见小脑和蚓部发育不全。由于患者在注射期间哭闹,眼部和/或眼肌血流量增加或代谢增加可能是导致这种眼眶表现的原因。或者,眼眶中HMPAO摄取增强可能反映了与FXS相关的病理情况,因为FXS患者可能存在视觉运动异常。据我们所知,尚无报告记录HMPAO在眼眶中的这种摄取情况。此外,MRI结果正常但脑灌注减少的显影表明,使用HMPAO进行脑SPECT成像有助于检测FXS患儿的脑部异常。