Gallant Rachel, McNall-Knapp Rene Y, Khan Osman
Children's Hospital of Los Angeles, University of Southern California, 4650 Sunset Blvd, Los Angeles, CA 90027 USA.
Division of Pediatric Hematology Oncology, University of Oklahoma Health Sciences Center, 1200 N. Children's Ave., Suite 14500, Oklahoma City, OK 73104 USA.
Radiol Case Rep. 2018 Oct 30;14(2):137-140. doi: 10.1016/j.radcr.2018.09.022. eCollection 2019 Feb.
Patients with hemophilia suffer from repeated episodes of hemarthrosis leading to chronic inflammation and synovitis. Radiosynovectomy is an effective nonsurgical modality that can reduce inflammation, pain, and hemarthrosis in such cases. We describe an adolescent male with severe Hemophilia A, who developed arterial vasculitis and perivasculitis targeting the brachiocephalic, right common carotid, and right subclabvian arteries occurring within few days after difficult Phosphorus-32 radiosynovectomy, possibly as a complication of the procedure. Despite prophylaxis with recombinant FVIII therapy, he developed chronic synovitis and underwent radionuclide synovectomy with P-32 injection to the left ankle and right knee. Five days later, he developed pain in the lower right neck and right upper chest. Computed tomography and magnetic resonance imaging and angiography demonstrated inflammation involving the arteries of the right thoracic inlet. Geiger-Mueller meter indicated increased radioactivity not only in the left ankle and right knee but also in the right upper chest. Detection of radioisotope at the right thoracic inlet corresponding to the area of vasculitis was indicative of likely deposition of the P-32 isotope in an area exposed to maximum cardiac output and increased blood flow, leading to subclavian, carotid, and innominate arteritis with surrounding edema.
血友病患者反复出现关节积血，导致慢性炎症和滑膜炎。放射性滑膜切除术是一种有效的非手术治疗方法，可减轻此类病例的炎症、疼痛和关节积血。我们描述了一名患有严重甲型血友病的青少年男性，在艰难的磷 - 32放射性滑膜切除术后数天内，发生了以头臂动脉、右颈总动脉和右锁骨下动脉为靶点的动脉血管炎和血管周围炎，这可能是该手术的并发症。尽管采用重组FVIII疗法进行了预防，他仍出现了慢性滑膜炎，并接受了向左脚踝和右膝关节注射P - 32的放射性核素滑膜切除术。五天后，他出现右下颈部和右上胸部疼痛。计算机断层扫描、磁共振成像和血管造影显示右胸廓入口动脉有炎症。盖革 - 米勒计数器显示不仅左脚踝和右膝关节放射性增加，右上胸部放射性也增加。在与血管炎区域相对应的右胸廓入口处检测到放射性同位素，表明P - 32同位素可能沉积在心脏输出量最大且血流增加的区域，导致锁骨下动脉、颈动脉和无名动脉炎并伴有周围水肿。
