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Ilizarov外固定架联合全膝关节置换术治疗血友病性膝关节屈曲僵直2例

2019-05-24 唐宏宇 王海彬 何伟 中国骨与关节损伤杂志

血友病是一种X染色体连锁的隐性遗传病,由于凝血因子缺乏而容易出血,以关节、肌肉出血多发。反复关节出血可导致慢性滑膜炎、关节软骨受损、关节周围组织纤维化和骨质破坏,出现关节疼痛和关节活动受限,形成血友病性关节炎,晚期导致严重的畸形,活动度降低和功能受限。血友病性关节炎可累及全身关节,以膝、踝关节多见。通过完备的围手术期管理,终末期血友病膝关节炎可采取全膝关节置换手术治疗。对于血友病膝关节严重屈曲畸形

血友病是一种X染色体连锁的隐性遗传病,由于凝血因子缺乏而容易出血,以关节、肌肉出血多发。反复关节出血可导致慢性滑膜炎、关节软骨受损、关节周围组织纤维化和骨质破坏,出现关节疼痛和关节活动受限,形成血友病性关节炎,晚期导致严重的畸形,活动度降低和功能受限。血友病性关节炎可累及全身关节,以膝、踝关节多见。通过完备的围手术期管理,终末期血友病膝关节炎可采取全膝关节置换手术治疗。对于血友病膝关节严重屈曲畸形患者,外固定架的使用尚未见报道,笔者介绍于2015-03收治的Ilizarov外固定架联合全膝关节置换术治疗终末期血友病性膝关节屈曲僵直的2例患者,取得良好的疗效,报道如下。

病例报道

病例1,男,25岁。因“左膝关节活动受限15年”入院,约10岁时诊断为“血友病甲型”,患者3个月前曾在本院行右侧全膝关节置换术。入院查体:左侧膝关节屈曲畸形,屈曲僵直70°,伸屈活动度-70°~130°。实验室检查血浆凝血因子Ⅷ活性2.3%,活化部分凝血活酶时间(APTT)84s,凝血因子Ⅷ抑制物0.09BU/ml。X线片提示骨质疏松,膝关节严重变形磨损。根据患者病情制定治疗计划,第一步使用外固定架牵伸,待关节基本拉伸伸直后再行膝关节置换术。安装外固定架术前将血浆Ⅷ因子水平补充至60%,手术采取全身麻醉,手术安装过程要求微创操作,钉道无明显出血,安装过程顺利(图1)。术后根据公斤体重测算,定期补充Ⅷ因子,术后前3d每天2次,维持血浆Ⅷ因子30%水平,以后每天1次或隔天补充,维持Ⅷ因子约10%水平,维持钉道干燥,无渗血,无明显肿胀瘀斑。术后第2天开始每天调整牵伸器,使膝关节逐步伸直。牵伸速度根据患者的耐受程度调整,开始为软组织水平1mm/d(连杆延伸3~4mm/d),分3或4次拧动。在调整过程中,持续观察患肢末端血运、感觉情况,如出现血管、神经危象及时调整其屈曲角度,待患肢适应后再逐步牵伸。定期清除骨圆针针孔处痂皮及分泌物。16d后膝关节屈曲挛缩矫正满意,输注一定量Ⅷ因子后,拆除外固定。拆除外固定架用长腿石膏托固定维持角度,拆除术后第5天行全膝关节置换术,使用Smith&nephew公司后稳定型旋转平台假体。术中残留20°屈曲畸形,术后指导锻炼,压腿。术后1年复查患者左膝疼痛消失,膝关节基本伸直,伸屈活动度-5°~60°(图2)。术后1年膝关节HSS评分由术前40分上升到82分。



病例2,男,25岁。因“双膝关节肿痛、活动受限12年”入院,约11岁时诊断为“血友病甲型”,2008年开始以轮椅代步。入院查体:双侧膝关节屈曲畸形,右膝屈曲僵直30°,伸屈活动度-30°~100°,左膝屈曲僵直60°,伸屈活动度-60°~100°。实验室检查:血浆凝血因子Ⅷ活性1.8%,活化部分凝血活酶时间(APTT)91.3s,凝血因子Ⅷ抑制物0.30BU/ml。患者双膝X线片检查提示双膝严重骨质疏松,膝关节严重变形磨损,骨缺损。根据患者病情制定治疗计划,第一步行右侧全膝关节置换+左膝外固定架牵伸,待左膝关节基本拉伸伸直后再行膝关节置换术。手术当天术前将血浆Ⅷ因子水平补充至100%,采取全身麻醉,手术过程顺利。第1次术后根据公斤体重测算,定期补充Ⅷ因子,术后前3d每天3次,维持血浆Ⅷ因子80%以上水平,术后4~14d每天2次,Ⅷ因子水平由80%递减至30%,维持手术切口和钉道干燥、无渗血、无明显肿胀瘀斑。术后第2天开始左膝每天调整牵伸器,调整方式同病例1,3d后膝关节屈曲挛缩矫正满意,输注一定量Ⅷ因子后拆除外固定。拆除外固定架用长腿石膏托固定维持角度,拆除术后第3天行全膝关节置换术,使用Link公司铰链型超小号膝关节假体,股骨外侧髁采取螺钉结构性植骨填充缺损。术中完全伸直膝关节,术后左膝佩带支具保护3个月,指导锻炼。术后1年复查双膝疼痛缓解,功能良好,左膝伸屈活动度-10°~80°。术后1年膝关节HSS评分由术前18分上升到71分。

讨论

血友病因凝血功能障碍,曾是人工关节置换手术的禁区,手术的风险主要是术中、术后出血问题。由于人凝血因子浓缩制剂的问世,围手术期补充凝血因子达到止血水平,并严密监测,血友病患者能够安全接受人工关节置换。此项工作的开展,需以骨科为主导,有经验的血液科医生配合,以及麻醉科等相关医疗团队的协助。本院从2014年开展第1例血友病人工关节置换,至今已超过20例关节,均取得良好的效果,因随访时间不长,暂未报道。笔者参照2012年世界血友病联盟《血友病管理指南》(第2版)以及国内北京大学人民医院的经验,制定本院围手术期管理规范,患者均安全度过围手术期,取得了良好的效果。多种疾病可导致膝关节屈曲挛缩畸形,Giles等将膝关节屈曲畸形分为三度,小于10°为轻度,10°~30°为中度,大于30°为重度畸形。重度畸形手术复杂,其常伴随着髋关节的屈曲畸形、骨质缺损、骨质疏松等,重度膝关节屈曲挛缩畸形行全膝关节置换效果相对较差,曾一度被学者列为全膝关节置换的禁忌证。随着医疗技术和骨科材料医学的发展,重度膝关节挛缩畸形手术的开展越来越普遍。由于重度膝关节屈曲畸形多数病程长,膝关节后侧间隙挛缩显著,包括血管及神经的挛缩。所以除了一般膝关节置换风险外,神经麻痹、血管损伤、伤口愈合并发症的发生率更高。褚亚明等报道使用后稳定或半限制性骨水泥型膝关节假体治疗严重屈曲畸形膝关节,取得良好疗效,大多数是屈曲畸形不超过40°的膝关节。

使用外固定牵伸器矫正严重屈膝挛缩畸形是顺应生物学生长规律的有效方法。李林等报道使用自行设计的一种新型膝关节外固定牵伸器治疗23例因小儿麻痹后遗症、类风湿性关节炎、强直性脊柱炎导致的膝关节屈曲挛缩患者,取得良好疗效;秦泗河等应用Ilizarov技术对49例52个膝关节重度屈曲挛缩畸形患者进行治疗,疗效满意。

鉴于外固定牵伸器对膝关节屈曲挛缩的特殊作用,以及此2例膝关节的屈曲挛缩严重程度(70°和60°),笔者设想首先应用Ilizarov技术矫正或部分矫正屈曲畸形,创造良好的软组织条件,二期再行关节置换术。然对于血友病关节炎导致的严重屈膝挛缩畸形病例,因其钉道的出血管理和护理困难,外支架的使用未见报道。笔者探索性地将外固定支具应用于血友病膝关节炎患者,牵伸期间严密监测和补充凝血因子Ⅷ,维持血浆Ⅷ因子在10%水平,钉道总体保持干燥,期间偶见有渗新鲜血液,及时补充凝血因子后出血停止。病例1牵伸2周达到预期效果,行膝关节表面置换术,使用普通后稳定型膝关节假体;病例2牵伸1个月,因骨质及软组织条件更差,选择髁限制性膝关节假体,术后患者功能均得到显著改善,病例1术后随访3年,病例2术后随访1年,效果优良。

原始出处:

唐宏宇,王海彬,何伟,张庆文,陈镇秋,唐立明,陈鹏.Ilizarov外固定架联合全膝关节置换术治疗血友病性膝关节屈曲僵直2例[J].中国骨与关节损伤杂志,2019(04):438-439.

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    2019-05-29 yxch48
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