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病例漫谈107:青年人并发混合型股骨髋臼撞击综合征和非特异性髋关节滑膜炎的罕见病例—一则病例报告 [复制链接]

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Introduction
Acetabular labrum damage leads to loss of its function, early appearance of groin pain, loss of hip function and early hip osteoarthritis development. Numerous pathomechanism have been proposed to explain the labrum damage, including the femoroacetabular impingement (FAI). Minimal bone changes of the acetabulum and/or femoral head, reduce the distance between them, leading to premature contact-impact of the femoral head-neck junction on the edge of the acetabulum, damaging labrum and labrum adjacent cartilage, wich are early signs of hip arthritis. Three types of FAI have been described: the first one is cam type with a morphological changes at the femoral head-neck junction in the form of cam deformmity. The secoond one is pincer type, in which the patological changes are localized exclusively to the acetabulum as a global (coxa profunda, protrusio acetabuli) or local overcoverage (retroversio acetabuli) of the femoral head, where the motion of the hip leads to the impact of the femoral neck on the edge of the acetabulum and consequent damage to the acetabular labrum. The third, mixed and the most common type of FAI is the combination of the previous two. Synovitis of the hip is a reaction of the articular synovium on the systemic or local pathological substrate in the form of synovial joint fluid production or specific hypertrofic synovium reaction that is seen in pathological conditions such as pigmented villonodular synovitis, tuberculosis or rheumatoid pannus. In the literature we found no case of hypertrofic non-specific synovitis combined with any kind of FAI.
前言
髋臼盂唇损伤导致功能丧失,早期会出现腹股沟区疼痛,髋关节功能丧失及髋关节骨性关节炎早期发展。针对盂唇损伤的产生原因,已经提出了许多病理机制,其中包括股骨髋臼撞击综合征(FAI)。髋臼和/或股骨头骨之间发生微小变化,两者距离缩小,导致股骨头颈连接处和髋臼边缘之间发生早期接触碰撞,并损伤盂唇和盂唇相邻软骨,这都是髋关节炎的早期症状。文中描述了三种FAI类型:第一种是凸轮型,是股骨头颈连接处出现凸轮样残缺的形态变化。第二种是钳夹型,其病理变化位于髋臼(髋臼过深,髋臼前突)或股骨头局部覆盖(髋臼后倾),这些部位髋关节的活动导致髋臼边缘的股骨颈受到撞击,同时造成髋臼盂唇受损。第三种是前两种类型的混合型,也是FAI最常见的类型。髋关节滑膜炎是机体在全身或局部产生关节滑膜关节液或特异性肥厚性滑膜反应的病理学反应,可见于色素沉着绒毛结节性滑膜炎,肺结核或风湿性血管翳等病变。在已有文献中,我们尚未发现肥厚性非特异性滑膜炎与任一种FAI类型并发的病例。  

We presented a patient with nonspecific chronic hypertrofic synovitis of the hip, combined with a mixed form of FAI, who was surgically treated.
我们报道了一例肥厚性慢性非特异性髋关节滑膜炎并发混合型FAI的患者接受手术治疗的病例。      

Case report
A 20-year-old male patient suffered from pain in the left groin, which lasted two years before the surgery. The patient was treated in several orthopedic institutions with non-steroid anti-inflammatory drugs, physiotherapy, skin traction, and suggested total hip replacement. Six months before the surgery, during the ultrasound examination of the hip joint, synovial fluid in the hip was asserted for which injection of corticosteroids into the joint was administered. After that, the symptoms became more pronounced, the patient began to limp visibly, walking on flat surfaces was difficult, and climbing up the stairs was practically impossible. At clinical examination the patient walked with the left leg in external rotation, with highlighted limps on that leg, Trendeleburg sign was positive on the left leg, highlighted weakness of muscles of the thigh and the left gluteal region, active flexion of the hip was possible up to 70°, internal rotation up to 5°, adduction up to 10° in the hip flexion of 70°. Impingement test was positive in the hip flexion of 30–70°. The patient brought computed tomography (CT) image of the hips and nuclear magnetic resonance (NMR) that were made before the application of corticosteroids into the joint and which showed that the bone and soft tissue structures of the hips were normal. Biochemical and laboratory tests were also within normal limits. Standardized anteroposterior radiography of the hips showed characteristics findigns for femoroacetabular impingement reported earlic in literature: (Figure 1) a strong mutual retroversion of the acetabulum 12° on the left and 11° on the right hip , mutual positive sign of ischial spine projection, the angle α was 46° on the right and 44° on the left, acetabular index, mutual 10°, mutual Wiberg angle 36°, a kolodyaphyseal angle of 126° that was asserted bilaterally.
病例报告
一名20岁男性患者,左腹股沟疼痛,手术前此疼痛已持续两年。患者在多个骨科机构接受了非甾体类抗炎药治疗,物理治疗及皮牵引治疗,并被建议接受全髋关节置换术。术前六个月,超声引导下在髋关节滑液中注射皮质类固醇激素进行治疗。此后,患者症状愈加恶化,跛行明显,平地行走困难,几乎不能爬楼。临床检查显示患者行走时左腿外旋且跛行明显,左腿Trendeleburg试验呈阳性,大腿和臀部左侧的肌肉明显萎缩,髋关节主动屈曲可达70°,内旋可达5°,髋关节屈曲为70°时内收可达10°。 髋关节屈曲30-70°时,撞击实验呈阳性。关节注射皮质类固醇激素之前,患者提供的髋关节的计算机断层扫描(CT)图像和核磁共振(NMR)图像显示髋关节处的骨和软组织结构均正常。生化化验检查结果也在正常范围内。标准化的髋关节前后位X线片显示了文献中已报道的股骨髋臼撞击综合征的特征:(图1)髋关节双侧髋臼后倾明显,左侧为12°,右侧为11°,坐骨棘投影呈阳性,右侧α为46°,左侧α为44°,两者髋臼指数为10°,Wiberg角为36°,双侧kolodyaphyseal角为126°。

Fig.1 – Standardized anteroposterior x-ray of the hips showing bilaterally positive sign of the ischial spine (white arrow), and acetabular retroversion (black arrow).

图1 – 标准化的髋关节前后位X线片显示双侧坐骨棘呈阳性(白色箭头),且髋臼后倾(黑色箭头)。  


In profile, Dunn-Ripstein-Müller's radiographs of the hips, the angle α on the left was 64° (normal value of this angle is below 50°), and on the right 42° (Figure 2).
剖面上,Dunn-Ripstein-Müller 髋关节X线片显示左侧α为64°(正常值应低于50°),右侧为42°(图2)。

Fig.2 – Standardized profile Dunn-Rippstein-Müller x-ray images of the hips: the left hip (right picture) – angle α 64°; the right hip (left picture) – angle α 42°.

图2 – 髋关节标准化剖面Dunn-Rippstein-MullerX线片:左侧髋关节(右图)-α为64°;右髋关节(左图)-α为42°。


Such clinical and radiographic findings revealed the existence of mixed-type of FAI on the left hip, due to which we proposed surgical treatment to the patient. The patient was operated on. It was planned to lift the acetabular labrum from its base, to osteotomize overcovered anterior and superior edge of the acetabulum, then, reinsert acetabular labrum in a new slot, and osteotomize cam deformity at the femoral head-neck junction in its antero-superior region. Lateral hip incision and transtrohanteric flip osteotomy of the hip were done, and after anterior ''Z'' hip capsulotomy unexpected hypertrophic synovial hip reaction was asserted (Figure 3).
这些临床和影像学结果表明左髋关节存在FAI混合型症状,因此我们建议患者接受手术治疗。随后,患者进行了手术治疗。手术计划将髋臼盂唇从基底切除,于髋臼边缘的前部和上部截骨,然后重新在一个新位置插入髋臼盂唇,并对股骨头颈连接处前上部的凸轮型畸形行截骨术。于髋关节外侧切口,并行经转子翻转截骨术,前部行“Z”型髋关节囊切开术后,意外地发现了肥厚性髋关节滑膜炎的存在(图3)。

Fig.3 – Intraoperative image: hypertrophied synovitis of the joint capsule on the anterior and superior femoral neck and head (black arrow), and femoral head (yelow arrow).

图3 – 术中图像:股骨头颈前位和上位(黑色箭头)以及股骨头(黄色箭头)处的肥厚性关节囊滑膜炎。


Entire extra-acetabular part of the joint was filled with synovial tissue which had fully and seriously damaged the anterior and the superior part of the acetabular labrum. Partial hip synoviectomy was performed, tissue samples were sent to histopathological analysis, the damaged parts of the labrum was resected, without possibility to be reinserted, anterior and superior part of the acetabular edge were osteotomized to reduce acetabular retroversion. Osteochondroplasty of the cam prominence at the femoral head-neck junction was done (Figure 4), and thus, intraoperatively, hip flexion of 90° and internal rotation of 30° was obtained. After joint capsule reconstruction, osteotomized part of the greater trochanter was re-fixed with two cortical screws and the operational wound was closed.

髋臼外部被滑膜组织充满,严重损伤了髋臼盂唇前部和上部。行局部髋关节滑囊切除术,取组织样本进行组织病理学分析,切除盂唇损伤部位,但不能被重新插入,切除髋臼边缘前部和上部以减小髋臼后倾。于股骨头颈连接处对凸轮型突出行骨软骨成形术(图4),由此,术中髋关节能达到屈曲90°,内旋30°。关节囊重建后,大转子截骨部分以两个皮质骨螺钉进行重新固定,并缝合伤口。  

Fig.4 – Intraoperative picture: osteochondroplasty of the cam deformity of the femoral head-neck junction (black arrow), and femoral head (yellow arrow).

图4 – 术中图像:股骨头颈连接处(黑色箭头)和股骨头(黄色箭头)处凸轮畸形的骨软骨成形术。


Postoperatively, the patient was reimbursed with 400 mL of blood; low molecular weight heparin thromboprophylaxis for seven days was introduced, followed by oral anticoagulant therapy for up to six weeks after the surgery. On the first post-operative day active exercises in bed started and walking on the crutches was allowed from the second day with touching the tip of toes of the operated leg for six weeks after the surgery. Histopathological findings confirmed a nonspecific chronic hypertrophic synovitis of the hip. Two years after the surgery, the patient was asymptomatic, without limping on the left leg and with normal life activities. Flexion of the operated hip was 90°, internal rotation 25°, adduction 30°, impingement test was negative. On the anteroposterior radiographs of the hips, the acetabular angle of retrovesion was reduced up to 4°, the angle α was 40° and 49° on the profile of Dunn-Rippsetin-Müller radiographs of the left hip.
术后,患者输400ml血,持续口服抗凝药六周,之后,接受了7天的低分子量肝素抗凝治疗。术后第一天允许患者在床上进行活动锻炼,第二天可依靠拐杖行走,术后六周可触摸行手术的左腿脚趾前端。病理结果证实为肥厚性慢性非特异性髋关节滑膜炎。术后两年,患者症状解除、左腿不再跛行,日常活动正常。手术治疗后,髋关节屈曲90°,内旋25°,内收30°,撞击实验呈阴性。髋关节前后位X线片显示髋臼后倾角减小到4°,Dunn-Rippsetin-Müller左髋关节X线片显示α角分别为40°和49°。  

Discussion
Damages of the acetabular labrum cause pain and partial loss of function of the hip in young adults. There is a number of reasons for labrum lesions, such as mechanical trauma of the hip (hip dyslocations, acetabular fractures) and in patients with FAI, otherwise, it is the result of biological specific or nonspecific synovial hypertrophic pannus production  17–20 . Morphological bone changes in the area of the acetabulum and /or proximal femur in patients with FAI reduce physiological distance between these anatomical structures of the hip, lead to premature contact-impact of the femoral neck on the edge of the acetabulum, causing damage of the acetabular labrum and acetabular cartilage adjacent to it, but never lead to mechanical irritation of the articular capsule and its hypertrophic response.
讨论
青年人髋臼盂唇受损会诱发疼痛和髋关节部分功能丧失。盂唇病变的病因很多,如髋关节机械性创伤(髋关节脱位,髋臼骨折),另一方面,FAI患者的发病原因是生物特异性或非特异性的滑膜肥厚形成的血管翳。FAI患者的髋臼和/或股骨近端骨骼发生微小变化,缩小了髋关节结构之间的间隙,导致股骨颈与髋臼边缘的过早接触碰撞,髋臼盂唇和相邻髋臼软骨的损伤,但不会造成关节囊肥厚反应和机械性刺激。

We presented a patient with a clear clinical and radiographic picture of mixed form of FAI with normal biochemical, CT and NMR findings in which the subjective symptoms suddenly worsened after inta-articular application of corticosteroids in the hip joint. We found intraoperatively and verified histologically, a nonspecific reaction of the hypertrophic synovium, an unexplained etiology, that filled the hip joint, further decreasing the space between the edge of the acetabulum and the anterior and superor part of the femoral neck, with additional deterioration of the patient's subjective complaints, hip movements and irreversible damage of the acetabular labrum without the possibility to be reinserted but only partially resected. There were no reports in the literature to compare with our experience. We hypothesized, as a possible explanation, that the intraarticular administration of corticosteroids was the reason for the development of nonspecified hypertrophy of the hip synovium, but it needs furhter research.
本文报道了一例患者,临床和影像学图像清晰呈现为混合型FAI,生化检查结果正常,CT和核磁共振发现髋关节内糖皮质激素治疗后患者症状突然恶化。 我们在术中发现并经组织学证实了肥厚性滑膜的非特异性反应,其病因较难解释,此非特异性反应能填充髋关节,进一步缩小髋臼边缘和股骨颈前部与上部之间的间隙,同时还有患者主诉的其他恶化症状,髋关节移动及髋臼盂唇的不可逆性损伤,此损伤部位不能再生只能被部分切除。尚无与本文病例可进行比较的文献报道。我们认为引起髋关节滑膜非特异性肥厚性发展的可能病因是关节内注射的糖皮质激素,但这一假设还需进一步进行研究。

FAI has been, in the last 20 years, well-defined pathological and pathophysiological entity which is treated only surgically.
过去20年,人们已经明确了FAI的病理和病理生理学信息,并且只有手术能治疗FAI。

Conclusion
We believe that young adults with pain in the groin and with normal biochemical, CT and NMR parameters should not be exposed to unnecessary and inappropriate treatment (skin traction, total hip replacement or intra-articular corticosteroid application), but to be appropriately operated on.
结论
我们认为,青年人如果腹股沟疼痛,但生化、CT和核磁共振检查结果正常时,不应做不必要和不合理的治疗(皮牵引,全髋关节置换术或关节内皮质类固醇激素治疗),而应选择合适的手术方式进行治疗。

由MediCool医库软件 王露黔 编译

原文来自Vojnosanit Pregl


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