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病例漫谈106:非典型性髋关节疼痛:股骨髋臼撞击综合征(FAI)和骨样骨瘤共存 [复制链接]

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Introduction

Femoroacetabular impingement (FAI) is a well-known condition that causes mechanical hip pain in young adults and initiates inevitable damage within the joint, leading to secondary osteoarthritis. In the last decade, abnormal osseous morphology (femoral and acetabular) has been increasingly recognized as a cause of hip pain and, possibly, primary hip arthritis. Patients with FAI are increasingly being treated with surgical intervention and obtaining good short-term results. However, there are less common hip conditions in young patients, which can exhibit clinical features similar to FAI, but which do not require surgical treatment. One such example is an osteoid osteoma. Osteoid osteomas are small, benign tumours often accompanied by severe pain, including night pain. An intraarticular osteoid osteoma, on the other hand, lacks this characteristic night pain and is less responsive to a nonsteroidal anti-inflammatory drugs (NSAIDS) than a classical osteoid osteoma. Intraarticular osteoid osteomas also show little or no signs of sclerosis due to a lack of new periosteal bone formation. Hence, the radiolucent nidus is often overlooked on initial radiographs, computerized tomography (CT) scans and magnetic resonance imaging (MRI). In fact, approximately 21 % of intraarticular niduses are not identified and a further 29 % poorly identified, on initial MRI.

前言

股骨髋臼撞击综合征(FAI)是一种为人熟知的病症,会使年轻人出现力学性髋关节疼痛,诱发关节内无法避免的损害进而导致继发性骨关节炎。过去十年里,骨形态异常(股骨和髋臼)已逐渐被认为是引起髋关节疼痛的原因之一,以及引发原发性髋关节炎的可能原因。接受手术治疗的FAI患者数量正在增加,并且短期疗效良好。然而,年轻患者出现罕见髋关节症状,表现出与FAI类似的临床特征,但不需要手术治疗,这样的一种例子就是骨样骨瘤。骨样骨瘤是较小的良性肿瘤,常伴有剧烈疼痛,存在夜间痛。而关节内骨样骨瘤不存在夜间痛这一特点,以非甾体类抗炎药(NSAIDs)治疗的效果较经典的骨样骨瘤差。关节内骨样骨瘤因为没有新生的骨膜成骨,同样表现出硬化较少或无硬化。因此,初期X线、电脑断层扫描(CT)和磁共振成像(MRI)上的透明病灶往往被忽视。事实上,通过初期磁共振成像,大约21%的关节内病灶尚未确定,另外29%较难确定。


Herein, the case of a 52-year-old male who presented with hip pain is reported. This patient was initially misdiagnosed with FAI, but was ultimately found to have an osteoid osteoma. There are few case reports and case series in the literature, such as this, that document concurrent causes of atypical hip pain and delayed diagnosis of an osteoid osteoma. Nevertheless, radiologists and orthopaedics surgeons should consider osteoid osteoma as part of the differential when diagnosing FAI. Correlation of history, clinical examination and radiological findings are extremely important in order to avoid misdiagnosis and provide patients with appropriate treatment and pain relief.

本文报道了一例52岁男性患者,临床表现为髋关节疼痛。此患者起初被误诊为FAI,但最终发现存在骨样骨瘤。很少有病例报告和病例系列文献报道这类病例,正如本文呈现的非典型性髋关节疼痛的并发性病因,以及骨样骨瘤的诊断延误。不管怎么样,放射科和骨外科医生在诊断为FAI时,应该将骨样骨瘤纳入到鉴别诊断的考虑范围。患者的相关病史、临床检查和影像学检查对于避免误诊,并给予患者合理治疗以及缓解疼痛具有重要意义。


Case report

A 52-year-old healthy male presented to a sports orthopaedic surgeon with a 1.5-year history of progressive left hip pain (sudden onset), which was activity dependent. Specifically, this pain was localized to the anterolateral aspect of the left hip. Occasionally, this pain radiated down to the anteromedial aspect of the left thigh as well. Initially, pain started with brisk walking and running, but soon progressed to daily activities and antalgic gait. As the hip pain progressed, the patient also started experiencing night pain, but there were no constitutional symptoms of weight loss or night sweats. The patient’s pain was kept under control with naproxen, NSAID, as well as rest and protected weight bearing.

病例报告

一名52岁健康男性,有一年半活动依赖型进行性左髋关节疼痛史(突发性),找运动学科骨外科医生就诊。具体来说,这种疼痛位于左髋关节前外侧。有时,疼痛会放射到左大腿前内侧。最初,患者在快步行走和跑步时出现疼痛,但很快发展到日常活动,出现止痛步态。随着髋关节疼痛的加剧,患者开始出现夜间痛,但没有体重下降和盗汗的症状。患者一直以非甾体类抗炎药萘普生控制疼痛,同时休息并避免负重。  


On examination, the patient had moderate tenderness over the groin area, just above the level of the greater trochanter. Hip flexion was restricted to 90°, as compared to 110° on the contralateral side. Internal rotation was also limited to 0° at 90° hip flexion, as compared to 30° on the contralateral side. Neurovascular examination was normal. Anterior impingement test on hip flexion, adduction and internal rotation was positive. Radiograph and MRI (Figs. 1, 2) findings were consistent with FAI. Specifically, the patient’s MRI revealed a Cam-type impingement with an anterosuperior labral tear in the hip (Fig. 2). There was no arthritic change in the hip joint at this time, though there was evidence of oedema in the femoral neck (Fig. 2). The initial MRI also revealed no evidence of a nidus, which is characteristic of an osteoid osteoma, but did show non- specific, mild bone marrow oedema at the femoral neck. A bone scan was subsequently performed and found to be normal (image not provided). As a result, osteoid osteoma and stress fracture were both ruled out based on these initial findings.

检查结果表明,患者大转子上方的腹股沟区存在中度压痛。患者左侧髋关节屈曲与对侧屈曲110°相比,受限于90°。同样,髋关节屈曲为90°时,内旋较对侧的30°局限于0°。血管神经检查正常。髋关节屈曲前撞击测试、内收和内旋为阳性。X线及MRI(图. 1,2)检测结果符合FAI。具体来说,患者的MRI检测结果显示髋部存在凸轮型撞击和前上盂唇撕裂(图2)。此时,虽然股骨颈水肿,但髋关节尚未出现关节炎(图2)。初期MRI检测结果也没有显示骨样骨瘤的病灶特征,但显示股骨颈处存在非特异性、轻度骨髓水肿。随后进行的骨扫描显示正常(未提供图像)。因此,基于这些初期检测结果,排除了骨样骨瘤和应力性骨折。


Fig. 1 Pre-operative X-ray showing Cam lesion and medial femoral neck thickening (arrow) characteristic of FAI; the nidus on the medial femoral neck (chevron), which was missed initially, can also be discerned radiographically

图.1 术前X线显示FAI的凸轮型病变与股骨颈内侧增厚(箭头)特征;最初被忽视的股骨内侧颈部病灶(人字形),也可见于X线片


Fig. 2 Initial MRI coronal T1 image showing labral tear (arrow) and oedema

图.2 初期MRI冠状位T1图像显示盂唇撕裂(箭头)和水肿


Following the FAI diagnosis, an intraarticular injection of 2% lidocaine (5ml) with depomedrol (80mg) was administered in the hip joint. The patient experienced 20% pain relief immediately after injection, which lasted for 2 months, after which pain returned to baseline. Nonoperative modalities, such as physiotherapy, showed no benefit. Surgical intervention was discussed with the patient after non-operative treatments failed. A left hip arthroscopy was then performed 10 months following his initial visit. During surgery, the patient was placed in supine position on a traction table and traction was applied. Standard trochanteric anterolateral, distal anterolateral and mid-anterior portals were made and access to the joint was achieved using the Seldinger technique. The joint capsule was thickened, and synovium was inflamed. There was an anterosuperior labral tear accompanied by chondral wear, which was arthroscopically shaved and debrided. A bony bump at the femoral head–neck junction was noted consistent with Cam impingement. A head–neck osteoplasty was performed with a 5.5-mm burr. The hip was then tested for intraoperative range of motion (ROM), and no signs of impingement were noted. Standard follow-up visits were completed at 2 weeks, 6 weeks, 6 months and 1 year following surgery.

诊断为FAI后,于髋关节内注射2%利多卡因(5ml)和甲基泼尼松龙(80ml)。注射后,患者疼痛迅速缓解20%,这持续了2个月,之后疼痛又恢复到原水平。物理疗法等非手术治疗方法未见疗效。非手术治疗失败后,与患者进行了手术干预治疗的讨论。初诊10个月后,进行了左侧髋关节镜手术。术中,患者仰卧于牵引台,行牵引术。标准转子前外侧、远端前外侧和前中位路径均进行了手术,并通过Seldinger技术连接至关节。关节囊增厚,滑膜发炎。前上盂唇撕裂,同时伴有软骨磨损,这些均在关节镜下进行刮削和清理。股骨头颈骨接头处的骨肿块符合凸轮型撞击特征。以5.5 mm的钻孔行头颈骨成形术。然后测试术中髋关节的活动范围(ROM),并没有呈现撞击迹象。术后2周、6周、6个月和1年分别进行了随访。


During the 2 weeks post-operative period, the deep-seated hip pain resolved and the patient’s ROM (flexion and internal rotation) improved. However, the patient still had some anterior thigh pain. The anterior thigh and knee pain persisted at both the 6-month and 1-year follow-up visits and did not resolve with dedicated physiotherapy that utilized manual techniques (i.e. active-release therapy). Clinical examination at 1 year post-surgery was otherwise relatively benign. At this time, a repeat MRI was ordered to rule out labral re-tears and/or bony pathology in the left hip. The repeat MRI was performed 1.5 years post-surgery and showed a mild increase in degenerative changes in the left hip and post-operative changes at the femoral head-neck junction, but no new labral tears (Fig. 3a). Moderate bone marrow oedema was noted on T1 fat-suppressed weighted signals, as well as cortical thickening (sclerosis) along the inferomedial aspect of the femoral neck and an osteolytic nidus in the centre on T1 images (Fig. 3b). These findings were consistent with an osteoid osteoma. As a result, the patient was subsequently referred to an orthopaedic oncologist. A CT scan (Fig. 4) was ordered, which showed thickening of the medial neck cortex and a nidus, thus confirming the osteoid osteoma diagnosis. The patient was given another selective NSAID (meloxicam; 15 mg) for 2 weeks and reassessed. Of note, bone scan findings were not typical of an osteoid osteoma as no increased blood pool activity or hyperaemia was noted.

手术后2周内,髋关节深部疼痛得到缓解,患者ROM(屈曲和内旋)有所改善。然而,患者前大腿仍有一些疼痛。术后6个月和1年的随访均报道了前大腿和膝盖的疼痛,并且专业人工理疗手法处理后(即主动缓解疗法)没有缓解疼痛。另外,术后1年的临床检查相对良好。此时,需再次对左侧髋关节进行MRI检查以排除盂唇再撕裂和/或骨性病变。手术后1.5年,再次进行MRI检查,结果显示左侧髋关节存在轻度增加的退行性变化,术后股骨头颈骨接头处出现了变化,但没有再次出现盂唇撕裂(图.3a)。T1脂肪抑制加权信号显示中度骨髓水肿以及股骨颈内下方处皮质增厚(硬化),T1图像中心呈现溶骨性病灶(图3b)。这些检测结果均符合骨样骨瘤。因此,随后建议患者接受骨科肿瘤学医师治疗。进行CT扫描(图.4),结果显示颈内侧皮质增厚且有病灶,从而确诊为骨样骨瘤。患者服用了另一种非甾体类抗炎药(美洛昔康;15mg),服药2周后进行再评估。值得注意的是,骨扫描结果未显示血池摄取增加或充血,表明是非典型性骨样骨瘤。


Fig. 3 Repeat MRI image 1.5 years post-surgery. a Coronal T1 image showing post-operative changes at the femoral head–neck junction osteoplasty; b coronal T1 fat-suppressed image showing the nidus over the medial femoral neck with thickening of neck and bone marrow oedema

图3 术后1.5年再次进行磁共振成像的图像。a 冠状面T1图像显示术后股骨头颈骨成形术部位出现了变化;b冠状面T1脂肪抑制图像显示股骨内侧颈处出现病灶和增厚,同时显示颈骨髓水肿


Fig. 4 CT scan (2-mm axial cut) approximately 2 years post-surgery showing the nidus with surrounding sclerosis

图4 术后约2年时,CT扫描(2-mm轴向切口)显示病灶周围出现硬化


At the 2-week follow-up with the orthopaedic oncologist (2 years following the initial hip arthroscopy), the patient reported that his anterior thigh and knee pain had begun to resolve. Based on clinical examination, it was determined that the osteoid osteoma symptoms were subsiding. Nonsurgical management with meloxicam was continued for another 4 weeks. The patient was completely symptom free 2.5 years after the initial hip arthroscopy and had no restricted ROM or pain upon examination.

在与骨科肿瘤科医师进行第2周的随访中(首次髋关节镜手术2年后),患者称前大腿和膝盖的疼痛已经逐渐得到缓解。根据临床检查,确认骨样骨瘤症状消退。另服用美洛昔康4周进行非手术治疗。首次髋关节镜手术2.5年后,患者症状完全消失,并且ROM不受限制,不再疼痛。


Discussion

This case report highlights the importance of continuous work-up to uncover the cause of patient symptoms and to enhance patient outcomes. In the case presented herein, an intraarticular osteoid osteoma, which was the original source of pain, was overlooked on initial radiographs. However, persistence of the patient’s symptoms after sur- gery led the clinician to further investigate and seek the opinion of a multidisciplinary team; this eventually led to the correct diagnosis and improved patient outcome. Of note, up to 25 % of osteoid osteomas are located on the proximal part of the femur, and 5–12 % of all osteoid osteoma are intraarticular. Intraarticular osteoid osteomas remain a diagnostic challenge because of their atypical symptoms and atypical appearance on conventional radiographs. Intraarticular osteoid osteomas have little or no evidence of sclerosis because they often lack new periosteal bone formation. Hence, the radiolucent nidus is often overlooked on initial radiographs, CT and MRI. In addition, both femoroacetabular impingement (FAI) and intraarticular osteoid osteomas are potential sources of similar hip pain. Intraarticular osteoid osteomas, however, lack night pain typical of osteoid osteomas and are less responsive to NSAIDS; thus, misdiagnosis of an osteoid osteoma is highly possible. Increased FAI awareness (among both surgeons and physicians) and its prevalence in the asymptomatic general population make it is possible for other potential causes of pain to be missed as well. In the case described herein, the patient had an atypical clinical presentation of an intraarticular osteoid osteoma, which was already subsiding on the initial MRI and bone scan, and thus not detected when the patient initially consulted with the sports physician.

讨论

本病例报告强调了连续诊断性研究的重要性,通过这种方式可以发现与患者症状有关的病因并且提高治疗效果。此病例中,关节内骨样骨瘤是疼痛的病因,但在初期X射线检查时被忽视了。并且,术后患者症状持续,促使临床医生进一步展开了调查,并向多学科团队寻求意见,最终得到了正确诊断,提高了患者治疗效果。值得注意的是,超过25%的骨样骨瘤位于股骨近端,另外5-12%的骨样骨瘤位于关节内。关节内骨样骨瘤的诊断仍具有挑战性,因为此病症在传统X线片上症状和外观均不典型。关节内骨样骨瘤因为没有新的骨膜骨生成,表现出硬化较少或无硬化。因此,初期X线、CT和MRI上的透明病灶往往被忽视。此外,股骨髋臼撞击综合征(FAI)和关节内骨样骨瘤是引起相似髋关节疼痛的潜在病因。但关节内骨样骨瘤不存在关节外骨样骨瘤夜间痛这一典型症状,同时非甾体类抗炎药治疗效果较差;因此,骨样骨瘤误诊可能性较高。外科和内科医生对FAI认识的提高及FAI在无症状人群中的患病率,同样可能使医生错过疼痛的其他潜在病因。本文描述的病例中,患者关节内骨样骨瘤的临床表现不典型,在初期MRI和骨扫描已经有所消退,使得患者最初向运动学科医生就诊时未检测到。


Osteoid osteomas can also lead to reactive bone formation, causing Cam impingement and labral tears. As a result of this coexistence, osteoid osteomas become more challenging to diagnose. Moreover, MRIs are not the ideal modality for diagnosing osteoid osteomas; CT scans are much more sensitive and, thus, considered the gold standard for detecting and diagnosing osteoid osteomas both in adults and paediatric population. The initial musculoskeletal radiologist in this case did, in fact, notice bone marrow oedema and medial femoral neck thickening on the initial MRI; however, he ruled out stress fracture and osteoid osteoma based on the lack of nidus and the initial negative bone scan, thereby considering these as nonspecific findings. Of note, a negative bone scan is possible in the case of an intraarticular osteoid osteoma, whereas a bone scan for a typical extraarticular osteoid osteoma shows increased activity (i.e. ‘hot spots’). In this particular case reported, a repeat bone scan from 1 year and 8 months post-surgery showed increased osteoblastic bone turnover at the femoral neck, which is consistent with changes following arthroscopy, but not typical of active osteoid osteomas. Consequently, the patient showed significant improvement following treat- ment for the osteoid osteoma with anti-inflammatories and was completely pain free after 2.5 years.

骨样骨瘤也可导致反应性骨形成,导致凸轮型撞击和盂唇撕裂。这种共存使得骨样骨瘤的诊断更具挑战性。此外,MRI并非诊断骨样骨瘤的理想方法;CT扫描灵敏性更强,因此,被认为是检测和诊断成人及儿童骨样骨瘤的金标准。事实上,本病例中,最初放射科医生在MRI图像上发现了骨髓水肿和股骨颈内侧增厚;但是,因为未发现病灶以及最初骨扫描结果呈阴性,医生排除了应力性骨折和骨样骨瘤,从而认为这些症状为非特异性表现。值得注意的是,关节内骨样骨瘤病例可能存在骨扫描结果为阴性,而典型的关节外骨样骨瘤的骨扫描常显示异常聚集(即“热灶”)。这一特殊的病例报道中,术后8个月和1年的两次骨扫描显示股骨颈成骨细胞骨转换增加,这与关节镜术后变化一致,但不是典型的活动性骨样骨瘤。因此,患者服用抗炎药治疗骨样骨瘤,病情得到显著改善,术后2.5年疼痛完全治愈。


Our theory that the patient had an intraarticular osteoid osteoma that was slowly subsiding at the time of initial presentation was further consolidated by the fact that the patient had only partial relief of symptoms following the initial intraarticular injection. Because Cam impingement is a frequent source of symptoms in the same area and because intraarticular osteoid osteomas are generally not visible on radiographs, misdiagnosis is highly possible. It is important to be mindful of less common conditions that should be considered in the differential diagnosis for hip pain. These less common conditions include osteoid osteoma, pigmented villonodular synovitis (PVNS), synovial chondromatosis, osteonecrosis, femoral neck stress fractures and, least frequently, bone and soft tissue sarcomas.

我们认为,患者关节内骨样骨瘤在初期表现为缓慢消退,但之后进一步实变,这可以从患者经首次关节腔内注射后症状只得到部分缓解得到证实。由于凸轮型撞击是相同部位出现病症的常见病因,同时关节内骨样骨瘤在X线上通常不可见,因此很可能出现误诊。重点需注意一些罕见的症状,在鉴别诊断髋关节疼痛时应多做考虑。这些罕见症状包括骨样骨瘤、色素绒毛结节性滑膜炎(PVNS)、滑膜软骨瘤病、骨坏死、股骨应力性颈骨折以及更为少见的骨和软组织肉瘤。


Conclusion

As the awareness of FAI grows, it is tempting to diagnose impingement in patients who present with hip pain. However, the presence of FAI morphology does not necessarily mean that FAI is the primary source of hip pain. Other less common causes of hip pain, which are often overlooked, should also be included in the differential diagnosis, especially when atypical clinical and radiological signs are found. The outcome of this case thus emphasizes the importance of correlating patient history with both clinical and radiological findings in order to arrive at an accurate diagnosis and devise an effective treatment plan.

结论

随着对FAI认识的增强,对于髋关节疼痛患者,医生更容易地能诊断为撞击征。 然而,FAI的形态学表现并不一定意味着FAI是髋关节疼痛的主要病因。引起髋关节疼痛的其他不常见病因往往会被忽视,也应在鉴别诊断时进行考虑,特别是当发现临床和影像学检查结果具有非典型性时。此病例治疗结果强调了患者相关病史,临床和影像学检查结果对于准确诊断并制定有效治疗计划的重要性。


由MediCool医库软件 王露黔 编译

原文来自 Knee Surg Sports Traumatol Arthrosc

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