Arthroscopic Treatment of Diffuse Pigmented Villonodular Synovitis of the Knee: Complete Synovectomy and Septum Removal Midterm Results

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1 Original Article Arthroscopic Treatment of Diffuse Pigmented Villonodular Synovitis of the Knee: Complete Synovectomy and Septum Removal Midterm Results Sohrab Keyhani, MD 1 Seyyed Morteza Kazemi, MD 2 Jin Hwan Ahn, MD, PhD 3 René Verdonk, MD, PhD 4 Mehran Soleymanha, MD 5 1 Department of Orthopedic Surgery, Akhtar Orthopedic Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran 2 Bone Joint and Related Tissues Research Center, Akhtar Orthopedic Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran 3 Department of Orthopedic Surgery, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, Seoul, Korea 4 Department of Orthopedics and Traumatology, Ghent University, Ghent, Belgium 5 Department of Orthopaedics, Orthopaedic Research Center, Poursina Hospital, School of Medicine, Guilan University of Medical Sciences, Rasht, Iran Address for correspondence Mehran Soleymanha, MD, Poursina Medical and Educational Center, Rasht, Gilan Province, Iran ( drmehransoleymanha@gmail.com). JKneeSurg Abstract Keywords knee pigmented villonodular synovitis arthroscopic synovectomy The purpose of this study was to evaluate and describe the clinical results of complete arthroscopic synovectomy through the four arthroscopic portals in the knees affected by diffuse pigmented villonodular synovitis (DPVNS). Between 2009 and 2012, 21 patients (15 men and 6 women) with the diffuse form of PVNS of the knee were enrolled in the study after qualification. The clinical diagnosis was confirmed by magnetic resonance imaging and postsurgical pathologic examination. All patients underwent complete synovectomy through posteromedial, posterolateral, anteromedial, and anterolateral portals. Each patient was evaluated before treatment and followed up for a minimum of 5 years (range: months) using the Lysholm score and International Knee Documentation Committee (IKDC) score. Both Lysholm score and IKDC scores were significantly improved in all study participants. No cases of clinical recurrence, infection, joint stiffness, or neurovascular lesions were observed. This study showed that an attentive arthroscopic synovectomy is a safer alternative with better clinical outcomes, with no clinical recurrences. Pigmented villonodular synovitis (PVNS) is a rare, benign, and proliferative synovial tissue disorder. 1 Annual incidence of the intra-articular type has been estimated to be 1.8 to 2.2 per 1 million population, and among intra-articular forms, diffuse type represents 23% of cases. 2 Jaffe et al were the first to describe histological characteristics of PVNS back in PVNS affects the synovial lining of joints, tendon, and bursas, which is further characterized by synovial proliferation and hemosiderin deposition that can lead to bone and soft tissue invasion. The etiology of PVNS remains controversial, but the possible mechanism is thought to be cytogenetic aberration with a neoplastic origin. 4 The gross pathologic appearance of diffuse PVNS (DPVNS) is mostly an infiltrative mass that completely invades and thickens the synovium. The morphology of synovial hypertrophy consists of an irregular papillary or villous projection. Granowitz et al distinguished two subtypes of PVNS considering the involvement of the synovial tissue: localized PVNS (LPVNS) and DPVNS that affected the entire synovium. 5 The received January 15, 2018 accepted after revision March 20, 2018 Copyright by Thieme Medical Publishers, Inc., 333 Seventh Avenue, New York, NY 10001, USA. Tel: +1(212) DOI /s ISSN

2 diffuse form is rare and typically occurs as a monoarticular disease, with theknee being the most commonly affected site. 6 The diffuse form is more aggressive, with a high risk of recurrence which may lead to erosive joint destruction and activity limitations. 7 Overall recurrence rates of 26.7 and 24.6%, overall complication rates of 5.7 and 3.2%, and overall osteoarthritis rates of 20 and 17.1% have been reported, respectively, following open and arthroscopic synovectomies. 8 Symptoms of pain and swelling tend to have an insidious onset and a slower progression rate. Clinical diagnosis is difficult, since symptoms are nonspecific and can include motion limitation, diffuse pain, edema, and a palpable mass. 9 PVNS is determined by clinical features, joint aspiration, and magnetic resonance imaging (MRI). Histologically, it is characterized by the presence of inflammation, multinucleate giant cells, hemosiderin deposition, and lipid-laden macrophages. 10 The definitive treatment is the complete resection of affected tissue by open or arthroscopic synovectomy, the therapy of choice for LPVNS is arthroscopic partial synovectomy with excision of the lesion. Total synovectomy, arthroscopically or through an open arthrotomy, is the recommended treatment for DPVNS. 11 Compared with open synovectomy, arthroscopic synovectomy could ensure better preservation of kneefunction while producing acceptable local recurrence rates and fewer complications. 12 Hemarthrosis, knee stiffness, and reduced range of motion are the most common complications which were reported in following an open synovectomy The purpose of this study was to evaluate and describe the clinical results and outcomes of complete arthroscopic synovectomy through the four arthroscopic portals in the knee affected by DPVNS. This study hypothesis was that this approach allows extensive arthroscopic synovectomy with acceptable recurrence rates and good functional outcome without the postoperative complications seen following open procedures. Patients and Methods Between June 2009 and August 2012, 21 patients (15 men and 6 women) with histologically confirmed DPVNS of the knee were enrolled in the study. In all cases, PVNS was preliminary diagnosed with the aid of MRI and further confirmed via postsurgical pathological evaluation of the shaved synovium (on thebasis of common histopathological characteristic of the localized nodular and diffuse varieties 17 ). In all cases, complete synovectomy was performed through the four arthroscopic portals: posteromedial, posterolateral, anteromedial, and anterolateral portals. Inclusion criteria were as follows: clinical examination, an MRI of the affected knee, followed by a postsurgical pathologic confirmation of DPVNS, and finally the lack of any major degenerative changes. Exclusion criteria were as follows: DPVNS with extra-articular extension into bone and soft tissue (open techniques are especially important in the presence of extra-articular extension of disease, which cannot be accessed with an arthroscope 18 ), patient with a history of recurrence, or evidence of PVNS in any other joints than the knee, and signs of focal PVNS. Fig. 1 Preoperative magnetic resonance imaging evaluation. Presenting symptoms were mainly spontaneous swelling, diffuse nonspecific knee pain, and a decreased range of motion. Anteroposterior, lateral, and flexion weight-bearing radiographs of the knee were performed to detect and exclude other pathologies such as osteoarthritis. All patients were subjected to an MRI scan for better evaluation ( Fig. 1). An arthroscopy procedure was performed in which all patients synovial biopsies were examined for pathological changes postsurgically, furthermore. None of the patients received radiofrequency ablation after the shaving technique or for hemostasis. Patients were followed up for a minimum of 5 years (range: months) in which postoperative complications, clinical recurrences, and treatment details were recorded. An independent examiner reviewed the patients records before interviewing and examining the patients at final follow-up. All cases were operated by the same consultant surgeon in which signed informed consents were collected from all participants. Surgical Technique Arthroscopy was performed in supine position under spinal anesthesia while a tourniquet was used high on the thigh. The knee was flexed 90 degrees by draping the leg over the edge of the operative table. Standard lateral and medial portals were made. The 30 degrees arthroscope was used. First of all, complete synovectomy of the anterior compartment (anteromedial, medial gutter, notch area, anterolateral, and lateral gutter) was done. Then, medial submeniscal area with valgus force and lateral submeniscal area with the figure of four positions were meticulously shaved and cleaned. The posterior compartment was approached using the two portal techniques ( Fig. 2). The suprapatellar pouch region was completely shaved and accessory superior portals were used

Fig. 2 Making the portals: (A) posteromedial portal and (B) posterolateral portal (right knee). if needed. A posterolateral portal was made by transillumination from notch area.

3 Fig. 2 Making the portals: (A) posteromedial portal and (B) posterolateral portal (right knee). if needed. A posterolateral portal was made by transillumination from notch area. After trocar passage from the septum and by using arthroscope in this portal and under direct vision, posteromedial portal was made. That safe zone for septum passage is mid-third. 19 Medial genicular artery is in the upper third of the septum and popliteal neurovascular bundle is in the proximity of the distal third. The posteromedial compartment synovectomy and middle third septum removal were done. Moving the arthroscope to the posteromedial portal, posterolateral synovectomy was done ( Figs ). The synovial was shaved till postcapsule, and it is our suggestion that less damage postcapsule causes less chance of extravasation and probably compartment syndrome. Arthroscopic electroablation was done just when an oozing vessel seen. In this study, synovectomy wasperformed using a motorized shaver with a 5.5-mm full-radius blade (Karl Storz; Miami, FL). An intra-articular suction drain was used for 24 hours. Sterile compression dressing was applied to prevent hemarthrosis. The synovial tissue was sent for histopathological examination. Suture removal was done on day 14. No radiation strategy (extra-articular or intra-articular) was done postoperatively. Patients were followed up at monthly intervals for 3 months and then 3 and 6 monthly. At every visit, patients were examined for effusion, pain, and range of motion. Recurrence was diagnosed on the basis of clinical exam. MRI screening for recurrence was performed for all patients 2 years later. Rehabilitation After drain removal, the patients began physical therapy that consisted of isometric quadriceps contraction and active range of motion exercises. Patients were allowed weight bearing as tolerated. Therapeutic goals were set to achieve maximum range of motion, restore quadriceps muscle strength, and decrease swelling and pain in the early postoperative period. Statistical Analysis Statistical analyses were performed with SPSS software version 21 (IBM; Armonk, NY). The frequency, mean, and standard deviation were used to data description. First, Kolmogorov Smirnov s test was used to evaluate the normal distribution of scores. According to small sample size, the results of this test showed that the scores do not follow a normal distribution (p < 0.05). Therefore, data were analyzed using the Wilcoxon s test for comparing the preoperative and postoperative Lysholm knee scores and International Knee Documentation Committee (IKDC) score. A p < 0.05 was considered statistically significant. Results The 21 patients consisted of 15 men and 6 women. The mean age of patients was 32 years (range: years) and the average body mass index was 29 kg/m 2 (range: kg/m 2 ). Patients were followed up for a minimum of 5 years (range: months) and each patient was evaluated before treatment and at thefinal follow-up visit. Mean timefrom symptom onset to surgery was 8 months (range: 3 21 months). One patient reported a history of trauma. There were no complications during or after the arthroscopic procedure. Two patients displayed evidence of local recurrence without clinical signs during the follow-up period.

4 Fig. 3 Anterior compartment viewing from anterolateral portal: (A) beforesynovectomyand(b) after synovectomy (left knee). The limitation of joint motion, Lysholm knee joint function score, and IKDC score were improved significantly postoperatively ( Table 1). No evidence of swelling, infection, joint stiffness, and neurovascular lesions were detected. Discussion DPVNS is an uncommon clinical diagnosis. Surgical excision is the preferred method of treatment in DPVNS. The goals of surgery are to restore joint function and prevent the destruction of articular cartilage by the diseased synovium. However, the long-term success of surgery depends on the ability to resect the synovium completely. Synovectomy may be performed with either an arthroscopic or open arthrotomy technique. Regardless of the approach, complete resection is required to reduce the likelihood of recurrence. Gu et al reported no difference in recurrence rate among patients who underwent arthroscopic versus open complete synovectomy. 20 In this study, complete arthroscopic synovectomy was performed for 21 patients via two anterior and two posterior portals; with a minimum follow-up time of 5 years. Our patients had the good functional outcome as evidenced by their postsurgical range of motion and no major complications or recurrences. Many authors suggest that recurrence is due to an incomplete resection of the pathological tissue, but this is not easy to achieve arthroscopically. The recurrence rate in this study is significantly better than the best results with the open surgical approach. The reported rate of recurrence after open synovectomy for DPNVS of the knee varied in the literature (25 46%), 21,22 and the lowest rate was obtained by Flandry et al 23 that reported 8%. In this study, the possibility of full access to the joint cavity was provided by anterior and posterior arthroscopic portals and posterior septum removal. Zvijac et al demonstrated that total arthroscopic synovectomy with the use of both anterior and posterior portals may have a similar if not better prognosis compared with open procedures. 24 Arthroscopic surgery offers the ability to resect diseased synovium with minimal loss of function and faster recovery. Complications such as pain and joint stiffness are frequently noted after open synovectomy. 21,23 In this study, no radiation was used with synovectomy to decline the recurrence rate. De Visser et al showed that the

5 Fig. 4 Posteromedial compartment viewing from posterolateral transseptal portal: (A) before synovectomy and (B) after synovectomy (left knee). Fig. 5 Posterolateral compartment from posterolateral portal: (A) beforesynovectomyand(b) after synovectomy (left knee).

6 Fig. 6 Septum shaving, viewing from posterolateral portal (left knee). Table 1 Preoperative and postoperative evaluations Preoperative Postoperative Difference Median difference p-value a ROM < Lysholm score < IKDC score < Abbreviations: IKDC, International Knee Documentation Committee; ROM, range of motion. a Wilcoxon s test. use of radiation with surgery, compared with the use of surgery alone did not improve patient outcome. 25 In this study, the minimum follow-up time was 5 years. Ushijima et al demonstrated that the interval between initial treatment and recurrence ranged from 2 to 36 months, with a mean of 24 months. 26 This study has several limitations worth noting. First, MRI screening for recurrence was not done for all patients, therefore may have underestimated the true number of recurrence and asymptomatic residual disease could be missed. Second, only a small number of patients with a relatively short follow-up were studied. Different outcomes may be observed with longer follow-up. This technique is clinically relevant in that a well-done complete arthroscopic synovectomy can be safely used for treatment of DPVNS with low complication and recurrence rate. Authors Contributions S.K.: Developing the original idea and the protocol, and definition of intellectual content; R.V. and J.H.A.: Study supervision; M.S. and S.M.K.: Contribution to the development of the protocol; M.S.: Preparation and drafting of the article. All authors read and approved the final article. Note This study was approved by the Akhtar Hospital Research Ethic Committee in Tehran, Iran (No. 9820). Informed consent was obtained from all individual participants included in the study. Funding None. Conflict of Interest None. Conclusion Arthroscopic treatment of DPVNS through the four arthroscopic portals (anterior and posterior) has fewer complications and relatively shorter postoperative recovery time. It could be used, safely and easily, while ensuring satisfactory control of diffuse PVNS with low recurrence rates. Intraarticular DPVNS of the knee could be treated with attentive arthroscopic synovectomy without requiring for postoperative chemoradiotherapy. References 1 Schiller AL. Tumors and tumor like lesions involving joints. In: Kelley WN, Harris ED, Ruddy S, Sledge CB, eds. Textbook of Rheumatology. Philadelphia: WB Saunders; 1989: Myers BW, Masi AT. Pigmented villonodular synovitis and tenosynovitis: a clinical epidemiologic study of 166 cases and literature review. Medicine (Baltimore) 1980;59(03): Jaffe HL, Lichtenstein L, Sutro CJ. Pigmented villonodular synovitis, bursitis, and tenosynovitis. Arch Pathol 1941;31:

7 4 Kanamori M, Matsushita I, Yasuda T, Kimura T. Cytogenetic findings in a case of pigmented villonodular synovitis and a review of the literature. Toyama Med J 2008;19(01): Granowitz SP, D Antonio J, Mankin HL. The pathogenesis and longterm end results of pigmented villonodular synovitis. Clin Orthop Relat Res 1976;(114): Chiari C, Pirich C, Brannath W, Kotz R, Trieb K. What affects the recurrence and clinical outcome of pigmented villonodular synovitis? Clin Orthop Relat Res 2006;450(450): Rauh PB, Bernard J, Craig DM. Pigmented villonodular synovitis of the knee: average five-year follow-up of arthroscopic treatment. J South Orthop Assoc 2002;11(02): Rodriguez-Merchan EC. Review article: open versus arthroscopic synovectomy for pigmented villonodular synovitis of the knee. J Orthop Surg (Hong Kong) 2014;22(03): Dines JS, DeBerardino TM, Wells JL, et al. Long-term follow-up of surgically treated localized pigmented villonodular synovitis of the knee. Arthroscopy 2007;23(09): Flandry F, Hughston JC, McCann SB, Kurtz DM. Diagnostic features of diffuse pigmented villonodular synovitis of the knee. Clin Orthop Relat Res 1994;(298): Kubat O, Mahnik A, Smoljanović T, Bojanić I. Arthroscopic treatment of localized and diffuse pigmented villonodular synovitis of the knee. Coll Antropol 2010;34(04): Akinci O, Akalin Y, İncesu M, Eren A. Long-term results of surgical treatment of pigmented villonodular synovitis of the knee. Acta Orthop Traumatol Turc 2011;45(03): Nakahara H, Matsuda S, Harimaya K, et al. Clinical results of open synovectomy for treatment of diffuse pigmented villonodular synovitis of the knee: case series and review of literature. Knee 2012;19(05): Aurégan JC, Klouche S, Bohu Y, Lefèvre N, Herman S, Hardy P. Treatment of pigmented villonodular synovitis of the knee. Arthroscopy 2014;30(10): Colman MW, Ye J, Weiss KR, Goodman MA, McGough RL III. Does combined open and arthroscopic synovectomy for diffuse PVNS of the knee improve recurrence rates? Clin Orthop Relat Res 2013; 471(03): Jabalameli M, Jamshidi K, Radi M, Hadi H, Bagherifard A. Surgical outcomes of 26 patients with pigmented villonodular synovitis (PVNS) of the knee at a mean follow-up of 4 years: introducing a novel technique. Med J Islam Repub Iran 2014;28: Rao AS, Vigorita VJ. Pigmented villonodular synovitis (giant-cell tumor of the tendon sheath and synovial membrane). A review of eighty-one cases. J Bone Joint Surg Am 1984;66(01): Chin KR, Barr SJ, Winalski C, Zurakowski D, Brick GW. Treatment of advanced primary and recurrent diffuse pigmented villonodular synovitis of the knee. J Bone Joint Surg Am 2002;84-A(12): Kim SJ, Song HT, Moon HK, Chun YM, Chang WH. The safe establishment of a transseptal portal in the posterior knee. Knee Surg Sports Traumatol Arthrosc 2011;19(08): Gu HF, Zhang SJ, Zhao C, Chen Y, Bi Q. A comparison of open and arthroscopic surgery for treatment of diffuse pigmented villonodular synovitis of the knee. Knee Surg Sports Traumatol Arthrosc 2014;22(11): Johansson JE, Ajjoub S, Coughlin LP, Wener JA, Cruess RL. Pigmented villonodular synovitis of joints. Clin Orthop Relat Res 1982;163(163): Schwartz HS, Unni KK, Pritchard DJ. Pigmented villonodular synovitis. A retrospective review of affected large joints. Clin Orthop Relat Res 1989;247(247): Flandry FC, Hughston JC, Jacobson KE, Barrack RL, McCann SB, Kurtz DM. Surgical treatment of diffuse pigmented villonodular synovitis of the knee. Clin Orthop Relat Res 1994;300(300): Zvijac JE, Lau AC, Hechtman KS, Uribe JW, Tjin-A-Tsoi EW. Arthroscopic treatment of pigmented villonodular synovitis of the knee. Arthroscopy 1999;15(06): de Visser E, Veth RP, Pruszczynski M, Wobbes T, Van de Putte LB. Diffuse and localized pigmented villonodular synovitis: evaluation of treatment of 38 patients. Arch Orthop Trauma Surg 1999; 119(7-8): Ushijima M, Hashimoto H, Tsuneyoshi M, Enjoji M. Giant cell tumor of the tendon sheath (nodular tenosynovitis). A study of 207 cases to compare the large joint group with the common digit group. Cancer 1986;57(04):

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