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Pigmented Villonodular Synovitis: Extrasynovial Recurrence
Case Report With Video Illustration
Pigmented Villonodular Synovitis: Extrasynovial Recurrence Christopher M. Jobe, M.D., Anwar Raza, M.D., and Lee Zuckerman, M.D.
Abstract: A 32-year-old female athlete underwent arthroscopy for a second recurrence of pigmented villonodular synovitis (PVNS), which was extrasynovial, seen on magnetic resonance imaging. It was noted on arthroscopy that (1) the nodules moved medially with joint insufflation, (2) the nodules were less prominent than on magnetic resonance imaging, and (3) more than 95% of the recurrent tumor was hidden by neosynovium. We believe that the extrasynovial location is because of the more rapid proliferation of the neosynovium relative to the growth of the remaining tumor cells after the previous resection. In resecting pigmented villonodular synovitis with a high risk of recurrence, a layer of periarticular fat should be removed and the surgeon should be wary of change in position with insufflation.
P
igmented villonodular synovitis (PVNS) is a benign but often locally aggressive process of uncertain etiology. It has a high rate of local recurrence, especially the diffuse type as opposed to the localized type. Surgical treatment has concentrated on total excision of all detectable disease. Whereas disagreement has centered on the technique by which this can best be accomplished, arthroscopic versus open synovectomy, there is general agreement on the surgical goal.1-5 A recent case of recurrent PVNS presented findings, in the mode of recurrence, that have implications for the performance of both primary and repeat synovectomies, be they open or arthroscopic.
From the Departments of Orthopaedic Surgery (C.M.J., L.Z.) and Pathology (A.R.), School of Medicine, Loma Linda University, Loma Linda, California, U.S.A. Address correspondence to Christopher M. Jobe, M.D., Department of Orthopaedic Surgery, School of Medicine, Loma Linda University, 11406 Loma Linda Dr, Ste 218, Loma Linda, CA 92354, U.S.A. E-mail: [email protected] © 2011 by the Arthroscopy Association of North America 0749-8063/11322/$36.00 doi:10.1016/j.arthro.2011.06.023 Note: To access the video accompanying this report, visit the October issue of Arthroscopy at www.arthroscopyjournal.org.
CASE REPORT A 32-year-old woman, an avid jogger, underwent arthroscopic synovectomy for a second recurrence of PVNS of the right knee. The original resection was performed 3 years before the current surgery. The first diagnosis had been suggested on magnetic resonance imaging (MRI), which was performed because of mechanical symptoms while jogging and recurrent effusion.6,7 One week after the index MRI study, the patient underwent the original surgery, an extensive (8-portal) arthroscopic synovectomy. Early results were good, and the findings on a follow-up MRI study performed 9 months after surgery were considered normal. Nine months after the follow-up MRI study, she had some milder but suggestive symptoms that led to a second follow-up MRI study that showed the first recurrence. Two months later, she underwent a repeat arthroscopic synovectomy. In this second arthroscopy the disease was found to be much less extensive, and a less extensive synovectomy was performed. Symptoms recurred again about 8 months after the second synovectomy, and MRI confirmed recurrent PVNS. In this MRI study, one of the more striking features was the nodular disease seen in the suprapatellar pouch (Fig 1). This was particularly notable because at arthroscopy, the findings on insufflation of the joint were rather mild in terms of intra-articular
Arthroscopy: The Journal of Arthroscopic and Related Surgery, Vol 27, No 10 (October), 2011: pp 1449-1451
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FIGURE 1. MRI scan taken just before latest synovectomy. The 3 nodules in the suprapatellar pouch should be noted (arrows). There is a thin white line between the nodules and the arrows. This is the joint space. The thicker white line on the lateral (left) side is also joint space and allowed the nodules to withdraw medially with insufflation.
disease (Fig 2). In addition, there were no nodules in the anatomic positions suggested by the MRI study (Video 1, available at www.arthroscopyjournal.org).
FIGURE 3. (A) Disease unroofed within fat pad on anterior surface of femur. The underlying bone was unaffected. (B) Three suprapatellar nodules unroofed (arrows). These are compatible with those on the MRI scan in Fig 1. It should be noted how insufflation of the joint has moved them medially from where they were seen on MRI. Moreover, they recurred within fat.
FIGURE 2. Initial arthroscopic view of suprapatellar pouch with only 1 small bit of PVNS visible within joint. This image should be compared with Fig 1. The small arrow points to the small area of recurrence. The arrowhead points to a larger extrasynovial nodule that was moved medially by the arthroscopy fluid.
After more aggressive probing, we found that when we removed neosynovium from this interior surface of the suprapatellar pouch and from the anterior surface of the femur, we unroofed nodules of disease (Fig 3). We therefore performed a more extensive arthroscopic synovectomy, removing additional periarticular fat and areolar tissue in addition to the visible disease. Histologic analysis confirmed that this was again PVNS. The patient is currently asymptomatic 6 months after surgery and has taken up distance running again.
PIGMENTED VILLONODULAR SYNOVITIS DISCUSSION The natural history of the latest recurrence would appear to be as follows: During the next-to-last surgery, all visible disease was removed. Microscopic rests of PVNS, invisible with the arthroscope and not palpable, remained. Generation of neosynovium is a short-term process, 4 weeks, whereas the recurrence of PVNS is slower, a matter of months.8 The recurrent PVNS in this case was then intra-articular but primarily extrasynovial. Thus it would appear that in a patient with aggressive disease such as that in our patient, there are microscopic extensions into fat and areolar tissue. Bone and muscle seem to be more effective barriers in this case of early recurrence, although this disease can and will erode bone through pressure and penetrate fascia in long-term cases. In addition, in our case the PVNS seemed to excite in several areas very little fibrous reaction, which would make small rests less detectable by vision or palpation (Fig 4).
FIGURE 4. A thin layer of neosynovium (A) appears on the left, overlying fat (B), which in turn is overlying PVNS (C). One should note the lack of fibrous reaction to the nodule, which would have made the nodule more palpable and visible. (H&E stain and original magnification ⫻40.)
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The implications of this case for the open as well as the arthroscopic surgeon are as follows: (1) if there is an indication of an aggressive tendency such as diffuse type, recurrence, or aggressive histology, the surgeon should consider excising some otherwise normal-appearing fat and areolar tissue with the synovium; and (2) recurrent PVNS may be extrasynovial and move with joint insufflation from the position where it was seen on imaging studies.
CONCLUSIONS Recurrence of PVNS may be due to undetectable rests of tissue left in periarticular fat. Surgeons should consider removing a layer of periarticular fat and be aware that recurrences may be extrasynovial and change position with insufflation.
REFERENCES 1. Ottaviani S, Ayral X, Dougados M, Gossec L. Pigmented villonodular synovitis: A retrospective single-center study of 122 cases and review of the literature. Semin Arthritis Rheum 2011; 40:539-546. 2. Dines JS, DeBerardino TM, Wells JL, et al. Long-term follow-up of surgically treated localized pigmented villonodular synovitis of the knee. Arthroscopy 2007;23:930-937. 3. De Ponti A, Sansone V, Malcherè M. Result of arthroscopic treatment of pigmented villonodular synovitis of the knee. Arthroscopy 2003;19:602-607. 4. Sharma V, Cheng EY. Outcomes after excision of pigmented villonodular synovitis of the knee. Clin Orthop Relat Res 2009; 467:2852-2858. 5. Kramer DE, Frassica FJ, Frassica DA, Cosgarea AJ. Pigmented villonodular synovitis of the knee: Diagnosis and treatment. J Knee Surg 2009;22:243-254. 6. Frick MA, Wenger DE, Adkins M. MR imaging of synovial disorders of the knee: An update. Radiol Clin North Am 2007; 45:1017-1031. 7. Wan JM, Magarelli N, Peh WC. Imaging of giant cell tumour of the tendon sheath. Radiol Med 2010;115:141-151. 8. Martini AK. Animal experiment studies on the topic of regeneration of the synovial membrane following synovectomy of flexor tendons in the so-called no man’s land. Handchir Mikrochir Plast Chir 1986;18:199-203 (in German).
Pigmented Villonodular Synovitis: Extrasynovial Recurrence Christopher M. Jobe, M.D., Anwar Raza, M.D., and Lee Zuckerman, M.D.
Abstract: A 32-year-old female athlete underwent arthroscopy for a second recurrence of pigmented villonodular synovitis (PVNS), which was extrasynovial, seen on magnetic resonance imaging. It was noted on arthroscopy that (1) the nodules moved medially with joint insufflation, (2) the nodules were less prominent than on magnetic resonance imaging, and (3) more than 95% of the recurrent tumor was hidden by neosynovium. We believe that the extrasynovial location is because of the more rapid proliferation of the neosynovium relative to the growth of the remaining tumor cells after the previous resection. In resecting pigmented villonodular synovitis with a high risk of recurrence, a layer of periarticular fat should be removed and the surgeon should be wary of change in position with insufflation.
P
igmented villonodular synovitis (PVNS) is a benign but often locally aggressive process of uncertain etiology. It has a high rate of local recurrence, especially the diffuse type as opposed to the localized type. Surgical treatment has concentrated on total excision of all detectable disease. Whereas disagreement has centered on the technique by which this can best be accomplished, arthroscopic versus open synovectomy, there is general agreement on the surgical goal.1-5 A recent case of recurrent PVNS presented findings, in the mode of recurrence, that have implications for the performance of both primary and repeat synovectomies, be they open or arthroscopic.
From the Departments of Orthopaedic Surgery (C.M.J., L.Z.) and Pathology (A.R.), School of Medicine, Loma Linda University, Loma Linda, California, U.S.A. Address correspondence to Christopher M. Jobe, M.D., Department of Orthopaedic Surgery, School of Medicine, Loma Linda University, 11406 Loma Linda Dr, Ste 218, Loma Linda, CA 92354, U.S.A. E-mail: [email protected] © 2011 by the Arthroscopy Association of North America 0749-8063/11322/$36.00 doi:10.1016/j.arthro.2011.06.023 Note: To access the video accompanying this report, visit the October issue of Arthroscopy at www.arthroscopyjournal.org.
CASE REPORT A 32-year-old woman, an avid jogger, underwent arthroscopic synovectomy for a second recurrence of PVNS of the right knee. The original resection was performed 3 years before the current surgery. The first diagnosis had been suggested on magnetic resonance imaging (MRI), which was performed because of mechanical symptoms while jogging and recurrent effusion.6,7 One week after the index MRI study, the patient underwent the original surgery, an extensive (8-portal) arthroscopic synovectomy. Early results were good, and the findings on a follow-up MRI study performed 9 months after surgery were considered normal. Nine months after the follow-up MRI study, she had some milder but suggestive symptoms that led to a second follow-up MRI study that showed the first recurrence. Two months later, she underwent a repeat arthroscopic synovectomy. In this second arthroscopy the disease was found to be much less extensive, and a less extensive synovectomy was performed. Symptoms recurred again about 8 months after the second synovectomy, and MRI confirmed recurrent PVNS. In this MRI study, one of the more striking features was the nodular disease seen in the suprapatellar pouch (Fig 1). This was particularly notable because at arthroscopy, the findings on insufflation of the joint were rather mild in terms of intra-articular
Arthroscopy: The Journal of Arthroscopic and Related Surgery, Vol 27, No 10 (October), 2011: pp 1449-1451
1449
1450
C. M. JOBE ET AL.
FIGURE 1. MRI scan taken just before latest synovectomy. The 3 nodules in the suprapatellar pouch should be noted (arrows). There is a thin white line between the nodules and the arrows. This is the joint space. The thicker white line on the lateral (left) side is also joint space and allowed the nodules to withdraw medially with insufflation.
disease (Fig 2). In addition, there were no nodules in the anatomic positions suggested by the MRI study (Video 1, available at www.arthroscopyjournal.org).
FIGURE 3. (A) Disease unroofed within fat pad on anterior surface of femur. The underlying bone was unaffected. (B) Three suprapatellar nodules unroofed (arrows). These are compatible with those on the MRI scan in Fig 1. It should be noted how insufflation of the joint has moved them medially from where they were seen on MRI. Moreover, they recurred within fat.
FIGURE 2. Initial arthroscopic view of suprapatellar pouch with only 1 small bit of PVNS visible within joint. This image should be compared with Fig 1. The small arrow points to the small area of recurrence. The arrowhead points to a larger extrasynovial nodule that was moved medially by the arthroscopy fluid.
After more aggressive probing, we found that when we removed neosynovium from this interior surface of the suprapatellar pouch and from the anterior surface of the femur, we unroofed nodules of disease (Fig 3). We therefore performed a more extensive arthroscopic synovectomy, removing additional periarticular fat and areolar tissue in addition to the visible disease. Histologic analysis confirmed that this was again PVNS. The patient is currently asymptomatic 6 months after surgery and has taken up distance running again.
PIGMENTED VILLONODULAR SYNOVITIS DISCUSSION The natural history of the latest recurrence would appear to be as follows: During the next-to-last surgery, all visible disease was removed. Microscopic rests of PVNS, invisible with the arthroscope and not palpable, remained. Generation of neosynovium is a short-term process, 4 weeks, whereas the recurrence of PVNS is slower, a matter of months.8 The recurrent PVNS in this case was then intra-articular but primarily extrasynovial. Thus it would appear that in a patient with aggressive disease such as that in our patient, there are microscopic extensions into fat and areolar tissue. Bone and muscle seem to be more effective barriers in this case of early recurrence, although this disease can and will erode bone through pressure and penetrate fascia in long-term cases. In addition, in our case the PVNS seemed to excite in several areas very little fibrous reaction, which would make small rests less detectable by vision or palpation (Fig 4).
FIGURE 4. A thin layer of neosynovium (A) appears on the left, overlying fat (B), which in turn is overlying PVNS (C). One should note the lack of fibrous reaction to the nodule, which would have made the nodule more palpable and visible. (H&E stain and original magnification ⫻40.)
1451
The implications of this case for the open as well as the arthroscopic surgeon are as follows: (1) if there is an indication of an aggressive tendency such as diffuse type, recurrence, or aggressive histology, the surgeon should consider excising some otherwise normal-appearing fat and areolar tissue with the synovium; and (2) recurrent PVNS may be extrasynovial and move with joint insufflation from the position where it was seen on imaging studies.
CONCLUSIONS Recurrence of PVNS may be due to undetectable rests of tissue left in periarticular fat. Surgeons should consider removing a layer of periarticular fat and be aware that recurrences may be extrasynovial and change position with insufflation.
REFERENCES 1. Ottaviani S, Ayral X, Dougados M, Gossec L. Pigmented villonodular synovitis: A retrospective single-center study of 122 cases and review of the literature. Semin Arthritis Rheum 2011; 40:539-546. 2. Dines JS, DeBerardino TM, Wells JL, et al. Long-term follow-up of surgically treated localized pigmented villonodular synovitis of the knee. Arthroscopy 2007;23:930-937. 3. De Ponti A, Sansone V, Malcherè M. Result of arthroscopic treatment of pigmented villonodular synovitis of the knee. Arthroscopy 2003;19:602-607. 4. Sharma V, Cheng EY. Outcomes after excision of pigmented villonodular synovitis of the knee. Clin Orthop Relat Res 2009; 467:2852-2858. 5. Kramer DE, Frassica FJ, Frassica DA, Cosgarea AJ. Pigmented villonodular synovitis of the knee: Diagnosis and treatment. J Knee Surg 2009;22:243-254. 6. Frick MA, Wenger DE, Adkins M. MR imaging of synovial disorders of the knee: An update. Radiol Clin North Am 2007; 45:1017-1031. 7. Wan JM, Magarelli N, Peh WC. Imaging of giant cell tumour of the tendon sheath. Radiol Med 2010;115:141-151. 8. Martini AK. Animal experiment studies on the topic of regeneration of the synovial membrane following synovectomy of flexor tendons in the so-called no man’s land. Handchir Mikrochir Plast Chir 1986;18:199-203 (in German).