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Recurrent synovial chondromatosis of the knee after radical synovectomy and arthrodesis

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We describe a case of highly refractory synovial chondromatosis, which recurred despite four arthroscopic synovectomies, a chemical synovectomy, two open synovectomies and an arthrodesis. A review of the literature revealed one similar case. Both presented with marked joint stiffness suggesting a poor prognosis. Although arthrodesis may relieve short-term symptoms it does not prevent further recurrence of disease.
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VOL. 88-B, No. 5, MAY 2006 673
CASE REPORT
Recurrent synovial chondromatosis of the
knee after radical synovectomy and
arthrodesis
J. S. Church,
W. H. Breidahl,
G. C. Janes
From Perth
Orthopaedic and
Sports Medicine
Centre, Perth,
Western Australia
J. S. Church, BSc, FRCS(Tr &
Orth), Orthopaedic Fellow
17 Inman Road, London SW18
3BB, UK.
W. H. Breidahl, MRCP,
FRANZCR, Radiologist
Perth Radiology Clinic, 127
Hamersley Road, Subiaco 6008,
Western Australia, Australia.
G. C. Janes, FRACS(Orth),
Orthopaedic Surgeon
Perth Orthopaedic & Sports
Medicine Centre, 31 Outram
Street, West Perth 6005,
Western Australia, Australia.
Correspondence should be sent
to Mr J. S. Church; e-mail:
j.s.church@btinternet.com
©2006 British Editorial Society
of Bone and Joint Surgery
doi:10.1302/0301-620X.88B5.
17579 $2.00
J Bone Joint Surg [Br]
2006;88-B:673-5.
Received 22 December 2005;
Accepted 18 January 2006
We describe a case of highly refractory synovial chondromatosis, which recurred despite
four arthroscopic synovectomies, a chemical synovectomy, two open synovectomies and
an arthrodesis. A review of the literature revealed one similar case. Both presented with
marked joint stiffness suggesting a poor prognosis. Although arthrodesis may relieve
short-term symptoms it does not prevent further recurrence of disease.
Case report
In January 1996, a 24-year-old female teacher
and national-standard hockey player was seen
with a three-month history of progressive loss
of movement and locking of the right knee. On
examination, she had a diffusely swollen joint
with a fixed-flexion deformity of 45˚ and flex-
ion to 90˚. She underwent an arthroscopy at
her local hospital during which multiple chon-
dromata were removed. Histology of these
chondromata revealed variably sized nodules
of hyaline cartilage, some which which were
undergoing dystrophic calcification. Synovial
chondromatosis was diagnosed. Following
arthroscopy and two subsequent injections of
cortisone, the patient’s range of movement
improved and she was able to return to playing
hockey. However, within three months, her
symptoms returned with associated pain and
swelling. This persisted despite an attempted
chemical synovectomy with a course of yttrium
injections and two further arthroscopic debride-
ments. By November 1997, she had a fixed-
flexion deformity of 40˚ with flexion to only
50˚. An MRI scan revealed extensive synovial
fibrosis with a loose body, 4 cm x 4 cm, in the
posterior compartment of the knee (Fig. 1).
She was referred to the regional tertiary cen-
tre, where it was decided to perform an open
synovectomy and removal of loose bodies. His-
tology, following the procedure, confirmed the
diagnosis of recurrent synovial chondroma-
tosis. Following this, however, her pain and
reduced movement persisted and, in April
1998, she underwent a radical open synovec-
tomy. This included the removal of both the
menisci and cruciates, which improved her
pain with a range of movement from 10˚ to
Fig. 1
Sagittal proton density image through the
intercondylar region of the right knee. A
large, low-signal-intensity mass lies poste-
rior to the anterior cruciate ligament (arrow).
674 J. S. CHURCH, W. H. BREIDAHL, G. C. JANES
THE JOURNAL OF BONE AND JOINT SURGERY
100˚ of flexion. Unfortunately, the improvement was brief.
Over the following year she underwent a further arthro-
scopic debridement, daily continuous passive movement
and correction of the fixed-flexion deformity with a circular
frame. Despite this, she continued to experience constant
pain in the knee and a limited range of movement from 0˚
to 10˚ of flexion. An MRI scan showed significant intra-
articular fibrosis but no evidence of recurrence (Fig. 2).
In April 2001, arthrodesis of the knee was successfully
undertaken using a circular frame. The pain resolved and
she was able to return to an active lifestyle, which included
participating in triathlons. In April 2005, however, she pre-
sented with a nine-month history of progressive night pain,
which had not responded to anti-inflammatory medication.
A further MRI scan revealed a solid fusion, but with multi-
ple foci of nodular synovial proliferation in the medial and
lateral gutters, intercondylar notch and along the popliteus
tendon, consistent with a diagnosis of recurrent synovial
chondromatosis (Fig. 3).
Discussion
Synovial chondromatosis is a rare, benign condition charac-
terised by cartilaginous metaplasia of the intimal layer of
the synovial membrane of joints, tendons and bursae. Jaffe
described the condition in 1958,
1
and the knee is the most
commonly affected joint.
1-4
The early literature focused on
the primary management of this condition. Murphy et al
2
described 32 patients treated at the Mayo clinic with open
removal of loose bodies and affected synovium. There was
only one recurrence. Jeffreys
3
reviewed 17 cases treated sur-
gically, with no recurrences. His paper concluded that
removal of the loose bodies alone was preferable to syn-
ovectomy, because there was less post-operative stiffness
Fig. 2
Sagittal proton density image through the intercondylar region of the
right knee. There is extensive fibrosis in the intercondylar region and the
fat pad. Patella baja has developed.
Fig. 3a
Fig. 3b
MRI scans performed in April 2005, demonstrating, a) sagittal T2-
weighted fat-suppressed image after arthrodesis. The recurrent synovial
chondromatosis appears as an area of relatively high signal intensity pos-
teriorly (arrow) and, b) coronal T2-weighted fat-suppressed image dem-
onstrating no residual joint space.
RECURRENT SYNOVIAL CHONDROMATOSIS OF THE KNEE AFTER RADICAL SYNOVECTOMY AND ARTHRODESIS 675
VOL. 88-B, No. 5, MAY 2006
and complete synovectomy was ‘impracticable’. In 1989,
Coolican and Dandy
1
reported that arthroscopic removal
of loose bodies and the affected synovium achieved better
results than open procedures, and in the same year Dorf-
mann et al
5
concluded that arthroscopic removal of the
loose bodies alone gave satisfactory results. This was dis-
puted by Ogilvie-Harris and Saleh
6
in 1994, who found a
significant difference between the rates of recurrence after
arthroscopic removal of the loose bodies and arthroscopic
synovectomy. All recurrences were subsequently treated
successfully with an arthroscopic synovectomy.
One can, therefore, conclude that recurrence of synovial
chondromatosis is rare, particularly after synovectomy. The
majority of the reported recurrences have responded well to
a repeat procedure. We could only find one report of a
highly refractory synovial chondromatosis similar to our
case. This was the one failure reported by Coolican and
Dandy
1
in which the patient presented with a range of
movement from 45˚ to 90˚ of flexion. After three arthro-
scopic synovectomies and removal of loose bodies, an
arthrotomy and an open synovectomy, the patient still com-
plained of severe pain and stiffness. A manipulation under
anaesthetic was performed which resulted in a fracture of
the femur. When this had united, the patient was left with
20˚ of movement in the knee and an arthrodesis was
offered. No further information was available as to the
eventual outcome of this patient.
The primary complaint in both our case and that of
Coolican and Dandy
1
was of significant stiffness, both hav-
ing a range of movement from 45˚ to 90˚ of flexion at pre-
sentation. Synovial chondromatosis most commonly pre-
sents with pain and swelling in the affected joint.
1,3
Stiffness was reported as a secondary symptom in only two
of eight patients of Jeffreys,
3
all of whom complained pri-
marily of pain and swelling, and loss of extension was
noted in five of 18 knees by Coolican and Dandy.
1
In their
case of recurrence and in ours, the stiffness was severe and
was the primary complaint, suggesting a poor prognosis.
The stiffness in our case could be caused by a thickened,
metaplastic synovium coupled with the presence of multiple
loose bodies within the knee.
Arthrodesis has previously been reported to be a success-
ful salvage procedure for recurrent synovial chondromato-
sis. Harvey and Negrine
7
carried out an arthrodesis of the
distal interphalangeal joint of the middle finger and reported
no recurrence after three years. Hocking and Negrine
4
sub-
sequently described the successful outcomes of two arthro-
deses performed for synovial chondromatosis of the
subtalar joint at five years. To the best of our knowledge,
ours is the first outcome report of arthrodesis for refractory
synovial chondromatosis of the knee and is the first re-
ported case of recurrence in any joint after arthrodesis. Al-
though the arthrodesis initially provided significant pain
relief and functional improvement, it did not prevent recur-
rence of the synovial chondromatosis.
No benefits in any form have been received or will be received from a commer-
cial party related directly or indirectly to the subject of this article.
References
1. Coolican MR, Dandy DJ.
Arthroscopic management of synovial chondromatosis of
the knee: findings and results in 18 cases.
J Bone Joint Surg [Br]
1989;71-B:498-500.
2. Murphy FP, Dahlin DC, Sullivan CR.
Articular synovial chondromatosis.
J Bone
Joint Surg [Am]
1962;44-A:77-86.
3. Jeffreys TE.
Synovial chondromatosis.
J Bone Joint Surg [Br]
1967;49-B:530-4.
4. Hocking R, Negrine J.
Primary synovial chondromatosis of the subtalar joint affect-
ing two brothers.
Foot Ankle Int
2003;24:865-7.
5. Dorfmann H, De Bie B, Bonvarlet JP, Boyer T.
Arthroscopic treatment of synovial
chondromatosis of the knee.
Arthoscopy
1989;5:48-51.
6. Ogilvie-Harris DJ, Saleh K.
Generalized synovial chondromatosis of the knee: a
comparison of removal of the loose bodies alone with arthroscopic synovectomy.
Arthroscopy
1994;10:166-70.
7. Harvey FJ, Negrine J.
Synovial chondromatosis in the distal interphalangeal joint.
J Hand Surg [Am]
1990;15:102-5.
... Complete excision of the lesion is the standard treatment for synovial chondromatosis, however arthrodesis seems to be the definitive treatment for recurrent synovial chondromatosis [14]. One case report showed a similar treatment strategy where after removal of all loose bodies, a subtalar arthrodesis was done. ...
... 14 Owing to difficulty in accessing the posterior compartment arthroscopically, open approaches have traditionally been used to excise loose bodies of the posterior compartment. 15,16 For arthroscopic treatment of posterior disease, the use of accessory posterior portals and a trans-septal technique has been described to provide adequate surgical access while protecting the popliteal neurovascular bundle. 6 Release of the posterior capsule and knee flexion greater than 90 are required to protect the vessels. ...
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Synovial chondromatosis is a benign metaplastic disease of the synovial joints, characterized by the development of cartilaginous nodules in the synovium. Treatment generally includes open or arthroscopic loose body removal combined with a synovectomy. An all-arthroscopic approach has been described to minimize complications and reduce morbidity while providing adequate control of local disease. The purpose of this Technical Note is to describe our techniques and technical pearls that allow for adequate excision of disease while minimizing complications and disease recurrence. The combination of patient positioning, the establishment of multiple arthroscopic portals to ensure optimal visualization and freedom of instruments, the use of a leg holder, and the use of a variety of surgical instruments to facilitate loose body removal and synovectomy is critical to optimize clinical outcomes and minimize complications. Although technically demanding, our described technique can help facilitate extensive loose body removal and complete synovectomy.
... The definitive treatment for recurrences remains arthrodesis. Despite this, Church et al. have reported an isolated case where recurrence despite arthrodesis is still possible [16]. ...
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... 11 No obstante, se han publicado reportes que informan recidivas luego de la sinovectomía artroscópica inicial, la sinovectomía química, la sinovectomía abierta e incluso posartrodesis de rodilla. 12 No hallamos comunicaciones sobre la recidiva tras la resección amplia en el tobillo o el pie. ...
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A case of synovial chondromatosis of the distal interphalangeal joint of the long finger with an 8-year follow-up is presented. The lesion recurred 4 years after simple synovectomy, requiring a second operation with arthrodesis.
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Between 1971 and 1987, arthroscopy was performed in 39 patients with synovial chondromatosis of the knee; 29 of these patients (32 knees) were followed an average of 3.5 years. A good result was obtained in 78% of the cases. Removal of loose bodies was the only treatment in 31 of the 32 knees. A synovectomy was performed in one case. No synovectomies were performed secondarily. Only three patients required a second arthroscopic procedure. The essential prognostic factor for a good functional result is the condition of the femorotibial cartilage. We concluded that simple arthroscopic removal of cartilaginous bodies without synovectomy is the treatment of choice for synovial chondromatosis of the knee.
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We report the results of arthroscopic removal of loose bodies and abnormal synovium from 18 knees with primary synovial chondromatosis. After a mean of three years, six months (range one to 10 years), 14 knees were either symptom-free or had only minor symptoms. Three of these had required two arthroscopic operations. Three patients were improved but not cured and there was one failure. The results were better than the published results of open operation for this condition. Three patterns of macroscopic appearances were noted: four knees had large lesions covered by normal synovium, 10 had small fragments of cartilage lying in or on the synovium and four had only free fragments of cartilage in the joint cavity but none in, on, or under, the synovium. These three appearances may represent three different disease processes.
Article
1. Nineteen patients with articular synovial chondromatosis are reviewed. 2. The etiology, diagnosis and management of the condition are discussed. 3. It is concluded that extensive synovectomy is not justified; simple removal of loose bodies is the treatment of choice.
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Thirteen patients with generalized synovial chondromatosis of the knee were treated by either removal of the loose bodies alone (n = 5) or arthroscopic synovectomy (n = 8). The average follow-up was 38 months (range 23-61). There were three recurrences in the loose body removal group, which were subsequently treated by arthroscopic synovectomy. Statistical analysis of the results shows a significant improvement in pain, synovitis and effusion, range of movement, and function after either treatment. The group treated by arthroscopic synovectomy had significantly lower recurrence rates (p = 0.02). We recommend arthroscopic synovectomy for patients with generalized synovial chondromatosis. However, if loose bodies alone are removed, a recurrence can be successfully treated by an arthroscopic synovectomy.
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Primary synovial chondromatosis is a rare benign condition characterized by the formation of multiple cartilaginous nodules in the synovium of joints and on occasions tendon sheaths or bursae. A case of primary synovial chondromatosis affecting the subtalar joint is reported. The patient's brother developed the same condition affecting the same joint 2 years later. The proposed etiologies are discussed including the presence of the proto-oncogene C-erb B-2.