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J Orthop Sci (2010) 15:251–256

DOI 10.1007/s00776-009-1432-x

Case report

Mucoid degeneration of the anterior cruciate ligament associated with


subclinical instability in young patients
YASUO NIKI1, HIDEO MATSUMOTO2, HIROYUKI ENOMOTO1, YOSHIAKI TOYAMA1, and YASUNORI SUDA1
1
Department of Orthopaedic Surgery, Keio University, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan
2
Institute for Integrated Sports Medicine, Keio University, Tokyo, Japan

Introduction Case reports

Mucoid degeneration of the anterior cruciate ligament Case 1


(ACL) is an unusual entity that has been described only
since 1999.1 With increasing use of magnetic resonance A 23-year-old man presented with a 3-year history of
imaging (MRI) to evaluate the knee, a number of cases left knee pain that was exacerbated in terminal knee
with this entity have been accumulated to date,2–7 and extension and after long walks. There was no history of
characteristic features of MRI have been accepted as major knee trauma. The patient had visited his local
important diagnostic information. Past experience can physician several times who had failed to reach a defini-
be summarized as showing that the patients are middle- tive diagnosis. The patient was subsequently referred to
aged and they present with knee pain and limited our hospital for specialist treatment.
motion but without clinical ligament instability.2 On examination, no diagnostic instability was appar-
We report herein two cases of mucoid degeneration ent from the Lachman, pivot shift, and anterior/poste-
of the ACL in which the patients were in their twenties. rior drawer tests. Range of motion was 0°–130°, with
These cases differed substantially from previously pain on both terminal extension and flexion. MRI indi-
reported cases, not only in the much younger age but cated a bulky ACL with increased intraligamentous
also in that the patients exhibited substantial subclinical signal, particularly on T2-weighted and fat-suppressed
ACL insufficiency manifesting only in an anesthetized sequences (Fig. 1). Based on these characteristic MRI
condition. Despite their young age, both patients also findings, mucoid degeneration of the ACL was sus-
presented with intercondylar osteophyte, which was pected, and arthroscopic treatment was applied as
removed at the time of operation to avoid impingement reported previously.
of the ACL. The causal relation between minor ACL Under anesthesia, arthrometric examination using
insufficiency and such osteophyte formation warrants KT-2000 (MEDmetric, San Diego, CA, USA) revealed
further consideration. Although partial débridement of 3 mm of anterior displacement and 32 N/mm of ante-
degenerative ACL has been reported to alleviate knee rior terminal stiffness; but manual pivot shift testing
pain and is the common therapeutic strategy for this revealed no rotatory instability. On arthroscopic exami-
entity, patients should be carefully followed particularly nation, the posterolateral bundle (PLB) of the ACL was
for knee instability postoperatively owing to the inher- hypertrophic and bulbous, easily bulging into the lateral
ent loss of ACL integrity associated with mucoid wall of the notch in terminal extension (Fig. 2A). The
degeneration. PLB was arthroscopically removed; and the notch, nar-
rowed by osteophyte formation, was widened using a
motorized burr. Tendon fibers of the PLB were found
to be interspersed with a yellowish homogeneous mass.
Although the anteromedial bundle (AMB) of the
ACL remained completely intact, arthrometric analysis
revealed that anteroposterior (AP) instability had
increased by 1.5 mm postoperatively. Histological
Offprint requests to: Y. Niki examination revealed loose fibrocollagenous tendinous
Received: January 7, 2009 / Accepted: June 29, 2009 tissue containing fibrin deposits and myxedematous
252 Y. Niki et al.: Mucoid degeneration of the ACL

Fig. 1. Case 1. Coronal (A)


and sagittal (B) T2-weighted
and fat-suppressed imaging
shows increased intraliga-
mentous signal intensity of
the bulky anterior cruciate
ligament (ACL) with intact
ACL fibers

Fig. 2. Case 1. Arthroscopic view of the ACL shows a hyper- biopsy specimen from the ACL shows mucoid degeneration
trophic, bulbous posterolateral bundle (PLB) and the inter- of the ligament with loose fibrocollagenous tendinous tissue
condylar notch narrowed by osteophyte growing at the roof containing fibrin deposits and myxedematous changes (B)
and lateral wall of the notch (A). Histological evaluation of a

changes indicative of mucoid degeneration of the liga- flexion, squatting, and long walking. The patient played
ment (Fig. 2B). basketball at a recreational level. There was no history
As of 1 year postoperatively, the patient had gained of trauma or previous knee problems. The patient had
complete pain relief and full range of knee motion, with visited a local hospital, and an unusual intraligamentous
the Lysholm score improved from 69 to 95. MRI at this signal with intermediate intensity was identified on pro-
time revealed complete recovery of intensity in the ton-weighted imaging along with high-intensity signals
ACL on T2-weighted and fat-suppressed imaging, but on T2-weighted and fat-suppressed imaging (Fig. 4).
substantial attenuation of ACL fibers was apparent The patient was thus referred to our hospital for special-
(Fig. 3). AP instability measured using a KT-2000 ist treatment.
arthrometer revealed 4.5-mm side-to-side differences at On examination, no ligament instability was identi-
14 months postoperatively. fied by the Lachman, pivot shift, and anterior/posterior
drawer tests. Range of motion was 0°–120°, with pain at
maximum flexion and extension. Based on the char-
Case 2
acteristic MRI findings, mucoid degeneration of the
A 28-year-old woman presented with an 11-month ACL was suspected, and arthroscopic treatment was
history of left posterior knee pain during terminal knee performed.
Y. Niki et al.: Mucoid degeneration of the ACL 253

Fig. 3. Case 1. T2-weighted


imaging at 1 year postopera-
tively shows reduced signal
intensity of the ACL and
decreased fibers of low signal
with an intact ACL

Fig. 4. Case 2. Sagittal pro-


ton-weighted imaging shows
an enlarged ACL with inter-
mediate signal intensity (A).
T2-weighted and fat-sup-
pressed imaging shows an
enlarged ACL with entirely
increased signal intensity (B)

In the anesthetized condition, arthrometric examina- using semitendinosus tendon (Fig. 5B,C). In addition,
tion revealed 2.5 mm of anterior displacement and the notch osteophyte was removed to prevent impinge-
37 N/mm of anterior terminal stiffness, but rotatory ment of the reconstructed ACL. Histological examina-
instability was insignificant on pivot shift testing. On tion revealed fibrin deposits and myxedematous
arthroscopy, hypertrophic AMB of the ACL contacted changes, indicating mucoid degeneration of the
the lateral wall of the intercondylar notch, which was ligament.
narrowed by osteophyte formation (Fig. 5A). Only By 6 months postoperatively, the patient had com-
AMB was arthroscopically removed and reconstructed pletely recovered without any knee symptoms, with the
254 Y. Niki et al.: Mucoid degeneration of the ACL

Fig. 5. Case 2. Arthroscopic view of the ACL shows the phy shows the AMB fixed with an Endobutton (Smith &
anteromedial bundle (AMB) impinging on the lateral wall of Nephew, Memphis, TN, USA) and double spike plate (DSP)
the intercondylar notch (A). Only the AMB was reconstructed (Smith & Nephew) (C). Histology of the ACL shows myxede-
using double-looped semitendinosus tendon with the postero- matous changes throughout the ligament (D)
lateral bundle (PLB) preserved (B). Postoperative radiogra-

Lysholm score improved from 74 to 100. AP instability minal extension and flexion, with pain in extension
measured by arthrometry showed a 0.5-mm side-to-side dominant in patient 1 and pain in flexion dominant in
difference at 8 months postoperatively, at which time patient 2.
she had returned to playing basketball. To date, the mechanisms evoking knee pain associ-
ated with mucoid degeneration of the ACL remain a
matter of debate. According to previous studies,
Discussion increased volume of the ACL and resultant increases
in internal pressure within the notch have been con-
To the best of our knowledge, 13 cases of mucoid sidered a cause of knee pain during terminal flexion,3,8
degeneration of the ACL have been reported previ- whereas impingement of the ACL against the osteo-
ously.1–7 A review of these 13 cases indicates that mucoid phyte of the notch plays a role in knee pain during
degeneration of the ACL generally affects middle-aged terminal extension.9 At present, we think it more likely
patients with ages ranging from 35 to 60 years — in that increased tension on the degenerated ACL in ter-
contrast to the present two patients, who were in their minal flexion and extension result in knee pain as a
twenties. As in the previous 13 cases in the literature, previous biomechanical study using cadaveric knees
our two patients presented with knee pain during ter- revealed that the length of both the AMB and PLB
Y. Niki et al.: Mucoid degeneration of the ACL 255

was maximum with the knee in >0° of extension and some patients sustain ACL rupture subsequent to
in >140° of flexion,10 suggesting that in situ tension débridement of mucoid tissues.17,20 Actually, in patient
within the ACL is maximized with the knee in these 1, arthrometric AP instability was increased by 1.5 mm
positions. Increased tension in the degenerative ACL just after operation compared with preoperatively and
appears to be the only mechanism that would explain showed 4.5 mm of AP instability at 14 months postop-
knee pain in both terminal flexion and extension eratively; whereas in patient 2, instability was 0.5 mm at
simultaneously. 8 months postoperatively following successful ACL
Although the previous 13 cases in the literature were augmentation.
reportedly not associated with clinical instability of the In agreement with the past two reports, single bundle
ACL and exhibited negative results on Lachman testing, augmentation of either AMB or PLB should be consid-
the present two patients showed minor ACL insuffi- ered as a therapeutic option following débridement of
ciency and decreased terminal stiffness when measured the ACL, particularly when the patient is young and
in the anesthetized condition using a KT-2000 arthrom- participates in sport activities.2 Indeed, we performed
eter. An anesthetized condition might allow precise ACL augmentation in patient 2 for three reasons. First,
detection of the integrity of the ACL, not only as ante- as mucoid degenerative changes normally extend to
rior displacement but also as anterior terminal stiffness. both the AMB and PLB, as confirmed on MRI, the rest
Anterior terminal stiffness reportedly reflects mechani- of the bundle after débridement of either AMB or PLB
cal properties of the ACL11 and has been considered a is inevitably degenerative, and mechanical properties
useful indicator of ACL insufficiency.12,13 are possibly deteriorated. If the patient participates in
Several studies have suggested that minor instability sport activities, the remaining degenerative bundle may
of the knee in extension potentiates osteophyte forma- be easily loosened or ruptured, as seen in case 1. Second,
tion around the notch as a natural homeostatic mecha- most patients with mucoid degeneration of the ACL
nism to prevent instability during knee extension, show decreased activity and do not participate in any
independent of the presence of osteoarthritis (OA).14,15 sport activities due to knee pain, even though the
In the present two cases, a narrow notch associated with patients are young and previously participated in sport
an osteophyte impinging against the ACL was a common activities. This may explain why most patients with
pathological feature. Narvekar and Gajjar reported five mucoid degeneration do not complain of knee instabil-
similar cases of mucoid degeneration of the ACL, in ity. Young patients with higher levels of participation in
which notchplasty contributed to postoperative relief sport activities may experience ACL insufficiency after
from pain.3 We thus speculate that minor instability knee pain is relieved by débridement of the degenera-
resulting from mucoid degeneration of the ACL may tive ACL. Third, single bundle débridement followed
promote osteophyte formation in the roof and lateral by single bundle augmentation may offer substantial
wall of the notch, which in turn starts to impinge against advantages from the perspective of reduced surgical
the ACL and represents a potential cause of knee pain damage and rapid return to function compared with
in terminal extension. total ACL reconstruction. Current advances in single
In addition to the controversy of the mechanism bundle augmentation techniques may further encour-
underlying knee pain in mucoid degeneration of the age consideration of this procedure for young, active
ACL, the etiology of this entity remains contentious. patients.21
Several possible etiologies have been proposed to date, Although the pathogenesis and pain mechanisms in
including structural variations of the intercondylar mucoid degeneration of the ACL remain unclear, the
notch (e.g., notch wide index and notch area),16 micro- present two cases show that knee pain during terminal
trauma with cellular response leading to the release of extension and flexion was relieved after débridement of
a mucin substance and hyaluronic acid,17 herniation of either the AMB or PLB, as reported previously. Post-
synovial tissue,18 and displacement of synovial tissue operative arthrometric analysis revealed increased AP
during embryogenesis.6 An alternative theory has been instability in patient 1 and normal AP laxity in patient
postulated that mucoid degeneration and ganglion cyst 2. Whether single bundle augmentation is effective after
formation coexist on MRI and commonly represent partial débridement of the ACL remains unknown,
mucinous degenerative changes of connective tissue, however this procedure may offer a reasonable thera-
suggesting that similar mechanisms may contribute to peutic option, particularly in young, active patients.
the pathogenesis of both entities.19 However, as we described only two cases with a rela-
Regarding surgical treatment, partial resection of tively short follow-up period, larger numbers and longer
the ACL, as either the AMB or PLB, has been shown follow-up intervals are necessary to investigate pre-
to relieve pain without causing clinical instability cisely the clinical outcomes of ACL débridement with
postoperatively.3,4 However, two case reports have rec- or without augmentation of the ACL in patients with
ommended ACL augmentation or reconstruction, as mucoid degeneration of the ACL.
256 Y. Niki et al.: Mucoid degeneration of the ACL

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