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Title: Pigmented Villonodular Synovitis of 1st
metatarsophalangeal joint: a case report
Author: Chaitanya Dev Pannu Vivek Morey B. PrashantShishir Rastogi
PII: S0958-2592(14)00053-4DOI: http://dx.doi.org/doi:10.1016/j.foot.2014.05.001Reference: YFOOT 1322
To appear in: The Foot
Received date: 28-3-2014Revised date: 5-5-2014Accepted date: 8-5-2014
Please cite this article as: Pannu CD, Morey V, Prashant B, Rastogi S, PigmentedVillonodular Synovitis of 1st metatarsophalangeal joint: a case report, The Foot (2014),http://dx.doi.org/10.1016/j.foot.2014.05.001
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Pigmented Villonodular Synovitis of 1st metatarsophalangeal
joint: a case report
Chaitanya Dev Pannu2, Vivek Morey2, Prashant B2, Shishir
Rastogi1 1Professor and unit head, Department of Orthopedics, All India
institute of Medical sciences, New Delhi 2 M.S, Senior Resident, Department of Orthopedics, All India
institute of Medical sciences, New Delhi
Corresponding Author: Chaitanya Dev Pannu, Senior Resident,
Department of Orthopedics, All India institute of Medical sciences,
New Delhi. Email id; cdpannu@gmail.com Ph. No:-
+919968037279
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Pigmented Villonodular Synovitis of 1st Metatarsophalangeal
joint: a case report and literature review
Abstract: Pigmented villonodular synovitis is a common disease entity particularly in the
knee joint but its incidence in the foot is quite rare. A case of First
Metatarsophalangeal(MTP) joint Pigmented Villonodular Synovitis(PVNS), presented to
us with recurrence of symptoms after surgical excision done outside our institute. After
histological confirmation of recurrence of the disease, repeat open surgical excision was
performed. After being asymptomatic for two months she presented to us with recurrence
of symptoms for which hyperkeratotic plaque at the ventral aspect of the first MTP joint
was found to be responsible on physical examination . It was treated surgically by pairing
it and now patient is symptom free for last 1 year. It signifies the importance of the
histopathology in the diagnosis and recurrence of the PVNS and thorough physical
examination in the management of the foot pathologies.
Introduction
Jaffe et al in 1941 introduced the term pigmented villonodular synovitis for the first time1.
An inflammatory pathology of the lesion was suggested by them rather than neoplastic
and still it is the most accepted theory for its pathogenesis. He classified it into two forms
nodular and diffuse form.
Clinically
It is usually found in the young age group of 20-50 years and distribution between male
and female is almost equal2 . Average delay from the onset to the manifestation of signs
and symptoms is usually between 2-3 years and it progresses slowly3. Patient presents
with symptoms like pain and limitation of motion and signs like swelling, heat and
tenderness over the involved joint3. Rarely it involves more than one joint. Most of the
lab tests such as complete blood count, sedimentation rate are usually normal2.
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Pathology
Name of this disease entity itself depicts its pathological features3. Pigmented word
corroborates with the rusty or brown color which is mainly due to hemosiderin deposits in
stroma, macrophages and synovial lining cells. Presence of the hemosiderin signifies
repeated old hemorrhages. Villonodular word signifies that grossly synovium is nodular
in texture and microscopically it is composed of fingerlike (villous) masses of fibrous
stroma covered by hyperplastic lining cells. Fibrin masses may be found to be free or
adhered to the synovial linings. Free fibrin masses are referred as ‘Rice bodies’. Although
considered to be as benign disease entity there can be enormous mitotic figures with
normal configuration in the proliferating fibroblasts, macrophages and synovial lining
cells.
Osseous changes in the radiograph correlate with the pathological stage and type of the
disease. Localized nodular variety produces very less radiographic abnormalities and
X-rays usually shows no bony abnormality. In diffuse type of PVNS one can find
erosions of the subchondral bone in early phase and juxta-articular cysts may be found in
the late phase of disease.
Most commonly involved joint is Knee joint (80%) followed by hip(16%) and ankle(7%),
Wrist, shoulder and elbow2. According to english literature its incidence in foot is around
2% 4and rate of recurrence in the foot has been reported to be around 14.3%5. PVNS is
very rare in 1st MTP joint and the management of recurrence at this site has still not been
reported elaborately. Moreover this report also signifies the importance of thorough
physical examination and explains how corresponding symptoms can be caused by the
differential diagnosis of the disease itself even after complete treatment of the causative
disease and misled the treating physician. Such a confusion can easily be clarified with
the help of thorough physical examination.
Case report
A 17 year old female presented to our institution with diagnosis of pigmented
villonodular synovitis already confirmed with help of synovial biopsy done from 1st
metatarsophalangeal joint and open excision of PVNS done elsewhere. She now came
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with the chief complaints of excessive pain at 1st Metatarsophalangeal joint for last 6
months which increased while walking and relieved by taking rest and NSAIDS. A
careful physical examination was done and all the movements of the first
metatarsophalangeal joint were painful throughout the range of motion. Radiographs
were showing no signs of any bony pathology ( Fig 1) but in MRI ( Fig 2) recurrence was
documented after discussing it with an expert radiologist. Recurrence was also confirmed
by the histopathological analysis of the repeat biopsy. Thorough open excision of the
pathological synovium was carried out. After the surgical procedure patient remained
pain free for about 2 months. On follow up after 2 months she started having pain at the
first metatarsophalangeal joint and suspicion of the recurrence of the disease was
considered but after careful physical examination she was found to have a hyperkeratotic
plaque at the planter aspect of the first metatarsophalangeal joint. She was sent for the
opinion of an expert of dermatologist, who paired that corn. After that patient is
completely pain free after one year of the surgical management.
Discussion
Pigmented villonodular synovitis is usually a monoarticular disease and knee joint is most
commonly involved in upto 80% cases2. Involvement of ankle and foot joints is quite rare.
In the foot it usually occurs in the rear foot and ankle although there are few case reports
describing involvement of the MTP joints.
Carpintero et al4 reported five out of eight cases in the hindfoot and only three cases
were in the forefoot. Similarly in a series by Ghert et al6 only two cases involved
matatarsal region out of six cases. It has also been documented earlier that in the foot and
ankle region the disease is quite invasive and bone involvement is quite common and
hence radical treatment modality is sometimes required as described by Rochwerger et al7.
He did arthrodesis in six cases, one ankle synovectomy and one toe amputation in total of
eight cases and reported bony involvement in seven of eight cases.Foot is exposed to
direct ground reaction and unevenness while walking which makes it prone to the
repeated trauma which may be responsible for micro-hemorrhages in the joint and initiate
an inflammatory process.
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This report has brought to our notice that even if Magnetic Resonance Imaging aids
immensely in the diagnosis of the PVNS but for definitive diagnosis and for the
confirmation of the recurrence, histopathology should be considered to be the gold
standard and final confirmation test.
According to the english literature PVNS is a rare entity in the foot and PVNS of the
forefoot and the 1st metatarsal joint , although has also been reported earlier, still
considered to be very rare. This report signifies the importance of the differential
diagnosis of pain in the foot and particularly in the weight bearing part of the foot. This
can be a masquerading factor and provides a perfect example signifying the importance
of thorough physical examination. It suggests that a disease with high rate of recurrence
should be considered to have recurred only when other causes of such symptoms have
been ruled out.
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References 1.Jaffe HL, Lichtenstein L, Sutro CJ. Pigmented villonodular synovitis, bursitis and
tenosynovitis. Arch Pathol 1941;3i :731-76
2.Byers PD, Cotton RE, Deacon OW, et al. The diagnosis and treatment of pigmented
villonodular synovitis. J Bone Joint Surg [Br] 1968;50:290-305
3.Atmore WG, Dahlin DC, Ghormley AK. Pigmented villonodular synovitis: a clinical
and pathologic study. Minn Med 1956;39: 196-2
4.Carpintero P, Gascon E, Mesa M, Mesa M, JimenezC, Lopez U. Clinical and
radiographic features of pigmentedvillonodular synovitis of the foot: Report of eight
cases. JAPMA 2007 97: 415-419.
5.Sharma H, Jane MJ, Reid R. Pigmented villonodular synovitis of the foot and ankle:
Forty years of experience from the Scottish Bone Tumor Registry. J Foot Ankle
Surgery2006 45: 329-336.
6.Gher MA, Scully SP, Harrelson JM. Pigmented villonodular synovitis of the foot and
ankle: A review of six cases. Foot Ankle Int 1999 20: 326-330.
7.Rochwerger A, Groulier P, Curvale G, Launay F. Pigmented villonodular synovitis of
the foot and ankle: A report of eight cases. Foot Ankle Int 1999 20: 587-590.
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Figure
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Figure
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