Accepted Manuscript

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