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  • Postgraduate Medical Journal (November 1982) 58, 716-719

    Subcutaneous nodular synovitis following medial arthrotomy in a case of myxoidliposarcoma of the thigh

    J. MCCLUREB.Sc., M.D., D.M.J.Path., M.R.C.Path.

    PREMA IYERM.B., B.S., F.R.C.P.A.

    R. J. BAUZEM.B., B.S., F.R.A.C.S.

    T. MUKHERJEEM.B., B.S., D.G.O., M.D.

    The Adelaide Bone and Joint Research Unit and the Division of Tissue Pathology IMVS, Frome Road, Adelaide,South Australia

    SummaryA lesion of pigmented synovitis appeared as a nodulein the scar one year after arthrotomy and wasremoved one year later. It was clearly distinguishedfrom a concurrent myxoid liposarcoma on clinical andmorphological grounds (light and electron micros-copy).

    Introduction

    Pigmented synovitis is a well defined disorderaffecting joints, tendons and bursae in a diffuse ornodular form. A variety of names reflect site andmorbid anatomy: giant cell tumour of tendon sheath,localized nodular synovitis (Granowitz and Mankin,1967; Fraire and Fechner, 1972) and pigmentedvillonodular synovitis (Jaffe, Lichtenstein and Sutro,1941). Aetiology and pathogenesis are unknown, butall recognize the lesions to be similar, to be reactive,and very probably to result from repeated haemor-rhage. The changes in the synovium of haemo-philiacs are most often adduced in support of thisargument. We record this very unusual case of asubcutaneous pigmented lesion, arising one year afterarthrotomy, examined by light and electron micros-copy, to add to experience and knowledge of thisuncommon condition. We believe the lesion to havedeveloped in displaced synovium. Difficulty andinterest was added by a concurrent liposarcoma.

    Case reportA 28-year-old man injured his left knee. After

    several months of persistent pain and swelling, withepisodic collapsing and clicking, a torn left lateralmeniscus was removed. Recovery was uneventful.

    Requests for reprints to: Dr J. McClure, C/- IMVS, Box 14,Rundle Street Post Office, Adelaide, South Australia 5001.

    One year later he twisted his right knee. Hepresented himself after 14 months because of persist-ing symptoms. During the last 2 months he had alsonoticed a lump on the inner side of his right lowerthigh, which was confirmed by clinical examination.Plain X-rays of the knee were normal. A medialarthrotomy was performed; there was no abnormalityof the meniscus or synovium. The arthrotomy woundwas closed using 3 figure-of-eight vicryl suturesclosing the synovium and capsule in one layer. Acontinuous vicryl suture was placed in the subcutane-ous tissue and the skin was closed by a sub-cuticularprolene suture. The back of the knee was thenexplored: there was a multilobulated, well-encapsu-lated tumour 100x40 mm beneath the adductormagnus muscle belly.

    Recovery was uneventful, but he continued to havepain in his knee. Three months after the lastoperation a torn lateral meniscus was removed. Thesynovium was normal.One year later a 3 mm, mobile, painless lump was

    noted in the lower end of the medial arthrotomywound of the right knee, attached to the capsule butnot to the skin. A suture granuloma was diagnosed.One year later it was slightly painful, 10 mm indiameter and fairly mobile. The lump plus an ellipseof subcutaneous tissue and skin were excised. Therewas a smaller (3 mm diameter) nodule just beneaththe capsule in the line of the wound. This was alsoexcised together with a segment of capsule. Thewound healed well and recovery has been unevent-ful.

    PathologicalfindingsThe tumour from the back of the right knee

    exhibited the features of a myxoid variant of liposar-coma (Fig. 1). Lobulated and yellow-pink in colour,

    0032-5473/82/1100-0716 $02.00 1982 The Fellowship of Postgraduate Medicine

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  • Clinical reports 717

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    FIG. 1. There are numerous lipoblasts with pleomorphic nuclei and lipidic droplets of variable size in this myxoid variant of liposarcoma.H.E x 400.

    it was cellular, myxoid and vascular. The tumourcells had oval and stellate nuclei, pale cytoplasm andindistinct margins. Nuclear pleomorphism was mildand mitoses were sparse. There were proliferatinglipoblasts in various stages of differentiation andlarge multivacuolated forms. The capillaries formeda delicate plexiform pattern. Hyaluronidase-sensitiveacid mucopolysaccharides were demonstrated.

    Skin and subcutaneous tissues with a nodule in thelatter and a portion ofjoint capsule with an attachedsmaller nodule were received from the second opera-tion. Both nodules had brown cut surfaces and wereidentical microscopically. Septae of dense collagendemarcated cellular areas with mononuclear cells,multinucleated giant cells and small numbers offoam cells (Fig. 2). Haemosiderin was abundant. Thefeatures were identical to those of a nodular (teno)synovitis. Neither foreign material, nor acid and/oralcohol fast organisms nor fungi were found.

    Tissue enucleated from paraffin blocks was proc-essed in the conventional manner for transmissionelectron microscopy (TEM). The sections stainedwith uranyl acetate and lead citrate were examined ina JEM 100 CX electron microscope. Four types ofcell were identified. One conformed to the descrip-tion by Alguacil-Garcia, Krishnan and Goellner(1978) and Eisenstein (1968) of the type A synovio-cyte. This had a highly electron dense cytoplasm,

    abundant cytoplasmic filopodia, a high concentrationof mitochondria, a poorly developed rough endoplas-mic reticulum (RER) (Fig. 3) and a few lysosomes.The nuclei were either oval or elongated andoccasional cells were binucleated. The distinguishingfeature of these cells was the presence of a prominentnucleolus. The second type of cell was the type Bsynoviocyte of the above authors, portions of whichmay be seen in Figure 3.The cytoplasm was pale with well developed but

    sparse RER, usually with dilated cisternae andcomparatively few mitochondria. Very often thenucleus was highly indented with an inconspicuousnucleolus. Junctional complexes were seen betweenboth types of cell and often the type B cells weresurrounded either wholly or in part by basal laminarmaterial. Insinuations of collagen fibrils were alsoseen between both cell types. Haemosiderin granules,characteristically present in type B cells, were alsoseen in some type A cells. Type A and type B cellswere by far the commonest of all cell types.The third cell type was the multinucleated giant

    cell, consisting of voluminous cytoplasm, crowdedwith mitochondria, strips of RER and vesiclescontaining electron dense inclusions. The number ofnuclei varied greatly and some showed prominentnucleoli, a feature consistently seen in type A cells.Although they were surrounded by both type A andtype B cells the association between the giant cells

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  • 718 Clinical reports

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    FIG. 2. The nodular lesions are divided into lobules bounded by fibrous septae (S) and contain giant cells (GC) and mononuclear cells.H.E x 200.

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  • Clinical reports 719

    and the type A cells was more intimate since type Bcells were invariably separated from the giant cells bya thin basal lamina and/or collagen fibrils. Occasion-ally giant cells surrounded type A cells; the sugges-tion that there was incorporation was enhanced bythe breaking down of plasma membranes whichseparated them. In addition, continuity of cytoplasmbetween a type A cell and a giant cell was found.The fourth cell was a typical fibroblast seen mostly

    in the septae. These differed from the fusiform type Acell: they contained only moderate amounts of RER,had no concentration of mitochondria and cytoplasmwas less dense. Furthermore they were surrounded bybundles of well formed collagen.

    Discussion

    The interest of this case lies in the occurrence ofsubcutaneous, extracapsular proliferative lesions atthe site of a previous arthrotomy of the right knee.Extrinsic foreign body material and infection bymycobacteria or fungi were excluded as causes. Theappearances were identical on light and electronmicroscopy to pigmented synovitis, whether thecommon nodular tenosynovitis (so-called giant celltumour of tendon sheaths) or the very much lesscommon intra-articular localized nodular synovitis.

    Jaffe et al. (1941) collected a group of synoviallesions of similar appearance, named xanthoma,xanthogranuloma, giant cell fibrohaemangioma andsynovioma of joints, bursae and tendon sheaths.They gave them the descriptive title of pigmentedvillo-nodular synovitis (PVNS). Jaffe and hiscolleagues recognized that the intra-articular lesioncould be localized or diffuse but both were consi-dered manifestations of a common pathologicalprocess; a reactive proliferation of synoviocytes. Thisview was supported by Lichtenstein (1955) and morerecently by E