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Malignant Fibrous Histiocytoma at the Site of a Total Hip Arthroplasty

Schuh, Alexander*; Zeiler, Günther*; Holzwarth, Ulrich; Aigner, Thomas

Clinical Orthopaedics and Related Research: August 2004 - Volume 425 - Issue - p 218-222
SECTION II: ORIGINAL ARTICLES: Tumor
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Malignant fibrous histiocytoma is the most frequent sarcoma in adults. Predisposing factors for malignant fibrous histiocytoma are Paget’s disease, bone infarcts, malignant disorders of the hematopoetic system, or prolonged intake of corticosteroids. Malignant fibrous histiocytoma has been described as occurring with increasing frequency after endoprosthetic therapy and has been attributed to the implants or to their alloy constituents. Malignant fibrous histiocytoma at the site of an endoprosthesis of the hip constitutes a distinct rarity. To our knowledge, only 13 cases have been described to date. In this report, we present the case of a 66-year-old woman with rheumatoid joint disease. Eight years after primary endoprosthetic surgery, loosening of the implant with severe osteolysis of the surrounding bone required replacement surgery. Histopathologic evaluation of resected tissue revealed scar and granulation tissue and Grade 3 malignant fibrous histiocytoma. The patient died 1 year after revision arthroplasty because of diffuse pulmonary and cerebral metastases. In patients with loosening of a total hip endoprosthesis in combination with severe periprosthetic osteolysis an accompanying malignancy should be in the differential diagnosis. The histopathologic examination of the resected tissue should be obligatory.

From the *Clinic of Orthopaedic Surgery Rummelsberg, Schwarzenbruck; †Peter Brehm Chirurgie Mechanik, Weisendorf; and the ‡Department of Pathology, University of Erlangen- Nürnberg, Germany.

Received: April 25, 2002

Revised: September 24, 2002; January 13, 2003; November 7, 2003

Accepted: January 16, 2004

Correspondence to: Alexander Schuh, MD, Orthopädische Klinik Rummelsberg, Rummelsberg 71, 90592 Schwarzenbruck, Germany. Phone: 0049-9128-50-3333; Fax: 0049-9128-50-3150; E-mail: Schuh-Alexander@t-online.de.

Malignant fibrous histiocytoma is a rare mesenchymal malignancy in adults, presenting either as a soft tissue tumor or, more rarely, as an intraosseous lesion. It is the most frequent sarcoma in adults representing approximately 20–30% of all soft tissue sarcomas and 5% of all intraosseous malignancies.6,8,11–13

Intraosseous malignant fibrous histiocytoma is an usually aggressive tumor with often extensive destruction of the cortical bone seen on the radiographs. The periosteal reactions are irregular and matrix calcifications rarely are present. Histologically, malignant fibrous histiocytoma is a poorly differentiated neoplasm with often strongly pleomorphic and hyperchromatic nuclei. This basic tumor cell most likely derives from primitive mesenchymal cells such as fibroblast precursor cells.16,23 Predisposing factors of malignant fibrous histiocytoma are Paget’s disease, bone infarcts, malignant disorders of the hematopoetic system, and the prolonged intake of corticosteroids.13,14,21,22,39 At the site of endoprostheses of the hip, malignant fibrous histiocytoma is a rare condition with only 13 cases described.1,4,5,10,15–17,24,30,33–35,37

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CASE REPORT

In May 1999, a 66-year-old woman was admitted to the hospital for revision hip arthroplasty because of aseptic prosthetic loosening. The absence of purulent inflammation was confirmed by fine needle aspiration 3 weeks before revision surgery. The preoperative radiograph showed severe osteolysis in the region of the distal shaft of the prosthesis, and radiolucent lines around the cement-free socket with several broken screws (Fig 1). The original implantation of a noncemented Jörgensen prosthesis (Ti5A12,5Fe, Peter Brehm, Chirurgiemechanik, Weisendorf, Germany) was done elsewhere in 1991. In general, the patient had rheumatoid arthritis and was treated with high doses of corticosteroids for several years. She did not report any significant fever, night sweats, or weight loss before admission. She had no complaints until early 1999, when she first mentioned pulling pains in the region of the right thigh. After routine preoperative checkups by the anesthesiologists, revision arthroplasty of the right hip was done in May 1999. During the operation, loosening of the socket with three broken screws and marked formation of granulomatous tissue were found. After removal of the components of the socket and of the granulomatous tissue, the socket first was built up by autologous corticospongiose chips from the iliac crest to support a ring osteosynthesis using an acetabular reinforcement ring (Peter Brehm) and a polyethylene cup. The femur showed extensive destruction of the cortical bone on the dorsal side of the prosthesis tip. Again, extensive granulomatous tissue (approximately 10 × 10 × 2 cm) was found and removed. During removal of the endoprosthetic stem the femur fractured. The cortical bone of the femur seemed thin and very brittle. Therefore, a cement-free MRP-Titan-1 stem (Peter Brehm) with a ceramic head was implanted. The femur fracture was treated by seven cerclage wires. The postoperative radiograph is shown in Figure 2.

Fig 1.

Fig 1.

Fig 2.

Fig 2.

The tissue resected from the osteolytic femur was examined histopathologically (Fig 3). Extensive areas of granulation and scar tissue with extensive Ti debris deposited in the cytoplasm of histolytic cells and polyethylene particles surrounded by multinuclear foreign body giant cells were found (Fig 3A). Additionally, infiltrates by a tumor composed of pleomorphic mostly spindle-shaped cells with hyperchromatic nuclei and numerous, partly atypical mitoses were found consistent with the diagnosis of a Grade 3 malignant fibrous histiocytoma of the storiform-pleomorphic type (Fig 3B,C).

Fig 3.

Fig 3.

Subsequently, the radiographs of the pelvis from 1991 were obtained and reviewed (Fig 4). They showed severe osteoarthritis of the right hip with a severely deformed femoral head that interdigitated with the acetabulum without subchondral sclerosis or cyst formation. Additionally, small intramedullary erosions of the lateral proximal femoral corticalis were present.

Fig 4.

Fig 4.

In August 1999, the patient was admitted for right hemipelvectomy because of a tumor of the right hip protruding into the small pelvis. Histologic samples were collected from the proximal femur, the acetabulum, and from the intrapelvic tumor portions. The tumor was removed completely (R0-resection) and the postoperative recovery was uneventful. The histologic analysis of the resected lesion confirmed third-degree malignant fibrous histiocytoma.

In October 1999, CT scans showed diffuse pulmonary metastatic spread and the patient had a partial pulmonectomy in November 1999. In May 2000 cerebral metastases were removed; however, she died in September 2000.

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DISCUSSION

Sarcomas including malignant fibrous histiocytoma at the site of a hip or knee endoprosthesis are rare (Table 1).1–5,7,10,15,17,18,20,24–31,33–38

Table 1

Table 1

Some authors have tried to link the occurrence of malignant fibrous histiocytomas to the materials used for the implants or to the cement.19,32 Experimental studies in rats showed that the different materials used in total hip replacement are potentially carcinogenic, such as CoCr compounds,32 polymethylmethacrylate,19 or polyethylene.9 In all published cases, except one concerning malignancy associated with endoprostheses,10 CoCr alloy was used. The current case is the second reported malignant fibrous histiocytoma at the site of a Ti total hip arthroplasty.10

The histogenesis of a malignant fibrous histiocytoma is not conclusive, but an origin from undifferentiated mesenchymal cells has been suggested.13,39 Mesenchymal cells are the basic cell population of granulomatous tissue, frequently forming a tumorlike mass in areas of implant debris. Therefore, the question arises whether malignant fibrous histiocytoma, at least in some of the cases, might not be initiated or promoted by continuous irritation of the granulomatous tissue by metal particles.

The latency period between implantation of a total endoprosthesis and diagnosis of malignant fibrous histiocytomas was reported to range from 5 months to 15 years, with a median of 8.5 years (Table 1).34 The latency period in the current patient was 8 years. There are three published cases with a low latency period of only a few months.1,17,27 These short latency periods raise the question of whether malignancies associated with endoprostheses are not coincidental. One limitation of these case reports is that few studies4,10,17 show radiographs before implantation of the first prosthesis. The majority of these studies show only radiographs of the hip after loosening of the implant. Therefore, preexisting malignancy cannot be evaluated. The preoperative radiographs in the current patient suggest, similar to the case of Jacobs et al,17 that despite the initial radiographs showing only subtle alterations, an occult malignant lesion might have existed preoperatively. Therefore, small intramedullary erosions of the lateral cortex of the proximal femur suggest an intramedullary tumor in the current patient. Alternatively, intramedullary erosions could reflect disuse osteopenia in a patient with rheumatoid arthritis.

Additional examinations would have been needed to confirm an occult malignant lesion. Overall, because of the extremely low incidence of malignant fibrous histiocytoma in conjunction with total hip endoprostheses and the short latency period described in the literature, there are significant doubts whether malignant fibrous histiocytoma can be attributed to the orthopaedic implants or to their alloy constituents. More likely, at least in most cases, it is a coincidental phenomenon.10,16,23

The main problem at the time of the revision arthroplasty of the hip was that we were not aware of the possibility of a malignancy. Retrospectively, a diagnostic frozen section of the tissue would have been useful. At the time of revision arthroplasty of the hip there was no sign of an intrapelvic mass. The surgical manipulation may have promoted tumor spread and local growth within only 4 months. Obviously, this is a critical issue. Perhaps the best patient management would have been referral of the patient after the final diagnosis for tumor staging and resurgery within 4 months. The current case strongly supports the hypothesis of Solomon and Sekel30 that in the event of localized pain, significant swelling, and rapid osteolyses or loosening of the implants, particularly if it occurs years after primary surgery and septic loosening are ruled out, a malignancy must be considered.

Different forms of treatment have been described for malignant fibrous histiocytoma in connection with a total hip replacement, ranging from local extirpation of the tumor in conjunction with prior arterial embolization,33 radiation,15,24 exarticulation of the hip,33 or hemipelvectomy.17 The current case documents the difficulty of successful treatment of this condition.

We assume that the malignant fibrous histiocytoma did not occur as a result of the Ti alloy but was a coincindent condition in the current patient. Currently, few cases of malignancy related to implants are published. There are two possible explanations: not all cases are published or the incidence is very low. So far, it is not possible to estimate properly the incidence of malignant fibrous histiocytoma in total joint arthroplasty. Only an international prosthesis register is able to clarify whether malignant changes at sites of implants, such as malignant fibrous histiocytoma, are a consequence of or a coincidental disorder in connection with endoprosthesic implants.

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