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.
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.
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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