ABSTRACT:
Although the relationship between hip arthroplasty and the development of sarcoma
was first described in the literature about forty years ago, this association is extremely
rare. In the present case report, we describe the association between orthopedic implants
and soft tissue sarcoma in a 79-year-old man who underwent primary total hip arthroplasty
(THA) for coxarthrosis 24 years ago. In the present case report, we describe the clinical
evolution and the radiographic and histopathological findings of the lesion. In the
intraoperative period of the second revision surgery, loosening of the acetabular
and femoral components in association with extensive areas of necrosis and metallosis
was evidenced. We performed debridement of the hip and right thigh region and removed
the implants. Due to the extent of the lesion and to necrosis, it was not possible
to perform a new joint reconstruction. The histopathological diagnosis of high-grade
undifferentiated pleomorphic sarcoma associated with extensive areas of metallosis
was confirmed in tissue adjacent to the implant. The patient developed pulmonary metastases
and died 6 months after the diagnosis. Despite the rarity of this association, sarcomas
should be considered in the differential diagnosis of aseptic loosening, especially
in the presence of metallosis in the peri-implant tissue. To our knowledge, the 24-year
latency period between primary THA and the establishment of a sarcoma diagnosis is
one of the longest reported to date.
Keywords: arthroplasty, replacement, hip; hip prosthesis; metals; sarcoma.
RESUMO:
Apesar da relação entre artroplastia de quadril e o desenvolvimento de sarcoma ter
sido descrita pela primeira vez na literatura há cerca de quarenta anos, esta associação
é extremamente rara. No presente relato de caso, descrevemos a associação entre implante
ortopédico e sarcoma de partes moles em um homem de 79 anos submetido a artroplastia
primária total do quadril (ATQ) por coxartrose há 24 anos. Foram descritas a evolução
clínica e os achados radiográficos e histopatológicos da lesão. No intraoperatório
da segunda cirurgia de revisão, foi evidenciada soltura dos componentes acetabular
e femoral em associação com extensas áreas de necrose e de metalose. Foi realizado
desbridamento da região do quadril e da coxa direita e retirada dos implantes. Devido
à extensão da lesão e da necrose, não foi possível realizar nova reconstrução articular.
O diagnóstico histopatológico de sarcoma pleomórfico indiferenciado de alto grau associado
a extensas áreas de metalose foi estabelecido no tecido adjacente ao implante. O paciente
evoluiu com metástases pulmonares e faleceu 6 meses após o diagnóstico. Apesar da
raridade da associação, os sarcomas devem ser considerados no diagnóstico diferencial
das solturas assépticas, particularmente na presença de metalose no tecido peri-implantar.
Pelo nosso conhecimento o período de latência de 24 anos entre a ATQ primária e o
estabelecimento do diagnóstico de sarcoma é um dos mais longos relatado até o momento.
Palavras-chave: artroplastia de quadril; metais; próteses de quadril; sarcoma.
FIGURES
| Citation: Campos RS, Meohas W, Ferradosa AS, Costa AFAF, Guimarães JAM, Duarte MEL. High-grade Pleomorphic Sarcoma Associated with Metallosis in a Patient with Total Hip Arthroplasty. 59(Suppl S1):e17. doi:10.1055/s-0041-1735140 |
| Financial Support: There was no financial support from public, commercial, or non-profit sources. |
|
Conflict of Interests: The authors have no conflict of interests to declare. |
|
Study carried out at the Instituto Nacional de Traumatologia e Ortopedia (National Institute of Traumatology and Orthopedics), Rio de Janeiro, RJ, Brazil. |
| Received: December 10 2020; Accepted: June 02 2021 |
INTRODUCTION
The cellular response to debris particles resulting from the degradation of different types of materials constitutes the biological basis of peri-implant osteolysis that results in failures in total hip arthroplasties (THA).1 Wear debris from the components of the prosthesis activate the mono-cyte-macrophage system, promoting the release of proin-flammatory cytokines, in particular growth factors, interleukins, and the receptor activator of nuclear factor-κB ligand (RANKL). The integrated action of these cell signaling molecules, and inparticular the activation ofosteoclasto-genesis through the interaction of RANKL with its receptor activator of nuclear factor-κB (RANK), begins the osteolysis process. Activation of the RANK/RANKL system results in increased recruitment and activity of osteoclasts at the interface with the implant, leading to bone resorption, loosening, and prosthesis failure.1
The relationship between hip arthroplasty and the development of sarcoma was first reported in 1984.2 In a review covering a period of 30 years (1974–2003), the occurrence of 46 malignant tumors (41 sarcomas, 4 lymphomas, and 1 squamous cell carcinoma) related to THA was described.3
CASE REPORT
A 79-year-old white male with bilateral idiopathic coxarthrosis underwent primary THA in 1992 (right hip) and 1993 (left hip). In both surgeries, an uncemented acetabular component was used, made of titanium alloy coated with porous plasma, fixed with acetabular screws, polyethylene acetabular core, and uncemented femoral nail, made of titanium alloy and rough proximal surface, covered with hydroxyapatite and interchangeable cobalt chrome head. In January 2001, the patient presented with loosening of the acetabular component of the left hip and underwent a revision arthroplasty changing for an uncemented acetabular prosthesis. In September of the same year, due to the same diagnosis, he underwent another revision arthroplasty of the right hip, with maintenance of the nail and replacement of the acetabulum for a cemented component. During both surgical procedures, significant wear of the polyethylene and extensive foci of metallosis in the peri-implant tissue were observed. In February 2016, he presented with increased volume and severe pain in the right hip and gait difficulties. On physical examination, he presented with increased thigh volume without fluctuation, decreased range of motion, and was unable to bear weight on the limb, without neurological deficit. Two months after the onset of symptoms, a new revision procedure was performed on the right hip; infection was the main diagnostic hypothesis, and the differential diagnosis was pseudotumoral lesion caused by metal debris. Intraoperatively, the loosening of the acetabular and femoral components and extensive areas of tissue necrosis and metal debris affecting the entire periarticular region was observed, with no evidence of purulent discharge (►Fig. 1). The debridement of the hip and right thigh region was performed to remove the implants and to collect material for microbiological culture and histopathological evaluation. Due to the extent of tissue necrosis, it was not possible to perform a new joint reconstruction. The histopathological evaluation confirmed the diagnosis of high-grade undifferentiated pleomorphic sarcoma (UPS) and metallosis (►Fig. 2). The results of intraoperative microbiological cultures were negative. In July 2016, the radiography of the right hip showed a large tumoral mass in soft tissues and extensive destruction of the proximal third of the femur. (►Fig. 3). The patient evolved with weight loss and dyspnea, being diagnosed with pleural effusion and multiple metastatic nodules in the lungs (►Fig. 4), and died in August 2016, 24 years after primary THA.
DISCUSSION
The incidences of soft tissue sarcomas in the general population and in association with THA were estimated at between 0.5 and 2.0 per 100,000 individuals per year4 and at 1.43 per 100,000 individuals per year,3 respectively. The most frequent association with implants is high-grade UPS (malignant fibrous histiocytoma, according to the old nomenclature). However, the association between THA and other sarcomas such as osteosarcoma, angiosarcoma, and synovial sarcoma has also been described.5,6,7 In a large population study, it was shown that metal-to-metal hip implants are not associated with an overall increased risk of cancer or of cancer deaths over a median follow-up period of 7.4 years.8 However, this study did not include implants of the same type used in the present report (metal-on-polyethylene).
Considering the date of onset of the complaints of the patient that were compatible with neoplastic disease (local tumor, weight loss, and dyspnea) with the histological confirmation of the diagnosis of UPS, the interval between the primary THA and, therefore, the development of neoplasm was 24 years. This timeframe is considerably longer than the average time of 6.7 years3 for the development of soft tissue sarcomas associated with orthopedic implants.
Carcinogenesis induced by wear particles and metal corrosion was investigated in vitro by exposing fibroblasts to cobalt and chromium particles. The metal particles promoted the generation of reactive oxygen species that resulted in aneuplody, chromosome alterations, mitochondria fragmentation, and damage to the microtubule network that forms the cytoskeleton.9 In the clinical setting, despite reports suggesting this association, the mechanism by which metal particles induce fibrosis and stimulate mutagenesis in peri-prosthetic tissue has not yet been confirmed. In 2015, Sarhadi et al. found genetic abnormalities in periprosthetic tissue, including alterations in genes related to the developmentof malignant neoplasms. In addition, geneticanomalies were associated with a longer period of contact of the tissue with the implant.10
In the present report, we present a case of association between high-grade UPS and a THA performed 24 years ago, in a 79-year-old man with bilateral THA. Considering that THA is the gold standard procedure for the treatment of hip osteoarthritis, performed worldwide on a large scale, its association with the development of a malignant lesion is an extremely rare event. However, orthopedic surgeons and radiologists should be alerted and aware of the possibility of an association between alterations in peri-implant tissues caused by debris particles resulting from the degradation of metallic implants and the development of sarcomas in patients with THA loosening. In this regard, early diagnosis and immediate surgical treatment can prevent an unsatisfactory evolution, with the possible emergence of metastases and an unfavorable outcome for the patient.
