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Oncological and Functional Outcomes after Hemicortical Resection and Biological Reconstruction Using Allograft for Parosteal Osteosarcoma of the Distal Femur

Oncological and Functional Outcomes after Hemicortical Resection and Biological Reconstruction... Hindawi Sarcoma Volume 2022, Article ID 5153924, 10 pages https://doi.org/10.1155/2022/5153924 Research Article Oncological and Functional Outcomes after Hemicortical Resection and Biological Reconstruction Using Allograft for Parosteal Osteosarcoma of the Distal Femur 1,2 1,2 1,2 3 Olga D Savvidou , Stavros Goumenos, Ioannis Trikoupis, Angelos Kaspiris , 1,2 1,2 1,2 Dimitra Melissaridou, Panagiotis Gavriil, Jimmy Georgoulis, 1,2 and Panayiotis J Papagelopoulos First Department of Orthopedics, National and Kapodistrian University of Athens, Medical School, Zografou, Greece ATTIKON University General Hospital, Rimini 1, Chaidari, Athens, Greece Laboratory of Molecular Pharmacology, Group of Orthopaedic Research, Department of Pharmacy, School of Health Sciences, University of Patras, Patras, Rion 26504, Greece Correspondence should be addressed to Panayiotis J Papagelopoulos; pjporthopedic@gmail.com Received 25 January 2022; Accepted 3 May 2022; Published 2 June 2022 Academic Editor: Manish Agarwal Copyright © 2022 Olga D Savvidou et al.  is is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Background. Parosteal osteosarcoma (PAOS) is a surface osteosarcoma. Treatment options include wide excision and endo- prosthetic or allograft. However, due to the low local recurrence and metastasis rate, when it appears in the posterior surface of the distal femur, the lesion can be managed with hemicortical wide resection and biological reconstruction with hemicortical allograft.  e purpose of this study is to evaluate the oncological and functional outcomes of patients with parosteal osteosarcoma (PAOS) of the posterior cortex of the distal femur who underwent biological reconstruction after hemicortical resection. Methods. Eleven patients who underwent wide tumor resection and defect reconstruction of the posterior surface of the distal femur using hemicortical allograft were retrospectively studied. Local recurrence, metastasis, complications, and the functional outcome using the Musculoskeletal Tumor Society (MSTS) scoring system were evaluated. Results.  e average postoperative follow-up period was 53.64 months (range, 30 to 84 months). At the latest follow-up, all patients had no evidence of disease without metastases. One patient with local recurrence underwent revision surgery with ’bula autograft reconstruction.  e mean MSTS score was 93.45 ± 3.56. Conclusions. Treatment of patients with PAOS of the posterior aspect of the distal femur with hemicortical resection and allograft reconstruction has satisfactory oncological and functional outcome and low complication rates. posterior cortex of the distal femur is most commonly af- 1. Introduction fected, it can also be detected in the metaphysis of other long Osteosarcomas are high-grade intramedullary bone tumors. bones [2]. Limb salvage surgery can be achieved with wide excision and Wide tumor resection and endoprosthesis or bio- endoprosthetic reconstruction. Parosteal osteosarcomas logical reconstruction are the most commonly used (PAOSs) belong to surface osteosarcomas, a rare di”erent therapeutic approaches [2]. PAOSs are low-grade tumors clinicopathological entity of osteogenic tumors rather than a with low risk for metastasis and local recurrence; hence, subtype of intramedullary osteosarcomas [1]. PAOS of the distal femur can be managed with hemi- PAOSs are the most common surface tumor.  ey have cortical wide tumor excision and biological reconstruc- nd th predominance for young (2 –4 decade) females, ac- tion, avoiding the risks of endoprosthetic reconstruction counting for 4% to 6% of the osteosarcomas. Although the complications [3]. 2 Sarcoma )e purpose of this study was to evaluate the oncological plate± cortical screws were used. Needle track biopsy was and functional outcomes of hemicortical excision and bi- excised in all cases. ological reconstruction using allograft for the treatment of When the tumor was small and totally posterior, the PAOS of the posterior aspect of the distal femur. patient was in the prone position and a posterior approach was undertaken. Two or 3 cortical compression screws were used in the posterior-anterior direction to secure the allo- 2. Materials and Methods graft to the distal femur. Clinical, surgical, radiological, and follow-up information of When the tumor was large in size with extension to the eleven patients with PAO of the posterior cortex of the distal lateral or medial aspect of the distal femur, a lateral or medial femur that underwent wide hemicortical resection and re- approach was used and the patient was operated supine. A construction with hemicortical strut allograft in First Or- lateral or medial anatomical distal femoral plate± cortical thopaedic Department of “Attikon” University Hospital, screws were used for prophylactic fixation. Athens, Greece, from 2010 to 2018, were retrospectively In 4 patients, a posterior approach was used, with the collected. Only individuals that fulfilled the criteria defined patient in the prone position. A longitudinal incision at the by the World Health Organization (WHO) classification for posterior aspect of the distal thigh and knee was performed. Tumors of Soft Tissue and Bone [1] were included in the )e fascia and the popliteal space were carefully dissected, study. and the peroneal and the tibial nerve as well as the femoral Population characteristics such as age, sex, and BMI and artery and vein were identified and preserved. )e posterior tumor diagnostic features such as the primary location aspect of the distal femur was exposed, and using a thin blade histological grading and presence of metastasis, type of saw and osteotomes, the posterior cortex of the distal femur surgical intervention and tumor surgical margins, and pe- was resected. )e posterior cortex of a distal femur allograft riod of follow-up and local recurrence as well as functional was prepared to match the cortex defect of the host. Due to and oncological outcomes were anonymously received from the size of the tumor, 2 or 3 cortical compression screws the medical archives of our department. were used in a posterior-anterior direction to secure the Finally, 7 females and 4 males were included in the study. allograft to the host bone (Figures 1–4). )e mean age and BMI of the patients were 29 years (Min: In 7 patients, a lateral or medial approach was used, with 16, Max: 41, and SD: 7.46) and 25 kg/m the patient in the supine position. Meticulous dissection and (Min: 19.6, Max: 28, and SD: 2.76), respectively. Tumor assessment was based on identification of the neurovascular structures were under- computer tomography (CT), magnetic resonance imaging taken, and the distal femur was exposed. Using a thin blade (MRI), and histopathological results. Needle biopsy under saw and osteotomes, the cortex of the distal femur with the CT-guided was undertaken in all patients. A coaxial tech- tumor was resected. )e cortex of a distal femur allograft was nique was preferred as multiple cores could be obtained prepared to match the cortex defect of the host. An anatomic through a single bone window. CT scan provided infor- distal femoral plate (in 4 patients a lateral and in 3 patients a mation about the route and the precise location of neuro- medial plate)± cortical screws were used for prophylactic vascular structures and assisted in safely avoiding the fixation (Figures 5–7). )e size of resection was based on the preoperative abovementioned vital structures that were not affected by the tumor. )e choice of a needle biopsy method was based on imaging (CT and MRI) with at least 1 cm of tumor-free surgical margins. )e mean cortical resection was the planned surgical approach, keeping the biopsy track excisable. Regarding the posterior biopsy, it was planned 14.64± 5.5 cm. according to the site of the lesion, through a posteromedial A drain suction was used, and the wound was closed in or posterolateral approach. layers. Postoperatively, a posterior splint and later a brace All patients were diagnosed with well-differentiated were applied until radiographical union was demonstrated PAO of the distal femur that involved less than 60% of the on CT scan. Partial weight-bearing was started when more cortex circumference without intramedullary extension. than 50% union of the transverse and longitudinal osteot- None of the patients appeared distant metastatic disease omies appeared in the radiographic evaluation 6–12 weeks (Table 1). after operation, while full-weight bearing was allowed when allograft incorporation was achieved (average time 7.64 )e research complies with the 1964 Helsinki Declara- tion and its later amendments, and it was approved by the months (Min:6, Max: 12, SD: 1.81). Ethical Committee of ATTIKON University General Hos- None of the patients were lost during the follow-up. )e pital with the reference number of AD 232/19-04-2021. All average postoperative follow-up was 53.64 months (Min: 30, patients agreed to participate in the study and provided Max: 84, and SD: 16.94). None of the patients received written consent prior to publication. neoadjuvant chemotherapy. All surgical procedures, postoperative evaluation, and assessment were performed by the same surgical team. )e 3. Operative Technique and Follow-Up primary endpoint of our study was the achievement of )e operation was performed under a general anesthesia, satisfactory disease control. Secondary endpoints were as and a pneumatic tourniquet was used on the proximal thigh. follows: functional outcomes during the last follow-up, time to graft incorporation, and assessment of postoperative Depending on the location and the size of the lesion, 2 or 3 cortical compression screws or an anatomical distal femoral complications. Sarcoma 3 Table 1: Clinical and demographic characteristics of the included patients. Range of Time to Age BMI Follow-up Resection MSTS Complication Patient Gender Osteosynthesis flexion union (years) (kg/m ) (months) Length (cm) score type (Henderson) (degrees) (months)< 1 M 24 22.4 30 23 Plate + screws 95 95 — 7 2 M 19 26.7 32 14 Plate + screws 97 111 — 8 3 F 32 21 40 19 Plate + screws 94 98 — 6 4 F 27 27 44 9 Screws 91 102 — 6 5 F 31 24 49 20 Plate + screws 89 100 — 9 Type 5 6 F 16 20.8 84 11 Screws 90 97 12 (recurrence) 7 F 20 28 58 8 Screws 96 107 — 6 8 F 38 19.6 61 17 Plate + screws 93 108 — 7 9 M 41 23 68 20 Plate + screws 88 104 — 7 10 F 35 25.2 72 13 Plate + screws 96 116 — 9 11 M 34 27 52 7 Screws 99 118 — 7 (c) (d) (f) (e) (a) (b) Figure 1: Parosteal osteosarcoma (PAO) in a 16-year-old female (no. 6). Anteroposterior (a) and lateral (b) radiographs of the knee showing a parosteal osteosarcoma as a large-ossified opacity attached to the posterior cortex of the distal femoral metaphysis. Sagittal computerized tomography (CT) reformatted images, soft tissue window (c), and bone window (d) demonstrate the characteristic separation between the tumor and the intact femoral cortex (cleft sign) as well as the ossified thick stuck lytic areas are seen within the ossified mass which is surrounded by a thick hypodense rim representing cartilaginous tissue. A fat-suppressed T2w magnetic resonance image sagittal (e) and axial view (f) showing the densely ossified stuck centrally, the inhomogenous moderately, T2 hyperintense mass in the middle, and the hyperintense cartilaginous component in the periphery. )ere is no intramedullary extension of the tumor. (a) (b) (c) (d) (e) (f) Figure 2: (a) Representative photomicrograph for histological evaluation with hematoxylin-eosin staining (original magnification: 200X): hypocellular tumor with spindle cells showing mild atypia arranged in fascicles in desmoplastic collagenous stroma. )e histological results are compatible with parosteal osteosarcoma (PAOS). (b, c) Representative photomicrographs for immunohistochemical assessment displaying tumor cells positively immunostained for MDM-2 (original magnification: 400X) and CDK4 (original magnification: 400X), respectively, with nuclear location. (d) Representative photomicrograph of the slices for histological evaluation after tumor excision with hematoxylin-eosin staining (original magnification: 200X): parallel trabeculae of the well-formed woven bone with spindle neoplastic cells in collagenous stroma, while (e) the neoplastic population is characterized by mild cellularity and atypia (original magnification: 40X). (f) Representative photomicrograph for histological evaluation with hematoxylin-eosin staining showing clusters of moderate cellularity and moderate atypia in PAOS (original magnification: 40X). 4 Sarcoma (c) (a) (b) (d) (e) Figure 3: Intraoperative image of the popliteal fossa after tumor resection and identification of the tibia nerve and the popliteal artery (a). Hemicortical resection (b). )e resected specimen (c). Distal femoral allograft (d). )e part of the posterior femoral allograft matching the dimension of the posterior distal femoral defect after tumor excision (e). Two cortical screws were used to fix the allograft to the distal femur. (a) (b) Figure 4: Anteroposterior (a) and lateral (b) radiographs 6 months postoperatively. As for the primary endpoint assessment, all patients patients were examined every month until the osteotomies underwent sequential imaging staging (MRI) and computer had consolidated and then after every six months. Bridging across three of four cortices in biplanar radio- tomography (CT) scan for local and systematic disease progression and a CT scan of the chest every four months in graphs and CT scan findings were considered evidence of the first two years and every six months thereafter. Re- consolidation. garding the secondary endpoints of our study, functional Pathology specimens were evaluated to assess the ade- results were evaluated using the Musculoskeletal Tumor quacy of surgical margins. A wide surgical margin procedure Society (MSTS) score [4] and measuring the knee range of was documented when a cuff of normal tissue totally covered motion. the tumor. A marginal margin was recognized when a free )e time of the allograft incorporation was established margin of the normal cortex and the marrow was seen based on radiographic and CT scan findings of union. )e microscopically between the tumor and the bone . Sarcoma 5 (b) (c) (a) (d) (e) Figure 5: Computer tomography (CT) of the right femur in a 32-year-old male with parosteal osteosarcoma of the distal femur (a). Sagittal CT with multiple large-ossified masses adjacent to the posterior surface of the distal femur (b). Axial view displaying the tumor's broad-based stuck attached to cortical surface associated with dense central ossification and the cleft sign that separated the ground glass mass from the bone cortex, with no evidence of medullary involvement (c). Magnetic resonance imaging of the distal femur (sagittal (d) and axial (e) views) showing the round mass of low signal intensity on T1-weighted imaging. )e cortex appears intact along the deep surface of the lesion. (a) (b) (c) (d) (e) (f) (g) Figure 6: Intraoperative images showing (a) midline longitudinal incision at the posterior aspect of the distal femur and hemicortical resection. (b) Tumor resection. (c) Distal femur allograft. (d) Preparation of the allograft. (e) Matching the allograft to the bone deficit. (f) Allograft fixation with four cortical screws in the anteroposterior direction (g). )e anatomical distal femoral plate is used as a bridging plate. )e postoperative complications were classified as me- )e mean MSTS score was 93.45 (Min: 88, Max: 99, and chanical (types 1, 2, and 3) and nonmechanical (types 4 and SD: 3.56). All patients were ambulatory without any post- 5) according to the classification system regarding the failure operative pain or restriction of daily activities at the latest of limb salvage after biological reconstruction described by follow-up. )e mean range of knee flexion was 105.1 (Min: Henderson et al. [5]. 95, Max: 118, SD: 7.66). Radiologic findings showed successful union within the first postoperative year. )e average time of allograft in- 4. Results corporation was 7.64 months (Min:6, Max: 12, and SD: 1.81). As far as the postoperative complications, there were no Wide excision of the lesion with clear surgical margins was soft tissue (type 1), hardware failure (type 3), or implant- achieved in all patients based on histopathological exami- related infections (type 4) based on the Henderson criteria. nation. At the follow-up examination, no distant metastases Only one patient had local recurrence (case no. 6) and were detected. All patients had no evidence of disease at the underwent revision surgery with wide excision and latest follow-up evaluation (primary endpoint). 6 Sarcoma cases, the combination of clinical, radiological, and needle (a) (b) biopsy findings provided efficient diagnostic results. His- tological and immunohistochemical analysis revealed that the tumor was composed of hypocellular areas of spindle cells arranged in fascicles in desmoplastic collagenous stroma with parallel trabeculae of the well-formed woven bone. Spindle cells were characterized by minimal, or less frequently, moderate, atypia and limited mitotic activity. PAO demonstrates 90% survival at 10-year follow-up after wide resection [5]. After wide tumor excision, endo- prosthesis or different reconstruction techniques can be used. In our study, all patients were diagnosed with early- stage PAOS and were treated with hemicortical resection and biological reconstruction with the application of allo- graft. Moreover, prophylactic plating fixation was used in seven patients. )e main indications of plate utilization were the relatively localized tumor, the cortical involvement, and the limited soft tissue expansion [9–13]. We must highlight the fact that in patients with PAOS of the distal femur, due to the low grade of the tumor and the young age of the patients, hemicortical resection and reconstruction of the bone defect using polymethyl-methacrylate (PMMA) or biological ma- terials, such as fibular autografts, allografts, or pasteurized/ autoclaved/irradiated host bones, with or without prophy- Figure 7: Anteroposterior (a) and lateral radiographs (b) nine lactic fixation were correlated with good oncological and months postoperatively showing incorporation of the allograft at functional outcome [10–13]. the distal part. However, the proximal part of the allograft shows On the contrary, endoprosthetic replacements of the partial union. femoral defect after PAOS resection have demonstrated long-term survival and good functional outcome [14], but they were linked to the increased postoperative complication reconstruction using fibula autografts (Figures 8–11). )is rate including infection, aseptic loosening, mechanical patient remains free of the disease five years after operation failure, fracture of the prosthesis, or the adjacent bone [2, 15, (Table 1). 16]. Similarly, application of allografts allows mechanical and biological reconstruction after tumor excision, but they 5. Discussion are, also, associated with high rates of complications, in- cluding fracture, nonunion, transmission of disease, and Despite the fact that PAO is the most frequent surface os- teosarcoma, it is a rare bone tumor [6]. It is considered a infection [17]. Hemicortical resection of the distal femur in the early low-grade tumor that originates from the periosteum and appears with low risk for local recurrence and metastases. stage of PAOS of the distal femur was first described by Campanacci et al. in 1984 [18]. Later, this method was )e demographic characteristics of the patients included in our study are in agreement with the findings of the inter- established as a safe therapeutic procedure [10]. However, national literature as PAOS was primarily developed in due to the rarity of patients with PAO, there are limited young adult female patients. Indeed, the incidence of PAOS studies with efficient data that examined the functional is prominent in female individuals in their third decade of outcomes of this surgical intervention. Our results analysis demonstrated satisfactory oncological and functional re- life [6]. Moreover, studies reported that the most common location of the tumor is the posterior surface of the distal sults. Specifically, the mean MSTS and the range of knee flexion scores were 93.45 and 105.1 femoral metaphysis followed by the proximal tibial and , respectively. Our femoral metaphysis being in line with our results. Symptoms findings are in agreement with many studies that reported include a slow-growing painless mass at the beginning. satisfactory functional and oncological outcomes after During time, as the mass increases, the pain worsens. If the hemicortical resection of PAOS of the distal femur. In tumor is closed to a joint, the decreased range of motion particular, Lewis et al. reported very good oncological and might occur [7, 8]. functional outcomes without any complications using this Radiographically, the lesion appears as a large-lobulated technique in 6 patients with PAOS of the distal femur [11]. mass with large usually central bone formation and a thick Deijkers et al. analyzed 22 allograft hemicortical recon- mineralized stalk fixed onto the bone surface, without ex- struction procedures, also showing very satisfying onco- tension into the medullary cavity. logical and functional outcomes. All patients had good or Despite the fact that needle biopsy shows lower accuracy excellent MSTS scores that were accompanied by low than cytogenetic, molecular, immunohistochemical, or FISH complication rates. Furthermore, the incorporation rate of (fluorescence in situ hybridization) studies, in all of our 11 allograft in thirty months was observed in 100% Sarcoma 7 (a) (b) Figure 8: Parosteal osteosarcoma (PAO) in a 16-year-old female (no. 6). Anteroposterior (a) and lateral (b) radiographs four years after operation showing recurrence of PAO. postoperatively [12]. Agarwal et al. reviewed 10 patients who reconstruction method after wide excision of PAOS of the underwent hemicortical excision of the distal femur and posterior cortex of the distal femur, complications may reconstruction with autograft or allograft. In both methods, occur. )e most common complication is host bone the functional outcome was optimal without any major fracture and local recurrence, followed by nonunion and surgical complications or local recurrence [3]. Liu et al. also infection. Rarely allograft fractures can occur. )ese reported very good outcomes with relative low complication complications are associated directly with the size of the rates in 13 patients diagnosed with PAOS and managed with bone defect after wide excision and often require surgical reintervention [21]. None of our patients had a host bone wide excision and reconstruction with pasteurized hemi- cortical autograft and internal fixation. )e authors stated fracture, allograft fracture, nonunion, or infection. Local recurrence was observed in one patient and was managed that the technique, although was operational demanding, was safe and effective in selected patients [19]. with revision, and biological reconstruction with fibular Compared to endoprosthetic reconstruction after wide autograft. )is complication corresponded to the 9.1% of resection of PAOS of the distal femur, many studies dem- the patients included in the study being significantly lower onstrate, respectively, good functional and oncological than the rate of 17% that was referred in previous reports of outcomes. Funovic et al. compared 12 patients who un- the literature [22]. In addition, the revision intervention of derwent prosthetic reconstruction, with 11 patients who this patient was associated with very good functional results underwent biological reconstruction. )e authors concluded avoiding the application of endoprosthetic replacement. that the oncological and functional outcome was not altered Although the frequency of local recurrence was not cor- related with the surgical intervention or the histological between the two reconstruction methods [20]. Nevertheless, the increased rate of revision arthroplasty (58%) in the grade of the tumor [22], inadequate wide resection and endoprosthetic reconstruction group was observed when it dedifferentiated PAOS have been described as potential was compared to the biologic reconstruction group (18%) at negative predictors for its relapse [9, 22–25]. 10 years postoperatively [20]. In addition, Wilke et al. re- While hemicortical resection and allograft of the PAO of the ported that the comparison between 5 and 7 patients who posterior aspect of the distal femur avoid complications asso- underwent endoprosthetic and biological reconstruction ciated with wide distal femur resection and reconstruction such with allograft application, respectively, revealed the same as endoprosthesis, drawbacks such as a higher risk of a positive mean MSTS score (mean MSTS 23) and the similar rate of margin or recurrence should be addressed. Currently, three- complications and reoperation between the two groups [2]. dimensional measurements on the tumor before surgical re- Although in our study the application of hemicortical section using computer technology and establishing a 3-di- mensional model and a 3D printing osteotomy guide plate could allograft was not correlated with any complications sup- porting the evidence that it is a reliable biological assist in accurately resecting the tumor lesion [23]. 8 Sarcoma (a) (b) (c) (d) (e) Figure 9: Intraoperative images with complete excision of the posterior-medial part of the distal femur (a), the excised specimen (b), and polymethyl-methacrylate (PMMA) used to fill the osseous void (c). Postoperative radiographs of anteroposterior (d) and lateral (e) views showing the PMMA in the posterior-medial part of the distal femur. (a) (c) (b) Figure 10: (a) Autograft from the contralateral fibula was used and was cut in the longitudinal axis. (b) Two pieces of hemicortical fibula were inserted to the posterior-medial part of the distal femur after excision of the PMMA. (c) Postoperative anteroposterior radiographs of the knee showing the fibula autografts in place. Sarcoma 9 (c) (a) (b) (d) Figure 11: Anteroposterior (a) and lateral (b) radiographs of the knee 13 months postoperatively showing incorporation of the fibula autografts with no signs of recurrence. Magnetic resonance imaging of the distal femur (sagittal (c) and axial (d) views) showing the distal femur with the fibula autografts with no signs of recurrence, infection, or autograft fracture. )e results of this case series are in accordance with References those of the published literature. We acknowledge that [1] WHO, “WHO Classification of tumors,” in Soft tissue and despite the satisfying results, the study has several limi- bone tumours, pp. 410–417, International Agency for Research tations. 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Oncological and Functional Outcomes after Hemicortical Resection and Biological Reconstruction Using Allograft for Parosteal Osteosarcoma of the Distal Femur

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Hindawi Publishing Corporation
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Copyright © 2022 Olga D Savvidou et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
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10.1155/2022/5153924
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Abstract

Hindawi Sarcoma Volume 2022, Article ID 5153924, 10 pages https://doi.org/10.1155/2022/5153924 Research Article Oncological and Functional Outcomes after Hemicortical Resection and Biological Reconstruction Using Allograft for Parosteal Osteosarcoma of the Distal Femur 1,2 1,2 1,2 3 Olga D Savvidou , Stavros Goumenos, Ioannis Trikoupis, Angelos Kaspiris , 1,2 1,2 1,2 Dimitra Melissaridou, Panagiotis Gavriil, Jimmy Georgoulis, 1,2 and Panayiotis J Papagelopoulos First Department of Orthopedics, National and Kapodistrian University of Athens, Medical School, Zografou, Greece ATTIKON University General Hospital, Rimini 1, Chaidari, Athens, Greece Laboratory of Molecular Pharmacology, Group of Orthopaedic Research, Department of Pharmacy, School of Health Sciences, University of Patras, Patras, Rion 26504, Greece Correspondence should be addressed to Panayiotis J Papagelopoulos; pjporthopedic@gmail.com Received 25 January 2022; Accepted 3 May 2022; Published 2 June 2022 Academic Editor: Manish Agarwal Copyright © 2022 Olga D Savvidou et al.  is is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Background. Parosteal osteosarcoma (PAOS) is a surface osteosarcoma. Treatment options include wide excision and endo- prosthetic or allograft. However, due to the low local recurrence and metastasis rate, when it appears in the posterior surface of the distal femur, the lesion can be managed with hemicortical wide resection and biological reconstruction with hemicortical allograft.  e purpose of this study is to evaluate the oncological and functional outcomes of patients with parosteal osteosarcoma (PAOS) of the posterior cortex of the distal femur who underwent biological reconstruction after hemicortical resection. Methods. Eleven patients who underwent wide tumor resection and defect reconstruction of the posterior surface of the distal femur using hemicortical allograft were retrospectively studied. Local recurrence, metastasis, complications, and the functional outcome using the Musculoskeletal Tumor Society (MSTS) scoring system were evaluated. Results.  e average postoperative follow-up period was 53.64 months (range, 30 to 84 months). At the latest follow-up, all patients had no evidence of disease without metastases. One patient with local recurrence underwent revision surgery with ’bula autograft reconstruction.  e mean MSTS score was 93.45 ± 3.56. Conclusions. Treatment of patients with PAOS of the posterior aspect of the distal femur with hemicortical resection and allograft reconstruction has satisfactory oncological and functional outcome and low complication rates. posterior cortex of the distal femur is most commonly af- 1. Introduction fected, it can also be detected in the metaphysis of other long Osteosarcomas are high-grade intramedullary bone tumors. bones [2]. Limb salvage surgery can be achieved with wide excision and Wide tumor resection and endoprosthesis or bio- endoprosthetic reconstruction. Parosteal osteosarcomas logical reconstruction are the most commonly used (PAOSs) belong to surface osteosarcomas, a rare di”erent therapeutic approaches [2]. PAOSs are low-grade tumors clinicopathological entity of osteogenic tumors rather than a with low risk for metastasis and local recurrence; hence, subtype of intramedullary osteosarcomas [1]. PAOS of the distal femur can be managed with hemi- PAOSs are the most common surface tumor.  ey have cortical wide tumor excision and biological reconstruc- nd th predominance for young (2 –4 decade) females, ac- tion, avoiding the risks of endoprosthetic reconstruction counting for 4% to 6% of the osteosarcomas. Although the complications [3]. 2 Sarcoma )e purpose of this study was to evaluate the oncological plate± cortical screws were used. Needle track biopsy was and functional outcomes of hemicortical excision and bi- excised in all cases. ological reconstruction using allograft for the treatment of When the tumor was small and totally posterior, the PAOS of the posterior aspect of the distal femur. patient was in the prone position and a posterior approach was undertaken. Two or 3 cortical compression screws were used in the posterior-anterior direction to secure the allo- 2. Materials and Methods graft to the distal femur. Clinical, surgical, radiological, and follow-up information of When the tumor was large in size with extension to the eleven patients with PAO of the posterior cortex of the distal lateral or medial aspect of the distal femur, a lateral or medial femur that underwent wide hemicortical resection and re- approach was used and the patient was operated supine. A construction with hemicortical strut allograft in First Or- lateral or medial anatomical distal femoral plate± cortical thopaedic Department of “Attikon” University Hospital, screws were used for prophylactic fixation. Athens, Greece, from 2010 to 2018, were retrospectively In 4 patients, a posterior approach was used, with the collected. Only individuals that fulfilled the criteria defined patient in the prone position. A longitudinal incision at the by the World Health Organization (WHO) classification for posterior aspect of the distal thigh and knee was performed. Tumors of Soft Tissue and Bone [1] were included in the )e fascia and the popliteal space were carefully dissected, study. and the peroneal and the tibial nerve as well as the femoral Population characteristics such as age, sex, and BMI and artery and vein were identified and preserved. )e posterior tumor diagnostic features such as the primary location aspect of the distal femur was exposed, and using a thin blade histological grading and presence of metastasis, type of saw and osteotomes, the posterior cortex of the distal femur surgical intervention and tumor surgical margins, and pe- was resected. )e posterior cortex of a distal femur allograft riod of follow-up and local recurrence as well as functional was prepared to match the cortex defect of the host. Due to and oncological outcomes were anonymously received from the size of the tumor, 2 or 3 cortical compression screws the medical archives of our department. were used in a posterior-anterior direction to secure the Finally, 7 females and 4 males were included in the study. allograft to the host bone (Figures 1–4). )e mean age and BMI of the patients were 29 years (Min: In 7 patients, a lateral or medial approach was used, with 16, Max: 41, and SD: 7.46) and 25 kg/m the patient in the supine position. Meticulous dissection and (Min: 19.6, Max: 28, and SD: 2.76), respectively. Tumor assessment was based on identification of the neurovascular structures were under- computer tomography (CT), magnetic resonance imaging taken, and the distal femur was exposed. Using a thin blade (MRI), and histopathological results. Needle biopsy under saw and osteotomes, the cortex of the distal femur with the CT-guided was undertaken in all patients. A coaxial tech- tumor was resected. )e cortex of a distal femur allograft was nique was preferred as multiple cores could be obtained prepared to match the cortex defect of the host. An anatomic through a single bone window. CT scan provided infor- distal femoral plate (in 4 patients a lateral and in 3 patients a mation about the route and the precise location of neuro- medial plate)± cortical screws were used for prophylactic vascular structures and assisted in safely avoiding the fixation (Figures 5–7). )e size of resection was based on the preoperative abovementioned vital structures that were not affected by the tumor. )e choice of a needle biopsy method was based on imaging (CT and MRI) with at least 1 cm of tumor-free surgical margins. )e mean cortical resection was the planned surgical approach, keeping the biopsy track excisable. Regarding the posterior biopsy, it was planned 14.64± 5.5 cm. according to the site of the lesion, through a posteromedial A drain suction was used, and the wound was closed in or posterolateral approach. layers. Postoperatively, a posterior splint and later a brace All patients were diagnosed with well-differentiated were applied until radiographical union was demonstrated PAO of the distal femur that involved less than 60% of the on CT scan. Partial weight-bearing was started when more cortex circumference without intramedullary extension. than 50% union of the transverse and longitudinal osteot- None of the patients appeared distant metastatic disease omies appeared in the radiographic evaluation 6–12 weeks (Table 1). after operation, while full-weight bearing was allowed when allograft incorporation was achieved (average time 7.64 )e research complies with the 1964 Helsinki Declara- tion and its later amendments, and it was approved by the months (Min:6, Max: 12, SD: 1.81). Ethical Committee of ATTIKON University General Hos- None of the patients were lost during the follow-up. )e pital with the reference number of AD 232/19-04-2021. All average postoperative follow-up was 53.64 months (Min: 30, patients agreed to participate in the study and provided Max: 84, and SD: 16.94). None of the patients received written consent prior to publication. neoadjuvant chemotherapy. All surgical procedures, postoperative evaluation, and assessment were performed by the same surgical team. )e 3. Operative Technique and Follow-Up primary endpoint of our study was the achievement of )e operation was performed under a general anesthesia, satisfactory disease control. Secondary endpoints were as and a pneumatic tourniquet was used on the proximal thigh. follows: functional outcomes during the last follow-up, time to graft incorporation, and assessment of postoperative Depending on the location and the size of the lesion, 2 or 3 cortical compression screws or an anatomical distal femoral complications. Sarcoma 3 Table 1: Clinical and demographic characteristics of the included patients. Range of Time to Age BMI Follow-up Resection MSTS Complication Patient Gender Osteosynthesis flexion union (years) (kg/m ) (months) Length (cm) score type (Henderson) (degrees) (months)< 1 M 24 22.4 30 23 Plate + screws 95 95 — 7 2 M 19 26.7 32 14 Plate + screws 97 111 — 8 3 F 32 21 40 19 Plate + screws 94 98 — 6 4 F 27 27 44 9 Screws 91 102 — 6 5 F 31 24 49 20 Plate + screws 89 100 — 9 Type 5 6 F 16 20.8 84 11 Screws 90 97 12 (recurrence) 7 F 20 28 58 8 Screws 96 107 — 6 8 F 38 19.6 61 17 Plate + screws 93 108 — 7 9 M 41 23 68 20 Plate + screws 88 104 — 7 10 F 35 25.2 72 13 Plate + screws 96 116 — 9 11 M 34 27 52 7 Screws 99 118 — 7 (c) (d) (f) (e) (a) (b) Figure 1: Parosteal osteosarcoma (PAO) in a 16-year-old female (no. 6). Anteroposterior (a) and lateral (b) radiographs of the knee showing a parosteal osteosarcoma as a large-ossified opacity attached to the posterior cortex of the distal femoral metaphysis. Sagittal computerized tomography (CT) reformatted images, soft tissue window (c), and bone window (d) demonstrate the characteristic separation between the tumor and the intact femoral cortex (cleft sign) as well as the ossified thick stuck lytic areas are seen within the ossified mass which is surrounded by a thick hypodense rim representing cartilaginous tissue. A fat-suppressed T2w magnetic resonance image sagittal (e) and axial view (f) showing the densely ossified stuck centrally, the inhomogenous moderately, T2 hyperintense mass in the middle, and the hyperintense cartilaginous component in the periphery. )ere is no intramedullary extension of the tumor. (a) (b) (c) (d) (e) (f) Figure 2: (a) Representative photomicrograph for histological evaluation with hematoxylin-eosin staining (original magnification: 200X): hypocellular tumor with spindle cells showing mild atypia arranged in fascicles in desmoplastic collagenous stroma. )e histological results are compatible with parosteal osteosarcoma (PAOS). (b, c) Representative photomicrographs for immunohistochemical assessment displaying tumor cells positively immunostained for MDM-2 (original magnification: 400X) and CDK4 (original magnification: 400X), respectively, with nuclear location. (d) Representative photomicrograph of the slices for histological evaluation after tumor excision with hematoxylin-eosin staining (original magnification: 200X): parallel trabeculae of the well-formed woven bone with spindle neoplastic cells in collagenous stroma, while (e) the neoplastic population is characterized by mild cellularity and atypia (original magnification: 40X). (f) Representative photomicrograph for histological evaluation with hematoxylin-eosin staining showing clusters of moderate cellularity and moderate atypia in PAOS (original magnification: 40X). 4 Sarcoma (c) (a) (b) (d) (e) Figure 3: Intraoperative image of the popliteal fossa after tumor resection and identification of the tibia nerve and the popliteal artery (a). Hemicortical resection (b). )e resected specimen (c). Distal femoral allograft (d). )e part of the posterior femoral allograft matching the dimension of the posterior distal femoral defect after tumor excision (e). Two cortical screws were used to fix the allograft to the distal femur. (a) (b) Figure 4: Anteroposterior (a) and lateral (b) radiographs 6 months postoperatively. As for the primary endpoint assessment, all patients patients were examined every month until the osteotomies underwent sequential imaging staging (MRI) and computer had consolidated and then after every six months. Bridging across three of four cortices in biplanar radio- tomography (CT) scan for local and systematic disease progression and a CT scan of the chest every four months in graphs and CT scan findings were considered evidence of the first two years and every six months thereafter. Re- consolidation. garding the secondary endpoints of our study, functional Pathology specimens were evaluated to assess the ade- results were evaluated using the Musculoskeletal Tumor quacy of surgical margins. A wide surgical margin procedure Society (MSTS) score [4] and measuring the knee range of was documented when a cuff of normal tissue totally covered motion. the tumor. A marginal margin was recognized when a free )e time of the allograft incorporation was established margin of the normal cortex and the marrow was seen based on radiographic and CT scan findings of union. )e microscopically between the tumor and the bone . Sarcoma 5 (b) (c) (a) (d) (e) Figure 5: Computer tomography (CT) of the right femur in a 32-year-old male with parosteal osteosarcoma of the distal femur (a). Sagittal CT with multiple large-ossified masses adjacent to the posterior surface of the distal femur (b). Axial view displaying the tumor's broad-based stuck attached to cortical surface associated with dense central ossification and the cleft sign that separated the ground glass mass from the bone cortex, with no evidence of medullary involvement (c). Magnetic resonance imaging of the distal femur (sagittal (d) and axial (e) views) showing the round mass of low signal intensity on T1-weighted imaging. )e cortex appears intact along the deep surface of the lesion. (a) (b) (c) (d) (e) (f) (g) Figure 6: Intraoperative images showing (a) midline longitudinal incision at the posterior aspect of the distal femur and hemicortical resection. (b) Tumor resection. (c) Distal femur allograft. (d) Preparation of the allograft. (e) Matching the allograft to the bone deficit. (f) Allograft fixation with four cortical screws in the anteroposterior direction (g). )e anatomical distal femoral plate is used as a bridging plate. )e postoperative complications were classified as me- )e mean MSTS score was 93.45 (Min: 88, Max: 99, and chanical (types 1, 2, and 3) and nonmechanical (types 4 and SD: 3.56). All patients were ambulatory without any post- 5) according to the classification system regarding the failure operative pain or restriction of daily activities at the latest of limb salvage after biological reconstruction described by follow-up. )e mean range of knee flexion was 105.1 (Min: Henderson et al. [5]. 95, Max: 118, SD: 7.66). Radiologic findings showed successful union within the first postoperative year. )e average time of allograft in- 4. Results corporation was 7.64 months (Min:6, Max: 12, and SD: 1.81). As far as the postoperative complications, there were no Wide excision of the lesion with clear surgical margins was soft tissue (type 1), hardware failure (type 3), or implant- achieved in all patients based on histopathological exami- related infections (type 4) based on the Henderson criteria. nation. At the follow-up examination, no distant metastases Only one patient had local recurrence (case no. 6) and were detected. All patients had no evidence of disease at the underwent revision surgery with wide excision and latest follow-up evaluation (primary endpoint). 6 Sarcoma cases, the combination of clinical, radiological, and needle (a) (b) biopsy findings provided efficient diagnostic results. His- tological and immunohistochemical analysis revealed that the tumor was composed of hypocellular areas of spindle cells arranged in fascicles in desmoplastic collagenous stroma with parallel trabeculae of the well-formed woven bone. Spindle cells were characterized by minimal, or less frequently, moderate, atypia and limited mitotic activity. PAO demonstrates 90% survival at 10-year follow-up after wide resection [5]. After wide tumor excision, endo- prosthesis or different reconstruction techniques can be used. In our study, all patients were diagnosed with early- stage PAOS and were treated with hemicortical resection and biological reconstruction with the application of allo- graft. Moreover, prophylactic plating fixation was used in seven patients. )e main indications of plate utilization were the relatively localized tumor, the cortical involvement, and the limited soft tissue expansion [9–13]. We must highlight the fact that in patients with PAOS of the distal femur, due to the low grade of the tumor and the young age of the patients, hemicortical resection and reconstruction of the bone defect using polymethyl-methacrylate (PMMA) or biological ma- terials, such as fibular autografts, allografts, or pasteurized/ autoclaved/irradiated host bones, with or without prophy- Figure 7: Anteroposterior (a) and lateral radiographs (b) nine lactic fixation were correlated with good oncological and months postoperatively showing incorporation of the allograft at functional outcome [10–13]. the distal part. However, the proximal part of the allograft shows On the contrary, endoprosthetic replacements of the partial union. femoral defect after PAOS resection have demonstrated long-term survival and good functional outcome [14], but they were linked to the increased postoperative complication reconstruction using fibula autografts (Figures 8–11). )is rate including infection, aseptic loosening, mechanical patient remains free of the disease five years after operation failure, fracture of the prosthesis, or the adjacent bone [2, 15, (Table 1). 16]. Similarly, application of allografts allows mechanical and biological reconstruction after tumor excision, but they 5. Discussion are, also, associated with high rates of complications, in- cluding fracture, nonunion, transmission of disease, and Despite the fact that PAO is the most frequent surface os- teosarcoma, it is a rare bone tumor [6]. It is considered a infection [17]. Hemicortical resection of the distal femur in the early low-grade tumor that originates from the periosteum and appears with low risk for local recurrence and metastases. stage of PAOS of the distal femur was first described by Campanacci et al. in 1984 [18]. Later, this method was )e demographic characteristics of the patients included in our study are in agreement with the findings of the inter- established as a safe therapeutic procedure [10]. However, national literature as PAOS was primarily developed in due to the rarity of patients with PAO, there are limited young adult female patients. Indeed, the incidence of PAOS studies with efficient data that examined the functional is prominent in female individuals in their third decade of outcomes of this surgical intervention. Our results analysis demonstrated satisfactory oncological and functional re- life [6]. Moreover, studies reported that the most common location of the tumor is the posterior surface of the distal sults. Specifically, the mean MSTS and the range of knee flexion scores were 93.45 and 105.1 femoral metaphysis followed by the proximal tibial and , respectively. Our femoral metaphysis being in line with our results. Symptoms findings are in agreement with many studies that reported include a slow-growing painless mass at the beginning. satisfactory functional and oncological outcomes after During time, as the mass increases, the pain worsens. If the hemicortical resection of PAOS of the distal femur. In tumor is closed to a joint, the decreased range of motion particular, Lewis et al. reported very good oncological and might occur [7, 8]. functional outcomes without any complications using this Radiographically, the lesion appears as a large-lobulated technique in 6 patients with PAOS of the distal femur [11]. mass with large usually central bone formation and a thick Deijkers et al. analyzed 22 allograft hemicortical recon- mineralized stalk fixed onto the bone surface, without ex- struction procedures, also showing very satisfying onco- tension into the medullary cavity. logical and functional outcomes. All patients had good or Despite the fact that needle biopsy shows lower accuracy excellent MSTS scores that were accompanied by low than cytogenetic, molecular, immunohistochemical, or FISH complication rates. Furthermore, the incorporation rate of (fluorescence in situ hybridization) studies, in all of our 11 allograft in thirty months was observed in 100% Sarcoma 7 (a) (b) Figure 8: Parosteal osteosarcoma (PAO) in a 16-year-old female (no. 6). Anteroposterior (a) and lateral (b) radiographs four years after operation showing recurrence of PAO. postoperatively [12]. Agarwal et al. reviewed 10 patients who reconstruction method after wide excision of PAOS of the underwent hemicortical excision of the distal femur and posterior cortex of the distal femur, complications may reconstruction with autograft or allograft. In both methods, occur. )e most common complication is host bone the functional outcome was optimal without any major fracture and local recurrence, followed by nonunion and surgical complications or local recurrence [3]. Liu et al. also infection. Rarely allograft fractures can occur. )ese reported very good outcomes with relative low complication complications are associated directly with the size of the rates in 13 patients diagnosed with PAOS and managed with bone defect after wide excision and often require surgical reintervention [21]. None of our patients had a host bone wide excision and reconstruction with pasteurized hemi- cortical autograft and internal fixation. )e authors stated fracture, allograft fracture, nonunion, or infection. Local recurrence was observed in one patient and was managed that the technique, although was operational demanding, was safe and effective in selected patients [19]. with revision, and biological reconstruction with fibular Compared to endoprosthetic reconstruction after wide autograft. )is complication corresponded to the 9.1% of resection of PAOS of the distal femur, many studies dem- the patients included in the study being significantly lower onstrate, respectively, good functional and oncological than the rate of 17% that was referred in previous reports of outcomes. Funovic et al. compared 12 patients who un- the literature [22]. In addition, the revision intervention of derwent prosthetic reconstruction, with 11 patients who this patient was associated with very good functional results underwent biological reconstruction. )e authors concluded avoiding the application of endoprosthetic replacement. that the oncological and functional outcome was not altered Although the frequency of local recurrence was not cor- related with the surgical intervention or the histological between the two reconstruction methods [20]. Nevertheless, the increased rate of revision arthroplasty (58%) in the grade of the tumor [22], inadequate wide resection and endoprosthetic reconstruction group was observed when it dedifferentiated PAOS have been described as potential was compared to the biologic reconstruction group (18%) at negative predictors for its relapse [9, 22–25]. 10 years postoperatively [20]. In addition, Wilke et al. re- While hemicortical resection and allograft of the PAO of the ported that the comparison between 5 and 7 patients who posterior aspect of the distal femur avoid complications asso- underwent endoprosthetic and biological reconstruction ciated with wide distal femur resection and reconstruction such with allograft application, respectively, revealed the same as endoprosthesis, drawbacks such as a higher risk of a positive mean MSTS score (mean MSTS 23) and the similar rate of margin or recurrence should be addressed. Currently, three- complications and reoperation between the two groups [2]. dimensional measurements on the tumor before surgical re- Although in our study the application of hemicortical section using computer technology and establishing a 3-di- mensional model and a 3D printing osteotomy guide plate could allograft was not correlated with any complications sup- porting the evidence that it is a reliable biological assist in accurately resecting the tumor lesion [23]. 8 Sarcoma (a) (b) (c) (d) (e) Figure 9: Intraoperative images with complete excision of the posterior-medial part of the distal femur (a), the excised specimen (b), and polymethyl-methacrylate (PMMA) used to fill the osseous void (c). Postoperative radiographs of anteroposterior (d) and lateral (e) views showing the PMMA in the posterior-medial part of the distal femur. (a) (c) (b) Figure 10: (a) Autograft from the contralateral fibula was used and was cut in the longitudinal axis. 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SarcomaHindawi Publishing Corporation

Published: Jun 2, 2022

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