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Dedifferentiated Peripheral Chondrosarcoma: A Review of Radiologic Characteristics

Dedifferentiated Peripheral Chondrosarcoma: A Review of Radiologic Characteristics Hindawi Publishing Corporation Sarcoma Volume 2013, Article ID 505321, 6 pages http://dx.doi.org/10.1155/2013/505321 Clinical Study Dedifferentiated Peripheral Chondrosarcoma: A Review of Radiologic Characteristics 1 2 2 Eric R. Henderson, Elisa Pala, Andrea Angelini, 2 3 Eugenio Rimondi, and Pietro Ruggieri Orthopaedic Oncology, Dartmouth-Hitchcock Medical Center, eTh Geisel School of Medicine at Dartmouth, Lebanon, NH 03756, USA Istituto Ortopedico Rizzoli, 40136 Bologna, Italy University of Bologna, 40126Bologna,Italy Correspondence should be addressed to Pietro Ruggieri; pietro.ruggieri@ior.it Received 24 December 2012; Accepted 24 February 2013 Academic Editor: G. Letson Copyright © 2013 Eric R. Henderson 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. Introduction. Peripheral de-differentiated chondrosarcomas are among the rarest malignant mesenchymal tumors. This tumor’s descriptive radiographic characteristics are reported but objective quantification does not exist. This investigation surveyed imaging of peripheral de-differentiated chondrosarcomas to facilitate better recognition of these uncommon tumors. Methods. Database interrogation for peripheral de-differentiated chondrosarcomas was performed; 23 patients were identified and imaging for 18 was reviewed. A musculoskeletal radiologist reviewed all studies for mineralization characteristics; presence of pre-existing osteo- chondromas; preserved corticomedullary continuity; adjacent cortical obliteration; soft-tissue mass; tumor necrosis; and presence of a cartilage cap. Tumor luminance was measured with computer software. Results. Mineralization was present in 17 tumors. Pre- existing exostoses were evident in nine cases, corticomedullary continuity was preserved in three cases. There was no difference in mineralization or other characteristics based on tumor location. Mean tumor luminance was 94.9 candela/m . Conclusions. The imaging characteristics described for central de-differentiated chondrosarcomas are similar to the peripheral form of this tumor. Peripheral mineralization with a bimorphic pattern on CT scan and the presence of a soft-tissue mass should be considered worri- some for a peripheral de-differentiated chondrosarcoma, particularly in the setting of multiple hereditary exostoses. 1. Introduction on descriptive reporting of histologic subtypes and patient survival [3, 4, 6–18]. Dedifferentiated chondrosarcoma is an uncommon tumor A consistent radiographic appearance of conventional, that is known to arise from preexisting, low-grade cartilage central dedifferentiated chondrosarcoma is recognized and lesions [1–5]. This tumor demonstrates bimorphic histol- described [19]; as a result radiologists and surgeons trained ogywithawell-dieff rentiated cartilaginouscomponent and in musculoskeletal imaging are able to identify these lesions a dedifferentiated, noncartilaginous component [ 4]. These successfully. The typical radiographic description of a central lesions comprise approximately 11% of chondrosarcomas and dedifferentiated chondrosarcoma is a lesion that originates generally occur in association with a central chondrosarcoma within bone with an area of cortical breach and subsequent [3]. Because of its cartilaginous origin, dedifferentiated chon- soft-tissue mass demonstrating a bimorphic pattern with drosarcoma may also occur in the setting of a preexisting mineralized and unmineralized areas; pathologic fracture is exostosis; however, the occurrence is rare [6]. When single- common [19]. Unlike the more common, central lesions, institution, redundant reporting is considered, approximately peripheral dedifferentiated chondrosarcomas arise from pre- 60 discrete cases of peripheral dedifferentiated chondrosar- coma have been reported in limited series with an emphasis existing exostoses or, extracortically, and may appear as 2 Sarcoma a peripheral chondrosarcoma without the features of its ded- Tumor luminance was measured in an eo ff rt to objectively ifferentiated counterpart [ 19]. quantify tumor mineralization content on plain radiographs. Descriptive reporting of radiologic findings has been Luminance is a measurement of brightness with units of undertaken in some case reports and limited series; however, candela per square meter; it is measured on a scale of zero, or objective quantification of radiographic characteristics for completely black, to 255, or completely white. Luminance has peripheral dedifferentiated chondrosarcoma has not been been used in previous investigations to measure trabecular performed. eTh varied descriptions of this tumor have and so-t ft issue density with both radiography and ultrasound included comparisons to a normal osteochondroma, a low- [21, 22]. Radiographs intended for analysis were displayed grade chondrosarcoma, and a conventional dedifferentiated on a conventional light box (Dupix, Milano, Italy) and chondrosarcoma [19, 20]. The purposes of this investigation photographed with a 12-megapixel digital camera at 50 cm were to quantify and describe the radiographic findings of a range (Canon A1100IS, Canon USA, Lake Success, NY, USA). large series of patients with peripheral dedifferentiated chon- Digital images were saved as Joint Photographic Experts drosarcomas to determine whether this tumor has a distinct, Group (JPEG) files without compression and were opened recognizable radiographic appearance. with GNU Image Manipulation Program version 2.8 (GIMP Developers, Groton, MA, USA). The manual selection tool was used to trace the periphery of the tumor, and the histo- gram function was used to measure the mean luminance of 2. Patients and Methods thetumor.Theselection wastheninvertedtomeasure the After Investigational Review Board and Ethics Committee mean luminance of the surrounding soft tissues. Luminance of the soft tissues was subtracted from tumor luminance to approvals were obtained, the senior investigator’s institu- tional database was queried for patients treated between 1980 yield a measurement of net tumor luminance. es Th e numbers and 2012 with a diagnosis of peripheral dedifferentiated chon- were recorded in a spreadsheet (Microsoft Excel for Mac 2011, drosarcoma; 23 patients were identified. Five patients were Microsoft Inc., Redmond, WA, USA). Means were compared encounteredonlyinconsultation, andnoimaging studies statistically with the Student’s𝑡 test. were available, leaving 18 patients for consideration. The mean age of the patients at the time of operation was 46.4 years (range from 22.9 to 70.0 years). eTh re were 13 men 3. Results and ve fi women. Nine patients’ tumors arose from an exos- tosis in the setting of multiple hereditary exostoses (MHE), Plain radiographs revealed mineralization was seen in 16 of eight lesions arose from a preexisting solitary exostosis, 17 cases; in eight cases it appeared to be bimorphic (Table 1). and one lesion arose peripherally without an exostosis. eTh Mineralization appeared to occupy more than half of the cartilaginous component of the tumors was chondrosarcoma tumor area in 10 patients and less than half in six patients. A in all patients. The histologic subtype of the dedifferen- so-ft tissue mass was seen in 14 of 17 cases. Preexisting exos- tiated, noncartilaginous component was malignant b fi rous toses at the tumors’ origin could be identiefi d in eight of 17 histiocytoma-like (MFH) in 11 patients, osteosarcoma-like in cases; corticomedullary continuity appeared to be preserved vfi e patients, and spindle-cell sarcoma-like in two patients. in three cases. Thirteen cases showed evidence of adjacent eTh re was one lesion of the sternum, two of the scapula, three cortical erosion. No patients had a pathologic fracture. of the humerus, ve fi of the pelvis, four of the femur, two of the Dedifferentiated, noncartilaginous histologic subtypes of tibia, andone of thefibula. patients in this series included 11 with MFH-like components, Preoperative imaging studies of the lesion included plain vfi e with osteosarcoma-like components, and two patients radiographs alone for vfi e patients; a computed tomography with a spindle-cell sarcoma-like components. Mineralization (CT) scan alone for one patient; plain radiographs and CT wasseenin10of11patientswithMFH-liketumors, andinall for eight patients; and plain radiographs, CT, and magnetic patients with osteosarcoma-like and spindle-cell-like tumors, resonance imaging (MRI) for four patients. An attending- there was no statistical difference between groups ( Table 1). level musculoskeletal radiologist evaluated all imaging stud- There were eight axial lesions, seven of which demon- ies. Plain radiographs were assessed for the presence of min- strated mineralization. All appendicular lesions showed min- eralization, whether mineralization appeared to encompass eralization. There was no difference in the occurrence of min- greaterorlessthan50% of thetumor area,abimorphic eralization when results were divided by tumor location pattern of mineralization, the presence of a soft-tissue mass, (Table 2). evidence of a preexisting exostosis, evidence of preserved corticomedullary continuity when an exostosis was present, Computed tomography demonstrated mineralization in extracompartmental extension, and erosion of the adjacent 12 of 13 scans, it was thought to be bimorphic in 11 cases. cortex. CT and MRI scans were assessed for the presence of The only CT to not demonstrate mineralization was the case mineralization, whether mineralization was central, periph- which did not show mineralization on plain radiographs. eral, or both; a bimorphic pattern of mineralization; the pres- Cross-sectional imaging showed the mineralization to be ence of a so-t ft issue mass; evidence of a preexisting exostosis; peripheral-only in four cases and central and peripheral in preserved corticomedullary continuity; extracompartmental eight cases; there were no cases of central-only mineraliza- extension; erosion of the adjacent cortex; the presence and tion. A so-ft tissue mass was identiefi d in all 13 CT scans. A thickness of a cartilage cap, and necrosis. preexisting exostosis was seen in six CT scans; four of these Sarcoma 3 Table 1: Plain radiograph findings by histologic subtype. Mineralization Soft-tissue mass Exostosis Tumor Cortical Corticomedullary luminance Present <50% >50% Bimorphic Present Extracompartmental Present obliteration continuity MFH-like 9 2 7 4 10 10 8 7 1 103.9 subtype OSA-like 5 3 2 3 5 5 4 3 2 90.4 subtype SCS-like 21 1 1 2 2 1 1 0 67.6 subtype Abbreviations: malignant brou fi s histiocytoma (MFH); osteosarcoma (OSA); spindle-cell sarcoma (SCS). Table 2: Plain radiograph findings by tumor location. Mineralization Soft-tissue mass Exostosis Tumor Cortical Corticomedullary luminance Present <50% >50% Bimorphic Present Extracompartmental Present obliteration continuity Axial 6 2 4 5 7 7 8 5 2 93.2 Appendicular 10 4 6 3 10 10 5 6 1 95.8 Abbreviations: malignant brou fi s histiocytoma (MFH); osteosarcoma (OSA); spindle-cell sarcoma (SCS). Table 3: CT findings by histologic subtype. Mineralization Soft-tissue mass Exostosis Cortical Corticomedullary Cartilage Present Peripheral Central Bimorphic Present Extracompartmental Necrosis Present obliteration continuity cap MFH- like 66 2 6 7 7 7 2 2 1 1 subtype OSA- like 44 2 3 4 4 3 1 3 2 1 subtype SCS- like 222 2 2 2 1 0 1 0 1 subtype Abbreviations: malignant brou fi s histiocytoma (MFH); osteosarcoma (OSA); spindle-cell sarcoma (SCS). exostoses were identified on the corresponding plain radio- (𝑃 = 0.14 ), however, limited patient numbers precluded graphs, and two were not identified on plain radiographs. robust statistical analysis (Table 1). Corticomedullary continuity was preserved in three cases with identifiable exostoses. Central necrosis was identified in three CT and MRI studies performed with contrast. A car- 4. Discussion tilage cap was identified in three lesions. There was no dif- ference when results were divided by histologic subtype or Anderson and coauthors reported the earliest description of tumor location (Tables 3 and 4). a peripheral dedifferentiated chondrosarcoma and noted that Tumor luminance was measured on plain radiographs in itsradiographiccharacteristics were consistent with osteo- 14 cases. Mean tumor luminance without soft-tissue subtrac- chondroma [20]. Since that time small series of peripheral tion was 138.7 candela/m (range, 70.5 to 201.2). Mean adja- dedifferentiated chondrosarcomas have been published with cent soft-tissue luminance was 43.8 candela/m (range from descriptive accounts of this tumor’s radiographic appear- 11.1 to 140.0); therefore mean luminance for the tumors alone ance but without objective quantification of findings. The was 94.9 candela/m (range from 59.4 to 129.0), indicating present study demonstrates that mineralization is present in that tumor opacity on radiographs was approximately 37%. the majority of peripheral dedifferentiated chondrosarcomas When compared by histological subtype, MFH-like and OSA- (Figure 1). Mineralization patterns, best visualized with CT like tumor luminance showed no statistical difference ( 𝑃= scan, are usually central and peripheral or peripheral-only 0.49). When spindle cell-like tumors were compared to MFH- (Figure 2). A bimorphic mineralization pattern was demons- like andOSA-like,the dieff renceapproachedsignicfi ance trated more reliably with CT than plain radiographs; however, 4 Sarcoma Table 4: CT findings by tumor location. Mineralization Soft-tissue mass Exostosis Cortical Corticomedullary Cartilage Present Peripheral Central Bimorphic Present Extracompartmental Necrosis Present obliteration continuity cap Axial 6 6 5 5 7 7 7 2 2 2 2 Appen- 66 3 6 6 6 4 1 4 1 1 dicular Sin (a) (b) Figure 1: (a) AP and (b) lateral radiographs of tibia and fibula demonstrating a peripheral dedifferentiated chondrosarcoma with mineralization and a soft-tissue mass. scalloping, cortical destruction, and a so-ft tissue mass [ 23]. Garrison and coauthors were the first to describe a large series of secondary chondrosarcomas arising from osteo- chondromas. Radiographic features consistent with malig- nant degeneration included an indistinct superficial border, thepresenceofapartiallymineralized so-ft tissuemass, and frequent destruction of the underlying osteochondroma [13]. Wuisman and coauthors mentioned only blurring of the bone borders as an indicator of malignant transformation [24]. Ahmed and coauthors, in a series of 107 patients with secondary chondrosarcomas arising from exostoses, doc- umented irregular margins, heterogeneous mineralization, andaso-ft tissuemassaspositiveindicatorsofmalignant Figure 2: Axial CT scan of tibia and b fi ula demonstrating a periph- change in an osteochondroma [25]. Altay and coauthors eral dedifferentiated chondrosarcoma with central and periph- reported a series of 32 patients with malignant degeneration eral, bimorphic mineralization, destruction of the prior exostosis, of an osteochondroma but did not comment on radiological and a soft-tissue mass. features [26]. Mercuri and coauthors reported that the imaging characteristics of central dedifferentiated chondrosarcoma it cannot be relied upon as a definite indicator of tumor de- depended on the preexisting cartilage tumor [19]. They noted that when the noncartilaginous component was small, the differentiation. Obliterations of the preexisting exostosis or adjacent cortex are common ndin fi gs; however, the presence imaging ndin fi gs oen ft reflected a conventional chondro- sarcoma. When the dedifferentiated, noncartilaginous com- of a so-ft tissue mass on CT or MRI was the most consistent ponent was larger, however, the tumor often demonstrated radiographic feature associated with peripheral dedifferenti- ated chondrosarcomas (Figure 3). no discernible radiographic characteristics of a cartilaginous neoplasm. eTh two features they found most commonly on The radiographic characteristics of conventional, central plain radiographs were permeative osteolysis and a so-ft tissue chondrosarcomas are known and include deep endosteal Sarcoma 5 (a) (b) Figure 3: Axial MRI scan of tibia and b fi ula demonstrating a peripheral dedieff rentiated chondrosarcoma with soft-tissue mass and heterogeneous T2 weighted (a) and T1 weighted with contrast (b) enhancement. mass. eTh largest series to address radiographic features of dominate [2, 3, 16, 28]. Other reports have documented a central dedifferentiated chondrosarcomas was published by rate of MFH-like histologic subtypes greater than 50% in Littrell and coauthors [16]. eTh authors reported cortical the peripheral form of dedifferentiated chondrosarcoma, destruction, chondroid matrix, so-ft tissue mass, and tumor indicating that the peripheral form may have a predilection bimorphism were the most common ndings fi associated for this morphology [7, 12, 15]. with these tumors; as in the present study, CT was more This investigation has limitations that warrant discus- sensitive in detecting the mineralized component as well as sion. Our study details the radiologic ndin fi gs of eighteen demonstrating bimorphism. Radiographic characteristics patients treated over a 33-year interval, during which imaging described by Johnson and coauthors included a lytic lesion technology changed substantially, creating a heterogeneous with cortical destruction and a so-ft tissue mass [ 27]. mix of radiographic studies. While our case number is small Several case series of dedifferentiated chondrosarcoma and underpowered to truly ascertain statistical differences have included mixed reporting of central and peripheral in the radiographic appearances of these rare lesions, this tumors. Discrete accounts of radiologic features for peri- study represents the largest and only investigation dedicated pheral dedifferentiated chondrosarcoma include few case to peripheral dedifferentiated chondrosarcoma imaging, and reports and limited case series. Cortical destruction was the we believe that the results justify reporting. Measurement first described harbinger of de-differentiation [ 17]. Bertoni of tumor luminance in this investigation was an attempt to and coauthors published the earliest series of peripheral quantify tumor opacity, and therefore its mineralized content dedifferentiated chondrosarcomas and described a preexist- is relative to the surrounding soft tissues. Further investi- ing osteochondroma, cortical destruction, and a soft-tissue gations correlating this technique with quantitative CT are mass as consistent n fi dings among all tumors [ 6]. Staals required to determine its usefulness and validate the results; and coauthors described the largest series of peripheral ded- however, the authors believe that it may prove a useful tech- ifferentiated chondrosarcomas. Radiographs of all patients nique for quantitifying mineralization in the absence of demonstrated indistinct borders, heterogeneous mineraliza- advanced, three-dimensional imaging. The authors acknowl- tion, and a soft-tissue mass [ 7]. Bimorphic mineralization was edge that luminance in isolation does not provide radiologists notedinhalfoftheirpatientsand,similartothecurrentstudy, and surgeons with objective criteria for ruling periphe- was more evident with CT than plain radiographs. ral dedifferentiated chondrosarcoma in or out as a diagno- The current investigation confirms that the primary sis; however, it does provide an objective starting point for radiologic features of central dedifferentiated chondrosar- comparison to other tumors that could lead to such para- coma, so-t ft issue mass, heterogeneous mineralization, and meters. bimorphism are similar to the less common peripheral lesion morphology (Figure 1). Secondary features of central tumors including intramedullary mineralization with an 5. Conclusion extramedullary, radiolucent so-t ft issue mass and pathologic fracture [19] are uncommon with peripheral lesions, likely In general the imaging characteristics described for cen- due to the origin of the lesion outside of the medullary space. tral dedifferentiated chondrosarcomas are applicable to the Over one-half of the cases in the current series showed an peripheral form of this tumor. Peripheral mineralization with MFH-like histologic morphology. This finding diverges from a bimorphic pattern on CT scan and the presence of a soft- the largest reports of central dedifferentiated chondrosar- tissue mass should be considered worrisome for a peripheral coma where MFH-like features comprised from 4% to 22% of dedifferentiated chondrosarcoma, particularly in the setting the total cases, and osteosarcoma-like characteristics usually of multiple hereditary exostoses. 6 Sarcoma References [18] S. A. Voutsinas, “Spindle-cell sarcoma in patients who have osteochondromatosis. 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Dedifferentiated Peripheral Chondrosarcoma: A Review of Radiologic Characteristics

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Copyright © 2013 Eric R. Henderson 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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Hindawi Publishing Corporation Sarcoma Volume 2013, Article ID 505321, 6 pages http://dx.doi.org/10.1155/2013/505321 Clinical Study Dedifferentiated Peripheral Chondrosarcoma: A Review of Radiologic Characteristics 1 2 2 Eric R. Henderson, Elisa Pala, Andrea Angelini, 2 3 Eugenio Rimondi, and Pietro Ruggieri Orthopaedic Oncology, Dartmouth-Hitchcock Medical Center, eTh Geisel School of Medicine at Dartmouth, Lebanon, NH 03756, USA Istituto Ortopedico Rizzoli, 40136 Bologna, Italy University of Bologna, 40126Bologna,Italy Correspondence should be addressed to Pietro Ruggieri; pietro.ruggieri@ior.it Received 24 December 2012; Accepted 24 February 2013 Academic Editor: G. Letson Copyright © 2013 Eric R. Henderson 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. Introduction. Peripheral de-differentiated chondrosarcomas are among the rarest malignant mesenchymal tumors. This tumor’s descriptive radiographic characteristics are reported but objective quantification does not exist. This investigation surveyed imaging of peripheral de-differentiated chondrosarcomas to facilitate better recognition of these uncommon tumors. Methods. Database interrogation for peripheral de-differentiated chondrosarcomas was performed; 23 patients were identified and imaging for 18 was reviewed. A musculoskeletal radiologist reviewed all studies for mineralization characteristics; presence of pre-existing osteo- chondromas; preserved corticomedullary continuity; adjacent cortical obliteration; soft-tissue mass; tumor necrosis; and presence of a cartilage cap. Tumor luminance was measured with computer software. Results. Mineralization was present in 17 tumors. Pre- existing exostoses were evident in nine cases, corticomedullary continuity was preserved in three cases. There was no difference in mineralization or other characteristics based on tumor location. Mean tumor luminance was 94.9 candela/m . Conclusions. The imaging characteristics described for central de-differentiated chondrosarcomas are similar to the peripheral form of this tumor. Peripheral mineralization with a bimorphic pattern on CT scan and the presence of a soft-tissue mass should be considered worri- some for a peripheral de-differentiated chondrosarcoma, particularly in the setting of multiple hereditary exostoses. 1. Introduction on descriptive reporting of histologic subtypes and patient survival [3, 4, 6–18]. Dedifferentiated chondrosarcoma is an uncommon tumor A consistent radiographic appearance of conventional, that is known to arise from preexisting, low-grade cartilage central dedifferentiated chondrosarcoma is recognized and lesions [1–5]. This tumor demonstrates bimorphic histol- described [19]; as a result radiologists and surgeons trained ogywithawell-dieff rentiated cartilaginouscomponent and in musculoskeletal imaging are able to identify these lesions a dedifferentiated, noncartilaginous component [ 4]. These successfully. The typical radiographic description of a central lesions comprise approximately 11% of chondrosarcomas and dedifferentiated chondrosarcoma is a lesion that originates generally occur in association with a central chondrosarcoma within bone with an area of cortical breach and subsequent [3]. Because of its cartilaginous origin, dedifferentiated chon- soft-tissue mass demonstrating a bimorphic pattern with drosarcoma may also occur in the setting of a preexisting mineralized and unmineralized areas; pathologic fracture is exostosis; however, the occurrence is rare [6]. When single- common [19]. Unlike the more common, central lesions, institution, redundant reporting is considered, approximately peripheral dedifferentiated chondrosarcomas arise from pre- 60 discrete cases of peripheral dedifferentiated chondrosar- coma have been reported in limited series with an emphasis existing exostoses or, extracortically, and may appear as 2 Sarcoma a peripheral chondrosarcoma without the features of its ded- Tumor luminance was measured in an eo ff rt to objectively ifferentiated counterpart [ 19]. quantify tumor mineralization content on plain radiographs. Descriptive reporting of radiologic findings has been Luminance is a measurement of brightness with units of undertaken in some case reports and limited series; however, candela per square meter; it is measured on a scale of zero, or objective quantification of radiographic characteristics for completely black, to 255, or completely white. Luminance has peripheral dedifferentiated chondrosarcoma has not been been used in previous investigations to measure trabecular performed. eTh varied descriptions of this tumor have and so-t ft issue density with both radiography and ultrasound included comparisons to a normal osteochondroma, a low- [21, 22]. Radiographs intended for analysis were displayed grade chondrosarcoma, and a conventional dedifferentiated on a conventional light box (Dupix, Milano, Italy) and chondrosarcoma [19, 20]. The purposes of this investigation photographed with a 12-megapixel digital camera at 50 cm were to quantify and describe the radiographic findings of a range (Canon A1100IS, Canon USA, Lake Success, NY, USA). large series of patients with peripheral dedifferentiated chon- Digital images were saved as Joint Photographic Experts drosarcomas to determine whether this tumor has a distinct, Group (JPEG) files without compression and were opened recognizable radiographic appearance. with GNU Image Manipulation Program version 2.8 (GIMP Developers, Groton, MA, USA). The manual selection tool was used to trace the periphery of the tumor, and the histo- gram function was used to measure the mean luminance of 2. Patients and Methods thetumor.Theselection wastheninvertedtomeasure the After Investigational Review Board and Ethics Committee mean luminance of the surrounding soft tissues. Luminance of the soft tissues was subtracted from tumor luminance to approvals were obtained, the senior investigator’s institu- tional database was queried for patients treated between 1980 yield a measurement of net tumor luminance. es Th e numbers and 2012 with a diagnosis of peripheral dedifferentiated chon- were recorded in a spreadsheet (Microsoft Excel for Mac 2011, drosarcoma; 23 patients were identified. Five patients were Microsoft Inc., Redmond, WA, USA). Means were compared encounteredonlyinconsultation, andnoimaging studies statistically with the Student’s𝑡 test. were available, leaving 18 patients for consideration. The mean age of the patients at the time of operation was 46.4 years (range from 22.9 to 70.0 years). eTh re were 13 men 3. Results and ve fi women. Nine patients’ tumors arose from an exos- tosis in the setting of multiple hereditary exostoses (MHE), Plain radiographs revealed mineralization was seen in 16 of eight lesions arose from a preexisting solitary exostosis, 17 cases; in eight cases it appeared to be bimorphic (Table 1). and one lesion arose peripherally without an exostosis. eTh Mineralization appeared to occupy more than half of the cartilaginous component of the tumors was chondrosarcoma tumor area in 10 patients and less than half in six patients. A in all patients. The histologic subtype of the dedifferen- so-ft tissue mass was seen in 14 of 17 cases. Preexisting exos- tiated, noncartilaginous component was malignant b fi rous toses at the tumors’ origin could be identiefi d in eight of 17 histiocytoma-like (MFH) in 11 patients, osteosarcoma-like in cases; corticomedullary continuity appeared to be preserved vfi e patients, and spindle-cell sarcoma-like in two patients. in three cases. Thirteen cases showed evidence of adjacent eTh re was one lesion of the sternum, two of the scapula, three cortical erosion. No patients had a pathologic fracture. of the humerus, ve fi of the pelvis, four of the femur, two of the Dedifferentiated, noncartilaginous histologic subtypes of tibia, andone of thefibula. patients in this series included 11 with MFH-like components, Preoperative imaging studies of the lesion included plain vfi e with osteosarcoma-like components, and two patients radiographs alone for vfi e patients; a computed tomography with a spindle-cell sarcoma-like components. Mineralization (CT) scan alone for one patient; plain radiographs and CT wasseenin10of11patientswithMFH-liketumors, andinall for eight patients; and plain radiographs, CT, and magnetic patients with osteosarcoma-like and spindle-cell-like tumors, resonance imaging (MRI) for four patients. An attending- there was no statistical difference between groups ( Table 1). level musculoskeletal radiologist evaluated all imaging stud- There were eight axial lesions, seven of which demon- ies. Plain radiographs were assessed for the presence of min- strated mineralization. All appendicular lesions showed min- eralization, whether mineralization appeared to encompass eralization. There was no difference in the occurrence of min- greaterorlessthan50% of thetumor area,abimorphic eralization when results were divided by tumor location pattern of mineralization, the presence of a soft-tissue mass, (Table 2). evidence of a preexisting exostosis, evidence of preserved corticomedullary continuity when an exostosis was present, Computed tomography demonstrated mineralization in extracompartmental extension, and erosion of the adjacent 12 of 13 scans, it was thought to be bimorphic in 11 cases. cortex. CT and MRI scans were assessed for the presence of The only CT to not demonstrate mineralization was the case mineralization, whether mineralization was central, periph- which did not show mineralization on plain radiographs. eral, or both; a bimorphic pattern of mineralization; the pres- Cross-sectional imaging showed the mineralization to be ence of a so-t ft issue mass; evidence of a preexisting exostosis; peripheral-only in four cases and central and peripheral in preserved corticomedullary continuity; extracompartmental eight cases; there were no cases of central-only mineraliza- extension; erosion of the adjacent cortex; the presence and tion. A so-ft tissue mass was identiefi d in all 13 CT scans. A thickness of a cartilage cap, and necrosis. preexisting exostosis was seen in six CT scans; four of these Sarcoma 3 Table 1: Plain radiograph findings by histologic subtype. Mineralization Soft-tissue mass Exostosis Tumor Cortical Corticomedullary luminance Present <50% >50% Bimorphic Present Extracompartmental Present obliteration continuity MFH-like 9 2 7 4 10 10 8 7 1 103.9 subtype OSA-like 5 3 2 3 5 5 4 3 2 90.4 subtype SCS-like 21 1 1 2 2 1 1 0 67.6 subtype Abbreviations: malignant brou fi s histiocytoma (MFH); osteosarcoma (OSA); spindle-cell sarcoma (SCS). Table 2: Plain radiograph findings by tumor location. Mineralization Soft-tissue mass Exostosis Tumor Cortical Corticomedullary luminance Present <50% >50% Bimorphic Present Extracompartmental Present obliteration continuity Axial 6 2 4 5 7 7 8 5 2 93.2 Appendicular 10 4 6 3 10 10 5 6 1 95.8 Abbreviations: malignant brou fi s histiocytoma (MFH); osteosarcoma (OSA); spindle-cell sarcoma (SCS). Table 3: CT findings by histologic subtype. Mineralization Soft-tissue mass Exostosis Cortical Corticomedullary Cartilage Present Peripheral Central Bimorphic Present Extracompartmental Necrosis Present obliteration continuity cap MFH- like 66 2 6 7 7 7 2 2 1 1 subtype OSA- like 44 2 3 4 4 3 1 3 2 1 subtype SCS- like 222 2 2 2 1 0 1 0 1 subtype Abbreviations: malignant brou fi s histiocytoma (MFH); osteosarcoma (OSA); spindle-cell sarcoma (SCS). exostoses were identified on the corresponding plain radio- (𝑃 = 0.14 ), however, limited patient numbers precluded graphs, and two were not identified on plain radiographs. robust statistical analysis (Table 1). Corticomedullary continuity was preserved in three cases with identifiable exostoses. Central necrosis was identified in three CT and MRI studies performed with contrast. A car- 4. Discussion tilage cap was identified in three lesions. There was no dif- ference when results were divided by histologic subtype or Anderson and coauthors reported the earliest description of tumor location (Tables 3 and 4). a peripheral dedifferentiated chondrosarcoma and noted that Tumor luminance was measured on plain radiographs in itsradiographiccharacteristics were consistent with osteo- 14 cases. Mean tumor luminance without soft-tissue subtrac- chondroma [20]. Since that time small series of peripheral tion was 138.7 candela/m (range, 70.5 to 201.2). Mean adja- dedifferentiated chondrosarcomas have been published with cent soft-tissue luminance was 43.8 candela/m (range from descriptive accounts of this tumor’s radiographic appear- 11.1 to 140.0); therefore mean luminance for the tumors alone ance but without objective quantification of findings. The was 94.9 candela/m (range from 59.4 to 129.0), indicating present study demonstrates that mineralization is present in that tumor opacity on radiographs was approximately 37%. the majority of peripheral dedifferentiated chondrosarcomas When compared by histological subtype, MFH-like and OSA- (Figure 1). Mineralization patterns, best visualized with CT like tumor luminance showed no statistical difference ( 𝑃= scan, are usually central and peripheral or peripheral-only 0.49). When spindle cell-like tumors were compared to MFH- (Figure 2). A bimorphic mineralization pattern was demons- like andOSA-like,the dieff renceapproachedsignicfi ance trated more reliably with CT than plain radiographs; however, 4 Sarcoma Table 4: CT findings by tumor location. Mineralization Soft-tissue mass Exostosis Cortical Corticomedullary Cartilage Present Peripheral Central Bimorphic Present Extracompartmental Necrosis Present obliteration continuity cap Axial 6 6 5 5 7 7 7 2 2 2 2 Appen- 66 3 6 6 6 4 1 4 1 1 dicular Sin (a) (b) Figure 1: (a) AP and (b) lateral radiographs of tibia and fibula demonstrating a peripheral dedifferentiated chondrosarcoma with mineralization and a soft-tissue mass. scalloping, cortical destruction, and a so-ft tissue mass [ 23]. Garrison and coauthors were the first to describe a large series of secondary chondrosarcomas arising from osteo- chondromas. Radiographic features consistent with malig- nant degeneration included an indistinct superficial border, thepresenceofapartiallymineralized so-ft tissuemass, and frequent destruction of the underlying osteochondroma [13]. Wuisman and coauthors mentioned only blurring of the bone borders as an indicator of malignant transformation [24]. Ahmed and coauthors, in a series of 107 patients with secondary chondrosarcomas arising from exostoses, doc- umented irregular margins, heterogeneous mineralization, andaso-ft tissuemassaspositiveindicatorsofmalignant Figure 2: Axial CT scan of tibia and b fi ula demonstrating a periph- change in an osteochondroma [25]. Altay and coauthors eral dedifferentiated chondrosarcoma with central and periph- reported a series of 32 patients with malignant degeneration eral, bimorphic mineralization, destruction of the prior exostosis, of an osteochondroma but did not comment on radiological and a soft-tissue mass. features [26]. Mercuri and coauthors reported that the imaging characteristics of central dedifferentiated chondrosarcoma it cannot be relied upon as a definite indicator of tumor de- depended on the preexisting cartilage tumor [19]. They noted that when the noncartilaginous component was small, the differentiation. Obliterations of the preexisting exostosis or adjacent cortex are common ndin fi gs; however, the presence imaging ndin fi gs oen ft reflected a conventional chondro- sarcoma. When the dedifferentiated, noncartilaginous com- of a so-ft tissue mass on CT or MRI was the most consistent ponent was larger, however, the tumor often demonstrated radiographic feature associated with peripheral dedifferenti- ated chondrosarcomas (Figure 3). no discernible radiographic characteristics of a cartilaginous neoplasm. eTh two features they found most commonly on The radiographic characteristics of conventional, central plain radiographs were permeative osteolysis and a so-ft tissue chondrosarcomas are known and include deep endosteal Sarcoma 5 (a) (b) Figure 3: Axial MRI scan of tibia and b fi ula demonstrating a peripheral dedieff rentiated chondrosarcoma with soft-tissue mass and heterogeneous T2 weighted (a) and T1 weighted with contrast (b) enhancement. mass. eTh largest series to address radiographic features of dominate [2, 3, 16, 28]. Other reports have documented a central dedifferentiated chondrosarcomas was published by rate of MFH-like histologic subtypes greater than 50% in Littrell and coauthors [16]. eTh authors reported cortical the peripheral form of dedifferentiated chondrosarcoma, destruction, chondroid matrix, so-ft tissue mass, and tumor indicating that the peripheral form may have a predilection bimorphism were the most common ndings fi associated for this morphology [7, 12, 15]. with these tumors; as in the present study, CT was more This investigation has limitations that warrant discus- sensitive in detecting the mineralized component as well as sion. Our study details the radiologic ndin fi gs of eighteen demonstrating bimorphism. Radiographic characteristics patients treated over a 33-year interval, during which imaging described by Johnson and coauthors included a lytic lesion technology changed substantially, creating a heterogeneous with cortical destruction and a so-ft tissue mass [ 27]. mix of radiographic studies. While our case number is small Several case series of dedifferentiated chondrosarcoma and underpowered to truly ascertain statistical differences have included mixed reporting of central and peripheral in the radiographic appearances of these rare lesions, this tumors. Discrete accounts of radiologic features for peri- study represents the largest and only investigation dedicated pheral dedifferentiated chondrosarcoma include few case to peripheral dedifferentiated chondrosarcoma imaging, and reports and limited case series. Cortical destruction was the we believe that the results justify reporting. Measurement first described harbinger of de-differentiation [ 17]. Bertoni of tumor luminance in this investigation was an attempt to and coauthors published the earliest series of peripheral quantify tumor opacity, and therefore its mineralized content dedifferentiated chondrosarcomas and described a preexist- is relative to the surrounding soft tissues. Further investi- ing osteochondroma, cortical destruction, and a soft-tissue gations correlating this technique with quantitative CT are mass as consistent n fi dings among all tumors [ 6]. Staals required to determine its usefulness and validate the results; and coauthors described the largest series of peripheral ded- however, the authors believe that it may prove a useful tech- ifferentiated chondrosarcomas. Radiographs of all patients nique for quantitifying mineralization in the absence of demonstrated indistinct borders, heterogeneous mineraliza- advanced, three-dimensional imaging. The authors acknowl- tion, and a soft-tissue mass [ 7]. Bimorphic mineralization was edge that luminance in isolation does not provide radiologists notedinhalfoftheirpatientsand,similartothecurrentstudy, and surgeons with objective criteria for ruling periphe- was more evident with CT than plain radiographs. ral dedifferentiated chondrosarcoma in or out as a diagno- The current investigation confirms that the primary sis; however, it does provide an objective starting point for radiologic features of central dedifferentiated chondrosar- comparison to other tumors that could lead to such para- coma, so-t ft issue mass, heterogeneous mineralization, and meters. bimorphism are similar to the less common peripheral lesion morphology (Figure 1). Secondary features of central tumors including intramedullary mineralization with an 5. Conclusion extramedullary, radiolucent so-t ft issue mass and pathologic fracture [19] are uncommon with peripheral lesions, likely In general the imaging characteristics described for cen- due to the origin of the lesion outside of the medullary space. tral dedifferentiated chondrosarcomas are applicable to the Over one-half of the cases in the current series showed an peripheral form of this tumor. Peripheral mineralization with MFH-like histologic morphology. This finding diverges from a bimorphic pattern on CT scan and the presence of a soft- the largest reports of central dedifferentiated chondrosar- tissue mass should be considered worrisome for a peripheral coma where MFH-like features comprised from 4% to 22% of dedifferentiated chondrosarcoma, particularly in the setting the total cases, and osteosarcoma-like characteristics usually of multiple hereditary exostoses. 6 Sarcoma References [18] S. A. Voutsinas, “Spindle-cell sarcoma in patients who have osteochondromatosis. 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