Akio Sakamoto

BM Gallagher

CR Antonescu

EY Cheng

GR Mundy

H Zhang

JH Schwab

JL Noble

JM Woolfenden

K Sheah

Katsumi Harimaya

PR Algra

SH Estourgie

T Taoka

TA Smith

Y Nishida

Yoshiaki Fukutoku

Yoshihiro Matsumoto

Yoshinao Oda

Yukihide Iwamoto

World Journal of Surgical Oncology

English

Crossref

WORLD JOURNAL OF SURGICAL ONCOLOGY Sakamoto et al. World Journal of Surgical Oncology 2012, 10:214http://www.wjso.com/content/10/1/214CASE REPORT Open AccessMyxoid liposarcoma with negative features onbone scan and [18F]-2-fluoro-2-deoxy-D-glucose-positron emission tomographyAkio Sakamoto1,2*, Yoshiaki Fukutoku2, Yoshihiro Matsumoto2, Katsumi Harimaya2, Yoshinao Oda3and Yukihide Iwamoto2AbstractBackground: Myxoid liposarcoma occurs in middle age, and is characterized by extrapulmonary metastasis,including bone metastasis. Bone scans and [18F]-2-fluoro-2-deoxy-D-glucose-positron emission tomography(FDG-PET) are widely used for assessment of tumor extension, including vertebral metastasis. However, bothmethods have a low positive rate with regard to vertebral metastasis arising from myxoid liposarcoma. This isparticularly true for bone scans for intramedullary lesions that have no cortical involvement.Case presentation: We present the case of a 53-year-old male with myxoid liposarcoma in the leg. He had beentreated for multiple metastases over a ten-year period, and was experiencing back pain due to a pathologicalfracture in the second lumbar vertebra (L2). Magnetic resonance imaging of all the vertebrae showed abnormalsignal intensity suggestive of metastasis in eight vertebrae, and revealed extraskeletal extension in three vertebrae.Bone scans and FDG-PET were negative except for the L2 fracture which was indicated on a bone scan.Conclusions: Both bone scans and FDG-PET can be negative in cases of vertebral metastasis that arise frommyxoid liposarcoma, even when extraskeletal extensions are present. Similarly, even a fractured vertebra may notalways be visible on FDG-PET.Keywords: Myxoid liposarcoma, Bone scan, FDG-PET, Vertebral metastasisBackgroundMyxoid liposarcoma is one of the subtypes of liposar-coma. This subtype typically peaks in the fourth andfifth decades of life [1]. Myxoid liposarcoma is com-monly associated with TLS-CHOP fusion transcript[2,3]. In contrast to other soft-tissue sarcomas that showa tendency for metastasis to the lung, myxoid liposar-coma has a propensity to spread to extrapulmonary sites,including bone [4,5]. The frequency of bone metastasisarising from myxoid liposarcoma has been reported tobe 14% [1] and 17% [6].Bone scans and [18F]-2-fluoro-2-deoxy-D-glucose-positron emission tomography (FDG-PET) are useful for* Correspondence: akio@med.kyushu-u.ac.jp1Department of Orthopaedic Surgery, National Hospital Organization KokuraMedical Center, Kitakyushu, Fukuoka 802-0803, Japan2Department of Orthopaedic Surgery, Graduate School of Medical Sciences,Kyushu University, Fukuoka 812-8582, JapanFull list of author information is available at the end of the article© 2012 Sakamoto et al.; licensee BioMed CentCommons Attribution License (http://creativecreproduction in any medium, provided the ordetecting distant metastases from various tumors. Bonescans are sensitive to unusual bone remodeling activitybecause radioactive material is taken up by osteoblasts.On the other hand, FDG-PET can be evaluated using thestandardized uptake value (SUV). An increased SUVreflects increased glucose metabolism as a result of vari-ous factors such as increased glucose transporters, highlevels of hexokinase and a reduction in glucose-6-phosphatase [7,8]. FDG-PET has been reported to beuseful for the diagnosis of soft-tissue tumors [9].Bone scans and FDG-PET have been reported to lacksufficient sensitivity for the detection of vertebral metas-tases arising from myxoid liposarcoma. Among vertebralmetastases detected by magnetic resonance imaging(MRI), positive rates with bone scans and FDG-PEThave been reported to be 16% and 14%, respectively, in anumber of vertebrae [6]. It also has been reported thatthe positive rate with bone scans for vertebral metastasisral Ltd. This is an Open Access article distributed under the terms of the Creativeommons.org/licenses/by/2.0), which permits unrestricted use, distribution, andiginal work is properly cited.Sakamoto et al. World Journal of Surgical Oncology 2012, 10:214 Page 2 of 4http://www.wjso.com/content/10/1/214of various tumors is rather low for intramedullarylesions that have no cortical involvement [10]. In thecurrent study, we report the case of a man with myxoidliposarcoma with multiple vertebral metastases. Al-though some of the metastatic vertebrae had extendedextraskeletally, all the metastatic vertebral lesions werenegative on bone scans and FDG-PET, except for thefractured vertebra, which was visible on a bone scan.Case presentationA 43-year-old man with a tumor in his left leg was re-ferred to our institute. The diagnosis was myxoid liposar-coma based upon biopsy findings. Radiation andchemotherapy were administered. Consequent wide re-section with the surrounding normal tissue was per-formed. Two years after the initial surgery, when thepatient was 45 years of age, metastasis appeared in theright neck region and the retroperitoneum, and resectionof these lesions was performed. Seven years after the ini-tial surgery, when the patient was 50 years of age, lungmetastasis was found and resection was performed. Eightyears after the initial surgery, when the patient was 51years of age, recurrence was found in the thigh andinvolved the major vessels. Amputation at the thigh wascarried out. Histological diagnosis of myxoid liposarcomawas confirmed in each of the resected materials. Chemo-therapy was performed after every resection of metastaticand recurrent lesions. Ten years after the initial oper-ation, at 53 years of age, the patient experienced backpain. Plain radiographs showed a compression typefracture in the second lumbar vertebra. MRI of all thevertebrae showed abnormally high signal intensity onT2-imaging with fat suppression in eight vertebrae: thefifth cervical vertebra (C5), the seventh, tenth, eleventhand twelfth thoracic vertebrae (T7, T10, T11 and T12),the second and fourth lumbar vertebrae (L2 and L4),and the second sacral vertebra (S2). High signal inten-sity on the MRI was seen throughout the entireFigure 1 Magnetic resonance imaging shows high signal intensity onPathological fracture is evident in the second vertebra. Extraskeletal extensiand second sacral vertebra.vertebral body in L2 accompanied by deformity, sug-gestive of a pathological fracture. Extraskeletal exten-sion of the lesion was seen in the T12, L2 and S2vertebrae (Figure 1). Bone scans showed negative find-ings except for the L2 fracture. FDG-PET was negativefor all of the vertebrae, including the fractured L2 ver-tebra (Figure 2). These examinations of bone scans andFDG-PET were performed within one month after theMRI examination. With a diagnosis of vertebral metas-tasis arising from myxoid liposarcoma, radiation andchemotherapy were administered. The patient wasinformed that data from his case would be submittedfor publication, and his consent was obtained.ConclusionsMyxoid liposarcoma is characteristic in giving rise to extra-pulmonary metastasis, such as to the retroperitoneum, theaxilla and the mesentery, while other soft-tissue sarco-mas demonstrate a tendency to metastasize to the lung[3,11,12]. The overall rate of metastasis in myxoid lipo-sarcoma has been reported to be one-third [2,6]. Thefrequency of bone metastasis arising from myxoid lipo-sarcoma has been reported to be 14% [1] and 17% [6].The actual frequency of vertebral metastasis of myxoidliposarcoma is not clear, since a low metastatic rate of4.3% has also been reported [13].Bone scans are known to be more sensitive than plainradiographs for detecting bone metastasis [13]. A lowpositive rate with bone scans has been reported for ver-tebral metastasis arising from myxoid liposarcoma. In aprevious report, bone scans revealed three out of ninecases (33%) and five out of 32 vertebrae (16%), basedupon known metastasis as shown on MRI [6]. As for thesensitivity of bone scans, a lower positive rate for meta-static bone tumors in various malignancies has beenreported in cases of intramedullary lesions without cor-tical involvement [10]. In the current report, althoughthe vertebral lesions had extended extraskeletally ina T2-image with fat suppression in multiple vertebrae.on is visible in the eleventh thoracic vertebra, second lumbar vertebraFigure 2 Positive findings can be seen on the bone scan in the fractured second lumbar vertebra (L2) (A). Coronal section (B) and axialsections of twelfth thoracic vertebra (T12) (C), L1 (D), L2 (E) and the first sacral vertebra (S1) (F) in FDG-PET are shown. There are no obviousdifferences between FDG uptake in metastatic vertebrae of T12 (C), L2 (E), and the non-metastatic vertebrae of L1 (D) and S1 (F). FDG-PET,[18F]-2-fluoro-2-deoxy-D-glucose-positron emission tomography.Sakamoto et al. World Journal of Surgical Oncology 2012, 10:214 Page 3 of 4http://www.wjso.com/content/10/1/214three vertebrae, bone scans of the vertebrae were nega-tive in all cases except for the one vertebra that was frac-tured. The reason for this low positive rate in cases ofmyxoid liposarcoma is still not clear. In certain condi-tions such as myeloma or lesions confined to the mar-row, bone scans have shown low sensitivity [14]. It hasbeen suggested that hematogenously seeded intramedul-lary metastasis produces marrow replacement lesionswithout destroying the bone structure [15]. Moreover,myeloma cells promote osteoclast activity but inhibitosteoblast differentiation, which may explain the nega-tivity of bone scans [16].An increased uptake of FDG-PET in cells reflectsincreased glucose metabolism [7,8], and has been usedto evaluate the extent of disease. As for bone and soft-tissue tumors, it has been reported that malignanttumors tend to have a higher SUV than benign tumors[9]. A low positive rate of FDG-PET has been reportedin cases of vertebral metastasis of myxoid liposarcoma.In a previous report, FDG-PET revealed two out of sixcases (33%), and four out of 29 vertebrae (14%), basedupon known metastasis as shown on MRI [6]. The rea-son FDG-PET has a lower positive rate in cases of verte-bral metastasis of myxoid liposarcoma is not clear. It hasbeen assumed that the myxoid stroma in myxoid liposar-coma may prevent the labeled glucose from reachingcells in a sufficient quantity to be detected by the scan-ner [6]. Interestingly, in the current case, SUV was stillnegative in FDG-PET even in the fractured vertebra,whereas positive results were seen on the bone scan. Afracture site is usually positive on FDG-PET. The dis-crepancy between the results of the FDG-PET and thebone scan for the fractured vertebra could be associatedwith a specific biological mechanism, such as mild inhib-ition of the healing reaction at the fracture site.In summary, a case of myxoid liposarcoma with mul-tiple vertebral metastases is reported. Bone scans andFDG-PET were negative, even for lesions with extraske-letal extension, except for the L2 fracture which was vis-ible on a bone scan. Bone scans and FDG-PET can thusbe negative in cases of multiple vertebral metastases ofmyxoid liposarcoma, and even a fractured vertebra maynot be visible on FDG-PET.ConsentWritten informed consent was obtained from the patientfor publication of this case report and any accompanyingSakamoto et al. World Journal of Surgical Oncology 2012, 10:214 Page 4 of 4http://www.wjso.com/content/10/1/214images. A copy of the written consent is available forreview by the Editor-in-Chief of this journal.AbbreviationsFDG-PET: [18F]-2-fluoro-2-deoxy-D-glucose-positron emission tomography;MRI: magnetic resonance imaging; SUV: standardized uptake value;C: cervical vertebra; T: thoracic vertebra; L: lumbar vertebra; S: sacral vertebra.Competing interestsThe authors declare that they have no competing interests.Authors’ contributionsAS drafted the manuscript. AS, YF, YM, KH and YI administered thetreatment. OY and YI conceived the study, participated in its design andcoordination, and helped to draft the manuscript. All authors read andapproved the final manuscript.AcknowledgementsWe thank Miss K. Miller (Royal English Language Centre, Fukuoka, Japan) forrevising the English used in this manuscript.Author details1Department of Orthopaedic Surgery, National Hospital Organization KokuraMedical Center, Kitakyushu, Fukuoka 802-0803, Japan. 2Department ofOrthopaedic Surgery, Graduate School of Medical Sciences, KyushuUniversity, Fukuoka 812-8582, Japan. 3Department of Anatomic Pathology,Graduate School of Medical Sciences, Kyushu University, Fukuoka 812-8582,Japan.Received: 19 April 2012 Accepted: 26 September 2012Published: 9 October 2012References1. Schwab JH, Boland P, Guo T, Brennan MF, Singer S, Healey JH, AntonescuCR: Skeletal metastases in myxoid liposarcoma: an unusual pattern ofdistant spread. Ann Surg Oncol 2007, 14:1507–1514.2. 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N Engl J Med1974, 291:1041–1046.doi:10.1186/1477-7819-10-214Cite this article as: Sakamoto et al.: Myxoid liposarcoma with negativefeatures on bone scan and [18F]-2-fluoro-2-deoxy-D-glucose-positronemission tomography. World Journal of Surgical Oncology 2012 10:214.Submit your next manuscript to BioMed Centraland take full advantage of: • Convenient online submission• Thorough peer review• No space constraints or color ﬁgure charges• Immediate publication on acceptance• Inclusion in PubMed, CAS, Scopus and Google Scholar• Research which is freely available for redistributionSubmit your manuscript at www.biomedcentral.com/submit

Myxoid liposarcoma with negative features on bone scan and [18F]-2-fluoro-2-deoxy-D-glucose-positron emission tomography

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Myxoid liposarcoma with negative features on bone scan and [18F]-2-fluoro-2-deoxy-D-glucose-positron emission tomography

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