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Recurrent Alveolar Soft Part Sarcoma on Bone Scintigraphy He Huang, MD, and Yue Chen, MD Abstract: A 15-year-old girl experienced an alveolar soft part sarcoma in the right thigh 3 years ago, which was resected. Postsurgical recovery was uneventful until approximately 1 year ago when she began to feel mild local tenderness and gradual swelling around the surgical scars. The patient underwent 99mTc-MDP bone scintigraphy to evaluate osseous metastases. Although no lesion in the bone was identified, the images showed abnormally increased activity in the region of known focal lesion. Key Words: alveolar soft part sarcoma, MDP, bone scintigraphy (Clin Nucl Med 2016;41: 251–252)
REFERENCES 1. Anderson ME, Hornicek FJ, Gebhardt MC, et al. Alveolar soft part sarcoma: a rare and enigmatic entity. Clin Orthop Relat Res. 2005;438:144–148. 2. Folpe AL, Deyrup AT. Alveolar soft-part sarcoma: a review and update. J Clin Pathol. 2006;59:1127–1132. Received for publication August 9, 2015; revision accepted August 19, 2015. From the Department of Nuclear Medicine, the First Affiliated Hospital of Sichuan Medical University, Luzhou, Sichuan, People's Republic of China. Conflicts of interest and sources of funding: none declared. Correspondence to: Yue Chen, MD, Department of Nuclear Medicine, the First Affiliated Hospital of Sichuan Medical University, 15 TaiPing St, Jiangyang District, Luzhou, Sichuan 646000, People's Republic of China. E-mail: [email protected]. Copyright © 2015 Wolters Kluwer Health, Inc. All rights reserved. ISSN: 0363-9762/16/4103–0251 DOI: 10.1097/RLU.0000000000001015
3. Kayton ML, Meyers P, Wexler LH, et al. Clinical presentation, treatment, and outcome of alveolar soft part sarcoma in children, adolescents, and young adults. J Pediatr Surg. 2006;41:187–193. 4. Wang HW, Dai W, Qin XJ, et al. A new clinical manifestation for cheek alveolar soft-part sarcoma: a case report and review of the literature. J Oral Maxillofac Surg. 2014;72:817–822. 5. van Ruth S, van Coevorden F, Peterse JL, et al. Alveolar soft part sarcoma. a report of 15 cases. Eur J Cancer. 2002;38:1324–1328. 6. Casanova M, Ferrari A, Bisogno G, et al. Alveolar soft part sarcoma in children and adolescents: a report from the soft-tissue sarcoma Italian cooperative group. Ann Oncol. 2000;11:1445–1449. 7. Ju HU, Seo KW, Jegal Y, et al. A case of alveolar soft part sarcoma of the pleura. J Korean Med Sci. 2013;28:331–335. 8. Sood S, Baheti AD, Shinagare AB, et al. Imaging features of primary and metastatic alveolar soft part sarcoma: single institute experience in 25 patients. Br J Radiol. 2014;87:20130719. 9. Pennacchioli E, Fiore M, Collini P, et al. Alveolar soft part sarcoma: clinical presentation, treatment, and outcome in a series of 33 patients at a single institution. Ann Surg Oncol. 2010;17:3229–3233. 10. Ogura K, Beppu Y, Chuman H, et al. Alveolar soft part sarcoma: a singlecenter 26-patient case series and review of the literature. Sarcoma. 2012; 2012:907179. 11. Shi X, Jing H, Zhuang H, et al. Diffuse hepatic and splenic uptake of Tc-99m methylene diphosphonate on bone scintigraphy after intravenous administration of gadolinium-containing MRI contrast. Clin Nucl Med. 2011;36:178–182. 12. Lin Y, Lin WY, Kao CH, et al. Easy interpretation of heterotopic ossification demonstrated on bone SPECT/CT. Clin Nucl Med. 2014;39:62–63. 13. Chang HR, Kao CH, Lian JD, et al. Evaluation of the severity of traumatic rhabdomyolysis using technetium-99m pyrophosphate scintigraphy. Am J Nephrol. 2001;21:208–214. 14. Mitomo M, Miyazaki C, Mukai M, et al. Tc-99m MDP bone scintigraphy of myositis as a manifestation of chronic graft-versus-host disease after nonmyeloablative peripheral stem cell transplantation. Ann Nucl Med. 2005;19:41–45. 15. Martinez-Sanchis B, Cortes-Vizcaino V, Frontado-Morales L, et al. Intestinal uptake of (99m)Tc-MDP: a case report of protein-losing enteropathy correlated with pathology findings from autopsy. Ann Nucl Med. 2011;25: 139–141. 16. Kung JW, Yu JQ, Lee B, et al. Increased Tc-99m MDP accumulation in soft tissue caused by bicycle riding. Clin Nucl Med. 2004;29:279–280. 17. Demirel K, Sadic M, Korkmaz M, et al. Diffuse Myocardial Uptake of (99m)Tc-HDP in Multiple Myeloma. Nucl Med Mol Imaging. 2013;47: 208–211. 18. Marwah A, Kumar R, Dasan JB, et al. Soft tissue uptake of Tc99m-MDP in acute lymphoblastic leukemia. Clin Imaging. 2002;26:206–208. 19. Zaman MU, Fatima N, Sajjad Z, et al. Higher scrotal uptake ratio of (99m)TcMDP on bone scans in newly diagnosed prostate cancer: a reliable indicator of pelvic node metastasis. Ann Nucl Med. 2012;26:676–680. 20. Huang J, Chan S, Servase S, et al. A large pelvic soft tissue mass implied by subtle bone scan findings. Clin Nucl Med. 2014;39:402–405. 21. Kim DW, Jeong HJ, Park SA, et al. Gastric accumulation of bone seeking agent in a patient with advanced gastric cancer. J Korean Med Sci. 2007; 22:153–155. 22. Hu B, Liu Y, Cheng L, et al. SPECT/CT imaging of retroperitoneal extraskeletal osteosarcoma. Clin Nucl Med. 2014;39:200–202. 23. Ohta H, Komibuchi T, Nishiyama H, et al. 99mTc(V)-DMSA and 99m Tc-MDP uptake and no 67Ga-citrate uptake in a case of primary pulmonary leiomyosarcoma. Ann Nucl Med. 1992;6:191–193. 24. Horne T, Mogle P, Finsterbush A, et al. Increased uptake of 99mTc-MDP in calcified synovial sarcoma. Eur J Nucl Med. 1983;8:75–76. 25. Ali I, Johns W, Gupta SM. Visualization of hepatic metastases of medullary thyroid carcinoma on Tc-99m MDP bone scintigraphy. Clin Nucl Med. 2006; 31:611–613. 26. Ohta H, Ishii M, Endo K, et al. Scintigraphic evaluation of two cases with alveolar soft part sarcoma. Ann Nucl Med. 1988;2:89–93.
Clinical Nuclear Medicine • Volume 41, Number 3, March 2016 Copyright © 2016 Wolters Kluwer Health, Inc. All rights reserved.
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251
Huang and Chen
Clinical Nuclear Medicine • Volume 41, Number 3, March 2016
FIGURE 1. A 15-year-old woman presented to our hospital for mild local tenderness and swelling in the right thigh for approximately 1 year. Approximately 3 years ago, she had histologically confirmed alveolar soft part sarcoma in the same location, which was surgically resected. Possible recurrent disease was considered, and a 99mTc-MDP bone scintigraphy was performed to evaluate osseous metastasis. The whole-body images showed no abnormally increased activity in the bones. However, a large focus of moderately increased activity (arrows) in the lateral right upper thigh was noted.
FIGURE 2. On the SPECT images (A), the activity (black arrows) was heterogeneous. The activity corresponded to a heterogeneous soft tissue mass (white arrows) with both grit calcification and necrosis in some regions. The mass was measured approximately 7.5 6.0 11.5 cm in the right vastus lateralis muscle on the CT (B) and fusion (C) images. The findings were suggestive of a recurrent alveolar soft part sarcoma. Surgery was performed, and the lesion was resected. The pathological examination confirmed recurrent alveolar soft tissue sarcoma. The patient received further chemotherapy. Unfortunately, 3 months after the resection of the recurrent tumor from the right thigh, the patient developed multiple metastases in the liver and the lungs. Alveolar soft part sarcoma is a rare tumor, and little is known about its clinical features and proper management1 because of its rarity. It represents approximately 0.5% to 0.9% of all soft tissue sarcomas in adults and 0.8% to 1.8% of those in children.2–7 Alveolar soft part sarcoma often occurs in the deep soft tissues, most commonly in the lower extremity in relatively young patients,8–10 such as in our case. Although bone scintigraphy was mainly used to detect osseous lesions, elevated bone tracer activity can occurs in the soft tissue due to many benign11–16 or malignant17–25 etiologies. Previously, a case report had shown that increased MDP activity could occur in primary alveolar soft part sarcoma.26 Our findings indicate that bone tracer can also have an elevated accumulation in recurrent alveolar soft part sarcoma as well. 252
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© 2015 Wolters Kluwer Health, Inc. All rights reserved. Copyright © 2016 Wolters Kluwer Health, Inc. All rights reserved.
Recurrent Alveolar Soft Part Sarcoma on Bone Scintigraphy He Huang, MD, and Yue Chen, MD Abstract: A 15-year-old girl experienced an alveolar soft part sarcoma in the right thigh 3 years ago, which was resected. Postsurgical recovery was uneventful until approximately 1 year ago when she began to feel mild local tenderness and gradual swelling around the surgical scars. The patient underwent 99mTc-MDP bone scintigraphy to evaluate osseous metastases. Although no lesion in the bone was identified, the images showed abnormally increased activity in the region of known focal lesion. Key Words: alveolar soft part sarcoma, MDP, bone scintigraphy (Clin Nucl Med 2016;41: 251–252)
REFERENCES 1. Anderson ME, Hornicek FJ, Gebhardt MC, et al. Alveolar soft part sarcoma: a rare and enigmatic entity. Clin Orthop Relat Res. 2005;438:144–148. 2. Folpe AL, Deyrup AT. Alveolar soft-part sarcoma: a review and update. J Clin Pathol. 2006;59:1127–1132. Received for publication August 9, 2015; revision accepted August 19, 2015. From the Department of Nuclear Medicine, the First Affiliated Hospital of Sichuan Medical University, Luzhou, Sichuan, People's Republic of China. Conflicts of interest and sources of funding: none declared. Correspondence to: Yue Chen, MD, Department of Nuclear Medicine, the First Affiliated Hospital of Sichuan Medical University, 15 TaiPing St, Jiangyang District, Luzhou, Sichuan 646000, People's Republic of China. E-mail: [email protected]. Copyright © 2015 Wolters Kluwer Health, Inc. All rights reserved. ISSN: 0363-9762/16/4103–0251 DOI: 10.1097/RLU.0000000000001015
3. Kayton ML, Meyers P, Wexler LH, et al. Clinical presentation, treatment, and outcome of alveolar soft part sarcoma in children, adolescents, and young adults. J Pediatr Surg. 2006;41:187–193. 4. Wang HW, Dai W, Qin XJ, et al. A new clinical manifestation for cheek alveolar soft-part sarcoma: a case report and review of the literature. J Oral Maxillofac Surg. 2014;72:817–822. 5. van Ruth S, van Coevorden F, Peterse JL, et al. Alveolar soft part sarcoma. a report of 15 cases. Eur J Cancer. 2002;38:1324–1328. 6. Casanova M, Ferrari A, Bisogno G, et al. Alveolar soft part sarcoma in children and adolescents: a report from the soft-tissue sarcoma Italian cooperative group. Ann Oncol. 2000;11:1445–1449. 7. Ju HU, Seo KW, Jegal Y, et al. A case of alveolar soft part sarcoma of the pleura. J Korean Med Sci. 2013;28:331–335. 8. Sood S, Baheti AD, Shinagare AB, et al. Imaging features of primary and metastatic alveolar soft part sarcoma: single institute experience in 25 patients. Br J Radiol. 2014;87:20130719. 9. Pennacchioli E, Fiore M, Collini P, et al. Alveolar soft part sarcoma: clinical presentation, treatment, and outcome in a series of 33 patients at a single institution. Ann Surg Oncol. 2010;17:3229–3233. 10. Ogura K, Beppu Y, Chuman H, et al. Alveolar soft part sarcoma: a singlecenter 26-patient case series and review of the literature. Sarcoma. 2012; 2012:907179. 11. Shi X, Jing H, Zhuang H, et al. Diffuse hepatic and splenic uptake of Tc-99m methylene diphosphonate on bone scintigraphy after intravenous administration of gadolinium-containing MRI contrast. Clin Nucl Med. 2011;36:178–182. 12. Lin Y, Lin WY, Kao CH, et al. Easy interpretation of heterotopic ossification demonstrated on bone SPECT/CT. Clin Nucl Med. 2014;39:62–63. 13. Chang HR, Kao CH, Lian JD, et al. Evaluation of the severity of traumatic rhabdomyolysis using technetium-99m pyrophosphate scintigraphy. Am J Nephrol. 2001;21:208–214. 14. Mitomo M, Miyazaki C, Mukai M, et al. Tc-99m MDP bone scintigraphy of myositis as a manifestation of chronic graft-versus-host disease after nonmyeloablative peripheral stem cell transplantation. Ann Nucl Med. 2005;19:41–45. 15. Martinez-Sanchis B, Cortes-Vizcaino V, Frontado-Morales L, et al. Intestinal uptake of (99m)Tc-MDP: a case report of protein-losing enteropathy correlated with pathology findings from autopsy. Ann Nucl Med. 2011;25: 139–141. 16. Kung JW, Yu JQ, Lee B, et al. Increased Tc-99m MDP accumulation in soft tissue caused by bicycle riding. Clin Nucl Med. 2004;29:279–280. 17. Demirel K, Sadic M, Korkmaz M, et al. Diffuse Myocardial Uptake of (99m)Tc-HDP in Multiple Myeloma. Nucl Med Mol Imaging. 2013;47: 208–211. 18. Marwah A, Kumar R, Dasan JB, et al. Soft tissue uptake of Tc99m-MDP in acute lymphoblastic leukemia. Clin Imaging. 2002;26:206–208. 19. Zaman MU, Fatima N, Sajjad Z, et al. Higher scrotal uptake ratio of (99m)TcMDP on bone scans in newly diagnosed prostate cancer: a reliable indicator of pelvic node metastasis. Ann Nucl Med. 2012;26:676–680. 20. Huang J, Chan S, Servase S, et al. A large pelvic soft tissue mass implied by subtle bone scan findings. Clin Nucl Med. 2014;39:402–405. 21. Kim DW, Jeong HJ, Park SA, et al. Gastric accumulation of bone seeking agent in a patient with advanced gastric cancer. J Korean Med Sci. 2007; 22:153–155. 22. Hu B, Liu Y, Cheng L, et al. SPECT/CT imaging of retroperitoneal extraskeletal osteosarcoma. Clin Nucl Med. 2014;39:200–202. 23. Ohta H, Komibuchi T, Nishiyama H, et al. 99mTc(V)-DMSA and 99m Tc-MDP uptake and no 67Ga-citrate uptake in a case of primary pulmonary leiomyosarcoma. Ann Nucl Med. 1992;6:191–193. 24. Horne T, Mogle P, Finsterbush A, et al. Increased uptake of 99mTc-MDP in calcified synovial sarcoma. Eur J Nucl Med. 1983;8:75–76. 25. Ali I, Johns W, Gupta SM. Visualization of hepatic metastases of medullary thyroid carcinoma on Tc-99m MDP bone scintigraphy. Clin Nucl Med. 2006; 31:611–613. 26. Ohta H, Ishii M, Endo K, et al. Scintigraphic evaluation of two cases with alveolar soft part sarcoma. Ann Nucl Med. 1988;2:89–93.
Clinical Nuclear Medicine • Volume 41, Number 3, March 2016 Copyright © 2016 Wolters Kluwer Health, Inc. All rights reserved.
www.nuclearmed.com
251
Huang and Chen
Clinical Nuclear Medicine • Volume 41, Number 3, March 2016
FIGURE 1. A 15-year-old woman presented to our hospital for mild local tenderness and swelling in the right thigh for approximately 1 year. Approximately 3 years ago, she had histologically confirmed alveolar soft part sarcoma in the same location, which was surgically resected. Possible recurrent disease was considered, and a 99mTc-MDP bone scintigraphy was performed to evaluate osseous metastasis. The whole-body images showed no abnormally increased activity in the bones. However, a large focus of moderately increased activity (arrows) in the lateral right upper thigh was noted.
FIGURE 2. On the SPECT images (A), the activity (black arrows) was heterogeneous. The activity corresponded to a heterogeneous soft tissue mass (white arrows) with both grit calcification and necrosis in some regions. The mass was measured approximately 7.5 6.0 11.5 cm in the right vastus lateralis muscle on the CT (B) and fusion (C) images. The findings were suggestive of a recurrent alveolar soft part sarcoma. Surgery was performed, and the lesion was resected. The pathological examination confirmed recurrent alveolar soft tissue sarcoma. The patient received further chemotherapy. Unfortunately, 3 months after the resection of the recurrent tumor from the right thigh, the patient developed multiple metastases in the liver and the lungs. Alveolar soft part sarcoma is a rare tumor, and little is known about its clinical features and proper management1 because of its rarity. It represents approximately 0.5% to 0.9% of all soft tissue sarcomas in adults and 0.8% to 1.8% of those in children.2–7 Alveolar soft part sarcoma often occurs in the deep soft tissues, most commonly in the lower extremity in relatively young patients,8–10 such as in our case. Although bone scintigraphy was mainly used to detect osseous lesions, elevated bone tracer activity can occurs in the soft tissue due to many benign11–16 or malignant17–25 etiologies. Previously, a case report had shown that increased MDP activity could occur in primary alveolar soft part sarcoma.26 Our findings indicate that bone tracer can also have an elevated accumulation in recurrent alveolar soft part sarcoma as well. 252
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© 2015 Wolters Kluwer Health, Inc. All rights reserved. Copyright © 2016 Wolters Kluwer Health, Inc. All rights reserved.
