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Enhanced CT and FDG PET/CT in Primary Malignant Peripheral Nerve Sheath Tumor of the Uterine Cervix Aisheng Dong, MD,* Changjing Zuo, MD,* Yang Wang, MSc,Þ Zhijun Zhai, BSc,* and Mingjuan Xu, MDþ Abstract: Malignant peripheral nerve sheath tumor of the uterine cervix is extremely rare. A 45-year-old woman presented with intermittent vaginal bleeding for 1 month. Enhanced CT in the arterial and venous phases showed a hypervascular tumor in the uterine cervix. Colposcopy-directed biopsy revealed malignant peripheral nerve sheath tumor. FDG PET/CT showed intense FDG uptake (SUVmax, 8.8) of the tumor mimicking cervical cancer. Total laparoscopic radical hysterectomy, bilateral salpingo-oophorectomy, and pelvic lymphadenectomy were performed. Key Words: malignant peripheral nerve sheath tumor, uterine cervix, CT, FDG, PET/CT (Clin Nucl Med 2014;39: 825Y827)

Received for publication August 1, 2013; revision accepted October 3, 2013. From the Departments of *Nuclear Medicine, †Pathology, and ‡Obstetrics and Gynecology, Changhai Hospital, Second Military Medical University, Shanghai, China. Drs Aisheng Dong and Changjing Zuo contributed equally to the article. Conflicts of interest and sources of funding: Dr Aisheng Dong was sponsored by the Young Scholar Grant from the National Natural Science Foundation of China (81000601). Reprints: Mingjuan Xu, MD, Department of Obstetrics and Gynecology, Changhai Hospital, Second Military Medical University, 168 Changhai Rd, Yangpu District, Shanghai 200433, China. E-mail: [email protected]. Copyright * 2013 by Lippincott Williams & Wilkins ISSN: 0363-9762/14/3909Y0825

Clinical Nuclear Medicine

REFERENCES 1. Rha SE, Byun JY, Jung SE, et al. Neurogenic tumors in the abdomen: tumor types and imaging characteristics. Radiographics. 2003;23:29Y43. 2. Kransdorf MJ. Malignant soft-tissue tumors in a large referral population: distribution of diagnoses by age, sex, and location. AJR Am J Roentgenol. 1995;164:129Y134. 3. Bernstein HB, Broman JH, Apicelli A, et al. Primary malignant schwannoma of the uterine cervix: a case report and literature review. Gynecol Oncol. 1999;74:288Y292. 4. Lallas TA, Mehaffey PC, Lager DJ, et al. Malignant cervical schwannoma: an unusual pelvic tumor. Gynecol Oncol. 1999;72:238Y242. 5. Keel SB, Clement PB, Prat J, et al. Malignant schwannoma of the uterine cervix: a study of three cases. Int J Gynecol Pathol. 1998;17:223Y230. 6. Kim NR, Chung DH, Park CY, et al. Malignant peripheral nerve sheath tumor of the uterine cervix expressing both S-100 protein and HMB-45. J Obstet Gynaecol Res. 2009;35:1136Y1141. 7. Hamada K, Tomita Y, Qiu Y, et al. (18)F-FDG PET analysis of schwannoma: increase of SUVmax in the delayed scan is correlated with elevated VEGF/VPF expression in the tumors. Skeletal Radiol. 2009;38:261Y266. 8. Ahmed AR, Watanabe H, Aoki J, et al. Schwannoma of the extremities: the role of PET in preoperative planning. Eur J Nucl Med. 2001;28:1541Y1551. 9. Benz MR, Czernin J, Dry SM, et al. Quantitative F18-fluorodeoxyglucose positron emission tomography accurately characterizes peripheral nerve sheath tumors as malignant or benign. Cancer. 2010;116:451Y458. 10. Ferner RE, Golding JF, Smith M, et al. [18F]2-fluoro-2-deoxy-D-glucose positron emission tomography (FDG PET) as a diagnostic tool for neurofibromatosis 1 (NF1) associated malignant peripheral nerve sheath tumours (MPNSTs): a long-term clinical study. Ann Oncol. 2008;19:390Y394. 11. Salamon J, Derlin T, Bannas P, et al. Evaluation of intratumoural heterogeneity on (18)F-FDG PET/CT for characterization of peripheral nerve sheath tumours in neurofibromatosis type 1. Eur J Nucl Med Mol Imaging. 2013;40:685Y692. 12. Bredella MA, Torriani M, Hornicek F, et al. Value of PET in the assessment of patients with neurofibromatosis type 1. AJR Am J Roentgenol. 2007;189: 928Y935. 13. Derlin T, Tornquist K, Mu¨nster S, et al. Comparative effectiveness of 18F-FDG PET/CT versus whole-body MRI for detection of malignant peripheral nerve sheath tumors in neurofibromatosis type 1. Clin Nucl Med. 2013;38:e19Ye25. 14. Bertagna F, Bosio G, Biasiotto G, et al. Plurifocal malignant peripheral nerve sheath tumor demonstrated by 18F-fluorodeoxyglucose positron emission tomography/computed tomography. Jpn J Radiol. 2009;27:320Y323. 15. Chander S, Westphal SM, Zak IT, et al. Retroperitoneal malignant peripheral nerve sheath tumor: evaluation with serial FDG-PET. Clin Nucl Med. 2004; 29:415Y418.

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FIGURE 1. A 45-year-old woman presented with intermittent vaginal bleeding for 1 month. Pelvic ultrasound showed a tumor in the uterine cervix. Enhanced CT in the arterial (A) phase showed a hypervascular tumor (arrow) measuring 3.7  2.6 cm in the uterine cervix. Enhanced CT in the venous (B) phase showed progressive enhancement of the tumor. Colposcopy-directed biopsy revealed malignant peripheral nerve sheath tumor (MPNST). For staging of the malignant tumor, FDG PET/CT (Biograph 64; Knoxville, TN) was performed 1 hour after the injection of 389 MBq (10.5 mCi) of 18F-FDG with a blood glucose level of 5.8 mmol/L. Transverse (C) and sagittal (D) CT, corresponding PET (E and F), and fused (G and H) images showed intense FDG uptake (SUVmax, 8.8) of the tumor mimicking cervical cancer.

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MPNST of the Uterine Cervix

FIGURE 2. The patient underwent total laparoscopic radical hysterectomy, bilateral salpingo-oophorectomy, and pelvic lymphadenectomy. Grossly (A), the cut surface showed a solitary, well-circumscribed mass (arrows) in the uterine cervix. Photomicrograph (B, hematoxylin-eosin, original magnification 200) showed that the tumor was composed of spindled cells arranged in intersecting fascicles with scattered mitotic figures (arrows). Tumor cells were positive for S-100 protein (C, original magnification 100). Ki-67 staining showed that the proportion of the positive tumor cells was about 40% (D, original magnification 100). These findings were consistent with MPNST. Tumors of nerve sheath origin include benign schwannoma, neurofibroma, neurofibromatosis, and MPNST. Neurofibromas often undergo malignant degeneration to MPNSTs, particularly in cases of neurofibromatosis, whereas schwannomas rarely if ever undergo malignant degeneration.1 Malignant peripheral nerve sheath tumors are aggressive uncommon tumors, accounting for about 6% of all soft tissue sarcomas.2 They usually occur in the extremities and the trunk. Malignant peripheral nerve sheath tumor of the uterine cervix is extremely rare with only a few cases reported in the literature.3Y6 Patients with MPNSTs have a poor prognosis. Accurately discriminating benign lesions from malignant ones and staging are essential. Differentiating MPNST from neurofibroma and schwannoma is very difficult on CT and MRI.1 Neurofibroma and schwannoma typically show low FDG uptake.7Y9 SUVmax is significantly higher in MPNST compared with benign peripheral nerve sheath tumor.9 Several studies have shown that FDG PET or PET/CT is a sensitive technique in the detection of MPNSTs in patients with neurofibromatosis type 1.10Y13 In addition, FDG PET is able to guide biopsy and direct appropriate therapy in positive cases obviating repetitive surgery and biopsy in negative cases.12 FDG PET is helpful for staging, restaging, and posttherapy follow-up of MPNST.14,15

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CT in primary malignant peripheral nerve sheath tumor of the uterine cervix.

Malignant peripheral nerve sheath tumor of the uterine cervix is extremely rare. A 45-year-old woman presented with intermittent vaginal bleeding for ...
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