Recurrent malignant solid pseudopapillary neoplasms of the pancreas (SPNP) are rare tumors with unpredictable clinical and histopathological features. There is a lack of consensus regarding utilization of adjuvant modalities in conjunction with or in lieu of curative metastatectomy. We present a remarkable case where Yttrium-90 selective internal radiation therapy (Y-90 SIRT) was successfully utilized to elucidate underlying tumor biology and aid resection of a large multifocal recurrent metastatic SPNP in the right hemi-liver of a 59-year-old female. Thus, in cases where curative metastatectomy remains the treatment goal in management of recurrent and/or metastatic SPNPs, Y-90 SIRT is a safe and effective adjunct treatment to facilitate curative resection

Dyas, Adam R

Johnson, David Thor

Kumar Sharma, Piyush

Rubin, Erin

Schulick, Richard D

English

DigitalCommons@The Texas Medical Center

1Journal of Surgical Case Reports, 2020;9, 1–3doi: 10.1093/jscr/rjaa325Case ReportC A S E R E P O R TYttrium-90 selective internal radiotherapy as bridge tocurative hepatectomy for recurrent malignant solidpseudopapillary neoplasm of pancreas: case reportand review of literatureAdam R. Dyas1, David Thor Johnson2, Erin Rubin3, Richard D. Schulick1 andPiyush Kumar Sharma1,4,*1Department of Surgery, University of Colorado School of Medicine, Aurora, CO, USA , 2Department ofRadiology, University of Colorado School of Medicine, Aurora, CO, USA , 3Department of Pathology, University ofColorado School of Medicine, Aurora, CO, USA and 4Department of Oncology, University of Oxford & OxfordCancer Centre, Churchill Hospital, Oxford University Hospitals NHS Foundation Trust, Oxford, UK*Correspondence address. Department of Surgery, University of Colorado School of Medicine, Aurora, CO, USA; Department of Oncology, University ofOxford, UK. Tel: +44 79 5184 2827; E-mail: piyush.sharma@cuanschutz.edu, piyush.sharma@ucdenver.eduAbstractRecurrent malignant solid pseudopapillary neoplasms of the pancreas (SPNP) are rare tumors with unpredictable clinicaland histopathological features. There is a lack of consensus regarding utilization of adjuvant modalities in conjunction withor in lieu of curative metastatectomy. We present a remarkable case where Yttrium-90 selective internal radiation therapy(Y-90 SIRT) was successfully utilized to elucidate underlying tumor biology and aid resection of a large multifocal recurrentmetastatic SPNP in the right hemi-liver of a 59-year-old female. Thus, in cases where curative metastatectomy remains thetreatment goal in management of recurrent and/or metastatic SPNPs, Y-90 SIRT is a safe and effective adjunct treatment tofacilitate curative resection.INTRODUCTIONSolid pseudopapillary neoplasms of the pancreas (SPNP)represent a rare form of exocrine pancreatic tumor with well-documented predisposition in young women. These typicallypresent with abdominal pain and have a pathognomonicappearance on imaging as large encapsulated masses with intra-tumoral hemorrhage. Prognosis is excellent with reported sur-vival at 5 years being >95% with optimal surgical resection butaggressive behavior has been reported in some cases especiallyReceived: July 20, 2020. Accepted: July 27, 2020Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2020.This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited.For commercial re-use, please contact journals.permissions@oup.comwith male sex, vascular invasion, extra-pancreatic invasion,nodal metastasis, and Ki67 index > 4% [1]. In the latter groupthe disease-specific survival is significantly poorer with 5-yearsurvival reported to be around 35% [2] and more importantly,absence of these features, does not preclude aggressive behavior.Nevertheless, aggressive surgical metastatectomy remains thestandard of care with excellent long-term survival.We present an interesting case of SPNP with metachronoushepatic metastasis despite an R0 index resection, lack of2 A. R. Dyas et al.Figure 1: CT imaging of metastatic recurrent SPNP in right hepatic lobe before (A) and after (B) Y-90 treatment. (C) Characteristic pseudopapillae with hyaline stroma(H&E staining, ×4 magnification). (D) Confirmatory β-catenin immunostaining with negative chromogranin A staining (inset)histological features suggestive of aggressive behavior and longrecurrence free interval. The presence of bulky liver metastasiswith satellitosis and associated portocaval lymphadenopathyadded an extra layer of complexity to the surgical managementand merit discussion as there are no evidence-based guidelinesto direct decision making in this context.CASE REPORTA 59-year-old female presented with a 7.6 × 9.7 cm hetero-geneous mass involving the tail and body of the pancreas in2008. Since, the patient was undergoing evaluation for a lym-phoma in light of a recently discovered forehead lesion, anendoscopic ultrasound guided biopsy was performed to rule outan alternative diagnosis which confirmed the mass to be SPNP.She underwent a distal pancreatectomy and splenectomy withsegmental transverse colectomy and negative surgical margins.She recovered well from her operation and did not show anyevidence of recurrence up until December 2011, after which shewas lost to follow up.She presented again in November 2018 with a 3-monthhistory of progressive right upper quadrant abdominal pain.Computed tomography (CT) imaging revealed a large hypoat-tenuating 10.3 × 12.7 × 9.8 cm3 lesion in the right hemi-liver,with multiple satellite lesions (Fig. 1A). There was no radio-graphic evidence of vascular invasion but extensive portocavallymphadenopathy was noted. Liver core needle biopsy wasconsistent with recurrent solid pseudopapillary neoplasm. Aftera multidisciplinary discussion, it was recommended that sheundergo Yttrium-90 selective internal radiation therapy (Y-90SIRT) prior to resection and she received 118.4 Gy of treatmentdose from a single administration of Theraspheres in January2019. Posttreatment surveillance imaging in April revealed aremarkable interval decrease in size of the dominant mass to8.3 × 6.4 × 7.1 cm3 without identification of any new lesions andstable lymphadenopathy (Fig. 1B). Given the favorable responseto SIRT, long progression free interval and her excellent clinicalstatus, an open extended right hepatectomy and portocavalas well as left renal vein lymphadenectomy were performed.Notably, extensive sclerosis and fibrotic reaction secondary tothe Y-90 treatment made the portal dissection more challenging,but very accomplishable. She tolerated the procedure well andafter a week in hospital was discharged home. Final pathologywas consistent with multifocal solid pseudopapillary tumor,with negative margins and no evidence of tumor cells in thelymph nodes examined (Fig. 1C,D). There was visible treatmentresponse with 50% residual tumor viability. She remainssymptom free without any evidence of residual or recurrentdisease.DISCUSSIONLocal recurrence or metachronous metastasis after radicalresection of SPNP have been reported in 15% of the casesand liver is the most commonly involved organ. This is likelyan underrepresentation because no evidence-based recom-mendations regarding postoperative surveillance exist. Basedon systematic review of reported cases, the mean follow-upduration falls way short of the time to recurrence and two-thirdsof the patients have a surveillance period < 5 years [3]. There aredocumented instances of metastatic progression in the liverYttrium-90 selective internal radiotherapy 3even after a hepatic metastatectomy and therefore a multidisci-plinary evaluation before undertaking a major liver resection iswarranted.There is conspicuous paucity of data regarding utilization ofadjuvant treatment of metastatic SPNP when metastatectomyis either technically complex or not feasible. Unlike in patientswith metastatic pancreatic adenocarcinoma, the role of systemicchemotherapy in this setting is unclear with conflicting reportsand absence of high-quality data to carry out any meaningfulsystematic meta-analysis. Other modalities which have beenemployed with varying degrees of success include liver trans-plantation [4], intensity modulated radiation therapy (IMRT) [5],selective internal radiotherapy (SIRT) [6], radiofrequency abla-tion (RFA) [7] and even hyper thermic intraperitoneal chemother-apy [8].Transarterial chemoembolization (TACE) and transarterialembolization (TAE) are commonly used for a variety of hepatictumors and metastatic lesions but their use in SPNP metastasishas largely remained unexplored. Matsuda et al. [9] utilizedTACE in combination with systemic combination chemotherapy(5-fluorouracil, doxorubicin and mitomycin-C) with palliativeintent in a patient with bilobar hepatic metastasis. They per-formed chemoembolization using doxorubicin in the right hemi-liver and intra-arterial infusion of doxorubicin alone in the lefthemi-liver. Following this, the right-sided lesions decreased involume (mean reduction 28%, range 13–54%), while the left-sidedlesions showed an increase in size. Prasad et al. [10] reporteda similar case where a 40-year-old female presented withwidespread bilobar hepatic metastasis 4 years after undergoinga distal pancreatectomy for SPNP. She underwent TACE withgemcitabine and lipiodol followed by gel foam embolization andhad 90% reduction in size and enhancement of the lesions onrepeat imaging performed a month after treatment.We opted to perform Y-90 SIRT prior to surgical resectionbecause the extensive involvement of the right hemi-liver man-dated a formal large hepatectomy and it was deemed impera-tive to rule out occult metastatic lesions in the remaining liverprior to a large parenchymal resection. Furthermore, the ensuingcompensatory left hemi-liver hypertrophy decreased the riskof post-hepatectomy liver insufficiency. It can be argued thatany potential technical advantage derived from the reduction intumor volume was countervailed by the post-SIRT inflammatorychanges, which made the portal dissection more exacting. Nev-ertheless, this case illustrates that since long-term survival withmetastatectomy is the rule rather than exception, use of TACE orany adjunct modalities may be employed safely and effectivelyas a bridge to surgical metastatectomy.CONFLICT OF INTEREST STATEMENTNone declared.FUNDINGNone.REFERENCES1. Machado MC, Machado MA, Bacchella T, Jukemura J, AlmeidaJL, Cunha JE. Solid pseudopapillary neoplasm of the pan-creas: distinct patterns of onset, diagnosis, and prognosis formale versus female patients. Surgery 2008;143:29–34.2. Law JK, Ahmed A, Singh VK, Akshintala VS, Olson MT, RamanSP, et al. A systematic review of solid-pseudopapillary neo-plasms: are these rare lesions? Pancreas 2014;43:331–7.3. Papavramidis T, Papavramidis S. Solid pseudopapillarytumors of the pancreas: review of 718 patients reported inEnglish literature. J Am Coll Surg 2005;200:965–72.4. Sumida W, Kaneko K, Tainaka T, Ono Y, Kiuchi T, AndoH. Liver transplantation for multiple liver metastases fromsolid pseudopapillary tumor of the pancreas. J Pediatr Surg2007;42:e27–31.5. Zauls JA, Dragun AE, Sharma AK. Intensity-modulated radi-ation therapy for unresectable solid pseudopapillary tumorof the pancreas. Am J Clin Oncol 2006;29:639–40.6. Krug S, Bartsch DK, Schober M, Librizzi D, Pfestroff A,Burbelko M, et al. Successful selective internal radiotherapy(SIRT) in a patient with a malignant solid pseudopapillarypancreatic neoplasm (SPN). Pancreatology 2012;12:423–7.7. Sugito K, Kusafuka T, Hoshino M, Inoue M, Ikeda T, Hagi-wara N, et al. Application of radiofrequency ablation forgiant solid pseudopapillary tumor of the pancreas. Pediatr Int2010;52:e29–31.8. Honore C, Goere D, Dartigues P, Burtin P, Dumont F, Elias D.Peritoneal carcinomatosis from solid pseudopapillary neo-plasm (Frantz’s tumour) of the pancreas treated with HIPEC.Anticancer Res 2012;32:1069–73.9. Matsuda Y, Imai Y, Kawata S, Nishikawa M, Miyoshi S, SaitoR, et al. Papillary-cystic neoplasm of the pancreas withmultiple hepatic metastases: a case report. Gastroenterol Jpn1987;22:379–84.10. Prasad TV, Madhusudhan KS, Srivastava DN, Dash NR, GuptaAK. Transarterial chemoembolization for liver metastasesfrom solid pseudopapillary epithelial neoplasm of pancreas:a case report. World J Radiol 2015;7:61–5.

Yttrium-90 Selective Internal Radiotherapy As Bridge To Curative Hepatectomy For Recurrent Malignant Solid Pseudopapillary Neoplasm of Pancreas: Case Report and Review of Literature

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Yttrium-90 Selective Internal Radiotherapy As Bridge to Curative Hepatectomy for Recurrent Malignant Solid Pseudopapillary Neoplasm of Pancreas: Case Report and Review of Literature

Yttrium-90 Selective Internal Radiotherapy As Bridge to Curative Hepatectomy for Recurrent Malignant Solid Pseudopapillary Neoplasm of Pancreas: Case Report and Review of Literature

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