Stereotactic Radiotherapy in the Treatment of Paraneoplastic Vasculitis in Oligometastatic Renal Cell Carcinoma
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Case Report Stereotactic Radiotherapy in the Treatment of Paraneoplastic Vasculitis in Oligometastatic Renal Cell Carcinoma Laura Burgess 1,* , Marissa Keenan 2, Alan Liang Zhou 3 , Kiefer Lypka 3, Delvina Hasimja Saraqini 3 , Jeff Yao 4, Samuel Martin 5, Christopher Morash 4, James Watterson 4, Christina Canil 6 and Robert MacRae 1 1 Department of Radiology, Division of Radiation Oncology, University of Ottawa, 501 Smyth Road, Ottawa, ON K1H8L6, Canada; [email protected] 2 Department of Medicine, Division of Rheumatology, University of Ottawa, 501 Smyth Road, Ottawa, ON K1H8L6, Canada; [email protected] 3 Department of Medicine, University of Ottawa, 501 Smyth Road, Ottawa, ON K1H8L6, Canada; [email protected] (A.L.Z.); [email protected] (K.L.); [email protected] (D.H.S.) 4 Department of Surgery, Division of Urology, University of Ottawa, 501 Smyth Road, Ottawa, ON K1H8L6, Canada; [email protected] (J.Y.); [email protected] (C.M.); [email protected] (J.W.) 5 Faculty of Medicine, University of Ottawa, 501 Smyth Road, Ottawa, ON K1H8L6, Canada; [email protected] 6 Department of Medicine, Division of Medical Oncology, University of Ottawa, 501 Smyth Road, Ottawa, ON K1H8L6, Canada; [email protected] * Correspondence: [email protected] Abstract: Approximately 20% of renal cell carcinoma (RCC) is diagnosed because of paraneoplastic manifestations. RCC has been associated with a large variety of paraneoplastic syndromes (PNS), but it is rarely associated with PNS vasculitis. We present a case of a previously healthy male who presented with systemic vasculitis; bitemporal headaches, diplopia, polyarthritis, palpable Citation: Burgess, L.; Keenan, M.; purpura, tongue lesion, peri-orbital edema, scleritis, chondritis and constitutional symptoms. He was Zhou, A.L.; Lypka, K.; Hasimja subsequently found to have oligometastatic RCC. Both his primary lesion and site of oligometastasis Saraqini, D.; Yao, J.; Martin, S.; Morash, C.; Watterson, J.; Canil, C.; were treated with stereotactic radiotherapy (SBRT) and resulted in the resolution of his vasculitis, as et al. Stereotactic Radiotherapy in the well as sustained oncologic response. This is the first case to demonstrate that effective sustained Treatment of Paraneoplastic Vasculitis treatment for PNS vasculitis due to oligometastatic RCC is possible with SBRT. in Oligometastatic Renal Cell Carcinoma. Curr. Oncol. 2021, 28, Keywords: stereotactic radiotherapy (SBRT); renal cell carcinoma (RCC); paraneoplastic vasculitis; 1744–1750. https://doi.org/10.3390/ paraneoplastic syndrome; oligometastases curroncol28030162 Received: 5 April 2021 Accepted: 30 April 2021 1. Introduction Published: 7 May 2021 The classic triad in renal cell carcinoma (RCC) of flank pain, gross hematuria and a palpable mass is seen in approximately 10% of cases [1]. Secondary to improved diagnos- Publisher’s Note: MDPI stays neutral tic imaging, most RCCs are detected incidentally. However, approximately 20% of RCC with regard to jurisdictional claims in patients are diagnosed as a result of paraneoplastic manifestations [2]. A wide range of para- published maps and institutional affil- iations. neoplastic syndromes (PNS) are associated with RCC, but PNS vasculitis is very rare [3]. Herein, we review a case of a previously healthy man diagnosed with oligometastatic RCC following an atypical presentation of systemic vasculitis who demonstrated sus- tained and effective resolution of his vasculitis following treatment with stereotactic radiotherapy (SBRT). Copyright: © 2021 by the authors. Licensee MDPI, Basel, Switzerland. 2. Case This article is an open access article A 60-year-old male with a past medical history significant for asthma, allergic rhinitis, distributed under the terms and conditions of the Creative Commons and dyslipidemia presented to the emergency department with a three-day history of Attribution (CC BY) license (https:// binocular diplopia, right eye pain, diffuse joint pain and edema in the feet, ankles and creativecommons.org/licenses/by/ wrists bilaterally, and a rash on the feet tracking up to the groin. Two months prior to 4.0/). this presentation, he developed persistent bi-frontal and bi-occipital headaches, fevers, Curr. Oncol. 2021, 28, 1744–1750. https://doi.org/10.3390/curroncol28030162 https://www.mdpi.com/journal/curroncol Curr. Oncol. 2021, 28, FOR PEER REVIEW 2 Curr. Oncol. 2021, 28 1745 presentation, he developed persistent bi-frontal and bi-occipital headaches, fevers, night sweats, and a swollen nose that failed two courses of outpatient antibiotics. Other outpa- tientnight investigations sweats, and including a swollen nose a computed that failed tomography two courses of outpatient(CT) head, antibiotics. CT venogram, Other and lum- bar puncture;outpatient investigations they were non-contributory. including a computed tomography (CT) head, CT venogram, and lumbar puncture; they were non-contributory. He was found to have right peri-orbital edema and conjunctival injection, Figure 1A, He was found to have right peri-orbital edema and conjunctival injection, Figure1A, withwith preserved preserved extra-ocular extra-ocular movements movements and and no no ptosis. ptosis. He He had had a circular a circular 1 cm ×1cm1 cm × 1cm white lesionwhite on his lesion lateral on his right lateral tongue, right tongue, Figure Figure 1B,1 B,edema/erythema edema/erythema of of the the nasal nasal bone bone and and upper cartilage,upper and cartilage, cervical and cervicallymphadenopathy. lymphadenopathy. He He was was noted noted to to havehave bilateral bilateral synovitis synovitis of the metatarsophalangealof the metatarsophalangeal joints, joints, ankles, ankles, elbows, elbows, wrists, wrists, and metacarpophalangeal metacarpophalangeal joints. joints. His His purpuric rash was palpable, non-blanching, and extended from his lower extremities purpuricto his buttocks.rash was The palpable, remainder non-blanching, of his exam was normal. and extended from his lower extremities to his buttocks. The remainder of his exam was normal. FigureFigure 1. (A 1.) (RightA) Right peri-orbital peri-orbital edema edema andand conjunctival conjunctival injection injection at presentation; at presentation; (B) The circular(B) The circular 1 cm × 11 cmcm× white1 cm white lesion lesion on onhis his late lateralral right right tonguetongue at at presentation. presentation. Initial investigations revealed a normocytic anemia (Hb 115 g/L) and neutrophilic leukocytosisInitial investigations (white blood revealed cell count 20.2a normocyt× 109/L, neutrophilsic anemia 17.3 (Hb× 101159/L). g/L) Renal and func- neutrophilic leukocytosistion was maintained; (white blood however, cell count urine studies 20.2 × revealed109/L, neutrophils hematuria and 17.3 non-nephrotic × 109/L). Renal pro- function was teinuriamaintained; (2.48 g/day). however, Liver enzymesurine studies revealed re avealed mixed transaminitis hematuria (AST and 90 non-nephrotic U/L, ALT pro- teinuria123 U/L, (2.48 ALP g/day). 242 U/L, Liver GGT enzymes 448 U/L). revealed Inflammatory a mixed markers transaminitis were elevated (AST (erythrocyte 90 U/L, ALT 123 sedimentation rate 122 mm/h, C-reactive protein > 190 mg/L). CT orbit revealed soft U/L, tissueALP thickening,242 U/L, GGT fat stranding, 448 U/L). and Inflammatory enhancement in themarkers right pre-septal were elevated region. CT(erythrocyte head sed- imentationand neck rate revealed 122 mm/hr, bilateral C-reactive prominent neck protein lymphadenopathy, > 190 mg/L). more CT orbit extensive revealed on the soft tissue thickening,right. CT fat abdomen stranding, and pelvis and enhancement revealed a 1.9 cm in indeterminate the right pre-septal isodense to region. hypodense CT head and necklesion revealed in the bilateral mid to lower prominent pole of the neck left kidney, lymp Figurehadenopathy,2A. He was more pan-cultured, extensive started on the right. CT abdomenon piperacillin/tazobactam, and pelvis revealed and admitted a 1.9 cm to inde internalterminate medicine. isodense Infectious to diseases hypodense and lesion in rheumatology were consulted. The differential remained broad and included infectious, the midautoimmune, to lower and pole malignant of the left aetiologies. kidney, Figure 2A. He was pan-cultured, started on pip- eracillin/tazobactam, and admitted to internal medicine. Infectious diseases and rheuma- tology were consulted. The differential remained broad and included infectious, autoim- mune, and malignant aetiologies. Curr.Curr. Oncol. Oncol.2021 2021, 28, 28, FOR PEER REVIEW 17463 FigureFigure 2. 2. ((AA)) AxialAxial CTCT revealingrevealing indeterminateindeterminate 18 mm left renal lesion; ( (BB)) Axial Axial dynamic dynamic gradient gradient echo echo sequence sequence demonstratingdemonScheme 17 17. mm mm T2 T2 hypointense hypointense left left renal renal lesion lesion in in keeping keeping with with renalrenal cellcell carcinoma.carcinoma. AnAn extensiveextensive infectious work-up returned negative; negative; including including blood blood cultures, cultures, urine urine cultures,cultures, stoolstool cultures,cultures, stoolstool ova and parasites, Chlamydia/Gonorrhea, and and serologies serologies for for HIV,HIV, hepatitishepatitis B,B, hepatitishepatitis C,C, cytomegalovirus,cytomegalovirus, Epstein-Barr virus, virus, lyme, lyme, and and syphilis. syphilis. An An autoimmuneautoimmune work-upwork-up revealed a positive RF (123 kIU/L), kIU/L), anti-CCP anti-CCP (250 (250 U), U), weakly weakly posi- posi- tivetive ANAANA (1:80,(1:80, speckled),speckled), ENAENA positivepositive forfor