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Primary Renal Carcinoid Tumor: a Radiologic Review Radiology Case Reports Volume 9, Issue 2, 2014 Primary renal carcinoid tumor: A radiologic review Leslie Lamb, MD, Msc, Bsc; Wael Shaban, MBBCH, MD, PhD Carcinoid tumor is the classic famous anonym of neuroendocrine neoplasms. Primary renal carcinoid tumors are extremely rare, first described by Resnick and colleagues in 1966, with fewer than a total of 100 cases reported in the literature. Thus, given the paucity of cases, the clinical and histological behav- ior is not well understood, impairing the ability to predict prognosis. Computed tomography and (occa- sionally) octreotide studies are used in the diagnosis and followup of these rare entites. A review of 85 cases in the literature shows that no distinctive imaging features differentiate them from other primary renal masses. The lesions tend to demonstrate a hypodense appearance and do not usually enhance in the arterial phases, but can occasionally calcify. Octreotide scans do not seem to help in the diagnosis; however, they are more commonly used in the postoperative followup. In addition, we report a new case of primary renal carcinoid in a horseshoe kidney. Case report Imaging findings 40-year-old male initially presented to a community hos- pital with a 20-lb weight loss over a few months. In retro- CT of the abdomen and pelvis, done in the portal ve- spect, the patient recalled mild left-flank discomfort and nous phase, demonstrated a solid, hypodense, 4.5-cm renal fatigue, but denied any hematuria. Blood work revealed an mass containing calcifications, located in the posterior as- elevated serum glucose, and he was diagnosed with type 2 pect of the medial diabetes. Further workup included ultrasound, which re- portion of the left vealed a tumor in his retroperitoneum abutting a left moi- renal moiety of ety of a horseshoe kidney. the horseshoe The patient’s past medical history was significant for re- kidney (Fig. 1). cently diagnosed type 2 diabetes and a knee ligamentous The mass did not injury at the age of 14. Medications included metformin, enhance strongly diamicron, ventolin, and symbicort. His family history con- in the venous sisted of a maternal grandfather requiring a nephrectomy; phase, and there the patient was unsure of the cause. There was no known was no apparent family history of renal-cell carcinoma. metastatic dis- Physical examination was unremarkable, with no flank or ease. Subsequent abdominal pain. All biochemical and hematological MRI attempted workup was normal, including CBC, LFTs, creatinine, and to further charac- calcium. terize the renal mass; it revealed an enhancing left renal upper pole Citation: Lamb L, Shaban W, Primary renal carcinoid tumor: A radiologic review. Radiology Case Reports. (Online) 2014;2;923. mass measuring Copyright: © 2014 The Authors. This is an open-access article distributed under the approximately terms of the Creative Commons Attribution-NonCommercial-NoDerivs 2.5 License, 4.1 x 3.8 cm, which permits reproduction and distribution, provided the original work is properly cited. Commercial use and derivative works are not permitted. which demon- The authors are both in the Department of Medical Imaging, The Ottawa Hospital, University of Ottawa, Ottawa ON, Canada. Contact Dr. Lamb at [email protected]. Figure 1. Computed tomography imaging of axial slices, unen- Competing Interests: The authors have declared that no competing interests exist. hanced (A) and enhanced (B), showing left renal carcinoid (circled DOI: 10.2484/rcr.v9i2.923 in red) in horseshoe kidney. RCR Radiology Case Reports | radiology.casereports.net! 1! 2014 | Volume 9 | Issue 2 Primary renal carcinoid tumor: A radiologic review Figure 2. MRI Discussion axial T1 Dixon VIBE out of phase Etiology and demographics (A), T1 VIBE Carcinoid tumor is the classic famous anonym of neuro- gadolinium- endocrine neoplasms, first described in 1888 by Lubarsch enhanced fat- (1). It gained its name from the earlier description of be- saturated (B), and nign behavior of the lesion despite its malignant appear- T1 FLASH ance under microscopy (2). It arises from a wide variety of gadolinium- tissues and organs, most commonly from specialized endo- enhanced fat- crine cells in the gastrointestinal and respiratory tracts, with saturated se- prevalence values of 66.9% and 24.5%, respectively (3). quences (C) The tumors produce hormones and protein products asso- showing left renal mass (circled in ciated with specific clinical symptoms, and their malignant yellow) in a potential varies by location and cell type. Primary renal horsehoe kidney. carcinoid tumors are extremely rare; they were first de- scribed by Resnick and colleagues in 1966 (4), with fewer than a total of 100 cases reported in the literature (5, 6). Thus, given the paucity of cases, the clinical and histologi- cal behavior is not well understood, impairing the ability to predict prognosis. Carcinoid tumors arise from neuroendocrine cells and are believed to originate from enterochromafin or amine precursors and decarboxylation cells with malignant poten- tial; however, neuroendocrine cells are not identified in the kidney or renal pelvis (7-10). Although renal carcinoid tu- mors exhibit morphologic and immunohistochemical fea- tures consistent with a hindgut neuroendocrine phenotype, the precise pathogenesis is controversial (11). Several hy- potheses have been proposed, on the basis that these tu- mors arise from interspersed neuroendocrine cells associ- strated a heterogeneous signal intensity on both T1 and ated with acquired and/or congenital abnormalities. The T2-weighted images with areas of bright signal on T1- first hypothesis suggests that chronic inflammation induces weighted images, likely due to intralesional hemorrhage metaplasia of the pyelocalciceal urothelium (12-14). The (Fig. 2). Although the tumor abutted the psoas muscle, it did second suggests that they are metastases from an unknown not appear to invade it. primary (15). The third is that the neural crest or pancre- atic cells have been misplaced or abnormally migrated dur- Management ing embryogenesis (14, 16). The fourth suggests concurrent A pre-operative chest radiograph was negative for meta- congenital renal abnormalities (17, 18). And the last hy- static disease. The patient was taken to the operating room pothesis suggests activation of gene sequences in multipo- and underwent an uncomplicated partial left nephrectomy. tent primitive stem cells (5, 19, 20-28). Intra-operatively, it was obvious that the mass did not in- vade the psoas muscle. Pathology revealed a well- Methods differentiated neuroendocrine tumor, its histology compati- We performed an extensive literature search for all re- ble with carcinoid. Numerous immunostains were per- ported renal primary carcinoid tumors. In total, we re- formed. The neoplastic cells were negative for inhibin, viewed 85 cases, with one unpublished case from our insti- TTF-1, keratin 903, carbonic anhydrase IX, PAX-2, RCC tution. We evaluated demographical, clinical, histopa- antigen, S-100, CD10, CK7, and CK20. The tumor cells thological, and prognostic data, with a focus on radiologic were strongly and diffusely positive for neuroendocrine findings. markers, including synaptophysin, chromogranin A, and CD56. The cells were also positive for vimentin and weakly Demographics for racemase (AMACR). The proliferation index, as dem- Of the 85 cases, 50 were males (59%) and 35 females onstrated by Ki-67 staining, was low, at 1-2%. (41%), with ages ranging from 12 to 75. The average age of diagnosis was 47.7 years, and the median age was 49 years. Followup Forty-eight tumors were right-sided, and 37 were left-sided. Postoperative CT thorax, octreotide study, endoscopy, Of the 85 cases, 50 were between 0-4 cm, 34 cases were and colonoscopy were all negative, with no source of gas- more than 4 cm, and one size was not specified. trointestinal tumor identified. The patient is now approxi- Several cases in the literature have reported coexisting mately two years post surgery, with no evidence of local renal anomalies such as horseshoe kidney and teratomas, recurrence or metastatic disease. suggesting that this disease may perhaps be more common RCR Radiology Case Reports | radiology.casereports.net! 2! 2014 | Volume 9 | Issue 2 Primary renal carcinoid tumor: A radiologic review when there are predisposing embryological factors (17, 5). literature were octreotide studies performed, 60% of which Of the 85 cases, 19 were in horseshoe kidneys (22.3%) and were 1 to 2 months after surgical resection (and were 10 in teratomas (11.8%). Of those not in horseshoe kid- negative). neys, 51 of 66 cases were in the renal parenchyma (77.3%), Thus, given the paucity of cases, octreotide sensitivity is 3 were in the renal pelvis (4.5%), 3 in the renal hilum not well established. Although it has a high stated sensitiv- (4.5%), and 8 were not specified. ity in detecting gastrointestinal tumors, relatively little is known about renal carcinoids such as the somatostatin re- Presentation ceptor prevalence. Further, a known limitation of scintig- Patients with carcinoid tumors usually present with non- raphy with octreotide to evaluate primary kidney carcinoids specific symptoms related to the mass such as pain, obstruc- is that the normal renal uptake of tracer material may ob- tion, a palpable abnormality, or hematuria. Rarely are scure a suspicious lesion (36). symptoms related to hormone production and result in CT is most often used for postoperative surveillance, but carcinoid syndrome, first described in 1957 by Pernow and it occasionally lacks the accuracy needed for postoperative Waldenstrom (29), and characterized by skin flushing and staging and monitoring. Long-term followup is suggested, telangiectasia, diarrhea, abdominal pain, cardiac valvular as new metastases have been reported as long as 7 years lesions, and bronchoconstriction. Carcinoid syndrome was after resection (6). Thus, the octreotide scan could poten- found in only 4 of the 85 cases. tially serve as an adjunct for staging and surveillance of These tumors are often discovered incidentally, as in 26 metastatic disease.
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