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Constipation, the sole presentation of primary renal carcinoid tumor: A case report:

Constipation, the sole presentation of primary renal carcinoid tumor: A case report: Primary renal carcinoid tumors are quite rare. The pathogenesis of these tumors is unknown due to lack of enterochromaffin cells in the kidney. Because of nonspecific clinical manifestations and radiologic features, they are commonly misdiagnosed. Hence, Primary renal carcinoid tumors should be considered in differential diagnosis of any renal mass. In the present case, a 26-year-old woman was presented with a renal mass and constipation. After partial nephrectomy, diagnosis of carcinoid tumor was confirmed. Keywords Carcinoid tumor, renal mass, renal carcinoid tumor, constipation Date received: 4 April 2019; accepted: 28 August 2019 Hemoglobin (Hb) = 13 g/dL, Hematocrit (HCT) = 42.3%, Introduction white blood cell (WBC) = 9.26 × 10 /µL (Neutrophil = 76%, Carcinoid tumors are neuroendocrine tumors that arise Lymphocyte = 22%, Eosinophil = 3%, Monocyte = 4%, from the enterochromaffin cells. Primary renal carcinoid Basophils = 1%), Platelets = 351 × 10 /mL, blood urea tumors (PRCTs) are externally rare because the entero- nitrogen (BUN) = 13 mg/dL, Creatinine = 08 mg/dL, Na = 143 chromaffin cells are absent in the renal parenchyma. mEq/L, K = 4.9 mEq/L, lactate dehydrogenase (LDH) = 154 The clinical manifestations of PRCTs are usually non- Iu/L, and Ferritin = 25.7 ng/mL. specific, and sometimes they are found incidentally on Liver function tests and urinalysis were normal. Chest radiologic studies. We herein report a case of PRCT in a X-ray was normal, too. Abdominopelvic ultrasonography young female who was presented with constipation as the showed a 30 mm × 33 mm echogenic mass in middle pole sole symptom of the tumor. Department of Urology, Shiraz University of Medical Sciences, Shiraz, Iran Case presentation 2 Department of Internal Medicine, Shiraz University of Medical Sciences, Shiraz, Iran A 26-year-old woman was presented with a history of con- Department of Pathology, Transplant Research Center, Shiraz stipation for 2 months. The patient had negative history of University of Medical Sciences, Shiraz, Iran fever, flashing, diarrhea, sweating, gross hematuria, and Corresponding author: any other urinary tract symptoms. Her post-medical history Mehdi Salehipour, Department of Urology, Faghihi Hospital, Shiraz was unremarkable. Physical examination revealed mild University of Medical Sciences, P.O. Box:7461684873, Shiraz, abdominal distension. 7134844119, Iran. The laboratory findings were as follows: Email: Salehipourmehdi@Yahoo.com Creative Commons CC BY: This article is distributed under the terms of the Creative Commons Attribution 4.0 License (http://www.creativecommons.org/licenses/by/4.0/) which permits any use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages (https://us.sagepub.com/en-us/nam/open-access-at-sage). 2 Rare Tumors Figure 2. Sections from renal mass show hyper cellular tumor with trabecular and nesting pattern. Individual tumor cells show salt and pepper nuclei (H&EX 250). Figure 1. Abdominopelvic spiral CT scan: a 38 mm × 30 mm × 43 mm hypodense solid mass was found in midpole of the left kidney that enhanced after administration of contrast material. of the left kidney. Abdominopelvic spiral CT scan demon- strated a 38 mm × 30 mm × 43 mm hypodense solid mass in the midpole of the left kidney that enhanced after adminis- tration of contrast material (Figure 1). Thus, with a prelimi- nary diagnosis of renal malignancy, she underwent left Figure 3. IHC shows positive chromogranin (diffuse and strong). partial nephrectomy through left flunk incision. Histo-pathologic examination indicated monomorphic cells with trabecular and nesting pattern (Figure 2). Carcinoid tumors occur in various organs of the body, Individual cells showed indistinct cytoplasmic borders and with the gastrointestinal tract being the most common site nuclei with salt and pepper stippled chromatin. No mitosis which accounts for 73%, while the respiratory tract or necrosis was seen. Immunohistochemistry was positive accounts for 25% of them. The testis and prostate are the for chromogranin and synaptophysin and negative for other most common sites of carcinoid tumors in the genitouri- renal cell carcinoma (RCC) markers (Figure 3). Proliferative 1,2 nary tract. PRCT is a very rare but a well-defined entity. index by Ki67 was low (Figure 4). Resnick et al. reported the first PRCT in 1966. The patho- After diagnosis of carcinoid tumor, octreotide scan was genesis of PRCT is uncertain because the neuroendocrine performed that showed two focal zones of increased radi- cells are not found in the renal parencyme. Several theo- otracer uptakes in the fourth and eighth segments of the liver. ries regarding the pathogenesis of PRCT have been postu- The patient’s chromogranin A was normal, so decision was lated including: made for radiofrequency (RF) ablation of the liver lesions. At the 6-month follow-up, the patient remained well. 1. Intestinal metaplasia of the pyelocalyceal urothe- 1,4 lium due to chronic inflammation; 2. Metastases from unknown primary origin to the Discussion 2,4 kidney; 3. Intrapped neural crest or pancreatic cells; Carcinoid tumors are low-malignant potential neuroendo- 1 1,4,6 4. Primitive stem cell differentiation. crine tumors arising from the enterochromaffin cells. Salehipour et al. 3 Radioactive octreotide is a synthetic somatostatin ana- logue that binds the somatostatin receptors (SR) with high affinity. Although octreotide scintigraphy has a highly reported sensitivity in detecting carcinoid tumors of gastro- intestinal origin, its role in PRCTs is not well established since little is known about the prevalence of SR in renal carcinoid tumors. On the contrary, normal renal paren- chyma can uptake the trace material and so obscure a suspi- 5,6,10 cious lesion. Thus, SRS is not routinely used in pre-operative evaluation of PRCTs. Complete surgical excision by radical or partial nephrec- tomy in association with lymphadenectomy is the treatment 1,5,6 of choice of PRCTs. Whenever the diagnosis of PRCTs is made, metastatic work-up for hidden tumors in other sites must be done. SRS is an important and useful tool in the evaluation of metastatic carcinoid tumors. Figure 4. IHC for Ki67 is low (<3%). Given the paucity of cases, the role of chemotherapy, somatostatin analogues, and targeted therapy with tyrosine Carcinoid tumors of the kidney have been reported in kinase inhibitors has not been well established in treatment some congenital renal anomalies, among which the most of metastatic renal carcinoid tumors. The prognosis of 6 2 common anomaly is horseshoe kidneys. Romero et al. PRCTs seems to be good. The most important prognostic showed that 17.8% of renal carcinoid tumors were associated factor is tumor stage at presentation. Tumors confined to with horseshoe kidneys, 14.3% with teratomas, and 18% the kidney, with size less than 4 cm and age younger than 40 with polycystic kidney disease. In another study, 25% of were found to have a good prognosis. PRCTs arising within renal carcinoid tumors were associated with the horseshoe a horseshoe kidney tend to have less aggressive behavior kidney. The relative risk of carcinoid tumors in individuals compared with primary carcinoid tumors originating from with horseshoe kidneys has been calculated between 62 and a normal kidney. 120. Increased occurrence of renal carcinoids in the horse- In conclusion, PRCTs are very rare and should be con- shoe kidney might be related to the existence of aberrant epi- sidered in differential diagnosis of other renal masses. 1,4 thelium or teratomatous elements in those kidneys. Hansel Surgical resection is currently the only treatment. There is et al. reported that PRCTs were mainly seen in patients limited data about the role of other therapeutic modalities younger than 50 years old and there was no tendency in gen- such as chemotherapy and targeted therapy in the treatment der and location. On the contrary, Omiyale and Venyo of PRCTs. After diagnosis of PRCTs, metastatic work up showed that carcinoid tumors associated with the horseshoe must be performed to find occult metastases in other organs. kidneys tend to have a male predilection which could be due Close and long-term follow-up is essential because the to the higher incidence of the horseshoe kidneys in men. indolent course of the disease. The clinical presentations of PRCTs are often nonspe- cific and can divided into either localized symptoms such Acknowledgements as hematuria and flank pain, or systemic symptoms such as The authors would like to thank Nasrin Shokrpour English fever, flashing, diarrhea, weight loss, perspiration and con- Department for his editorial assistance in the Research Consulting 1,3 stipation. Systemic symptoms could be explained to be Center (RCC) of Shiraz University of Medical Sciences for due to production and secretion of several hormones. For improving the use of English in the manuscript. example, constipation might be due to the secretion of pep- tide YY. On the contrary, 25–30% of renal carcinoids Conflict of interest were diagnosed as an incidental finding during radiologic The author(s) declared no potential conflicts of interest with investigations. Imaging study of choice for evaluation of respect to the research, authorship, and/or publication of this the renal masses including renal carcinoid tumors is the article. helical CT scan. PRCTs show variable and nonspecific findings in the helical CT scan. They may be hyperdense, Ethical approval hypodense, or isodense in comparison with normal renal Ethical approval to this research was obtained from Shiraz 4,5 parenchyma. After administration of contrast material, University of Medical Science research committee. PRCT may be demonstrated marked, minimal, or no 1,5 enhancement. Punctate calcification is repeated in some 1 Funding patients. Somatostatin receptor scintigraphy (SRS) is a useful imaging modality for staging and detecting the The author(s) received no financial support for the research, recurrence or metastasis of carcinoid tumors. authorship, and/or publication of this article. 4 Rare Tumors Informed consent 5. Lamb L and Shaban W. Primary renal carcinoid tumor: a radiologic review. Radiol Case Rep 2014; 9(2): 923. Written informed consent was obtained from the patient for her 6. Shurtleff BT, Shvart O and Rajfer J. Carcinoid tumor of the anonymized information to be published in this article. kidney: case report and review of the literature. Rev Urol 2005; 7(4): 229–233. ORCID iDs 7. Krishnan B, Truong LD, Saleh G, et al. Horseshoe Mehdi Salehipour https://orcid.org/0000-0002-8780-6490 kidney is associated with an increased relative risk of pri- Alireza Makarem https://orcid.org/0000-0001-8821-7973 mary renal carcinoid tumor. J Urol 1997; 157(6): 2059– References 8. Hansel DE, Epstein JI, Berbescu E, et al. Renal carcinoid tumor: a clinicopathologic study of 21 cases. Am J Surg 1. Omiyale AO and Venyo AK-G. Primary carcinoid tumors Pathol 2007; 31(10): 1539–1544. of the kidney: a review of the literature. Adv Urol 2013; 9. Noh HK, Kwon BS, Kim YH, et al. Peptide YY producing 2013(e7): 579396. strumal carcinoid tumor of the ovary in a postmenopausal 2. Romero FR, Rais-Bahrami S, Permpongkosol S, et al. woman: a rare cause of chronic constipation. Obstet Gynecol Primary carcinoid tumors of the kidney. J Urol 2006; 176(6 Sci 2017; 60(6): 602–607. Pt 1): 2359–2366. 10. Shi W, Johnson CF, Buchanan KD, et al. Localization of neu- 3. Resnick ME. Renal carcinoid producing the carcinoid syn- drome. Med Times 1996; 94: 895–896. roendocrine tumors with [111n] DTPA—octreotide scintig- 4. Weng CL, Chen CJ, Lin SP, et al. Primary renal carcinoid raphy (Octreoscan): a comparative study with CT and MR tumor. J Radiol SCI 2011; 36: 237–240. imaging. QJM 1998; 9: 295–301. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Rare Tumors SAGE

Constipation, the sole presentation of primary renal carcinoid tumor: A case report:

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Abstract

Primary renal carcinoid tumors are quite rare. The pathogenesis of these tumors is unknown due to lack of enterochromaffin cells in the kidney. Because of nonspecific clinical manifestations and radiologic features, they are commonly misdiagnosed. Hence, Primary renal carcinoid tumors should be considered in differential diagnosis of any renal mass. In the present case, a 26-year-old woman was presented with a renal mass and constipation. After partial nephrectomy, diagnosis of carcinoid tumor was confirmed. Keywords Carcinoid tumor, renal mass, renal carcinoid tumor, constipation Date received: 4 April 2019; accepted: 28 August 2019 Hemoglobin (Hb) = 13 g/dL, Hematocrit (HCT) = 42.3%, Introduction white blood cell (WBC) = 9.26 × 10 /µL (Neutrophil = 76%, Carcinoid tumors are neuroendocrine tumors that arise Lymphocyte = 22%, Eosinophil = 3%, Monocyte = 4%, from the enterochromaffin cells. Primary renal carcinoid Basophils = 1%), Platelets = 351 × 10 /mL, blood urea tumors (PRCTs) are externally rare because the entero- nitrogen (BUN) = 13 mg/dL, Creatinine = 08 mg/dL, Na = 143 chromaffin cells are absent in the renal parenchyma. mEq/L, K = 4.9 mEq/L, lactate dehydrogenase (LDH) = 154 The clinical manifestations of PRCTs are usually non- Iu/L, and Ferritin = 25.7 ng/mL. specific, and sometimes they are found incidentally on Liver function tests and urinalysis were normal. Chest radiologic studies. We herein report a case of PRCT in a X-ray was normal, too. Abdominopelvic ultrasonography young female who was presented with constipation as the showed a 30 mm × 33 mm echogenic mass in middle pole sole symptom of the tumor. Department of Urology, Shiraz University of Medical Sciences, Shiraz, Iran Case presentation 2 Department of Internal Medicine, Shiraz University of Medical Sciences, Shiraz, Iran A 26-year-old woman was presented with a history of con- Department of Pathology, Transplant Research Center, Shiraz stipation for 2 months. The patient had negative history of University of Medical Sciences, Shiraz, Iran fever, flashing, diarrhea, sweating, gross hematuria, and Corresponding author: any other urinary tract symptoms. Her post-medical history Mehdi Salehipour, Department of Urology, Faghihi Hospital, Shiraz was unremarkable. Physical examination revealed mild University of Medical Sciences, P.O. Box:7461684873, Shiraz, abdominal distension. 7134844119, Iran. The laboratory findings were as follows: Email: Salehipourmehdi@Yahoo.com Creative Commons CC BY: This article is distributed under the terms of the Creative Commons Attribution 4.0 License (http://www.creativecommons.org/licenses/by/4.0/) which permits any use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages (https://us.sagepub.com/en-us/nam/open-access-at-sage). 2 Rare Tumors Figure 2. Sections from renal mass show hyper cellular tumor with trabecular and nesting pattern. Individual tumor cells show salt and pepper nuclei (H&EX 250). Figure 1. Abdominopelvic spiral CT scan: a 38 mm × 30 mm × 43 mm hypodense solid mass was found in midpole of the left kidney that enhanced after administration of contrast material. of the left kidney. Abdominopelvic spiral CT scan demon- strated a 38 mm × 30 mm × 43 mm hypodense solid mass in the midpole of the left kidney that enhanced after adminis- tration of contrast material (Figure 1). Thus, with a prelimi- nary diagnosis of renal malignancy, she underwent left Figure 3. IHC shows positive chromogranin (diffuse and strong). partial nephrectomy through left flunk incision. Histo-pathologic examination indicated monomorphic cells with trabecular and nesting pattern (Figure 2). Carcinoid tumors occur in various organs of the body, Individual cells showed indistinct cytoplasmic borders and with the gastrointestinal tract being the most common site nuclei with salt and pepper stippled chromatin. No mitosis which accounts for 73%, while the respiratory tract or necrosis was seen. Immunohistochemistry was positive accounts for 25% of them. The testis and prostate are the for chromogranin and synaptophysin and negative for other most common sites of carcinoid tumors in the genitouri- renal cell carcinoma (RCC) markers (Figure 3). Proliferative 1,2 nary tract. PRCT is a very rare but a well-defined entity. index by Ki67 was low (Figure 4). Resnick et al. reported the first PRCT in 1966. The patho- After diagnosis of carcinoid tumor, octreotide scan was genesis of PRCT is uncertain because the neuroendocrine performed that showed two focal zones of increased radi- cells are not found in the renal parencyme. Several theo- otracer uptakes in the fourth and eighth segments of the liver. ries regarding the pathogenesis of PRCT have been postu- The patient’s chromogranin A was normal, so decision was lated including: made for radiofrequency (RF) ablation of the liver lesions. At the 6-month follow-up, the patient remained well. 1. Intestinal metaplasia of the pyelocalyceal urothe- 1,4 lium due to chronic inflammation; 2. Metastases from unknown primary origin to the Discussion 2,4 kidney; 3. Intrapped neural crest or pancreatic cells; Carcinoid tumors are low-malignant potential neuroendo- 1 1,4,6 4. Primitive stem cell differentiation. crine tumors arising from the enterochromaffin cells. Salehipour et al. 3 Radioactive octreotide is a synthetic somatostatin ana- logue that binds the somatostatin receptors (SR) with high affinity. Although octreotide scintigraphy has a highly reported sensitivity in detecting carcinoid tumors of gastro- intestinal origin, its role in PRCTs is not well established since little is known about the prevalence of SR in renal carcinoid tumors. On the contrary, normal renal paren- chyma can uptake the trace material and so obscure a suspi- 5,6,10 cious lesion. Thus, SRS is not routinely used in pre-operative evaluation of PRCTs. Complete surgical excision by radical or partial nephrec- tomy in association with lymphadenectomy is the treatment 1,5,6 of choice of PRCTs. Whenever the diagnosis of PRCTs is made, metastatic work-up for hidden tumors in other sites must be done. SRS is an important and useful tool in the evaluation of metastatic carcinoid tumors. Figure 4. IHC for Ki67 is low (<3%). Given the paucity of cases, the role of chemotherapy, somatostatin analogues, and targeted therapy with tyrosine Carcinoid tumors of the kidney have been reported in kinase inhibitors has not been well established in treatment some congenital renal anomalies, among which the most of metastatic renal carcinoid tumors. The prognosis of 6 2 common anomaly is horseshoe kidneys. Romero et al. PRCTs seems to be good. The most important prognostic showed that 17.8% of renal carcinoid tumors were associated factor is tumor stage at presentation. Tumors confined to with horseshoe kidneys, 14.3% with teratomas, and 18% the kidney, with size less than 4 cm and age younger than 40 with polycystic kidney disease. In another study, 25% of were found to have a good prognosis. PRCTs arising within renal carcinoid tumors were associated with the horseshoe a horseshoe kidney tend to have less aggressive behavior kidney. The relative risk of carcinoid tumors in individuals compared with primary carcinoid tumors originating from with horseshoe kidneys has been calculated between 62 and a normal kidney. 120. Increased occurrence of renal carcinoids in the horse- In conclusion, PRCTs are very rare and should be con- shoe kidney might be related to the existence of aberrant epi- sidered in differential diagnosis of other renal masses. 1,4 thelium or teratomatous elements in those kidneys. Hansel Surgical resection is currently the only treatment. There is et al. reported that PRCTs were mainly seen in patients limited data about the role of other therapeutic modalities younger than 50 years old and there was no tendency in gen- such as chemotherapy and targeted therapy in the treatment der and location. On the contrary, Omiyale and Venyo of PRCTs. After diagnosis of PRCTs, metastatic work up showed that carcinoid tumors associated with the horseshoe must be performed to find occult metastases in other organs. kidneys tend to have a male predilection which could be due Close and long-term follow-up is essential because the to the higher incidence of the horseshoe kidneys in men. indolent course of the disease. The clinical presentations of PRCTs are often nonspe- cific and can divided into either localized symptoms such Acknowledgements as hematuria and flank pain, or systemic symptoms such as The authors would like to thank Nasrin Shokrpour English fever, flashing, diarrhea, weight loss, perspiration and con- Department for his editorial assistance in the Research Consulting 1,3 stipation. Systemic symptoms could be explained to be Center (RCC) of Shiraz University of Medical Sciences for due to production and secretion of several hormones. For improving the use of English in the manuscript. example, constipation might be due to the secretion of pep- tide YY. On the contrary, 25–30% of renal carcinoids Conflict of interest were diagnosed as an incidental finding during radiologic The author(s) declared no potential conflicts of interest with investigations. Imaging study of choice for evaluation of respect to the research, authorship, and/or publication of this the renal masses including renal carcinoid tumors is the article. helical CT scan. PRCTs show variable and nonspecific findings in the helical CT scan. They may be hyperdense, Ethical approval hypodense, or isodense in comparison with normal renal Ethical approval to this research was obtained from Shiraz 4,5 parenchyma. After administration of contrast material, University of Medical Science research committee. PRCT may be demonstrated marked, minimal, or no 1,5 enhancement. Punctate calcification is repeated in some 1 Funding patients. Somatostatin receptor scintigraphy (SRS) is a useful imaging modality for staging and detecting the The author(s) received no financial support for the research, recurrence or metastasis of carcinoid tumors. authorship, and/or publication of this article. 4 Rare Tumors Informed consent 5. Lamb L and Shaban W. Primary renal carcinoid tumor: a radiologic review. Radiol Case Rep 2014; 9(2): 923. Written informed consent was obtained from the patient for her 6. Shurtleff BT, Shvart O and Rajfer J. Carcinoid tumor of the anonymized information to be published in this article. kidney: case report and review of the literature. Rev Urol 2005; 7(4): 229–233. ORCID iDs 7. Krishnan B, Truong LD, Saleh G, et al. Horseshoe Mehdi Salehipour https://orcid.org/0000-0002-8780-6490 kidney is associated with an increased relative risk of pri- Alireza Makarem https://orcid.org/0000-0001-8821-7973 mary renal carcinoid tumor. J Urol 1997; 157(6): 2059– References 8. Hansel DE, Epstein JI, Berbescu E, et al. Renal carcinoid tumor: a clinicopathologic study of 21 cases. Am J Surg 1. Omiyale AO and Venyo AK-G. Primary carcinoid tumors Pathol 2007; 31(10): 1539–1544. of the kidney: a review of the literature. Adv Urol 2013; 9. Noh HK, Kwon BS, Kim YH, et al. Peptide YY producing 2013(e7): 579396. strumal carcinoid tumor of the ovary in a postmenopausal 2. Romero FR, Rais-Bahrami S, Permpongkosol S, et al. woman: a rare cause of chronic constipation. Obstet Gynecol Primary carcinoid tumors of the kidney. J Urol 2006; 176(6 Sci 2017; 60(6): 602–607. Pt 1): 2359–2366. 10. Shi W, Johnson CF, Buchanan KD, et al. Localization of neu- 3. Resnick ME. Renal carcinoid producing the carcinoid syn- drome. Med Times 1996; 94: 895–896. roendocrine tumors with [111n] DTPA—octreotide scintig- 4. Weng CL, Chen CJ, Lin SP, et al. Primary renal carcinoid raphy (Octreoscan): a comparative study with CT and MR tumor. J Radiol SCI 2011; 36: 237–240. imaging. QJM 1998; 9: 295–301.

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Rare Tumors – SAGE

Published: Oct 23, 2019

Keywords: Carcinoid tumor; renal mass; renal carcinoid tumor; constipation

There are no references for this article.