Small bowel intussusception secondary from intraluminal high grade epithelioid cell carcinoma of unknown origin with concurrent metastatic renal ...
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Int J Case Rep Images 2019;10:101003Z01TC2019. Chuang et al. 1 www.ijcasereportsandimages.com CASE REPORT PEER REVIEWED | OPEN ACCESS Small bowel intussusception secondary from intraluminal high grade epithelioid cell carcinoma of unknown origin with concurrent metastatic renal cell cancer Tzu-Yi (Arron) Chuang, Havish Srinath, Hassan Malik, Paul Tingey, Raphael Varghese ABSTRACT Article ID: 101003Z01TC2019 Intussusception is an unusual cause of intestinal obstruction especially in adult populations. ********* Malignant lesion accounts for approximately 30– doi: 10.5348/101003Z01TC2019CR 35% of small bowel intussusception in the adult population. We presented here a rare clinical case of a 66-year-old demented male with small bowel intussusception from intraluminal tumor INTRODUCTION of unknown origin with concurrent metastatic renal cell carcinoma. Intussusception is an unusual cause of intestinal obstruction, in which accounts for approximately Keywords: High grade epithelioid carcinoma, 1–5% of bowel obstructions. Intussusception is rare Intraluminal tumor, Intussusception, Meta- in the adult population, which carries about 5% of all static renal cell carcinoma cases of intussusceptions [1, 2]. Among of all adult intussusceptions, malignant lesion accounts for How to cite this article approximately 30–35% of small bowel intussusception [2, 3]. The pathophysiology of intussusception secondary Chuang TY, Srinath H, Malik H, Tingey P, Varghese to tumor (either intraluminal or extraluminal lesion) R. Small bowel intussusception secondary is associated with alteration of the normal peristaltic from intraluminal high grade epithelioid cell activity caused by lesions serving as a lead point [2]. carcinoma of unknown origin with concurrent We here presented a rare clinical case of a 66 years old metastatic renal cell cancer. Int J Case Rep Images demented male with small bowel intussusception from 2019;10:101003Z01TC2019. histologically-demonstrated intraluminal tumor of unknown origin, with concurrent radiologically-proven renal cell carcinoma. Tzu-Yi (Arron) Chuang1, Havish Srinath1, Hassan Malik2, Paul Tingey3, Raphael Varghese4 Affiliations: 1Surgical Principle House Officer, Department CASE REPORT of General Surgery, Ipswich Hospital, Ipswich, Queensland, Australia; 2Surgical Registrar, Department of General Sur- A 66-year-old cachectic male with history of dementia gery, Ipswich Hospital, Ipswich, Queensland, Australia; 3Sur- was initially brought in to emergency department with gical Intern, Department of General Surgery, Ipswich Hospi- tal, Ipswich, Queensland, Australia; 4General and Colorectal nausea and vomiting and abdomen distension. His past Surgical Consultant, Department of General Surgery, Ip- medical history included Korsakoff dementia secondary to swich Hospital, Ipswich, Queensland, Australia. alcohol abuse, malnutrition, vitamin D deficiency and iron Corresponding Author: Tzu-Yi (Arron) Chuang, 7/141 Sta- deficiency. Initially, he was admitted under the medical tion Road, Sunnybank, Queensland 4109, Australia; Email: team as gastroeneteritis. His blood test result revealed Thomas0227@hotmail.com that he had normal electrolyte balance with potassium of 4.8 (3.5–5.2 mmol/L), sodium of 135 (135–145 mmol/L), Chloride of 95 (95–110 mmol/L); normal kidney function Received: 18 November 2018 with creatinine of 78 (64–108 umol/L) and eGFR of 89 Accepted: 17 January 2019 (>60 ml/min/1.73 m2); haemoglobin of 119 (135–180g/L); Published: 15 February 2019 and white cell count of 14.9 (4.0–11.0x109/L). His urine is International Journal of Case Reports and Images, Vol. 10, 2019. ISSN: 0976-3198
Int J Case Rep Images 2019;10:101003Z01TC2019. Chuang et al. 2 www.ijcasereportsandimages.com clear with 10 leukocytes, 10 erythrocytes and 10 epithelial cells, suggesting no sign of urinary infection. As symptoms progressed, a CT abdomen/pelvis was conducted which demonstrated small bowel intussusception at left hemi- abdomen with the impression of a mass within the lead point (Figure 1). There was also an incidental finding suggestive of right renal cell carcinoma invading the right renal vein and probable right adrenal and right lower lobe pulmonary nodule metastasis (Figure 2). During admission, the patient had spontaneously resolved his Figure 2(A and B): Right renal mass with radiological proven intussusception and was discharged back to care facility renal cell cancer. with plan of outpatient follow up. Patient returned to the emergency department a few days later with worsening of symptoms. Repeated CT abdomen/pelvis again revealed recurrent intussusception in the right iliac fossa. He was admitted under general surgery and a nasogastric tube and in dwelling urinary catheter were inserted. His bowel obstruction had intermittently resolved and reoccurred. Decision of palliative intention surgery was discussed with his next of kin for the purpose of resolving his acute surgical problem. Optimisation of his cachectic status was planned prior to his surgery (pre-operative albumin of 27 g/L). Total parenteral nutrition was initiated with daily electrolyte check to avoid refeeding syndrome. His operation occurred on the nineteenth day of his second admission. Laparoscopy was initially conducted and mid small Figure 3: (A) (Left) Small bowel intussusception from the mass bowel intussusception with dilated proximal bowel and (B) (Right) The intraluminal mass post-enterotomy. collapsed distal bowel was identified. It was deemed to be high risk to reduce intussusception laparoscopically. Therefore, small right sided Lanz incision was made to deliver small bowel. A large palpable mass was found at the proximal side of intussusception (Figure 3A and B). A longitudinal enterotomy was made and a 60x38x40 mm intra-luminal polypoid and lobular tumor was resected by pedunculated stapled approach (Figure 4). A limited resection of small bowel where tumor was attached was performed. The primary anastomosis as functional end- to-end was conducted with PROXIMATE linear cutters Figure 1: Intussusception at left abdomen causing small bowel Figure 4: Staple resection of intraluminal mass with appropriate obstruction (yellow arrow). This picture also demonstrated margin. right renal mass. International Journal of Case Reports and Images, Vol. 10, 2019. ISSN: 0976-3198
Int J Case Rep Images 2019;10:101003Z01TC2019. Chuang et al. 3 www.ijcasereportsandimages.com (TLC) 75 mm stapler and oversewn with 3–0 PDS. Distal of 2–5% of small bowel metastatic melanoma deposit enterotomy was closed transversely with 3–0 PDS. Small in patients with malignant melanoma of the skin [6]. bowel was then carefully examined again via laparoscopy Renal cell carcinomas constitute 7.1% of cancers that approach. No mesenteric deposits or other deposits of metastasize to the small intestine. Other forms of tumor were noted. cancer that commonly metastasize to the small intestine He was closely observed in intensive care unit one include lung, cancers of the head and neck, breast, day post-operation. He recovered uneventfully from the and oesophagus [2]. Intraluminal metastases usually surgery. However, he had a prolonged hospital admission presented with gastrointestinal bleeding due to tumor with urinary tract infection and slow recovery. During invasion to intestinal vessels, and some may present with admission, a CT abdomen/pelvis was repeated 2 weeks obstructive symptoms or perforation [5, 7–9]. post-surgery for ongoing abdominal pain. It was found In our case, the exact primary of intraluminal carcinoma that the metastatic lesion at the right lung base has was unable to be identified. This has complicated the significantly increased in size from 1.5 to 4.2 cm in 1 diagnostic dilemma and delayed potential treatment as month, and there was also enlargement of his right renal immunohistochemistry tests required a long time for mass. results. Post-operative histology of the intraluminal tumor Few potential origins of his small bowel tumor was unfortunately non-specific. The histology testing were proposed. Firstly, his radiological proven primary demonstrated that the tumor is high grade epithelioid right renal cancer metastasized to small bowel, right malignancy of unknown origin. Its morphological adrenal and right chest. Secondly, primary lung cancer features are not typical of a renal cell carcinoma or metastasized to small bowel, right kidney, right adrenal a primary small intestinal adenocarcinoma. Further and right chest. Thirdly, small bowel tumor is the primary immunohistochemistry had been performed. cancer which metastasis to right kidney, right adrenal The neoplastic cells stain with SALL4 (typically positive and right chest. Finally, there were synchronous primary in germ cell tumors), CDX2 (focally positive in carcinoma tumors. of colorectal origin), cytokeratin markers AE1/AE3 and These theories had all been shown by previous case CK8/18, markers for CK7, CK20, S100m, PAX8, GATA3, reports. Numerous case reports had demonstrated renal Hep Par1, TTF1, CD34, chromogranin, synaptophysin, cell carcinoma can metastasize to small bowel causing MUM1, inhibin, desmin, and Melan-A were all negative. intussusceptions [1–3, 5, 7]. All negative immunohistochemistry results have left the Dilege et al. [8] had published a case report of diagnosis of tumor’s origin undetermined. an adult intestine intussusception from epithelioid His case was discussed in colorectal multidisciplinary type mesenchymeal tumor (gastrointestinal stromal team meeting. As there was no definitive diagnosis of tumors, also known as GISTs) as primary tumor of the metastatic renal cell carcinoma, we were unable to offer small intestine. GISTs are a heterogeneous groups of palliative chemotherapy. Given his interval progression mesenchymal tumors which arise from the gastrointestinal of metastatic cancer and poor prognosis, palliative tract. The small intestine is the second most common site approach with possible palliative radiation therapy was of GISTs. Guner et al. [10] also had published a case report offered with agreement of patient’s next of kin. Patient to demonstrate metastasized sarcomatoid carcinoma of was subsequently discharged from hospital back to care the lung to cause intestinal obstruction. However, none facility on day 36 of his admission. of these immunohistochemistry results were consistent with our histology report. Based on the clinical picture, the idea of metastatic DISCUSSION primary renal carcinoma metastasize to small bowel is more favorable. Unfortunately, no formal pathological Intussusception in adults is a rare cause of intestinal diagnose of RCC was able to be performed as patient obstruction. It is often difficult to diagnose as it has diverse and his family declined kidney biopsy when palliation presentations and varied symptoms. Radiological studies treatment was offered. had been found to be useful in preoperative diagnosis, It was recommended that the mainstay of this including abdominal ultrasound, plain abdominal films, condition is surgical resection of lesions to reduce upper gastrointestinal contrast series, barium enema or intussusceptions as it not only gives symptoms relief but CT abdomen/pelvis. Abdominal CT has been considered also potentially improves survival benefit [2, 5]. as the most useful tool to diagnose intussusception with a reported accuracy of 58–100% [4] It is extremely rare that intussusception is caused CONCLUSION by intraluminal tumor. It was noted that small bowel tumors are more common from secondary cancer than Intussusception caused by intraluminal polypoid small primary and they occur equally in jejunum and ileum bowel tumor from metastatic cancer is extremely rare. [5]. Melanomas are the most common form of cancer The exact pathophysiological of intraluminal polypoid to metastasise to the small bowel. There is an incidence tumor from metastatic cancer is uncertain. Surgical International Journal of Case Reports and Images, Vol. 10, 2019. ISSN: 0976-3198
Int J Case Rep Images 2019;10:101003Z01TC2019. Chuang et al. 4 www.ijcasereportsandimages.com intervention of complete excision is recommended by and interpretation of data, Drafting the article, Revising authors not only for palliative symptoms relief but also it critically for important intellectual content, Final potentially survival benefit. approval of the version to be published Havish Srinath – Substantial contributions to conception and design, Acquisition of data, Analysis REFERENCES and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final 1. Hegde RG, Gowda HK, Agrawal RD, Yadav VK, approval of the version to be published Khadse GJ. Renal cell carcinoma presenting as small Hassan Malik – Substantial contributions to conception bowel obstruction secondary to a metastatic ileal and design, Acquisition of data, Analysis and intussusception. J Radiol Case Rep 2014;8(4):25–31. interpretation of data, Drafting the article, Revising 2. Bellio G, Cipolat Mis T, Kaso G, Dattola R, Casagranda B, Bortul M. Small bowel intussusception from renal it critically for important intellectual content, Final cell carcinoma metastasis: A case report and review of approval of the version to be published the literature. J Med Case Rep 2016;10(1):222. Paul Tingey – Substantial contributions to conception and 3. Sasaki M, Murakami Y, Takesue Y, et al. Double design, Acquisition of data, Analysis and interpretation intussusceptions in the small intestine caused by of data, Drafting the article, Revising it critically for metastatic renal cell carcinoma: Report of a case. Surg important intellectual content, Final approval of the Today 2006;36(1):85–8. version to be published 4. Somma F, Faggian A, Serra N, et al. Bowel Raphael Varghese – Substantial contributions to intussusceptions in adults: The role of imaging. conception and design, Acquisition of data, Analysis Radiol Med 2015;120(1):105–17. 5. Vani M, Nambiar A, Geetha K, Kundil B. Metastatic and interpretation of data, Drafting the article, Revising renal cell carcinoma causing small intestinal polyps it critically for important intellectual content, Final with intussusception: A report of two cases. J Clin approval of the version to be published Diagn Res 2017;11(4):ED13–5. 6. Gatsoulis N, Roukounakis N, Kafetzis I, Gasteratos Guarantor of Submission S, Mavrakis G. Small bowel intussusception due The corresponding author is the guarantor of submission. to metastatic malignant melanoma. A case report. Techniques in Coloproctology 2004;8 Suppl 1:s141–3. Source of Support 7. Sridhar SS, Haider MA, Guindi M, Moore MJ. A None. case of small bowel obstruction due to intraluminal metastases from metastatic renal cell cancer. Oncologist 2008;13(2):95–7. Consent Statement 8. Tiwari P, Tiwari A, Vijay M, Kumar S, Kundu AK. Written informed consent was obtained from the patient Upper gastro-intestinal bleeding – Rare presentation for publication of this case report. of renal cell carcinoma. Urol Ann 2010;2(3):127–9. 9. Dilege E, Coșkun H, Kaya C, et al. A rare case of Conflict of Interest adult intestinal intussusception: Epithelioid type Authors declare no conflict of interest. mesenchymal tumor of the small intestine. Turk J Gastroenterol 2008;19(2):121–4. 10. Guner A, Karyagar S, Livaoglu A, Kece C, Kucuktulu Data Availability U. Small bowel intussusception due to metastasized All relevant data are within the paper and its Supporting sarcomatoid carcinoma of the lung: A rare cause Information files. of intestinal obstruction in adults. Case Rep Surg 2012;2012:962683. Copyright © 2019 Tzu-Yi (Arron) Chuang et al. This article is ********* distributed under the terms of Creative Commons Attribution License which permits unrestricted use, Acknowledgements distribution and reproduction in any medium provided We would like to thank emergency, pathology, urology, the original author(s) and original publisher are properly medical oncology and palliative department colleagues’ credited. Please see the copyright policy on the journal contribution in this patient’s care. website for more information. Author Contributions Tzu-Yi (Arron) Chuang – Substantial contributions to conception and design, Acquisition of data, Analysis International Journal of Case Reports and Images, Vol. 10, 2019. ISSN: 0976-3198
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