Harnanan, Dave

Maharaj, Ravi

Mohammed, Wayne

Raghunanan, Barry

Rambally, Rakesh

Sookdeo, Vandana Devika

Warner, Wayne A

Journal of Surgical Case Reports

English

Ravi Maharaj

Barry Raghunanan

Wayne Mohammed

Rakesh Rambally

Vandana Devika Sookdeo

Dave Harnanan

Wayne A Warner

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A rare case of massive lower gastrointestinal bleeding from a ruptured splenic artery aneurysm

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Washington University School of MedicineDigital Commons@BeckerOpen Access Publications2018A rare case of massive lower gastrointestinalbleeding from a ruptured splenic artery aneurysmRavi MaharajUniversity of the West IndiesBarry RaghunananUniversity of the West IndiesWayne MohammedUniversity of the West IndiesRakesh RamballySangre Grande HospitalVandana Devika SookdeoUniversity of the West IndiesSee next page for additional authorsFollow this and additional works at: https://digitalcommons.wustl.edu/open_access_pubsThis Open Access Publication is brought to you for free and open access by Digital Commons@Becker. It has been accepted for inclusion in OpenAccess Publications by an authorized administrator of Digital Commons@Becker. For more information, please contact engeszer@wustl.edu.Recommended CitationMaharaj, Ravi; Raghunanan, Barry; Mohammed, Wayne; Rambally, Rakesh; Sookdeo, Vandana Devika; Harnanan, Dave; and Warner,Wayne A., ,"A rare case of massive lower gastrointestinal bleeding from a ruptured splenic artery aneurysm." Journal of Surgical CaseReports.2018,2. 1-4. (2018).https://digitalcommons.wustl.edu/open_access_pubs/6536AuthorsRavi Maharaj, Barry Raghunanan, Wayne Mohammed, Rakesh Rambally, Vandana Devika Sookdeo, DaveHarnanan, and Wayne A. WarnerThis open access publication is available at Digital Commons@Becker: https://digitalcommons.wustl.edu/open_access_pubs/6536Journal of Surgical Case Reports, 2018;2, 1–4doi: 10.1093/jscr/rjy003Case ReportC A S E R E PORTA rare case of massive lower gastrointestinal bleedingfrom a ruptured splenic artery aneurysmRavi Maharaj1, Barry Raghunanan1,*, Wayne Mohammed2,Rakesh Rambally3, Vandana Devika Sookdeo1, Dave Harnanan1,and Wayne A. Warner4,5,61Department of Clinical Surgical Sciences, University of the West Indies, Eric Williams Medical SciencesComplex, Champ Fleurs, Trinidad and Tobago, 2Department of Paraclinical Science, University of the WestIndies, Eric Williams Medical Sciences Complex, Champ Fleurs, Trinidad and Tobago, 3Sangre Grande Hospital,Sangre Grande, Trinidad and Tobago, 4Division of Oncology, Siteman Cancer Center St. Louis, MO 63110, USA,5Department of Cell Biology and Physiology, Washington University School of Medicine, St. Louis, MO 63110, USA,and 6MedSeq HealthCare Solutions/A Fighting Chance Foundation, Trincity, Trinidad and Tobago*Correspondence address. Department of Clinical Surgical Sciences, University of the West Indies, Eric Williams Medical Sciences Complex, Uriah ButlerHighway, Champ Fleurs, Trinidad and Tobago. Tel: (868) 382-2712; E-mail: bray_raghunanan@yahoo.comAbstractSplenic artery aneurysms (SAAs) are an extremely rare cause of asymptomatic massive lower gastrointestinal bleeding withless than a handful of patients surviving such a presentation. A 24-year-old female presented in shock after multiple epi-sodes of massive rectal bleeding. Imaging revealed a heterogeneous mass arising from the tail of the pancreas eroding intothe splenic ﬂexure of the colon. Further episodes of bleeding led to an exploratory laparotomy. Intraoperatively, a suspectedneoplastic process arising from the tail of the pancreas with contiguous involvement of the splenic ﬂexure of the colon andthe greater curvature of the stomach was noted. Distal pancreaticosplenectomy, gastric wedge resection with segmental col-ectomy and primary anastomosis were performed. Histology revealed a SAA with rupture into the colon. This case reportshows that en-bloc resection of a ruptured SAA can be performed with success in the emergency setting.INTRODUCTIONSplenic artery aneurysms (SAAs) are the third most commonintra-abdominal aneurysm after those of the aortic and iliacartery [1]. The female to male ratio is 4:1 with rupture occurringmore frequently in men [2]. They are mostly asymptomatic,with occasional symptoms such as left upper quadrant/epigas-tric pain, hypovolemic shock and bleeding from the gastro-intestinal tract arising in about 5% of patients [3].We describe the case of a 24-year-old female of African ances-try who presented with a SAA. This case highlights the risk factors,radiologic and intraoperative ﬁndings, histologic diagnosis and sur-veillance of a SAA patient in the setting of a developing country.CASE REPORTA 24-year-old Trinidad and Tobago female national of Africanancestry, without any known co-morbidities, presented to theReceived: September 25, 2017. Accepted: January 7, 2018Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018.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.com1Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy003/4837045by Washington University in St. Louis useron 13 February 2018emergency department with a 1-day history of massive rectalbleeding accompanied by dark red clots. En route to the hos-pital, she suffered a catastrophic bleed and upon arrival was inshock with complaints of mild abdominal pain, syncope andsevere lethargy.On examination, the patient was somewhat alert andoriented. Presenting vitals reﬂected class III hypotensive shockwhich responded adequately to aggressive ﬂuid resuscitation.Hourly urine output was adequate and the nasogastric tubeaspirate and free drainage were blood free. Her abdomen wassoft with mild epigastric tenderness but no signs of perotinism.An ill-deﬁned, supraumbilical fullness was noted and dark redblood and stool appeared on digital rectal exam. Biochemicalassessments, electrocardiogram, abdominal and chest X-rayswere all unremarkable. A pregnancy test was negative.A triphasic, contrast enhanced computed tomography (CT)scan revealed a 6 cm × 5.2 cm well deﬁned, heterogeneous,hypodense mass arising from the pancreatic tail, with anextremely enhancing component (200 Hounsﬁeld units) on itslateral side, a feeder vessel arising from the splenic artery anda small splenic infarct (Fig. 1). Fascial margins of the mass werepoorly deﬁned from the splenic hilum and splenic ﬂexure ofthe large intestine (Fig. 2). CT imaging was supportive of a dif-ferential diagnosis of a solid pseudopapillary tumor of the tailof the pancreas with invasion of the colonic wall and compres-sion of the stomach.Once stable, the patient was counseled about possible inter-ventions. Eight hours post-admission, after transfusion with twounits of packed red blood cells, her vitals remained stable,adequate hourly urine output resumed and the bleeding abated.However an hour later, she had an enormous episode of rectalbleeding losing ~1.5 L of blood. She became acutely unresponsiveand unstable and was rushed to the operating room while beingresuscitated with crystalloids, colloids and uncross-matchedblood.An exploratory laparotomy revealed the presence of bloodin the distal ileum and large bowel. Further dissection uncov-ered a mass adherent to the posterior stomach that arose frommiddle and distal pancreas and involved the distal transversecolon. With a differential diagnosis of cancer and with consultby the operating team, the decision for deﬁnitive surgery wasmade. An en-bloc resection of the mass, distal pancreatectomyand splenectomy, resection of the cuff of the distal transversecolon and a wedge resection of stomach were performed.Histopathology conﬁrmed the specimen to a SAA, with a1 cm rupture through the colonic wall (Fig. 3A and B). On micro-scopic examination, the wall of the colon showed attenuationand loss of the muscle layers with bleeding into the lumen ofthe colon (Figs 4 and 5).The patient remained stable throughout the surgery and herpost-operative period. The patient was asymptomatic 2 yearspost-surgery.An exhaustive search of the PubMed/Medline and EMBASEmedical databases from inception (1946–present) was performed.The search strategy was ‘(developing countries, Latin America,Trinidad and Tobago and Caribbean) AND (‘splenic artery’ OR‘spleen artery aneurysm’) AND ‘ruptured aneurysm’’. No cita-tions were found after limiting ﬁnal retrieval to English language,humans and case reports. To the best of our knowledge, this isthe ﬁrst case report of a SAA in the Caribbean and Latin America.DISCUSSIONThe prevalence of SAA is low [4], and pelvic rectal bleed sec-ondary to SAA is extremely rare (<10 reported cases) [5]. Whilepatients can present with SAA at any age it is much more com-monly observed in patients 50–60 years. Our patient presenta-tion at 24 years is extremely rare. While the etiology is oftenunknown it has been associated with a myriad of systemic dis-eases including hypertension, cirrhosis, arteritis and collagenvascular disease. Interestingly, our patient did not have a his-tory of these conditions.CT angiography diagnostic regimen is a powerful minimallyinvasive technique for the evaluation of blood vessel diseaseand related conditions, such as aneurysms or blockages.Catheter directed angiography as a diagnostic tool for massivelower gastrointestinal bleeding (GI) bleeds is also widely usedin conjunction with angiographic coiling and a stent for immedi-ate therapeutic relief. In most developing countries, endovascularmanagement approaches, including availability of endovascularequipment and blood products, for lower GI bleeding are notreadily available. In such low-resource environments, emer-gency patients with massive GI bleeds are managed expect-antly. If bleeding persists, open surgical management is theonly option.Treatment for SAA should be undertaken when there is anincreased risk of rupture, perforation diameter greater than2 cm, and in symptomatic, transplant patients and in youngerFigure 1: CT image showing the ‘ying yang’ sign of the SAA. White arrow showsthe origin from the splenic artery. Black arrow shows a splenic infarct.Figure 2: CT shows the point of communication (free rupture) between theaneurysm and splenic ﬂexure of the colon.2 | R. Maharaj et al.Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy003/4837045by Washington University in St. Louis useron 13 February 2018or pregnant women [6]. Those presenting with lower GI representa distinct entity and management commences with adequateresuscitation and appropriate investigations. Endovascular coilembolization is an attractive ﬁrst line treatment [5, 7]. However,it is not the preferred clinical approach for the managementof giant aneurysms due to suboptimal occlusion of the aneur-ysm, inﬂammatory process and increased risk of embolization.Aneurysmectomy, pancreatectomy and splenectomy approacheshave been reported [8].In conclusion, SAAs are an extremely rare cause of lower GIbleeding. Contrast CT for ambiguous cases of lower gastrointes-tinal bleeding may be warranted, especially in low-resourcecenters without endoscopy or selective angiography services, toavoid the high-mortality rate associated with undiagnosedlesions.ACKNOWLEDGEMENTSThe authors acknowledge the assistance of Kim Lipsey, MedicalLibrarian, Becker Medical Library, Washington UniversitySchool of Medicine-St. Louis, MO, USA. The contents of thismanuscript are solely the responsibility of the authors and donot necessarily represent the views of the afﬁliating institutions.CONFLICT OF INTEREST STATEMENTNone declared.FUNDINGW.A.W. was supported by Washington University School ofMedicine, GSAS/CGFP Fund 94028C.REFERENCES1. Al-Habbal Y, Christophi C, Muralidharan V. Aneurysms ofthe splenic artery—a review. Surgeon 2010;8:223–31. doi:10.1016/j.surge.2009.11.011.2. Ierardi AM. Endovascular retreatment of a splenic arteryaneurysm reﬁlled by collateral branches of the left gastricFigure 3: En-bloc specimen. (A) Hemorrhage into the colonic lumen from the aneurysmal communication. (B) The specimen showing free rupture of the aneurysminto the colonic lumen.Figure 4: Wall of the colon showing attenuation and loss of the muscle layers.The wall of the aneurysm is indicated by the arrowhead (H&E 4×).Figure 5: Point of rupture of the aneurysm into the lumen of the colon. Thelumen of the aneurysm contains blood clot (arrowhead) (H&E 4×).A rare case of massive lower gastrointestinal bleeding from a ruptured splenic artery aneurysm | 3Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy003/4837045by Washington University in St. Louis useron 13 February 2018artery: a case report. J Med Case Reports 2014;8:436 doi:10.1186/1752-1947-8-436.3. Jones E, Finney GG. Splenic artery aneurysms a reappraisal. ArchSurg 1968;97:640–7. doi:10.1001/archsurg.1968.01340040136027.4. Agrawal GA, Johnson PT, Fishman EK. Splenic artery aneur-ysms and pseudoaneurysms: clinical distinctions and CTappearances. AJR Am J Roentgenol 2007;188:992–9. doi:10.2214/ajr.06.0794.5. Ek ET, Moulton CA, Mackay S. Catastrophic rectal bleeding froma ruptured splenic artery aneurysm. ANZ J Surg 2003;73:361–4.6. Abbas MA. Splenic artery aneurysms: two decades experi-ence at Mayo clinic. Ann Vasc Surg 2002;16:442–9. doi:10.1007/s10016-001-0207-4.7. Zubaidi A. Rupture of multiple splenic artery aneurysms: acommon presentation of a rare disease with a review of lit-erature. Saudi J Gastroenterol 2009;15:55–8. doi:10.4103/1319-3767.45061.8. Quandalle P, Rousseau B, L’Hermine C, Ribet M. [Massive rec-tal hemorrhage by colonic ﬁstulization of a pancreatitislesion]. J Chir (Paris) 1985;122:665–9.4 | R. Maharaj et al.Downloaded from https://academic.oup.com/jscr/article-abstract/2018/2/rjy003/4837045by Washington University in St. Louis useron 13 February 2018

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A rare case of massive lower gastrointestinal bleeding from a ruptured splenic artery aneurysm

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