A Hokama

A Huycke

A Sadatomo

B Abboud

C Venugopal

D Pappas

DA Iannitti

H Al-Jahdali

H Kinoshita

HM Jennifer

J Delamarre

JW Lim

K Asakura

M Kirsch

M Ujihara

PR Liebman

RS Faberman

S Alqahtani

S Rao

SS Thethy

T Brandon

T Pinto Pais

WJ Salyers

Masato Yamadera

Yoshiki Kajiwara

Eiji Shinto

Ryota Hokari

Hideyuki Shimazaki

Junji Yamamoto

Kazuo Hase

Hideki Ueno

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CASE REPORT Open AccessSmall intestinal Crohn’s disease withhepatic portal venous gas: a case reportMasato Yamadera1*, Yoshiki Kajiwara1, Eiji Shinto1, Ryota Hokari2, Hideyuki Shimazaki3, Junji Yamamoto1,Kazuo Hase1 and Hideki Ueno1AbstractAn 80-year-old man presented in another hospital with acute abdominal pain; computed tomography indicatedhepatic portal venous gas (HPVG) and small intestinal thickening. He was then transferred to our hospital, where wediagnosed idiopathic inflammation and stenosis of the ileum. Because the patient’s abdominal symptoms were mildand his general condition was good, we chose to administer conservative therapy. His condition improved and wedischarged him from our hospital. However, he was hospitalized again 9 days later because his abdominal pain hadrecurred and was worse. We performed a laparoscopic partial resection of the ileum 3 weeks after the patients’ initialpresentation. Macroscopically, longitudinal ulcers were observed near the stenosis of the ileum; the segment of thesmall intestine that contained the ulcers was removed, and subsequent pathological findings indicated Crohn’s diseaseof the small intestine. The post-operative course was favorable, and the patient was discharged on post-operative day9. Such serendipitous diagnosis of small intestinal Crohn’s disease in an elderly patient with hepatic portal venous gasis rare; to our knowledge, this is the first of such case in which laparoscopic surgery was performed.Keywords: Hepatic portal venous gas, Crohn’s disease, Laparoscopic surgeryBackgroundHepatic portal venous gas (HPVG) can occur alongsidevarious gastrointestinal diseases and is often associatedwith a severe clinical course [1]. Recently, laparoscopicsurgery for Crohn’s disease (CD) is increasing, and it hasbeen shown to be a safe and practical approach inselected patients [2]. However, the report of laparoscopicsurgery for CD with HPVG was not found so far as wesearched. Additionally, the peak age of CD onset occursfrom 15–20 years, and elderly onset is rare [3]. Herein,we describe the case of an 80-year-old man with smallintestinal CD who developed small intestinal obstructionwith HPVG and who underwent laparoscopic surgicaltreatment.Case presentationAn 80-year-old-man presented in another hospital withabdominal pain and nausea; he was transferred to ourhospital for further examination and treatment, afterHPVG was detected by enhanced abdominal computedtomography (CT). With regard to the patient’s medicalhistory, he had previously undergone appendectomy, hadbeen treated for pulmonary tuberculosis in the remote past,and had suffered tuberculous cervical lymphadenitis at theage of 76 years. His familial history was unremarkable. CTrevealed marked HPVG, wall thickening in a part of thesmall intestine, and minor ascites in the pelvic space. Atfirst admission, his body temperature was 38.8 °C, bloodpressure was 138/84 mmHg, and pulse rate was 109 beatsper minute (bpm). Physical examination revealed slightabdominal distention with mild tenderness of the lowerabdomen; however, no signs of peritoneal irritation werefound. As the patient was in good general health, we choseto treat him using conservative therapy. The next day, noHPVG was identified upon CT scanning, and his abdom-inal pain had diminished. He restarted oral feeding at thatpoint, and was discharged on the tenth hospital day. Weplanned to follow him up as an outpatient; however, he washospitalized again 9 days after discharge because his ab-dominal pain had recurred and was worse.At readmission, his body temperature was 37.3 °C, bloodpressure was 90/56 mmHg, and pulse rate was 92 bpm. He* Correspondence: masatoyamadera@yahoo.co.jp1Department of Surgery, National Defense Medical College, Tokorozawa,Saitama 359-8513, JapanFull list of author information is available at the end of the article© 2016 The Author(s). Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made.Yamadera et al. Surgical Case Reports  (2016) 2:66 DOI 10.1186/s40792-016-0193-yshowed slight abdominal distention and rebound tender-ness of the lower abdomen, but no signs of abdominalguarding. Concerning laboratory findings, his white bloodcell count was 16,800/μl, with 91.6 % neutrophils; he had aC-reactive protein level of 1.5 mg/dl. Acid-fast bacterialtesting of gastric juice and sputum was negative, as was theQuantiFERON™ TB-2G test for tuberculosis. Plain radio-graphs of the abdomen showed the note gas in the smallintestine, and colonoscopic examination revealed a linearand a circular ulcer in the ileum approximately 40 cm orallyfrom the Bauchin valve; a stricture prevented further pas-sage of the scope (Fig. 1a, b). Gastrografin examination ofthe small intestine showed constriction of the terminalileum (Fig. 2a, b), while the jejunum was normal. Enhancedabdominal CT at readmission revealed that the HPVG(Fig. 3a, b) had not recurred (Fig. 3c); however, the thicken-ing of the ileum wall, the edematous change, and the minorascites in the pelvic space had not improved (Fig. 3d). Nointraperitoneal air was found.We presumptively diagnosed idiopathic partial ileitis withstenosis; prompted by the unresponsiveness to conservativetherapy, we performed surgery. Upon laparoscopy, wefound minor serous ascites and wall stiffness at the 20-cmsegment of the mid-to-distal ileum. A 25-cm segment ofthe small intestine that included the disease was resectedlaparoscopically (Fig. 4). There were no adhesions through-out the peritoneal space, and the other parts of bowel werenormal. The resected specimen had a thickened small in-testinal wall. Linear ulcers were found approximately 5 cmon either side of the stricture, which was located at themesentery side of small intestine. The dissection marginwas free of ulceration. Pneumatosis cystoides intestinaliswas not found (Fig. 5). Pathological examination revealedFig. 1 Fiber colonoscopy showing a linear ulcer (a) and a circular ulcer (b) in the ileum, 40 cm from the ileum endFig. 2 Enema examination showing the stenosis at the small intestinenear the terminal ileum (a, b; arrow)Yamadera et al. Surgical Case Reports  (2016) 2:66 Page 2 of 6that the submucosal layer alongside the linear ulcer con-tained epithelioid cell granuloma. Furthermore, inflamma-tion spanned the entire depth of the intestinal wall, withfocal infiltration of neutrophils, lymphocytes, and plasmacells; this strongly indicated CD (Fig. 6). After surgery, thepatient had an uneventful recovery and was dischargedon post-operative day 9. He has continued to do verywell following his discharge. Follow-up gastroscopy andcolonoscopy have revealed no CD-like lesions in any otherpart of the gastrointestinal tract, from the esophagus tothe anus.DiscussionHPVG is characterized by linear or dendric radiolucenciesalong the periphery of the liver on CT images. This is incontrast to biliary air, which is central and almost neverextends to the periphery. Gas in the portal venous systemis likely transported to small peripheral branches in theliver as a result of the centrifugal flow of the portal venousblood [4]. With regard to the mechanism of HPVG, it hasbeen suggested that elevated intraluminal pressure causedby constriction of the intestine can allow bowel gas toenter the portal venous circulation through either micro-scopic mucosal perforations or disease invasions deeperthan the submucosal layer [1]. HPVG has been reportedin various abdominal diseases, namely, gastric ulcer, in-flammatory bowel disease, bowel ischemia, acute enteritis,and simple bowel obstruction. Occasionally, the symptomFig. 3 Contrast-enhanced abdominal computed tomography showing hepatic portal venous gas (HPVG) (a) and thickening of the ileum wall(b; arrow). Upon readmission to our hospital, the HPVG disappeared (c) but the ileum wall thickening did not improve (d; arrow)Fig. 4 Operative findings showing inflammatory thickening of thesmall intestine 30 to 50 cm from the terminal ileumFig. 5 Macroscopic appearance of the resected specimen. Theintestine wall was remarkably thickened. Linear ulcer and stenosiswere observedYamadera et al. Surgical Case Reports  (2016) 2:66 Page 3 of 6Fig. 6 Histopathological analysis showing inflammatory cell invasion through all layers of the intestine (a; hematoxylin and eosin stain ×5) andepithelioid granulomas (b; hematoxylin and eosin stain, object lens ×40)Table 1 Reported cases of Crohn’s disease (CD) with HPVGNo. Author Year Age Sex Symptoms Duration betweenonset of CD and HPVGOperation Outcome1 Pappas et al. [12] 1984 36 M Left-sided lower abdominalpain, tenesmus, liquid stoolsAt onset Conservative therapy Alive2 Huycke et al. [13] 1985 22 M Abdominal pain 6 years Ileocolic resection Alive3 Kirsch et al. [14] 1990 26 F Epigastric pain, chills, nausea,vomitingAt onset Conservative therapy Alive4 Venugopal et al. [10] 1990 27 F Nausea, vomiting, fever 5 years Partial resection of theileumAlive5 Delamarre et al. [15] 1991 70 M Abdominal pain, fever 4 years Conservative therapy Alive6 al-Jahdali et al. [16] 1994 40 F Abdominal pain, nausea 20 years Partial resection of theileumNotdescribed7 Brandon et al. [17] 2000 59 F Nausea, vomiting, loose stool,chills vague abdominal painAt onset Right hemicolectomy Notdescribed8 Thethy et al. [18] 2005 58 F Malaise, rigors, bloody diarrhea,vague perianal painNot described Partial colectomy Alive9 Salyers et al. [4] 2007 24 M Abdominal pain, nausea Not described Conservative therapy Alive10 Alqahtani et al. [19] 2007 26 F Right-sided upper quadrantabdominal pain3 years Conservative therapy Alive11 Hokama et al. [20] 2009 44 M Severe abdominal pain, vomiting 28 years Partial resection of smallintestineAlive12 Lim et al. [9] 2011 32 M Lower abdominal pain, nausea,vomiting, fever12 years Ileocecal resection Alive13 Ujihara et al. [21] 2013 54 F Abdominal pain, nausea 36 years Conservative therapy Alive14 Rao et al. [22] 2013 52 F Abdominal pain, vomiting 11 years Elective colonicresectionAlive15 Pinto Pais et al. [23] 2014 43 F Abdominal pain, vomiting, fever At onset Conservative therapy Alive16 Cunningham et al. [24] 2014 27 F Fever, rigors, abdominal pain,vomiting, diarrheaNot described Conservative therapy Alive17 Our case – 80 M Nausea, abdominal pain At onset Partial resection of theileumAliveYamadera et al. Surgical Case Reports  (2016) 2:66 Page 4 of 6is iatrogenic [5–7]. HPVG is a critical sign, with anassociated mortality rate of 25 to 39 % [6–8]. Therefore,the condition has been considered an indication forurgent surgical intervention, especially if the HPVGwas caused by bowel necrosis, abscess, or massivedamage to the mucosa of the alimentary canal [6]. Con-versely, Lim et al. [9] suggested that treatment ofpatients with HPVG should be based on the underlyingdisease, as well as the patient’s clinical condition. More-over, Venugopal et al. [10] reported that HPVG does notnecessarily indicate necrotic bowel in patients with CD;Kinoshita et al. [6] demonstrated that approximately 4 % ofHPVG cases occur alongside CD and that such casesalmost always have a good clinical course. Our case corrob-orates this claim.HPVG is likely to occur in the elderly [6, 11]. In contrast,CD usually begins in young people—only around 5 % ofCD cases have an age of onset of 65 years or older [3]. Forthis reason, in the present case, we suspected intestinaltuberculosis, intestinal ischemia, or post-operative adhesivesmall bowel obstruction rather than inflammatory boweldisease, including CD. However, repeat examinations fortuberculosis were all negative, and some macroscopic find-ings indicated CD in the resected specimen. Ultimately, wediagnosed CD based on histopathological findings in theresected specimen.CD with HPVG has been reported in the literature;we carried out a search of PubMed wherein we cross-referenced the terms “Crohn's disease” and “hepaticportal venous gas.” We found 17 cases including ourown (Table 1) [4, 9, 10, 12–24]. That is, new-onsetsmall intestinal CD in an elderly patient presenting withHPVG is rare; to our knowledge, this case was the firstfor which laparoscopic surgery was performed. It hasbeen suggested that laparoscopic intervention com-bined with treatment is useful in cases of bowel diseasethat lead to HPVG if (1) no peritoneal irritation signsare noted, and (2) the patient’s physical condition isstable; these two criteria describe our case well. More-over, partial resection of the intestine that contains theaffected area is a valid, reliable therapy for HPVG incases where it may lead to more serious disease.ConclusionsWe have reported the first case of small intestinal CDwith HPVG treated laparoscopically.ConsentWritten informed consent was obtained from the patientfor publication of this case report and any accompanyingimages. A copy of the written consent form is available forreview by the Editor-in-Chief of this journal upon request.Abbreviationsbpm, beats per minute; CD, Crohn’s disease; CT, computed tomography;HPVG, hepatic portal venous gasCompeting interestsThe authors declare that they have no competing interests.Authors’ contributionsMY and HU carried out the surgery and post-operative management. MYdrafted the manuscript and YK and HU critically revised the manuscript. RHcarried out the medical diagnosis. HS carried out the pathological diagnosis.ES, JY, KH, and HU participated in the study design and coordination andhelped to draft the manuscript. All authors read and approved the finalmanuscript.AcknowledgementsWe thank all staffs, who were employed at National Defense Medical College,for treating and caring for this patient.Author details1Department of Surgery, National Defense Medical College, Tokorozawa,Saitama 359-8513, Japan. 2Department of Internal Medicine, NationalDefense Medical College, Tokorozawa, Saitama 359-8513, Japan.3Department of Pathology and Laboratory Medicine, National DefenseMedical College, Tokorozawa, Saitama 359-8513, Japan.Received: 8 March 2016 Accepted: 21 June 2016References1. Liebman PR, Patten MT, Manny J, Benfield JR, Hechtman HB.Hepatic—portal venous gas in adults: etiology, pathophysiology and clinicalsignificance. Ann Surg. 1978;187(3):281–7.2. Jennifer HM, Priscilla M, Stefan DH. Minimally invasive surgery forinflammatory bowel disease. Inflamm Bowel Dis. 2015;21(6):1443–58.3. Asakura K, Nishiwaki Y, Inoue N, Hibi T, Watanabe M, Takebayashi T.Prevalence of ulcerative colitis and Crohn’s disease in Japan. J Gastroenterol.2009;44(7):659–65.4. Salyers WJ, Mansour A. Portal venous gas following colonoscopy and smallbowel follow-through in a patient with Crohn’s disease. Endoscopy. 2007;39Suppl 1, e130.5. Abboud B, El Hachem J, Yazbeck T, Doumit C. Hepatic portal venous gas:physiopathology, etiology, prognosis and treatment. World J Gastroenterol.2009;15(29):3585–90.6. Kinoshita H, Shinozaki M, Tanimura H, et al. Clinical features andmanagement of hepatic portal venous gas: four case reports andcumulative review of the literature. Arch Surg. 2001;136(12):1410–4.7. Iannitti DA, Gregg SC, Mayo-Smith WW, Tomolonis RJ, Cioffi WG, Pricolo VE.Portal venous gas detected by computed tomography: is surgeryimperative? Dig Surg. 2003;20(4):306–15.8. Faberman RS, Mayo-Smith WW. Outcome of 17 patients with portal venousgas detected by CT. AJR Am J Roentgenol. 1997;169(6):1535–8.9. Lim JW, Kim KJ, Ye BD, et al. Enterovenous fistulization: a rare complicationof Crohn’s disease. World J Gastroenterol. 2011;17(47):5227–30.10. Venugopal C, Colley DP. Portal venous gas in a patient with Crohn’s disease.J Clin Gastroenterol. 1990;12(5):595–7.11. Sadatomo A, Koinuma K, Kanamaru R, et al. Hepatic portal venous gas afterendoscopy in a patient with anastomotic obstruction. World J GastrointestSurg. 2015;7(2):21–4.12. Pappas D, Romeu J, Tarkin N, Dave PB, Messer J. Portal vein gas in a patientwith Crohn’s colitis. Am J Gastroenterol. 1984;79(9):728–30.13. Huycke A, Moeller DD. Hepatic portal venous gas after colonoscopy ingranulomatous colitis. Am J Gastroenterol. 1985;80(8):637–8.14. Kirsch M, Bozdech J, Gardner DA. Hepatic portal venous gas: an unusualpresentation of Crohn’s disease. Am J Gastroenterol. 1990;85(11):1521–3.15. Delamarre J, Capron JP, Dupas JL, Deschepper B, Jouet-Gondry C, Rudelli A.Spontaneous portal venous gas in a patient with Crohn’s ileocolitis.Gastrointest Radiol. 1991;16(1):38–40.16. Al-Jahdali H, Pon C, Thompson WG, Matzinger FR. Non-fatal portal pyaemiacomplicating Crohn’s disease of the terminal ileum. Gut. 1994;35(4):560–1.Yamadera et al. Surgical Case Reports  (2016) 2:66 Page 5 of 617. Brandon T, Bogard B, ReMine S, Urban L. Early recognition of hepatic portalvein gas on CT with appropriate surgical intervention improves patientsurvival. Curr Surg. 2000;57(5):452–5.18. Thethy SS, Potter MA. An unusual complication of Crohn’s colitis. Gut. 2005;54(6):768. 96.19. Alqahtani S, Coffin CS, Burak K, Chen F, MacGregor J, Beck P. Hepatic portalvenous gas: a report of two cases and a review of the epidemiology,pathogenesis, diagnosis and approach to management. Can JGastroenterol. 2007;21(5):309–13.20. Hokama A, Nagahama M, Kishimoto K, et al. Pneumatosis intestinalis andhepatic portal venous gas in Crohn’s disease. J Crohns Colitis. 2009;3(4):313–4.21. Ujihara M, Ando T, Ishiguro K, et al. Hepatic portal venous gasfollowing colonoscopy in a patient with Crohn’s disease. Nagoya J MedSci. 2013;75(3-4):273–8.22. Rao S, Tkacz JN, Farraye FA. Portal venous gas after colonoscopy in twopatients with Crohn’s disease. Gastrointest Endosc. 2013;77(2):316–8.23. Pinto Pais T, Pinho R, Carvalho J. Hepatic portal venous gas and intestinalpneumatosis as initial presentation of Crohn’s disease: first case report. JCrohns Colitis. 2014;8(10):1329–30.24. Cunningham G, Cameron G, De Cruz P. Hepatic portal venous gas inCrohn’s disease. BMJ Case Rep. 2014; doi: 10.1136/bcr-2014-206244.Submit your manuscript to a journal and beneﬁ t from:7 Convenient online submission7 Rigorous peer review7 Immediate publication on acceptance7 Open access: articles freely available online7 High visibility within the ﬁ eld7 Retaining the copyright to your article    Submit your next manuscript at 7 springeropen.comYamadera et al. Surgical Case Reports  (2016) 2:66 Page 6 of 6

Surgical Case Reports

Small intestinal Crohn’s disease with hepatic portal venous gas: a case report

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Small intestinal Crohn’s disease with hepatic portal venous gas: a case report

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