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Resolving Sphincter of Oddi Incontinence for Primary Duodenal Crohn’S

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Resolving Sphincter of Oddi Incontinence for Primary Duodenal Crohn’S CASE REPORT – OPEN ACCESS International Journal of Surgery Case Reports 4 (2013) 149–152 View metadata, citation and similar papers at core.ac.uk brought to you by CORE Contents lists available at SciVerse ScienceDirect provided by Elsevier - Publisher Connector International Journal of Surgery Case Reports j ournal homepage: www.elsevier.com/locate/ijscr Resolving sphincter of Oddi incontinence for primary duodenal Crohn’s disease with strictureplasty ∗,a a a a a G. Alemanno ,A.Sturiale ,F.Bellucci ,F.Giudici , F. Tonelli Digestive Surgery Unit, Department of Clinical Physiopathology, University of Florence Medical School, Careggi Hospital, Italy a r t i c l e i n f o a b s t r a c t Article history: INTRODUCTION: Crohn’s involvement of duodenum is a rare event and may be associated to proteiform Received 9 November 2012 symptoms and uncommon pathological aspects which make diagnosis and treatment complex. Accepted 16 November 2012 PRESENTATION OF CASE: The peculiar aspect of this case was a suspected duodeno-biliary fistula. The Available online 23 November 2012 patient (female, 22 years old) was affected by duodenal Crohn’s disease. Magnetic resonance imaging showed a dilated common bile duct, whose final part linked to a formation containing fluid, and character- Keywords: ized by filling of the contrast medium in the excretory phase. Abdominal ultrasound showed intra-hepatic Duodenal Crohn’s disease and intra-gallbladder aerobilia. At surgery, the duodenum was mobilized showing an inflammatory stric- Sphincter of Oddi incontinence Strictureplasty ture and a slight dilatation of the common bile duct, with no signs of fistulas. The opened duodenum was anastomized side to side to a transmesocolic loop of the jejunum. After surgery, the general condition of the patient improved. DISCUSSION: Only two cases of fistula between a narrow duodenal bulb and the common bile duct have been described in literature and the Authors were not be able to verify the occurrence of a duodenal biliary fistula at surgery. The association between duodenal Crohn’s disease and Sphincter of Oddi incontinence is a very rare finding with different etiology: chronic intestinal pseudo-obstruction, common bile duct stones, progressive systemic sclerosis. CONCLUSION: The treatment to resolve Sphincter of Oddi incontinence for primary duodenal Crohn’s disease is not clear. Strictureplasty could be the treatment of choice, because, resolving the stricture, the duodenal pressure is likely to decrease and the reflux through the incontinent sphincter can be avoided. © 2012 Surgical Associates Ltd. Published by Elsevier Ltd. Open access under CC BY-NC-ND license. 2 1. Introduction the main radiographical features. Obstruction is the most com- mon complication of duodenal CD, but fistulae arising from the Crohn’s disease (CD) can affect the entire gastrointestinal tract, duodenum are extremely rare. It is commonly believed that the but gastroduodenal involvement is rarely observed. First described duodenal fistulae originate only from other primary localizations 3 in 1937 by Gottlieb and Alpert, duodenal localization occurs in 0.5% of diseased small or large bowel. Submitting patients with duode- 1 to 4% of patients with CD. In nearly all cases epigastric pain or dys- nal CD to X-ray examination, filling of pancreatic or biliary ducts pepsia are the predominant symptoms, which mimic peptic ulcer or may occur. This may be due to either fistula formation or reflux 4 non-ulcer dyspepsia. In more advanced phases of the disease, most through a damaged ampulla of Vater. Consequences of this reflux patients have symptoms due to obstructive lesions, such as epi- can be an obstruction of the duodenal portion of the bile duct and 5,6 gastric distress, anorexia, nausea, vomiting and weight loss. Only a pancreatitis. 1 few have haematemesis or maelena. Every part of the duodenum The most frequent indication for surgery is gastroduodenal can be involved, but the second part is most commonly affected. obstruction, whereas major hemorrage, extensive fistula formation Different types of ulcer, such as aphthous, longitudinal, transverse, or suspicion of malignancy may warrant resection. Dilatation of and deep, may occur. Abnormal folds, stenosis and ulceration are strictures and strictureplasty may be viable treatment options in selected patients with stenosing duodenal CD. 2. Presentation of case Abbreviations: CD, Crohn’s disease; ESR, erythrocyte sedimentation rate; CRP, C-reactive protein; UGI, upper gastro intestinal; MRI, magnetic resonance imaging; CKK, cholecystokinin; SO, Sphincter of Oddi. Female, 22 years old, with duodenal CD, onset at age of 8, with ∗ Corresponding author at: Careggi Hospital, Digestive Surgery Unit, Largo Bram- diffuse abdominal pain and malabsorption syndrome. At the age billa 3, 50134 Florence, Italy. Tel.: +39 055 7947449; fax: +39 055 7947449. of 11, she was hospitalized for persistent abdominal pain associ- E-mail address: [email protected] (G. Alemanno). a ated with dyspepsia, constipation, fatigue and 7-kg weight loss in Authors’ contribution: All authors contributed equally to this work: G.A., F.B. 3 months, and a BMI of 15.9. Blood tests showed an increased ERS, and A.S. collected the data, G.A. and A.S. analyzed data, G.A., A.S., F.G, F.B and F.T. wrote the manuscript, G.A. and F.T. supervised all the manuscript. CRP 15 mg/dL, Hb 12.1 g/dL and normal WBC, glucose, coagulation, 2210-2612 © 2012 Surgical Associates Ltd. Published by Elsevier Ltd. Open access under CC BY-NC-ND license. http://dx.doi.org/10.1016/j.ijscr.2012.11.016 CASE REPORT – OPEN ACCESS 150 G. Alemanno et al. / International Journal of Surgery Case Reports 4 (2013) 149–152 Fig. 1. Abdominal ultrasound: intragallbladder and intrahepatic aerobilia (A and B). serum protein, amylase. Upper gastro intestinal (UGI) endoscopy opacification of the main biliary duct and of some intrahepatic was performed with difficulty to overcome the superior duode- branches (Fig. 2). Considering duodenal stricture, the failure of nal knee, showing a “granulomatous and easily bleeding duodenal previous medical therapy and the suspicious of a biliary-digestive mucosa, with multiple ulcers covered with fibrin.” The biopsy con- fistula, we decided to perform surgery. firmed the suspected diagnosis of duodenal CD. Colonoscopy was Surgery was carried out in July 2011. The duodenum was com- negative. From the age of 11–16, the patient was treated with aza- pletely mobilized showing an inflammatory stricture, starting near thioprine and corticosteroid therapy, and reported a relative good the pylorus until the III portion, many gallbladder adhesions, and condition with some exacerbation episodes. At the age of 19, the a slight dilatation of the common bile duct, without signs of fistu- patient began Thalidomide therapy because of poor response to las. The duodenum was opened longitudinally for a length of about previous drugs. In 2009, due to exacerbation of symptoms, feed- 8 cm confirming the widespread inflammatory involvement of the ing difficulties and weight loss, an MRI was performed and showed duodenal mucosa and wall thickening, up to 8 mm. The papilla was a dilated common bile duct (9 mm) with its final part communi- found within the inflammatory context and was probed for some cating with a formation (about 1 cm in diameter) containing fluid, centimeters, penetrating easily both into the Wirsung and the bil- and characterized by filling of the contrast medium in the excre- iary duct, from which clear mucus and bile flowed out (Fig. 3). The tory phase. This situation was attributable to a biliary inflammatory opened duodenum was anastomized side to side to a transmeso- stricture. A new UGI endoscopy was performed: the duodenal bulb colic loop of the jejunum, taken about 20 cm from Treitz. and the second duodenal portion appeared deformed and stenotic About 25 cm from the Roux anastomosis there were three steno- with hyperemic and ulcerated mucosa. A pneumatic dilatation sis, that were treated with 12 cm Finney strictureplasty (Fig. 4). was performed. At the age of 21, the patient suspended treat- Below this, there were other 2 short jejunal strictures which were ment with Thalidomide and began Infliximab with poor results. treated with Heineke-Mikulicz strictureplasties. After surgery, the In 2011 another exacerbation of symptoms occurred, namely: diff- general condition of the patient improved. Post-operative therapy used abdominal pain, dyspepsia, weight loss, diarrhea, and fever. was performed with 5-ASA. Twelve months after the operation Abdominal ultrasound showed intrahepatic and intragallbladder there was a complete remission of symptoms with no difficulties aerobilia (Fig. 1). Gastro-duodenal follow-through contrast X-ray in intaking food nor signs of occlusion. At abdominal ultrasound no documented a rigid, retracted and shortened duodenum, and slight signs of intrahepatic and intragallbladder aerobilia were found. Fig. 2. Opacification of main biliary duct and intrahepatic branches (A) and duodenal strictures (B) are visible at gastro-duodenal follow-through contrast X-ray. CASE REPORT – OPEN ACCESS G. Alemanno et al. / International Journal of Surgery Case Reports 4 (2013) 149–152 151 10,11 12–14 duodenum, gallbladder and bile ducts. Sphincter of Oddi motility is controlled by cholecystokinin (CCK), which is released into the bloodstream by the duodenal mucosa, as a response to duodenal luminal acid and nutrients. Oddi’s Sphincter plays an important role in decreasing the basal pressure and the amplitude of phasic waves. The cause of SO incontinence is uncertain, and one potential mechanism may be a defect of the neural connec- tions that coordinate the interaction between the duodenum, the biliary tract and the SO. There is evidence that SO dysfunction may be a part of a generalized motor disorder of the gastrointestinal 15 tract, such as small intestinal dysmotility. In one study, patients with irritable bowel syndrome and SO dysfunction demonstrated paradoxical responses to CCK more often than patients with SO dys- 16 function alone.
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