Báo cáo khoa học: "Adenocarcinoma of the third portion of the duodenum in a man with CREST syndrome"
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- World Journal of Surgical Oncology BioMed Central Open Access Case report Adenocarcinoma of the third portion of the duodenum in a man with CREST syndrome Georgios Anastasopoulos, Athanasios Marinis*, Christos Konstantinidis, Theodosios Theodosopoulos, Georgios Fragulidis and Ioannis Vassiliou Address: Second Department of Surgery, Areteion University Hospital, 76 Vassilisis Sofia's Ave, 11528, Athens, Greece Email: Georgios Anastasopoulos - drmarinis@gmail.com; Athanasios Marinis* - sakisdoc@yahoo.com; Christos Konstantinidis - drchriskons@yahoo.gr; Theodosios Theodosopoulos - theodosios@vodafone.net.gr; Georgios Fragulidis - foreo@otenet.gr; Ioannis Vassiliou - ianvass@otenet.gr * Corresponding author Published: 1 October 2008 Received: 3 June 2008 Accepted: 1 October 2008 World Journal of Surgical Oncology 2008, 6:106 doi:10.1186/1477-7819-6-106 This article is available from: http://www.wjso.com/content/6/1/106 © 2008 Anastasopoulos et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Abstract Background: CREST (Calcinosis, Raynaud's phenomenon, Esophageal dysmotility, Sclerodactyly and Telangiectasias) syndrome has been rarely associated with other malignancies (lung, esophagus).This is the first report of a primary adenocarcinoma of the third portion of the duodenum in a patient with CREST syndrome. Case presentation: A 54-year-old male patient with CREST syndrome presented with colicky postprandial pain of the upper abdomen, diminished food uptake and a 6-Kg-body weight loss during the previous 2 months. An ulcerative lesion in the third portion of the duodenum was revealed during duodenoscopy, with a diagnosis of adenocarcinoma on biopsy specimen histology. The patient underwent a partial pancreatoduodenectomy. No adjuvant therapy was instituted and follow-up is negative for local recurrence or metastases 21 months postoperatively. Conclusion: CREST syndrome has been associated with colon cancer, gastric polyps, familial adenomatous polyposis (FAP) syndrome and Crohn's disease; however, this is the first report of a primary adenocarcinoma of the duodenum in a patient with CREST syndrome. However, any etiologic relationship remains to be further investigated. Background Case presentation CREST syndrome (Calcinosis, Raynaud's phenomenon, A 54-year-old man was referred to our clinic because of Esophageal dysmotility, Sclerodactyly and Telangiecta- colicky postprandial pain of the upper abdomen during sias) has been rarely associated with other malignancies the last month, with aggravation 5 days before admission. (lung, esophagus), whereas duodenal adenocarcinoma The patient had diminished the amount of food uptake has never been reported to be associated with this subtype and had also a 6-Kg-body weight loss during the previous of systemic sclerosis [1-3]. This is the first report of a pri- 2 months. His past medical history included CREST syn- mary adenocarcinoma of the third portion of the duode- drome under cortisone therapy during the last year and num in an adult male patient with CREST syndrome. primary repair of the left ureter and the small intestine Page 1 of 4 (page number not for citation purposes)
- World Journal of Surgical Oncology 2008, 6:106 http://www.wjso.com/content/6/1/106 due to traumatic perforation in a car accident 17 years ago. Physical examination revealed a pale, malnourished patient and an unremarkable abdomen without tender- ness or distension. Besides an anemia, the other blood investigations, as well as tumor markers, were within nor- mal limits. Plain abdominal radiographs showed no path- ological findings, while contrast-enhanced series demonstrated a stricture in the transition area from the second to the third portion of the duodenum (Figure 1). Abdominal computed tomography showed a mass in the duodenum, without evidence of metastases (Figure 2). Thorax computed tomography was negative for metas- tases as well. Gastroduodenoscopy revealed an ulcerated lesion between the second and third portion of the duo- denum and the biopsies showed an adenocarcinoma. Colonoscopy was normal. Figure 2 (arrows) in the duodenum Computed tomography of the abdomen depicting a mass Laparotomy, through a bilateral subcostal incision, Computed tomography of the abdomen depicting a revealed a large tumor locally invading the third portion mass (arrows) in the duodenum. of the duodenum and possibly the pancreas. Therefore, a partial pancreatoduodenectomy (Whipple's procedure) was carried out. Histological examination revealed a low- grade duodenal adenocarcinoma of maximal diameter 4 cm, which infiltrated the duodenal wall and the fibro-adi- pose tissue of the mesentery. Metastasis was found in one out of ten lymph nodes examined, while the surgical mar- gins were negative for microscopic disease. The patient's postoperative course was complicated with a peripancreatic-remnant collection and a lower respiratory infection, which were treated conservatively. No additional therapy was instituted and 21 months after the operation the patient is alive, without any evidence of local recurrence or metastases. Discussion Primary duodenal adenocarcinoma represents a relatively rare clinical entity. Duodenum constitutes the most pre- dominant site (55%) of occurrence of adenocarcinoma throughout the small intestine, followed by the jejunum (18%) and ileum (13%) [4]. The distribution pattern of adenocarcinoma within the duodenum is reported to par- allel the length of each portion. The first portion is affected in 15%, the second portion in 40% and the third and fourth portion in 45% of the cases [5]. Most of these neoplasms are asymptomatic until they become large in size. Partial duodenal obstruction, with associated symptoms of crumpy postprandial pain of the Figure 1 num transition of the series to the third portion of the in the Barium upper GIsecondshowing a stricture (arrow) duode- upper abdomen, nausea and vomiting, is the most com- Barium upper GI series showing a stricture (arrow) mon mode of presentation. Hemorrhage, usually indo- in the transition of the second to the third portion of lent, is the second most common mode of presentation. the duodenum. Due to their location these tumors cause non-specific and vague symptoms leading to a delay of diagnosis [6]. Peri- Page 2 of 4 (page number not for citation purposes)
- World Journal of Surgical Oncology 2008, 6:106 http://www.wjso.com/content/6/1/106 ampullary tumors may cause obstructive jaundice or pan- sclerodactyly and telangiectasias and is associated with a creatitis. better prognosis [14]. Barium upper GI series and endoscopy of the upper gas- The association of this syndrome with malignancies is trointestinal (GI) tract establish the diagnosis with a sen- extremely rare. A case of CREST syndrome and adenocar- sitivity of 81,9% and 88%, respectively [7], while cinoma of the lung has been reported [1]. It is also sug- colonoscopy and thoraco-abdominal computed tomogra- gested that patients with scleroderma and Barrett's phy are carried out to exclude any synchronous or meta- metaplasia have an increased risk of complications, such static cancer. as strictures or adenocarcinoma [2]. Another case of pro- gressive systemic sclerosis (CREST syndrome), sarcoidosis The only possibility of cure is obtained by a radical resec- and esophageal adenocarcinoma in a 50-year-old Japa- tion. The percentage of resectability with a curative intent nese female has been reported [3]. Finally, we report the is reported to range from 50% to 73% [7]. For the tumors first case of a male patient with CREST syndrome and duo- located in the first and the second portion of the duode- denal adenocarcinoma. num, a Whipple's procedure is considered necessary for the radical resection of the tumor and its lymphatic drain- Conclusion age. For tumors of the third and fourth portion, a segmen- A relationship between duodenal adenocarcinoma and tal resection of the duodenum with the appropriate limited cutaneous scleroderma has not yet been described lymphadenectomy is performed with a curative intent [8]. and in our case is considered a matter of coincidence. Chemotherapy and radiation are considered to have only Whether underlying mechanisms exist (immunologic, a small contribution to the overall survival or the disease- genetic, etc) remains to be elucidated with further free survival of these patients [9]. research. Primary duodenal adenocarcinoma is an aggressive tumor Competing interests with an overall 5-year survival rate of about 25% [7], The authors declare that they have no competing interests. which can be significantly improved up to 54% after cur- ative resection [8]. The most important prognostic factors Authors' contributions are the stage of the disease and the location of the tumor. GA, IV and CK carried out the surgical procedure and con- Tumors with negative margins in the surgical specimen, tributed to the design of the study; GA and AM gathered which are located in the first and second portions of the the data, drafted the manuscript and critically revised it; duodenum, are considered to have a better prognosis. IV, TT and GF revised and finally approved the manuscript Other series report that histologic grade, depth of inva- for been published. All authors read and approved the sion, tumor size and metastases to regional lymph nodes final manuscript. influence the survival rate [7-10]. Consent Primary duodenal adenocarcinoma has been associated Written informed consent was obtained from the patient with colon cancer, gastric polyps, especially villous and for publication of this case report and any accompanying tubulovillous adenomas, familial adenomatous polyposis images. A copy of the written consent is available for (FAP) and Crohn's disease. It has been stated that primary review by the Editor-in-Chief of this journal. duodenal adenocarcinoma is one of the main causes of death in patients with FAP [11]. A case of an early duode- References nal adenocarcinoma from a Brunner's gland has been 1. Salvant EE, Carter JM, Armstrong EM, Polk OD Jr, Austin KI: CREST syndrome: a variant of progressive systemic sclerosis, associ- reported [12]. Another case of primary duodenal adeno- ated with interstitial pulmonary fibrosis and malignancy. carcinoma in a patient with neurofibromatosis type 1 has South Med J 1988, 81:1185-1187. 2. Katzka DA, Reynolds JC, Saul SH, Plotkin A, Lang CA, Ouyang A, also been reported [13]. Jimenez S, Cohen S: Barrett's metaplasia and adenocarcinoma of the esophagus in scleroderma. Am J Med 1987, 82:46-52. Limited cutaneous scleroderma or CREST syndrome con- 3. Maenawa Y, Nogami R: A case of progressive systemic sclerosis associated with sarcoidosis and esophageal adenocarcinoma. sists one of the two main subsets of systemic sclerosis, J Dermatol 1993, 20:45-48. which is a multisystemic disorder of unclear pathogenesis, 4. Howe JR, Kamel LH, Menck HR, Scott-Conor C: Adenocarcinoma of the small bowel. Review of the National Cancer Data characterized by inflammatory, vascular and fibrotic Base, 1985–1995. Cancer 1999, 86:2593-2706. changes of the skin and various internal organ systems (GI 5. Coit DG: Cancer of the small intestine. In Cancer: Principles and tract, lungs, heart and kidneys) and involves immuno- Practice of Oncology Edited by: De Vita Jr VT, Hellman S Rosenberg SA. Philadelphia: Lipincott, Williams & Wilkins; 2001:1204-1206. logic mechanisms leading to vascular endothelial damage 6. McCarron EC, Gibbs J, Fakih M, Nava HR, Rajput A: Duodenal ade- and fibroblast activation. CREST syndrome consists of cal- nocarcinoma: vague complaints, delayed diagnosis and poor cinosis, Raynaud's phenomenon, esophageal dysmotility, outcome. Proceedings in the ASCO (Gastrointestinal Cancers Sympo- sium) 2004, 171A:. Page 3 of 4 (page number not for citation purposes)
- World Journal of Surgical Oncology 2008, 6:106 http://www.wjso.com/content/6/1/106 7. Sohn TA, Lillemoe KD, Cameron JL, Pitt HA, Kaufman HS, Hruban RH, Yeo CJ: Adenocarcinoma of the duodenum: factors influ- encing long-term survival. J Gastrointest Surg 1998, 2:79-87. 8. Bakaeen F, Murr MM, Sarr MG, Thompson GB, Farnell MB, Nagorney DM, Farley DR, van Heerden JA, Wiersema LM, Schleck CD, Dono- hue JH: What prognostic factors are important in duodenal adenocarcinoma? Arch Surg 2000, 135:635-642. 9. Tocchi A, Mazzoni G, Puma F, Mizzini M, Cassini D, Bettelli E, Tagliacozzo S: Adenocarcinoma of the third and fourth por- tions of the duodenum: results of surgical treatment. Arch Surg 2003, 138:80-85. 10. Ryder NM, Ko CY, Hines OS, Gloor B, Reber HA: Primary duode- nal adenocarcinoma: a 40-year experience. Arch Surg 2000, 135:1070-1075. 11. Groves CJ, Saunders BP, Spigelman AD, Phillips : Duodenal cancer in patients with familial adenomatous polyposis (FAP): results of a 10-year prospective study. Gut 2002, 50:636-641. 12. Akino K, Kondo Y, Ueno A, Yamazaki K, Hosokawa M, Shimozi H, Adachi T, Honda S, Ichiyanagi S, Akahonai Y, Fujisawa Y, Takahashi H, Arimura Y, Endo T, Imai K: Carcinoma of duodenum arising from Brunner's gland. J Gastroenterol 2002, 37:293-296. 13. Joo YE, Kim HS, Choi SK, Rew JS, Park CS, Kim SJ: Primary duode- nal adenocarcinoma associated with neurofibromatosis type 1. J Gastroenterol 2002, 37:215-219. 14. Fauci AS, Braunwald E, Isselbacher KJ, Wilson JD, Martin JB, Kasper DL, Hauser SL, Longo DL: Systemic Sclerosis (Scleroderma). In Harrison's Principles of Internal Medicine. Companion Handbook McGraw-Hill International Editions; 1998:880-882. Publish with Bio Med Central and every scientist can read your work free of charge "BioMed Central will be the most significant development for disseminating the results of biomedical researc h in our lifetime." Sir Paul Nurse, Cancer Research UK Your research papers will be: available free of charge to the entire biomedical community peer reviewed and published immediately upon acceptance cited in PubMed and archived on PubMed Central yours — you keep the copyright BioMedcentral Submit your manuscript here: http://www.biomedcentral.com/info/publishing_adv.asp Page 4 of 4 (page number not for citation purposes)
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