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 Table of Contents  
CASE REPORT
Year : 2013  |  Volume : 27  |  Issue : 3  |  Page : 225-226

Adenomyoma of ampulla of pancreas secreting CA-19-9: A case report


Department of Surgery, Regional Institute of Medical Sciences (RIMS), Imphal, India

Date of Web Publication19-Feb-2014

Correspondence Address:
Sapam Opendro Singh
Department of Surgery, Regional Institute of Medical Sciences (RIMS), Imphal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-4958.127400

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  Abstract 

A 70-year-old patient presented with episode of colicky upper abdominal pain associated with vomiting and fever since 4 days. While investigating the patient, CECT abdomen showed features of neoplastic lesion in head of pancreas and CA-19-9 level was > 2000 U/ml, and it was a resectable disease. Whipple's procedure was carried out. Histopathological report showed features suggestive of ampullary adenomyoma.
Adenomyoma of ampulla is very rare with only a few cases reported till now, and adenomyoma secreting CA-19-9 has not been reported till now in literature.

Keywords: Ampullary adenomyoma, CA-19-9, Whipple′s procedure


How to cite this article:
Singh SO, Khan T, Singh L S, Babu Singh T Y. Adenomyoma of ampulla of pancreas secreting CA-19-9: A case report. J Med Soc 2013;27:225-6

How to cite this URL:
Singh SO, Khan T, Singh L S, Babu Singh T Y. Adenomyoma of ampulla of pancreas secreting CA-19-9: A case report. J Med Soc [serial online] 2013 [cited 2020 Oct 26];27:225-6. Available from: https://www.jmedsoc.org/text.asp?2013/27/3/225/127400


  Introduction Top


Adenomyoma of the gastrointestinal (GI) tract is a rare benign lesion. It arises from pancreatic metaplasia or a displaced pancreatic anlage during embryogenesis. The term ectopic pancreas is used if the differentiation is into normal pancreas. If the differentiation is into various abnormally arranged pancreatic elements, various terms are used including adenomyoma, myoepithelial hamartoma, adenomyosis, and foregut choristoma. [1] Adenomyoma occurs mainly in the stomach. [2] The small bowel is the second most frequent location, usually in the peri-ampullary area or ileum. [1] The most common clinical presentations of adenomyoma in the small bowel are intussusception and intestinal or biliary obstruction, depending on the location of the lesions. [1] Ampullary adenomyoma is a rare presentation, and this often leads to a clinical misdiagnosis of carcinoma leading to extensive surgery. Pre-operative diagnosis of this condition is extremely difficult. To best our knowledge, ampullary adenomyoma secreting CA-19-9 has not been reported till now. We hereby report a case of ampullary adenomyoma secreting CA-19-9 leading to diagnostic dilemma.


  Case Report Top


A 70-year-old male patient came with acute upper abdominal pain associated with vomiting since 4 days and fever since 3 days. He had similar history of pain abdomen on and off since 4 years. There was no history of malena or jaundice. Patient has undergone cholecystectomy 1 year back. On general physical examination, patient was having mild icterus, and rest of the general physical examination was within normal limits. His abdominal examination revealed tender right hypochondrium.

On investigation, his complete hemogram was within normal limits, except raised ESR of 50. Liver function tests showed mild elevation of bilirubin of 2.2 mg/dl, apart from which rest of the liver function was within normal limits. His kidney function was deranged at time of admission with creatinine of 3.2, which came back to normal limits after 1 week of treatment. Rest of his blood profiles, ECG, and chest X-ray was normal.

USG abdomen revealed post-cholecystectomy status with mildly dilated CBD of 6.5 mm diameter and prominent intra-hepatic biliary radicles. His MRCP showed CBD measuring 20 mm in diameter and gradual distal tapering without shouldering. Pancreas and its main duct were normal. CECT abdomen showed small focal isodense lesion about 1.5 cm diameter in anterior aspect of head of pancreas suggesting neoplastic lesion, and the disease was resectable. His serum CA-19-9 level was > 2000 U/mL, and serum CEA was 1.7 ng/mL.

On the basis of investigations, it was diagnosed as a case of carcinoma head of pancreas. Patient underwent pylorus sacrificing pancreaticoduodenectomy (Whipple's procedure), and feeding jejunostomy was done. Patient recovered well in post-operative period without any complications.

His histopathology report revealed histologically unremarkable pancreatic tissue [Figure 1]. Ampulla of Vater was lined by columnar epithelium and sub-epithelium showing pseudoinvasion by well-formed glands and intervening stroma showing desmoplastic reaction. These findings were suggestive of ampullary adenomyoma. Supra pancreatic and common hepatic lymph nodes showed features of reactive lymphadenitis.
Figure 1: Micrograph picture of adenomyoma of ampulla showing normal pancreatic tissue and benign mucous glands in ampulla

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His serum CA-19-9 levels repeated after one month post-operatively was reduced to 6.0 U/mL.


  Discussion Top


Benign tumors of ampulla of Vater are extremely rare. Only few cases of adenomyoma of ampulla have been reported all over the world. [3],[4],[5] Most cases of adenomyoma of ampulla have been misdiagnosed as malignant tumors based on the clinical and radiological findings and has lead to extensive surgical resection like Whipple's procedure, which has got a high rate of morbidity and mortality. Kayahara et al. had described a case of an adenomyoma of the ampulla of Vater in a 42-year-old female. She was treated successfully by laparatomy and duodenotomy, incorporating cholangio manometry and cholangiography as well as intra-operative frozen section examination of a transduodenal papillectomy specimen led to the diagnosis of adenomyoma of the ampulla. The patient was doing well 38 months postoperatively. [6]

In our case, the CECT findings and raised CA-19-9 levels lead us to diagnose this case as carcinoma head of pancreas and perform such extensive procedure like Whipple's procedure. CA-19-9, being a tumor marker of pancreatic cancer, has not been described previously to be raised in ampullary adenomyoma. And, also in our case, CA-19-9 levels drops down dramatically after removal of tumor, and the patient is doing well till today.


  Conclusion Top


Adenomyoma of ampulla is a very rare disease. Adenomyoma secreting CA-19-9 is still rarer and is not found in the literature. Adenomyoma of ampulla is a diagnostic problem preoperatively, particularly when CA 19-9 is raised in the blood. If we could have diagnosed adenomyoma preoperatively, Whipple's procedure could have been avoided. We conclude that adenomyoma of ampulla can be considered a possibility in a suspected case of ca head of pancreas with raised CA-19-9 level.

 
  References Top

1.Ryan A, Lafnitzegger JR, Lin DH, Jakate S, Staren ED. Myoepithelial hamartoma of the duodenal wall. Virchows Arch 1998;432:191-4.  Back to cited text no. 1
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2.Vandelli A, Cariani G, Bonora G, Padovani F, Saragoni L, Dell'Amore D. Adenomyoma of the stomach: Report of a case and review of the literature. Surg Endosc 1993;7:185-7.  Back to cited text no. 2
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3.Ligorred L, Morollon MJ, Aragon J, Gonzalez G, Munilla E, Garcia Omedes A, et al. Obstructive jaundice from an Adenomyoma of the Ampulla of Vater. Rev Esp Enferm Dig 1997;89:411-2.  Back to cited text no. 3
    
4.Hammarstrom LE, Holmin T, Stenram U. Adenomyoma of the Ampulla of Vater; an uncommon cause of bile duct obstruction. Surg Laparosc Endosc 1997;7:388-93.  Back to cited text no. 4
    
5.Chand EM, Caudell P. Pathologic quiz case. Patient with duodenal strictures and a mass at the head of the pancreas. Arch Pathol Lab Med 2001;125:701-2.  Back to cited text no. 5
[PUBMED]    
6.Kayahara M, Ohta T, Kitagawa H, Miwa K, Urabe T, Murata T. Adenomyomatosis of the papilla of the vater: A case illustrating diagnostic difficulties. Dig Surg 2001;18:39-42.  Back to cited text no. 6
    


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Abstract
Introduction
Case Report
Discussion
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