Fundal Variant of Adenomyomatosis of the Gall Bladder
Transcription
Fundal Variant of Adenomyomatosis of the Gall Bladder
Case Report DOI: 10.17354/cr/2015/69 Fundal Variant of Adenomyomatosis of the Gall Bladder: An Uncommon Entity Archana Shivamurthy1, Roumina Hasan1, Sushmitha Malpe Gopal1, Shaila Talengala Bhat2, Tanvi Shetty1 Assistant Professor, Department of Pathology, Melaka Manipal Medical College, Manipal University, Manipal, Karnataka, India, 2Professor and Head, Department of Pathology, Melaka Manipal Medical College, Manipal University, Manipal, Karnataka, India 1 Adenomyomatosis of the gallbladder is a benign and degenerative condition of the gallbladder. It is an incidental finding in gall bladder specimens resected for chronic cholecystitis or cholelithiasis. It frequently occurs after 3rd or 4th decade of life and is often an incidental finding in cholecystectomy specimens resected for chronic cholecystitis or cholelithiasis. Patients with adenomyomatosis are usually asymptomatic it can be classified into three types: Segmental, fundal and diffuse types. The fundal variant is uncommon compared to the other two types. Here, in we present a case of a fundal variant of adenomyomatosis of the gall bladder in a 65-year-old male patient. Keywords: Adenomyomatosis, Fundus, Gall bladder INTRODUCTION Adenomyomatosis of the gall bladder is an uncommon benign and hyperplastic condition of the gall bladder characterized by the proliferation of its mucosal lining.1 The incidence ranges from 2% to 8.7%. It is found to occur more frequently after 3rd or 4th decade of life. However, few cases have also been reported in pediatric population.2 A female predominance has also been noted. Adenomyomatosis is an incidental finding in gall bladder specimens resected for chronic cholecystitis or cholelithiasis. Patients with adenomyomatosis are usually asymptomatic. 1,2 Jutras in 1960 used the term “hyperplastic cholecystoses” to described what is now termed as adenomyomatosis.3 There are three different variants described in adenomyomatosis. These include segmental, fundal, and diffuse type. soft, non-tender, and no mass was palpable. Investigations revealed hemoglobin of 14 g/dl, the total count of 6.6 × 103/µL. Ultrasound examination of the abdomen and pelvis showed grade one fatty infiltration of liver and chronic cholecystitis. The patient was hence taken up for laparoscopic cholecystectomy, and the specimen was sent for histopathological examination. Pathological Findings On macroscopic examination, the cholecystectomy specimen weighed 9 g and measured 6 cm × 3.5 cm. The cut section showed focal bile stained, denuded mucosa. The fundus of the gall bladder showed a multicystic mass in the wall measuring 1 cm × 1 cm (Figure 1). A single lymph CASE REPORT A 65-year-old male patient came with complaints of vague abdominal discomfort associated with lower abdominal pain since 1-month. There was no history of fever, nausea, vomiting, diarrhea, yellowish discoloration of the eyes and urine. On physical examination, the abdomen was Access this article online Month of Submission Month of Peer Review Month of Acceptance Month of Publishing : : : : 02-2015 03-2015 03-2015 04-2015 www.ijsscr.com Figure 1: Gall bladder with focal denuded mucosa. Fundus shows a multicystic mass Corresponding Author: Dr. Archana Shivamurthy, Assistant Professor, Department of Pathology, Basic Science Building, Manipal University Campus, Manipal - 576 104, Karnataka, India. Phone: +91-9880455094. E-mail: [email protected] IJSS Case Reports & Reviews | April 2015 | Vol 1 | Issue 11 55 Shivamurthy, et al.: Adenomyomatosis of Gall Bladder node was also identified at the neck of the gallbladder. No stones were identified. On microscopic examination, focally ulcerated, hyperplasytic gall bladder mucosa was observed. Rokitansky-Aschoff sinuses or outpouchings of the mucosa were seen penetrating into and through the muscularis propria (Figures 2 and 3). The lining consisted of columnar epithelial cells with no atypical features. Focal pylori metaplasia was also found. Few glands were irregularly shaped and cystically dilated surrounded by proliferating smooth muscle cells (Figures 3, 4a and b). None of the glands showed features of malignancy. The mucular layer and serosa showed few congested vessels and lymphoplasmacytic infiltrate. Section from the lymph node showed reactive changes. Hence, a diagnosis of adenomyomatosis-fundal variant with chronic cholecystitis was rendered. DISCUSSION Of the three variants of adenomyomatosis, segmental form is the most common, followed by the fundal Figure 2: Proliferation on glandular structures (H and E ×40) variant and the diffuse type.2 The fundal variant is often difficult to appreciate on radiology as the fundus of the gallbladder is insufficiently visualized, because of the intestinal gas. Even in the present case the lesion was not detected on ultrasonography. It has also been suggested that computerized tomography may help to distinguish ademyomatosis of fundal type from localized chronic cholecystitis.4,5 Both clinically and pathologically the fundal type differs from the other two types of adenomyomatosis. This type has a lower incidence of gall stones. No stones were detected in our case on gross examination. The grade of inflammation is also found to be of lower grade in these cases.2,4 Carcinoma of the gall bladder is also found to be less commonly associated with the fundal variant.6 No atypical features were observed in our case. On histopathological examination, adenomyomatosisof the gall bladder has hyperplastic mucosa, lobules of glandular parenchyma, cystically and irregularly dilated glands lined by cuboidal to columnar epithelium with no atypical features. Smooth muscle cell bundles are seen in the surrounding stroma.1,7 On immunohistochemistry, similar to the biliary epithelium, the epithelial cells in adenomyomatosis show positivity for cytokeratin-7 and cytokeratin-20 and the smooth muscle cell cells for alphasmooth muscle actin.2,4 The differential diagnosis includes adenoma and adenocarcinoma of the gall bladder, various other polpoidal lesions such as hyperplatic and adenomatous polyps. Cholecystectomy is the treatment of choice for the fundal type of adenomyomatosis if patients do not respond to medical therapy. The latter is initiated only when adenomyomatosis is detected on radiology.2,8 a b Figure 3: Proliferation of smooth muscle cells surrounding irregularly dilated glandular structures (H and E ×40) 56 Figure 4: (a and b) Irregularly dilated glandular structures lined by columnar epithelium surrounded by proliferation of smooth muscle cell (H and E ×40) IJSS Case Reports & Reviews | April 2015 | Vol 1 | Issue 11 Shivamurthy, et al.: Adenomyomatosis of Gall Bladder CONCLUSION Fundal adenomyomatosis is a rare entity. It is often detected incidentally. Due to its rare potential of developing into benign or malignant tumor, it should be always born in mind during evaluation of cholecystectomy specimens. ACKNOWLEDGMENT We wish to thank all the technical staff of the Department of Pathology, Manipal University. REFERENCES 1. Il’chenko AA, Orlova IuN. [Article in Russian] [Gallbladder adenomyomatosis]. Eksp Klin Gastroenterol 2010;86-91. 2. Ozgonul A, Bitiren M, Guldur ME, Sogut O, Yilmaz LE. Fundal variant adenomyomatosis of the gallbladder: Report of three cases and review of the literature. J Clin Med Res 2010 19;2:150-3. 3. Jutras JA. Hyperplastic cholecystoses; Hickey lecture, 1960. Am J Roentgenol Radium Ther Nucl Med 1960;83:795-827. IJSS Case Reports & Reviews | April 2015 | Vol 1 | Issue 11 4. Kim BS, Oh JY, Nam KJ, Cho JH, Kwon HJ, Yoon SK, et al. Focal thickening at the fundus of the gallbladder: Computed tomography differentiation of fundal type adenomyomatosis and localized chronic cholecystitis. Gut Liver 2014;8:219-23. 5. Kim JH, Jeong IH, Han JH, Kim JH, Hwang JC, Yoo BM, et al. Clinical/pathological analysis of gallbladder adenomyomatosis; type and pathogenesis. Hepatogastroenterology 2010;57:420-5. 6. Ootani T, Shirai Y, Tsukada K, Muto T. Relationship between gallbladder carcinoma and the segmental type of adenomyomatosis of the gallbladder. Cancer 1992;69:2647-52. 7. Cetinkursun S, Surer I, Deveci S, Demirbag S, Saglam M, Atabek C, et al. Adenomyomatosis of the gallbladder in a child. Dig Dis Sci 2003;48:733-6. 8. Ram MD, Midha D. Adenomyomatosis of the gallbladder. Surgery 1975;78:224-9. How to cite this article: Shivamurthy A, Hasan R, Gopal SM, Bhat ST. Fundal Variant of Adenomyomatosis of the Gall Bladder: An Uncommon Entity. IJSS Case Reports & Reviews 2015;1(11):55-57. Source of Support: Nil, Conflict of Interest: None declared. 57