Adenomyomatosis-a case report

Ahmed Ourfali; Othman Maimani; Afaf M El Shafi; Minhajul Bari; Kamal Ezzeldin

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CASE REPORT

Year : 1997 | Volume : 3 | Issue : 3 | Page : 144-146

Adenomyomatosis-a case report

Ahmed Ourfali¹, Othman Maimani², Afaf M El Shafi³, Minhajul Bari¹, Kamal Ezzeldin⁴
¹ Department of Gastroenterology, Al Noor Specialist Hospital, Makkah Al Mukarramah, Saudi Arabia
² Department of Surgery, Al Noor Specialist Hospital, Makkah Al Mukarramah, Saudi Arabia
³ Department of Histopathology, Al Noor Specialist Hospital, Makkah Al Mukarramah, Saudi Arabia
⁴ Department of Radiology, Al Noor Specialist Hospital, Makkah Al Mukarramah, Saudi Arabia

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Date of Submission	09-Apr-1996
Date of Acceptance	12-Jul-1997

How to cite this article:
Ourfali A, Maimani O, El Shafi AM, Bari M, Ezzeldin K. Adenomyomatosis-a case report. Saudi J Gastroenterol 1997;3:144-6

How to cite this URL:
Ourfali A, Maimani O, El Shafi AM, Bari M, Ezzeldin K. Adenomyomatosis-a case report. Saudi J Gastroenterol [serial online] 1997 [cited 2022 Jun 30];3:144-6. Available from: https://www.saudijgastro.com/text.asp?1997/3/3/144/33924

The term hyperplastic cholecystosis encompasses a group of disorders of the gallbladder including adenomyomatosis and cholesterolosis ^[1] . Adenomyomatosis may be focal or diffuse with hyperplasia of the muscle and mucosa of the gallbladder. The projection of pouches of mucous membrane through the weak points in the muscle coat produces the Rokitansky-Aschoff sinuses or crypts. These are probably identical with intramural diverticula, and may contain pigmented stones ^[2] . They may opacify on cholecystography. They are best seen after gallbladder contraction when they appear as a "halo" or "ring" of opacified beads around the gallbladder. Localized adenomyomatosis is responsible for the appearance of a pharyngeal cap at the gallbladder fundus. Ultrasonographic features of adenomyomatosis include thickening of the gallbladder wall and round, anechoic areas in the thickened wall representing stones within the diverticula ^[3] [Figure - 1].

Case Report

A 28-year-old Sudanese male attended the gastroenterology clinic with complaints of intermittent epigastric and right upper quadrant pain bearing no relation to food and without vomitting. There was nothing remarkable in the history and physical examination. The blood chemistry, complete blood count, X-ray, and EKG examinations were all normal.

Upper GI endoscopy was normal except that the antral biopsy yielded Helicobacter pylori

ch a course of metronidazole, bismuth citrate, and amoxycillin was given for two weeks. The patient revisited the clinic after four months with the same complaints along with postprandial vomitting, especially after fatty meals. Examination and routine investigation were again normal. Upper GI endoscopy was repeated, which revealed two antral ulcers measuring 7.0 and 5.0 mm at the 3 and 6 o' clock positions with a normal duodenum. Biopsies from ulcer sites showed superficial gastritis and were positive for Helicobacter pylori.

He was stated on a course of cimetidine, amoxycillin, and antacids for two weeks followed by cimetidine and antacids for another six weeks and was advised to have ultrasonography of the abdomen. After four weeks he returned without improvement. The abdominal ultrasonography was highly suggestive of adenomyomatosis, showing thickening of the gallbladder wall with some lucent and hyperechoic areas suggestive of diverticula [Figure - 2].

To confirm this diagnosis, oral cholecystography was performed which showed a ring of opacified beads around the gallbladder i.e., its typical appearance. During this period, upper GI endoscopy was repeated and found that the ulcers had healed. Biopsies at this time were normal.

The patient underwent an uneventful laparascopic cholecystectomy. Histopathology showed muscular hypertrophy, encrusted stones, and irregular tubular structures within the wall lined by cuboidal epithelium (i.e. Rokitansky-Aschoff sinuses). These findings correlated well with the ultrasonographic and cholecystographic picture of adenomyomatosis. He was discharged after four days and remained well in the seven-month postoperative follow up period.

Discussion

The etiology of gallbladder adenomyomatosis is not known. Its incidence varies from 2 to 33% ^{[4],[5],[6],[7]} . It is usually an asymptomatic disease, discovered incidentally by cholecystography and/or ultrasonography. It is more often diagnosed by histopathologic examination ^[7] . It can cause right upper quadrant pain as in the present case, but the majority of the fundal type are asymptomatic ^[8],[9] .

A few cases have been reported indicating that this usually benign condition may transform into malignancy. This includes invasive or in-situ adenocarcinoma arising from the mucosa, squamous cell carcinoma, early carcinoma originating in and limited to the Rokitansky-Aschoff sinuses, or papillotubular adenocarcinoma also arising from the Rokitansky-Aschoff mucosa ^{[10],[11],[12],[13],[14],[15]} . The incidence of gallbladder carcinoma is significantly higher in the segmented variety of adenomyomatosis ^[16] .

Adenomyomatosis may also occur in the common bile duct where it may present with extrahepatic cholestatic jaundice ^[17] .

Oral cholecystography is still considered the "golden standard," but ultrasonography and computerized axial tomography are gaining ground as accurate diagnostic tools. A fatty meal given as a routine part of oral cholecystography is also recommended for detection of acalculous gallbladder disease; however, its routine use had been previously questioned ^[18] .

Ultrasonography is a worthwhile technique having a good correlation between sonographic, radiologic, and pathologic findings ^[19],[20] . The ultrasonographic appearance is specific in established disease of adenomyomatosis as its segmented forms can result in marked mural thickening in the waist of the gallbladder giving a characteristic hour-glass deformity in both the cholecystogram and the ultrasound examinations ^[21],[22] .

Computerized axial tomography is also a sensitive diagnostic tool. It shows variation in the mural density, marked differential enhancement of wall layers during dynamic liver scanning, and detection of Rokitansky Aschoff sinuses within the walls or characteristic "rosary sign" ^[23],[24] . The rosary sing is formed by enhanced proliferative mucosal epithelium with intramural diverticula surrounded by the unenhanced hypertropied muscularis of the gallbladder.

Conclusions

This condition is frequently asymptomatic but can present with abdominal pain or even obstructive jaundice; thus, it should be considered in the differential diagnosis of such symptoms. The increasing incidence of associated malignancy makes it imperative to diagnose such cases early. Ultrasonography can diagnose most of the cases with confirmation by oral cholecystography. These are both readily available and cost effective. Once the diagnosis is established, cholecystectomy is advised.

References

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