Ascariasis of gall bladder associated with xanthogranulomatous inflammation and cholelithiasis.

We report a rare case of ascariasis of gall bladder. The unusual features in this case were the presence of eggs of Ascaris lumbricoides in the lumen. Some of the eggs had evoked a foreign body reaction indicating chronicity. The bladder wall was unevenly thickened with yellowish white nodules and showed maximum thickness around the neck region. Microscopy showed predominantly xanthogranulomatous inflammation in the thickened parts of the wall.

INTRODUCTION

In the literature, cases have been documented of ascariasis of gall bladder associated with cholelithiasis.[1] The diagnosis was often based on the ultrasonographic findings of nonshadowing, echogenic, tubular structures within the bile ducts, with a longitudinal, central echo-free line representing the gastrointestinal tract of the worm,[2] or history of passage of worms by the patient.[1]

We encountered a case of ascariasis of gall bladder associated with cholelithiasis in a 39-year-old female. She gave a history of passage of worms, about 6 inches in length and whitish in color, for which she was treated outside few months back. Examination of the excised gall bladder showed histolological evidence of eggs in the lumen of the gall bladder with a striking xanthogranulomatous cellular response in the thickened wall of the gall bladder. These rare and interesting findings have prompted us to report this case.

CASE REPORT

A 39-year-old female patient came with complaints of pain in the abdomen and vomiting since 3–4 days. The patient gave a past history of the passage of worms and treatment given outside about 6–8 months back. Local examination revealed tenderness all over the abdomen with a lump in the right iliac fossa. Rest of the systemic examination was unremarkable. The laboratory investigations revealed a hemoglobin level of 10.2 g% and total leukocyte count 13100/mm3 with predominant neutrophilia (74%). Stool examination was noncontributory. Liver function tests showed serum bilirubin 0.68 mg%, serum glutamic oxaloacetic transaminase (SGOT) 17 IU/l, serum glutamic pyruvic transaminase (SGPT) 20 IU/l, and serum alkaline phosphatase 290 IU/l. Kidney function tests were within normal limits. The computed tomography (CT) scan showed a markedly distended gall bladder with a luminal diameter of 4.5 cm. There was marked thickening of the wall near the neck region, measuring approximately 1.2 cm in thickness. Luminal contents were suggestive of debris. A large calculus was seen at the neck of the gall bladder, measuring 1.2 cm in the greatest diameter.

The patient was posted for laparoscopic cholecystectomy. Intraoperatively, dense adhesions were noted between the gall bladder serosa and the hepatic capsule; hence a decision was taken to convert the procedure into open cholecystectomy. The abdomen was opened by right subcostal incision and the firm adhesions were dissected out. The gall bladder was submitted for histopathological examination.

Grossly, the gall bladder measured 12 × 5 × 3 cm. The external surface showed marked congestion and a large roughened area of adhesions [Figure 1a]. On cutting open, the lumen contained a blackish brown necrotic material and multiple brownish black-colored calculi, the largest (1.5 × 1.2 × 0.5 cm) of which was impacted at the neck. The wall of the gall bladder showed variation in the thickness. It was 0.5 cm at the tip of the distended part, while markedly thickened (2.5 cm) around the neck region. The thickened portion was grayish white and showed yellowish nodular areas [Figure 1b].

Figure 1

Gross photograph showing the (a) external surface with a large roughened area and (b) cut section showing impacted stone near the neck and necroinflammatory debris in the lumen with yellowish nodules in the wall

Multiple sections from the wall showed evidence of chronic inflammatory infiltrate of histiocytes admixed with lymphocytes and plasma cells. The histiocytes showed foamy, lipid-laden cytoplasm [Figure 2]. Foci of necrosis and inspissated bile along with polymorphs and nuclear debris were noted on the luminal side. In addition, the walls of eggs of nematodes with an outer crenated surface, indicating rugosities were seen. A foreign body-type giant cell reaction was noted in some of the broken egg shells. The walls of the eggs had crenated borders, were hence interpreted as Ascaris lumbricoides [Figure 3].

Figure 2

Photomicrograph showing chronic inflammation with infiltrationby lymphocytes and plasma cells along with foamy, lipid-laden macrophages admixed with lymphocytes and plasma cells (H and E, ×400)

Figure 3

Photomicrograph showing a foreign body-type giant cell reaction to the eggs of Ascaris lumbricoides (H and E, ×400)

DISCUSSION

A. lumbricoides is well documented in the hepatobiliary system; however, ascariasis of the gall bladder with the presence of eggs is seldom documented.[3] The gall bladder ascariasis is often associated with cholecystitis and cholelithiasis. The commonly encountered inflammatory response is chronic nonspecific inflammatory cells, i.e. lymphocytes, plasma cells, and histiocytes. Occasionally, the predominance of eosinophils has been noted in the literature.[1] The diagnosis of ascariasis is generally based on the history of passing worms with typical morphology. As in this case, in addition, examination of stool and bile for the presence of eggs or adult worms is of diagnostic importance.[4] In this case, although there was a history of passage of worms, worms were not found intraoperatively. However, this may be accounted for by the treatment given outside.

Xanthogranulomatosis is defined as the presence of chronic inflammatory cells, namely, lymphocytes and plasma cells along with foamy, lipid-laden macrophages. The exact etiology of this entity remains unelucidated.[5] However, bacterial infection is implicated in its pathogenesis by some authors.[6] It is interesting to note that in our case, there was xanthogranulomatous inflammatory response associated with thickening and yellowish appearance on the cut section, similar to the gross appearance of xanthogranulomatous pyelonephritis of kidney.[7] In the literature, xanthogranulomatous cholecystitis (XGC) has been reported by Chinese workers. It must be mentioned that the first case of XGC was described in 1981. The authors postulated that the lesion results due to ruptured Rokitansky–Aschoff sinuses with intramural extravasation of bile and subsequent xanthogranulomatous reaction. They have also reported a xanthogranulomatous response, which was associated with severe fibrosis causing difficulty in cholecystectomy, as was encountered in our case. Furthermore, it is stated that characteristic thickening associated with xanthogranulomatous inflammation may lead to erroneous diagnosis of carcinoma by imaging or during surgery.[8]

Although gall bladder ascariasis is well documented in the Indian literature,[9] the presence of eggs in the lumen and the xanthogranulomatous inflammation are unusual findings in this case.