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Idiopathic Perforation of the Gallbladder: A Novel Differential Diagnosis of Acute Abdomen

Estevão-Costa, José*; Soares-Oliveira, Miguel*; Lopes, José Manuel; Carvalho, José Luis*

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Journal of Pediatric Gastroenterology and Nutrition: July 2002 - Volume 35 - Issue 1 - p 88-89
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INTRODUCTION

Acute biliary tract disease is uncommon in childhood. Apart from bile duct perforations, spontaneous rupture of the gallbladder itself is very rare in pediatric age; all recorded cases were secondary to coexistent disease (1,2). A child presenting idiopathic gallbladder perforation (GP) is described herein.

CASE REPORT

A six-year-old boy was referred with suspicion of acute appendicitis. He complained of vomiting and abdominal pain for the last 24 hours. Axillary temperature was 38°C and the abdomen was painful and tender. Complete blood count revealed leukocytosis. Abdominal ultrasonography demonstrated moderate free intraperitoneal fluid, mainly in the right iliac fossa and pelvis. An emergency laparotomy was performed with the presumptive diagnosis of peritonitis secondary to appendicitis but there was a 500 ml bile-stained ascites. Exploration of the abdominal cavity revealed a type I (3) oval perforation (2 cm) of the gallbladder infundibulum (Fig. 1). There were no gallstones or bile duct dilatation. Cholecystectomy was then performed and the child experienced an uneventful recovery.

FIG. 1.
FIG. 1.:
Surgical specimen showing an oval perforation of the gallbladder infundibulum.

Histological examination did not reveal inflammatory infiltrate or any other significant alteration of the gallbladder wall. The cultures of peritoneal fluid were negative. Postoperative biliary MRI was normal. At 15-month follow-up the child continued to be well.

DISCUSSION

Gallbladder perforation (GP) is a well-recognized condition in adults, usually located at the least vascularized fundus (4,5). It has been frequently reported vaguely as spontaneous or idiopathic, but almost all cases were in fact secondary to coexistent disease such as inflammation, trauma, or obstruction (1,3,5,7). Thus, a classification of GP is needed. We propose the criteria shown in Table 1.

TABLE 1
TABLE 1:
Gallbladder perforations

In children, biliary tract perforations are usually located at bile ducts, the gallbladder itself being the least common situation. There are few scattered reports of spontaneous GP in childhood; however, all recorded cases were secondary to obstruction (1,2,6,7).

Although we cannot entirely exclude a minor trauma, the present case was classified as idiopathic because comprehensive investigation did not disclose any inflammatory/infectious condition (local or systemic) or biliary tract obstruction; moreover, the location of GP at infundibulum and absence of histologic signs of ischemia did not suggest a vascular etiology. Thus, congenital weakness of the gallbladder wall could have been a potential explanation.

The diagnosis of spontaneous GP is often delayed or missed altogether, the majority being made only at surgery. Ultrasonography and computerized tomography may demonstrate abdominal fluid but lack specificity to diagnose GP, which can be easily detected on hepatobiliary scanning (HIDA or DISIDA) (2,7,8,9). Prompt recognition and early treatment are critical to prevent serious complications (2,6,8,9).

As far as we know, the present case is the first one of an idiopathic GP in children. This entity should be added to the list of causes of acute abdomen in childhood.

REFERENCES

1. Prévot J, Babut JM. Spontaneous perforations of the biliary tract in infancy. Prog Pediatr Surg 1971; 1:187–208.
2. Banani SA, Bahador A, Nezakatgoo N. Idiopathic perforation of the extrahepatic bile duct in infancy: pathogenesis, diagnosis and management. J Pediatr Surg 1993; 28:950–52.
3. Niemier OW. Acute free perforation of the gallbladder. Ann Surg 1934; 99:922–24.
4. Nomura T, Shirai Y, Hatakeyama K. Spontaneous gallbladder perforation without acute inflammation or gallstones. Am J Gastroenterol 1997; 92:895.
5. Greenwald G, Stine RJ, Larson RE. Perforation of the gallbladder following blunt abdominal trauma. Ann Emerg Med 1987; 16:452–54.
6. Soares-Oliveira M, Mariz C, Carvalho JL, Estevão-Costa J. Diaphragm of the gallbladder: a rare cause of acalculous acute cholecystitis. J Pediatr Gastroenterol Nutr 2000; 31:570–71.
7. Sharma R, Mondal A, Sen IB. Spontaneous perforation of the gallbladder during infancy diagnosed on hepatobiliary imaging. Clin Nucl Med 1997; 22:759–61.
8. Felice PR, Trowbridge PE, Ferrara JJ. Evolving changes in the pathogenesis and treatment of the perforated gallbladder. Am J Surg 1985; 149:466–73.
9. Ong CL, Wong TH, Rauff A. Acute gallbladder perforation-a dilemma in early diagnosis. Gut 1991; 32:956–58.
© 2002 Lippincott Williams & Wilkins, Inc.