Two Cases of Acute Pancreatitis in Infants Less Than One Year Old

Although acute pancreatitis is rare in children, especially in infants less than 1 year old, the condition is more common than has previously been considered (1,2). Acute pancreatitis is diagnosed on the basis of clinical findings of an episode of acute abdominal pain accompanied by increased amylase levels at biochemical testing. Abdominal ultrasound (US) and computed tomography (CT) can be useful for diagnosing cases with confusing clinical findings (3,4). Endoscopic retrograde cholangiopancreatography (ERCP) and magnetic resonance (MR) cholangiopancreatography have been the imaging modalities used for diagnosis of biliary tract abnormalities (5,6). In Western countries, acute pancreatitis in children has been attributed to infection, trauma, structural abnormalities, drugs, and systemic disease (7). In Japan, however, an abnormal pancreaticobiliary ductal junction is the most common cause of pancreatitis in children (8). We report two infants with acute pancreatitis. One was a 6-month-old Japanese girl with recurrent pancreatitis complicated by pseudocyst formation. The other was an 11-month-old Japanese boy with acute pancreatitis caused by a pancreaticobiliary tract abnormality. CASE REPORT Patient 1 was a girl who appeared to be well until 6 months of age, when she was admitted to our hospital because of frequent diarrhea, vomiting, and abdominal distension with an ascites. She had been ill for 7 days before admission. A stool culture revealed bacterial enteritis due to Klebsiella oxytoca. She had significantly elevated amylase, lipase, elastase, trypsin, and phospholipase A2 levels in her serum and ascitic fluids but no elevation of serum bilirubin. Abdominal US and CT showed enlargement of her pancreas with peripancreatic fluid collection. She showed clinical improvement after bowel rest and high-energy intravenous fluids for 2 months, in addition to administration of prophylactic antibiotics, nafamostat mesylate, and ulinastatin. Seven months later, abdominal CT showed pancreatic pseudocyst formation (Fig. 1), but ERCP showed no abnormalities in the pancreaticobiliary tract.FIG. 1.: Abdominal computed tomographic scan of patient 1 at 13 months of age showed a pancreatic pseudocyst.During the subsequent year, the patient had recurrent pancreatitis and required treatment for 8 months for abdominal pain, vomiting and dehydration. She had been given treatments with nasogastric decompression, bowel rest, intravenous fluid infusion, and prophylactic antibiotics, nafamostat mesylate, and ulinastatin. At 35 months of age, she had significantly elevated serum amylase, lipase, elastase, trypsin, and phospholipase A2 levels of 973 IU/l, 239 IU/l, 955 ng/dl, 1030 ng/dl, and 2640 ng/dl, respectively. Her urine amylase level was also elevated to 4040 IU/l. Abdominal US and CT demonstrated a very large pancreatic pseudocyst (Fig. 2) that required surgical intervention for the drainage and resection of the pseudocyst. We suggest that this condition may have been related to the unresolved pseudocyst. The postoperative course was uneventful. She has remained well for 14 months.FIG. 2.: Abdominal computed tomographic scan of patient 1 at 35 months of age showed an increase in size of the pancreatic pseudocyst.Patient 2 was an 11-month-old Japanese boy who was admitted to our hospital with abdominal pain, vomiting, diarrhea, a pale face, and dehydration with hypovolemic shock. He had had two previous episodes of acute enteritis with abdominal pain and diarrhea at 7 and 9 months of age. When admitted, he had significantly elevated serum amylase, lipase, elastase, trypsin, and phospholipase A2 levels of 1732 IU/l, 498 IU/l, 1,104 ng/dl, 2,130 ng/dl, and 2,192 ng/dl, respectively. His urine amylase level was extremely high at 21,530 IU/l. Abdominal US and CT showed pancreatic enlargement with surrounding inflammation and peripancreatic fluid. All of the abnormal laboratory findings including serum amylase were normalized after 6 days of treatment with nasogastric decompression, bowel rest, intravenous fluid infusion, prophylactic antibiotics, nafamostat mesylate, and ulinastatin. Although he continued to be asymptomatic, with no recurrence of pancreatitis, ERCP was performed when he was 16 months of age to find the underlying cause. An abnormal 15-mm-long common channel was revealed, formed by the distal common bile duct and pancreatic duct (Fig. 3).FIG. 3.: Endoscopic retrograde cholangiopancreatography of patient 2 at 16 months of age showed an abnormal 15-mm-long common channel (arrow) formed by the distal common bile duct and pancreatic duct. Dilatation of the common bile duct and intrahepatic duct was also seen.DISCUSSION Clinical awareness of acute pancreatitis in children has improved immensely over recent years (9). However, reports of acute pancreatitis in infants less than 1 year old have been scarce. Our patients had abdominal bouts of pancreatitis when one was 6 and the other 11 months of age. The cause of acute pancreatitis in children is different from that in adults. Furthermore, there is a significant difference in cause between Western countries and Asian countries. Tomomasa et al. (8) reviewed the cause of acute pancreatitis under 15 years old in Japan. An abnormal biliary tract (47%) was the most common cause of acute pancreatitis in Japanese children, and the other causes were infections (12%), trauma (9%), drugs (9%), and systemic disease (2%). Mader and McHugh (7) reviewed pediatric cases of acute pancreatitis in Western countries and reported greater diversity with no particular category predominant. Five different groups, trauma (22%), medications (13%), infections (11%), structural anomalies (15%), and systemic illness (13%) share distribution fairly evenly. In our patient 2, dilatation of the common bile duct with an abnormal pancreaticobiliary ductal junction was demonstrated by ERCP. Patient 1 had recurrent pancreatitis with the complication of pancreatic pseudocyst formation after an enteric infection. The traditional diagnosis of acute pancreatitis in adults was dependent on clinical features and laboratory findings. However, in infants, it can be difficult to diagnose, because the symptoms of acute pancreatitis in children differ from those that typically occur in adults. We should take special care when diagnosing pancreatitis in infants. Both of our patients had abdominal pain, vomiting, and diarrhea. Serum and urine amylase were extremely high, as were other pancreatic enzymes, including lipase, elastase, trypsin, and phospholipase A2. These enzymes are necessary when diagnosing and monitoring acute pancreatitis. Abdominal CT revealed enlargement of the pancreas in both patients. A pancreatic pseudocyst in patient 1 was revealed by abdominal US and CT. ERCP showed pancreaticobiliary tract abnormality in patient 2. Although there are technical difficulties, ERCP has been useful in diagnosing pancreatic and biliary duct abnormalities, even in small infants (5,10). Recently, the noninvasive method of MRCP has been used to diagnose choleductal cysts in children (6). However, because it cannot show abnormalities in infants 1 year old clearly, we must take care when using it. ERCP is an invasive procedure that usually necessitates the use of general anesthetics in children and is contraindicated in the acute stage of pancreatitis. Because of its ease, availability, and reliability, US is the first choice in a plan of appropriate investigations for the child with acute pancreatitis. There is no specific treatment for pancreatitis. Current management recommendations continue to be based on the concept of putting the pancreas at rest. Supportive measures include the administration of intravenous fluids to replace third-space fluid losses and avoidance of enteric feedings. Recent reports indicate that enzyme inhibitors have been effective in patients with acute pancreatitis and have been tested with good results in vitro (11,12). We used the combination of nafamost at (0.2 mg/kg per hour), ulinastatin (5000 IU/kg per day), and prophylactic broad-spectrum antibiotics, administered intravenously. The role of drugs such as nafamostat mesylate and ulinastatin is to inhibit proteases. In patient 2, clinical signs disappeared and his serum pancreatic enzyme levels were normalized with no deterioration. However, the complication of pseudocyst formation was present in patient 1. Until recently, the treatment of nonresolving, large pseudocysts has been surgical. However, percutaneous drainage of pseudocysts has now been shown to be a safe and effective treatment in most patients. The effectiveness of such protease inhibitors as nafamostat mesylate and ulinastatin in patients with acute pancreatitis is still controversial.

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