Rare cause of recurrent fever of unknown origin: Acute obstructive suppurative pancreatic ductitis after pancreaticojejunostomy

Abstract

An 82-year-old man, who underwent pancreatoduodenectomy 14 years earlier for bile duct cancer, was referred for overnight fever. He had been suffering from recurrent fever of unknown origin (FUO) and bacteremia that occurred once every one or two months for the last 3 years. Laboratory tests showed leukocytosis and elevation of Creactive protein (CRP), but his serum pancreatic amylase and lipase levels were in normal range. Computed tomography showed stones in the pancreatic duct without signs of acute pancreatitis (Fig. 1). Singleballoon enteroscopy showed a pancreaticojejunal anastomotic stricture (Fig. 2a), and, after a balloon dilatation, purulent discharge was observed from the anastomosis (Fig. 2b). Finally, two 7-Fr stents were placed across the pancreaticojejunostomy to treat the acute obstructive suppurative pancreatic ductitis (AOSPD) (Fig. 2c). Enterobacter cloacae was identified from blood cultures and pancreatic juice. His fever, leukocytosis, and elevated CRP subsided after 7 days of intravenous ceftriaxone. AOSPD did not recur thereafter, and no further intervention was required until he died of heart failure 16 months later.Acute obstructive suppurative pancreatic ductitis is defined as acute suppuration of the pancreatic duct without concurrent pancreatic abscess or infected pseudocyst. 1,2 Most of the AOSPD patients present with high fever and elevated CRP levels. 3,4 Unlike pancreatitis, pancreatic enzymes are often normal or only slightly elevated in patients with pancreatic ductitis. 3 The diagnosis of AOSPD is confirmed by the finding of pancreatic duct obstruction with evidence of infection, such as positive pancreatic juice culture or purulent pancreatic juice. 4 Although there is no standard treatment, prompt administration of antibiotics and pancreatic duct drainage is the mainstay of the treatment. 3,5 In this case, the patient suffered from recurrent FUO for 3 years. As this case indicates, AOSPD should be considered in the differential diagnosis of FUO, particularly in patients with previous history of pancreatic intervention.

A case of acute obstructive suppurative pancreatic ductitis complicated with acute cholangitis diagnosed only after the removal of a pancreatic duct stent

“…Although no fatal cases have been reported, AOSPD can lead to sepsis 3 and is a rare but important complication of CP. The reported causes of pancreatic duct obstruction in AOSPD include pancreatic duct obstruction due to CP, pancreatic cancer, and/or pancreatic surgery 1–5 . AOSPD is usually accompanied by fever and abdominal pain.…”

“…The reported causes of pancreatic duct obstruction in AOSPD include pancreatic duct obstruction due to CP, pancreatic cancer, and/or pancreatic surgery. 1 , 2 , 3 , 4 , 5 AOSPD is usually accompanied by fever and abdominal pain. In a retrospective review of 20 patients with AOSPD, a temperature of 37.5°C or higher was observed in 14/20 patients (70%), while abdominal pain was observed in 19/20 (95%).…”