Subcutaneous emphysema of the lower extremity and pneumoperitoneum after endoscopic retrograde cholangiopancreatography–related duodenal perforation

A 65-year-old woman with no record of chronic diseases was referred to the gastroenterology department for endoscopic retrograde cholangiopancreatography (ERCP) due to symptomatic choledocholithiasis. She complained of colic and epigastric pain lasting for a few days prior to the admission. A large gallstone causing common bile duct dilation was visualized on ultrasonography. No findings other than mild tenderness in the epigastric region were notable on physical examination. Her baseline laboratory findings showed slight alkaline phosphatase (ALP) and C-reactive protein (CRP) elevation (ALP, 218 U/l, reference range [RR], 35–105 U/l; CRP, 10.5 mg/l, RR <⁠5 mg/l). Total bilirubin, alanine transaminase, and aspartate transaminase levels were normal (0.29 mg/dl, 22 U/l, and 24 U/l, respectively). γ-Glutamyl transpeptidase concentration was not evaluated at baseline. The ERCP procedure itself appeared difficult to perform, as the major duodenal papilla was located deep in the diverticulum (Figure 1A). The papilla was cut with a needle knife, but unfortunately stent placement was unsuccessful. The patient was scheduled for another ERCP 5 days later, which was to be performed by another endoscopist. Upon presentation for the second ERCP, she complained of right calf tenderness lasting for 3 days. Edema and crepitus in that region were palpable on physical examination. These findings were suggestive of subcutaneous emphysema due to either gastrointestinal perforation or bacterial infection (gas gangrene). The patient denied any fever or trauma. During the second ERCP, a 12-mm gallstone was removed from the common bile duct and a 30-French fully covered self-expanding metallic stent (WallFlex Biliary RX, Boston Scientific, Marlborough, Massachusetts, United States) was placed due to a high clinical suspicion of microperforation (Figure 1B and 1C), even though no periampullary leak of contrast was seen. We immediately performed a computed tomography scan of the abdomen and lower extremities. It showed free gas in the abdominal cavity, most visible at the posterior surface of the anterior abdominal wall (a 27-mm thick layer) and around the right kidney—signs of gastrointestinal perforation. Also, numerous small gas bubbles were noted under the skin and under the fascia within the lower right limb almost along its entire length, reaching up to the ankle joint; these were considered features of subcutaneous emphysema (Figure 1D and 1E). The patient was transferred to the surgery department for further treatment. She underwent exploratory laparotomy, but no evident place of the perforation within the duodenum was found, possibly because it partially healed in between the 2 ERCP procedures. The surgery was performed without complications, and the patient was discharged home after a week and scheduled for biliary stent removal in our department in 1 month.

Duodenal perforation during ERCP is a rare complication (<⁠1% of all procedures), but it can be potentially life-threatening, as the in-hospital mortality rate is as high as 20%.¹ Subcutaneous emphysema in the course of ERCP-related duodenal perforation is even less frequently reported; less than 50 cases have been published to date. Cases of subcutaneous emphysema have been previously reported, mainly in the area of the abdominal and chest walls.^2,3 One of the unusual locations described includes the scrotum.^4,5 To our best knowledge, this is the first published case of ERCP-related emphysema of the lower extremity.