Clin J Gastroenterol DOI 10.1007/s12328-013-0408-8

CASE REPORT

Air in the main pancreatic duct associated with a pancreatic intraductal papillary mucinous neoplasm Satoshi Yamamoto • Kazuo Inui • Junji Yoshino Hironao Miyoshi • Takashi Kobayashi



Received: 20 February 2013 / Accepted: 15 July 2013 Ó Springer Japan 2013

Abstract A 62-year-old man was referred to our hospital after ultrasonographic mass screening detected a pancreatic cyst that proved to be an intraductal papillary mucinous neoplasm. Computed tomography additionally demonstrated air in the main pancreatic duct. Magnetic resonance cholangiopancreatography and endoscopic retrograde cholangiopancreatography delineated a filling defect in the main pancreatic duct in the body of the pancreas. The sphincter of Oddi was open. The main pancreatic duct was dilated by viscous mucin; air in the duct was attributed to consequent dysfunction of the sphincter. Laboratory findings included no significant abnormality. The patient has remained asymptomatic during follow-up. Of 25 previously reported cases with air in the duct, only 1 involved an intraductal papillary mucinous neoplasm. Keywords Intraductal papillary mucinous neoplasm  Air  Main pancreatic duct  Ultrasonography  Mass screening

Introduction Air is found less frequently in the main pancreatic duct (MPD) than in the biliary tract (pneumobilia). The most frequently suspected etiology of air in the pancreatic duct has been acute pancreatitis. We encountered a patient with

S. Yamamoto (&)  K. Inui  J. Yoshino  H. Miyoshi  T. Kobayashi Department of Gastroenterology, Second Teaching Hospital, Fujita Health University, 3-6-10 Otobashi, Nakagawa-ku, Nagoya 454-8509, Japan e-mail: [email protected]

air in the MPD associated with an intraductal papillary mucinous neoplasm (IPMN) initially detected by ultrasonographic mass screening. We found only 1 previously reported case of air in the MPD with this associated condition.

Case report A 62-year-old man was referred to our hospital because of a cystic lesion of the pancreas detected during ultrasonographic cancer screening in September 2007. No significant symptoms were present, and he had no history of cholelithiasis. On physical examination, the patient was afebrile, non-anemic, and non-icteric. His abdomen was soft, flat, and nontender. Laboratory values were slightly low or within normal limits, including serum amylase (28 IU/L; normal range 30–120 IU/L), carcionoembryonic antigen (0.9 ng/dL) and carbohydrate antigen 19–9 (11.4 U/mL). Abdominal ultrasonography disclosed a unilocular cyst, 14 mm in diameter, in the head of the pancreas and a strong echo with acoustic shadowing in the MPD in the body of the pancreas (Fig. 1). Computed tomography (CT) delineated the cyst and showed air in the MPD in the body of the pancreas (Fig. 2). No pneumobilia was detected. Magnetic resonance cholangiopancreatography (MRCP) demonstrated dilation of the branch duct in the head of the pancreas and a defect in the MPD in the body of the pancreas (Fig. 3). When endoscopic retrograde cholangiopancreatography (ERCP) was performed on an inpatient basis, dilation of the orifice of the duodenal papilla and abundant viscous fluid were seen. In the head of the pancreas, the pancreatic duct was 9 mm in diameter; in the body, it was 7 mm. Pancreatic juice was not examined cytologically.

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Clin J Gastroenterol

Fig. 1 Ultrasonographic findings. A cyst, 14 mm in diameter, is present in the head of the pancreas (panel a, arrowhead). Strong echoes with acoustic shadowing are seen in the main pancreatic duct in the body of the pancreas (panel b, arrows)

component. Obstructive jaundice in a patient with a cystic lesion in the head of the pancreas, an enhancing solid component of the cyst, or MPD diameter of C10 mm would indicate a high risk of malignant tumor and need for surgery according to current revised guidelines [1]. In summary, we diagnosed our patient with branch-type IPMN. We could not identify air in the MPD by ERCP (Fig. 4). A CT performed 7 months later showed a small amount of air in the MPD (Fig. 5). Another CT performed 18 months after diagnosis demonstrated more air in the duct (Fig. 6).

Discussion Fig. 2 Computed tomography demonstrates air in the main pancreatic duct in the body of the pancreas (arrow)

Fig. 3 Magnetic resonance cholangioportography demonstrates dilation of the duct branch in the head of the pancreas, and a filling defect in the main pancreatic duct in the body of the pancreas (arrow)

In this asymptomatic patient IPMN was not an indication for surgery, because MPD diameter was \10 mm and the cyst did not have any enhancing solid

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We searched for cases of air in the pancreatic duct reported from 1949 to 2012, using PubMed with ‘air’ and ‘pancreas’ as key words. The 25 cases identified [2–14] are summarized in Table 1 together with our own, totaling 26 cases. The median age was 46 years (range 40-75). The maleto-female ratio was 1.3:1. The most frequently suspected etiology of air in the duct was pancreatitis (10 patients, including 7 with acute pancreatitis and 3 with chronic pancreatitis). Acute cholangitis and/or cholelithiasis was considered etologic in 8 patients. Multiple possible etiologies were present in some patients. When present, acute symptoms required immediate diagnosis. Peer et al. [9] reported 3 cases of air in the MPD associated with duodenal obstruction, suggesting that air in the MPD should provoke suspicion of duodenal obstruction, given that congenital duodenal obstruction is commonly accompanied with abnormality of the sphincter of Oddi. Costa et al. [7] reported 11 patients with air in the pancreatic duct; among them, 91 % had a history

Clin J Gastroenterol

Fig. 4 Endoscopic findings. In panel a, duodenoscopy displays a widely open orifice of the papilla of Vater. In panel b, endoscopic retrograde cholangiopancreatography shows dilation of the main pancreatic duct, but detects no air

Fig. 5 Approximately 7 months later, computed tomography shows decreased air in the main pancreatic duct

Fig. 6 Computed tomography approximately 18 months after diagnosis shows increased air in the main pancreatic duct

of liver and/or pancreatic disease (gallstones with cholecystitis, cholangitis, chronic pancreatitis, or acute pancreatitis). In many cases, pancreatitis was part of the clinical picture. However, the literature does not clearly link pancreatitis with air in the MPD by proposing a mechanism responsible for such a connection. In a single case, pancreatic duct sphincterotomy performed for chronic pancreatitis might have allowed air to enter the duct [9]. Apart from our patient, air in the MPD was associated with IPMN in only 1 case, reported by Itai et al. [3]. We believe that air in the MPD in our patient with IPMN resulted from dysfunction of the sphincter of Oddi, which became patulous when the MPD was distended by viscous mucin produced by the tumor. Under such conditions, duodenal air would enter the MPD when intraduodenal pressure increased for any reason. Presence of air could vary in the MPD; air had decreased according to CT performed 7 months after initial evaluation. This variability could account for no finding of air by endoscopic retrograde cholangiopancreatography in our patient. Among patients with IPMN, air in the MPD should be a rare observation because a collection of tumor-derived mucin sufficient to interfere with sphincter function would need to coincide with elevated intraduodenal pressure. One might expect associated pneumobilia in a patient such as ours. However, among 13 patients with air in the MPD due to dysfunction of the sphincter of Oddi, 5 did not have pneumbilia, like our patient [2, 3, 6, 11]. Our experience suggests that IPMN should be considered in the differential diagnosis of asymptomatic air in the MPD, and that such air usually could be managed by follow-up with serial imaging.

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Clin J Gastroenterol Table 1 Reported cases of air in the main pancreatic duct Case

Report year

Author

Age/sex

Symptom

Primary disease

Cause of air image

1

1949

Long et al. [2]

68 years/F

Right upper quadrant pain

Calculi in the common duct

Pancreatcbiliary fistula

2

1986

Itai et al. [3]

75 years/M

No complaining

IPMN

Patulous papilla of Vater

3 4

1986 1986

Braver et al. [4] Radin et al. [5]

62 years/M 4 years/F

Abdominal pain Fever enlarged liver

Chronic pancreatitis/C Biliary ascariasis

After endoscopic sphincterotomy Worms migration

5

1987

Hughes et al. [6]

41 years/M

Hematemesis

Gastric ulcer

Pancreatic fistula

6

1991

Costa and Righetti [7]

57 years/F

Biliary colic

Cholelithiasis/acute pancreatitis

C/LLCD

7

1991

Costa and Righetti [7]

3S years/M

Biliary colic

Cholelithiasis/acute pancreatitis

C/LLCD

8

1991

Costa and Righetti [7]

66 years/F

Biliary colic

Cholelithiasis

C/after endoscopic sphincterotomy

9

1991

Costa and Righetti [7]

51 years/M

Unknown

Acute pancreatitis

PC J7 LLCD

10

1991

Costa and Righetti [7]

73 years/M

None

None

Patulous papilla of Vater/ peripapillary’polyp/PD dilatation

11

1991

Costa and Righetti [7]

66 years/M

Unknown

Acute cholangitis

Inflammatory papilla of Vater

12

1991

Costa and Righetti [7]

72 years/M

Unknown

Chronic pancreatitis/C

Patulous and enlarged papilla of Vater/PD dilatation

13

1991

Costa and Righetti [7]

27 years/F

Unknown

Cholangitis

CCR/HJ

14

1991

Costa and Righetti [7]

44 years/F

Biliary colic

Cholelithiasis

C/after endoscopic sphincterotomy

15

1991

Costa and Righetti [7]

45 years/M

Abdominal pain

Acute pancreatitis

Pancreatic duct non dilatation

16

1991

Costa and Righetti [7]

72 years/M

Abdominal pain

Chronic pancreatitis

Moderate papillary stenosis PD dilatation

17

1993

England et al. [8]

57 years/F

Epigastric pain

Acute pancreatitis/C

Injected during ERCP

18

2002

Peer et al. [9]

5 days/F

Vomiting

Duodenal obstruction

Refluxed bowel gas

19 20

2002 2002

Peer et al. [9] Peer et al. [9]

9 months/M 4 days/M

Vomiting Vomiting

Duodenal obstruction Duodenal obstruction

Refluxed bowel gas Refluxed bowel gas

21

2004

Abeygunasekera et al. [10]

29 years/F

Epigastric pain/diarrhea

Gastric ulcer

Pancreatic fistula

22

2007

Saperas et al. [11]

50 years/F

Epigastric/blood in stool

Duodenal ulcer

Pancreatic fistula

23

2008

Brindisi et al. [12]

53 years/M

Upper abdominal pain/ weight loss

Acute pancreatitis

Patulous papilla of Vater

24

2009

Tyagi et al. [13]

34 years/F

Abdominal discomfort

Acute biliary pancreatitis pseudocvst

No mention

25

2011

Our study

62 years/M

None

IPMN

Patulous papilla of Vater

26

2012

Kim et al. [14]

54 years/F

None

None

Innocent

Pt patient, IPMN intraductal papillary mucinous neoplasm, C cholecystectomy, LLCD laterolateral choledochojejunostomy, PCJ pancreaticocystjejunostomy, PD pancreatic duct, CCR choledochal cyst resection, HJ hepaticojejunostomy, ERCP endoscopic retrograde cholangiopancreatography Conflict of interest of interest.

The authors declare that they have no conflict

References 1. Tanaka M, Ferna´ndez-del Castillo C, Adsay V, Chari S, Falconi M, Jang JY, et al. International consensus guidelines 2012 for the

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management of IPMN and MCN of the pancreas. Pancreatology. 2012;12:183–97. 2. Long L, Evana AL, Blaine BC. Common duct calculus simulated by air bubble in pancreatic duct. South Surg. 1949;15: 501–4. 3. Itai Y, Ohtomo K, Kokubo T, Nagai H, Atomi Y, Kuroda A. CT demonstration of gas in dilated pancreatic duct. J Comput Assist Tomogr. 1986;10:1052–3.

Clin J Gastroenterol 4. Braver JM, Jones TB, Brooks JR. The sonographic appearance of the pancreatic duct following transampullary septectomy. J Ultrasound Med. 1986;5:459–60. 5. Radin DR, Vachon LA. CT findings in biliary and pancreatic ascariasis. J Comput Assist Tomogr. 1986;10:508–9. 6. Hughes JJ, Blunck CE. CT demonstration of gastropancreatic fistula due to penetrating gastric ulcer. J Comput Assist Tomogr. 1987;11:709–11. 7. Costa PL, Righetti G. Air in the main pancreatic duct: demonstration with US. Radiology. 1991;181:801–3. 8. England R, Schutz SM, Leung JW. Case of the month: ‘‘air in a view’’. Br J Radiol. 1996;69:681–2. 9. Peer S, Kiechl-Kohlendorfer U, Gassner I. Air in the main pancreatic duct revealed by abdominal ultrasound: an additional diagnostic sign in paediatric patients with duodenal obstruction. Clin Radiol. 2002;57:945–8.

10. Abeygunasekera S, Freiman J, Engelman J, Glenn D, Craig P. Gastropancreaticocolic fistula: complication of a benign ulcer. Gastrointest Endosc. 2004;59:450–2. 11. Saperas E, Miranda A, Armengol JR, Malagelada JR. Pneumopancreatogram after injection therapy for bleeding duodenal ulcer. Endoscopy. 2007;39 Suppl 1:E277 (Epub 2007 Oct 24). 12. Brindisi C, Calculli L, Casadei R, Pezzilli R. Air in the Wirsung duct. An unusual finding. JOP. 2008;9:534–7. 13. Tyagi P, Puri AS, Sachdeva S. Spontaneous air in the main pancreatic duct. Indian J Gastroenterol. 2009;28:87. 14. Kim YJ, Kim HK, Cho YS, Kim SS, Chae HS, Kim SK, et al. Air in the main pancreatic duct: a case of innocent air. World J Gastroenterol. 2012;18:5142–4.

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Air in the main pancreatic duct associated with a pancreatic intraductal papillary mucinous neoplasm.

A 62-year-old man was referred to our hospital after ultrasonographic mass screening detected a pancreatic cyst that proved to be an intraductal papil...
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