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CASE REPORT
Year : 2023  |  Volume : 22  |  Issue : 3  |  Page : 385-387  

Recurrent idiopathic pancreatitis complicating as emphysematous pancreatitis and gastroduodenal artery pseudoaneurysm: A rare case report


Department of Medicine, Sir Ganga Ram Hospital, New Delhi, India

Date of Submission29-Nov-2021
Date of Acceptance17-Dec-2022
Date of Web Publication4-Jul-2023

Correspondence Address:
Vinus Taneja
Room No. 1417, Medicine Office 4th Floor, Sir Ganga Ram Hospital, Rajinder Nagar, New Delhi - 110 060
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/aam.aam_245_21

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   Abstract 


Emphysematous pancreatitis (EP) is a rare and potentially fatal condition of the pancreas. It is associated with gas-forming bacteria and is characterized by the presence of gas in or around the pancreas. It is identified by a computed tomography scan of the abdomen. Although predisposing factors are not precisely known, diabetes mellitus, which predisposes to gas gangrene, is seen to be commonly associated with patients of EP. EP being potentially fatal requires immediate management. Surgery is generally indicated in EP. However, EP can also managed conservatively. In our case, the patient developed recurrent pancreatitis, the cause being idiopathic, and the second episode of acute pancreatitis was complicated by EP and gastroduodenal artery pseudoaneurysm.
Résumé
La pancréatite emphysémateuse (EP) est une condition rare et potentiellement mortelle du pancréas. Il est associé à des bactéries de formation de gaz et se caractérise par la présence de gaz dans ou autour du pancréas. Il est identifié par une tomodensitométrie calculée de l'abdomen. Bien que les facteurs prédisposants ne soient pas précisément connus, le diabète sucré, qui prédispose à la gangrène du gaz, est considéré comme communément associé aux patients du PE. EP étant potentiellement mortel nécessite une gestion immédiate. La chirurgie est généralement indiquée dans EP. Cependant, EP peut également gérer de manière conservatrice. Dans notre cas, le patient a développé une pancréatite récurrente, la cause étant idiopathique, et le deuxième épisode de pancréatite aiguë a été compliqué par l'EP et le pseudo-anévrisme de l'artère gastroduodénale.
Mots-clés: Pancréatite emphysémateuse, pseudo-anévrisme gastroduodénal, pancréatite

Keywords: Emphysematous pancreatitis, gastroduodenal artery pseudoaneurysm, pancreatitis


How to cite this article:
Taneja V, Shah D, Dessai R, Sondhi M, Nautiyal M, Garg A. Recurrent idiopathic pancreatitis complicating as emphysematous pancreatitis and gastroduodenal artery pseudoaneurysm: A rare case report. Ann Afr Med 2023;22:385-7

How to cite this URL:
Taneja V, Shah D, Dessai R, Sondhi M, Nautiyal M, Garg A. Recurrent idiopathic pancreatitis complicating as emphysematous pancreatitis and gastroduodenal artery pseudoaneurysm: A rare case report. Ann Afr Med [serial online] 2023 [cited 2023 Sep 28];22:385-7. Available from: https://www.annalsafrmed.org/text.asp?2023/22/3/385/380154




   Introduction Top


Recurrent acute pancreatitis is defined as more than two attacks of acute pancreatitis without any evidence of underlying chronic pancreatitis.[1] Common causes of recurrent pancreatitis are gallstones, alcohol intake, and idiopathic.[2] Other less common causes are autoimmune diseases, hypertriglyceridemia, hyperparathyroidism, tumors, infections, and drugs.[3] Emphysematous pancreatitis (EP) is a rare and potentially fatal condition of the pancreas.[4] It is associated with gas-forming bacteria and is characterized by the presence of gas in or around the pancreas.[2] It is identified by computed tomography (CT) scan of the abdomen. Although predisposing factors are not precisely known, diabetes mellitus, which predisposes to gas gangrene, is seen to be commonly associated with patients of EP.[5] Only a few scattered cases of EP have been reported to date most of them dealing with radiological features. Gastroduodenal artery (GDA) aneurysm is an extremely rare condition accounting for <1.5% of the total visceral artery aneurysm.[6] GDA aneurysm may be seen as associated with various conditions but chronic pancreatitis is the most common potential risk factor.[7] It is life-threatening with a potential risk of rupture in 20%–80% of cases.[8],[9] EP being potentially fatal requires immediate management. Surgery is generally indicated in EP.[10] However, EP can also managed conservatively.[11],[12] In our case, the patient developed recurrent pancreatitis, the cause being idiopathic, and the second episode of acute pancreatitis was complicated by EP and GDA pseudoaneurysm. EP was managed conservatively. To the best of our knowledge, acute pancreatitis complicating both EP and GDA pseudoaneurysm has not been reported to date.


   Case Report Top


A 36-year-old male was asymptomatic before 4 days when he complained of pain in the abdomen which was located in all quadrants, moderate-to-severe intensity, nonspasmodic, noncolicky, and nonradiating. He also complained of sudden-onset breathlessness for 3 days and high-grade fever for 1 day. On examination, the patient had a temperature of 101° F, pulse rate of 140 beats/min, blood pressure of 140/90 mmHg, and respiratory rate of 30/min. Respiratory system examination revealed decreased air entry at bilateral bases and bilateral basal crepitations. On per abdominal examination, the abdomen was distended with diffuse tenderness in right hypochondrium, left hypochondrium, umbilical, and hypogastrium regions. Neurological and cardiovascular examinations were unremarkable. He had acute pancreatitis 1 year ago. He was diabetic and hypertensive for 2 years. There was no history of alcohol intake or smoking. Family history was insignificant. Investigations revealed hemoglobin 12.4 g/dL, total leukocyte count 11,500/mm3, platelets 102,000/mm3, serum calcium 5.04 mg/dL, serum phosphorus 1.20/dL, aspartate aminotransferase 83 U/L, alanine aminotransferase 50 U/L, serum amylase 517 U/L, serum lipase 387 U/L, serum uric acid 8.50 mg/dL, serum triglycerides 310 mg/dL, and erythrocyte sedimentation rate was 50 mm at the end of the 1st h. CT abdomen showed acute edematous pancreatitis with peripancreatic inflammation, mild ascites, and bilateral pleural effusion (Mortele Modified CT Severity Index-4). He was started on intravenous antibiotics and managed conservatively. With the above line of management, his fever spikes subsided and his clinical condition showed improvement. Repeat serum amylase was 65 U/L and serum lipase was 51 U/L. After 5 days of the afebrile state, he again complained of high-grade fever. A repeat CT scan was done which showed necrosis in the head, uncinate process, and body and tail of the pancreas with associated intrapancreatic and peripancreatic fluid collection with internal air locules (Mortele Modified CT Severity Index-8), suggestive of EP. During the course of the hospital stay, the patient complained of severe abdominal pain and the passage of dark-colored stools. CT angiography abdomen was done which revealed a pseudoaneurysm of GDA. Transcatheter coil embolization was done. With continued antibiotic therapy, fever spikes decreased. EP was managed conservatively with antibiotics. The patient improved symptomatically; hence, he was discharged in stable condition.


   Discussion Top


Acute pancreatitis can be either interstitial or necrotizing. Acute interstitial pancreatitis is more common (70%–80%) and a milder form of disease, whereas acute necrotizing pancreatitis is a less common (20%–30%) and severe form of the disease having a mortality rate of up to 40%.[13] The extent of pancreatic necrosis correlates with organ failure and mortality.[14],[15] Our case is unique as there are very few cases of EP reported in the literature to date. EP is a rare and life-threatening variant of acute severe pancreatitis which is associated with gas-forming bacteria and is characterized by the formation of gas within and around the pancreas.[10],[11] It has been reported in association with emphysematous infections of the gallbladder, kidney, urinary bladder, and intestines.[5] Although there are no definite predisposing factors, it is seen to be occurring in patients of diabetes mellitus and those who are immunosuppressed like tuberculosis in persons with HIV infection.[5] Gas within the pancreas can rarely be seen in patients with a patulous ampulla of Vater, duodenal diverticulum, penetrating duodenal ulcer, or the following instrumentation.[16] A study done by Solanki et al. have reported that hyperglycemia coupled with insulin resistance causes increased production of reactive oxygen species in acinar cells and has a role in predisposing diabetics to acute pancreatitis.[17] Although our patient had hypertriglyceridemia, it was not a predisposing factor for acute pancreatitis as serum levels >1000 mg/dl predispose to pancreatitis.[18]

Second, EP was complicated by GDA aneurysm which is very rare and accounts for 1.5% of total visceral artery aneurysms.[6] GDA aneurysm can be a true aneurysm or pseudoaneurysm. It is commonly associated with chronic pancreatitis.[7] However, association with autoimmune disorders such as Takayasu arteritis or polyarteritis nodosa is also seen.[19] Gastrointestinal hemorrhage secondary to rupture of aneurysm is the most common manifestation seen in 52% of cases.[6] Hence, early diagnosis and intervention are required as it carries a high risk of rupture.[6],[8],[9] It is diagnosed by visceral angiography which is considered the gold standard diagnostic modality. It has the advantage that therapeutic intervention can also be done whenever needed. Hence, it serves both diagnostic as well as therapeutic purposes.[20] It has the utmost sensitivity of 100%, whereas a CT scan is 67% sensitive and ultrasonography is sensitive in 50% of cases. CT scan as compared to visceral angiography has the advantage that it is noninvasive.[6]

Third, our patient had recurrent pancreatitis caused by being idiopathic and the second episode of pancreatitis was complicated by EP and GDA pseudoaneurysm. Patients with acute pancreatitis may develop recurrent pancreatitis if the offending agent has not been removed or eliminated. When the cause of acute pancreatitis is not known it is termed idiopathic pancreatitis. In a study done by Zhang et al. and Gullo et al., it was shown that approximately 27% of the cases of recurrent pancreatitis are idiopathic in origin.[2],[21] As the prognosis of EP is very bad and early radiological detection may improve survival. CT scan is the imaging modality of choice. It is very sensitive and specific for the detection of gas, its anatomical location, and its extent.[16] The association of GDA pseudoaneurysm with EP makes this condition more life-threatening.

Finally, we managed EP conservatively with antibiotics. Treatment of EP usually requires surgical intervention.[10] However, it can also be managed conservatively.[11],[12] Although it carries the risk of high mortality, we managed conservatively as the patient was clinically stable.


   Conclusion Top


EP is a rare life-threatening complication of acute pancreatitis. In our case, we managed EP conservatively with antibiotics. Treatment of EP usually requires surgical intervention.[10] GDA aneurysm is a rare but potentially fatal complication of pancreatitis. Mortality and multiorgan failure in EP can be prevented if diagnosed early. Although surgery is generally indicated in EP, it can also be managed conservatively if the patient's clinical condition is stable.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

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Kedia S, Dhingra R, Garg PK. Recurrent acute pancreatitis: An approach to diagnosis and management. Trop Gastroenterol 2013;34:123-35.  Back to cited text no. 1
    
2.
Zhang W, Shan HC, Gu Y. Recurrent acute pancreatitis and its relative factors. World J Gastroenterol 2005;11:3002-4.  Back to cited text no. 2
    
3.
Mitchell RM, Byrne MF, Baillie J. Pancreatitis. Lancet 2003;361:1447-55.  Back to cited text no. 3
    
4.
Birgisson H, Stefánsson T, Andresdóttir A, Möller PH. Emphysematous pancreatitis. Eur J Surg 2001;167:918-20.  Back to cited text no. 4
    
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Grayson DE, Abbott RM, Levy AD, Sherman PM. Emphysematous infections of the abdomen and pelvis: A pictorial review. Radiographics 2002;22:543-61.  Back to cited text no. 5
    
6.
Habib N, Hassan S, Abdou R, Torbey E, Alkaied H, Maniatis T, et al. Gastroduodenal artery aneurysm, diagnosis, clinical presentation and management: A concise review. Ann Surg Innov Res 2013;7:4.  Back to cited text no. 6
    
7.
White AF, Baum S, Buranasiri S. Aneurysms secondary to pancreatitis. AJR Am J Roentgenol 1976;127:393-6.  Back to cited text no. 7
    
8.
Grego FG, Lepidi S, Ragazzi R, Iurilli V, Stramanà R, Deriu GP. Visceral artery aneurysms: A single center experience. Cardiovasc Surg 2003;11:19-25.  Back to cited text no. 8
    
9.
Gabelmann A, Görich J, Merkle EM. Endovascular treatment of visceral artery aneurysms. J Endovasc Ther 2002;9:38-47.  Back to cited text no. 9
    
10.
Ghidirim G, Gagauz I, Mişin I, Guţu E, Vozian M. Emphysematous necrotizing pancreatitis. Chirurgia (Bucur) 2005;100:293-6.  Back to cited text no. 10
    
11.
Ku YM, Kim HK, Cho YS, Chae HS. Medical management of emphysematous pancreatitis. J Gastroenterol Hepatol 2007;22:455-6.  Back to cited text no. 11
    
12.
Kvinlaug K, Kriegler S, Moser M. Emphysematous pancreatitis: A less aggressive form of infected pancreatic necrosis? Pancreas 2009;38:667-71.  Back to cited text no. 12
    
13.
Forsmark CE, Baillie J, AGA Institute Clinical Practice and Economics Committee, AGA Institute Governing Board. AGA institute technical review on acute pancreatitis. Gastroenterology 2007;132:2022-44.  Back to cited text no. 13
    
14.
Garg PK, Madan K, Pande GK, Khanna S, Sathyanarayan G, Bohidar NP, et al. Association of extent and infection of pancreatic necrosis with organ failure and death in acute necrotizing pancreatitis. Clin Gastroenterol Hepatol 2005;3:159-66.  Back to cited text no. 14
    
15.
Isenmann R, Rau B, Beger HG. Bacterial infection and extent of necrosis are determinants of organ failure in patients with acute necrotizing pancreatitis. Br J Surg 1999;86:1020-4.  Back to cited text no. 15
    
16.
Wig JD, Kochhar R, Bharathy KG, Kudari AK, Doley RP, Yadav TD, et al. Emphysematous pancreatitis. Radiological curiosity or a cause for concern? JOP 2008;9:160-6.  Back to cited text no. 16
    
17.
Solanki NS, Barreto SG, Saccone GT. Acute pancreatitis due to diabetes: The role of hyperglycaemia and insulin resistance. Pancreatology 2012;12:234-9.  Back to cited text no. 17
    
18.
Berglund L, Brunzell JD, Goldberg AC, Goldberg IJ, Sacks F, Murad MH, et al. Evaluation and treatment of hypertriglyceridemia: An Endocrine Society clinical practice guideline. J Clin Endocrinol Metab 2012;97:2969-89.  Back to cited text no. 18
    
19.
Tulsyan N, Kashyap VS, Greenberg RK, Sarac TP, Clair DG, Pierce G, et al. The endovascular management of visceral artery aneurysms and pseudoaneurysms. J Vasc Surg 2007;45:276-83.  Back to cited text no. 19
    
20.
Yeh TS, Jan YY, Jeng LB, Hwang TL, Wang CS, Chen MF. Massive extra-enteric gastrointestinal hemorrhage secondary to splanchnic artery aneurysms. Hepatogastroenterology 1997;44:1152-6.  Back to cited text no. 20
    
21.
Gullo L, Migliori M, Pezzilli R, Oláh A, Farkas G, Levy P, et al. An update on recurrent acute pancreatitis: Data from five European countries. Am J Gastroenterol 2002;97:1959-62.  Back to cited text no. 21
    




 

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