Top

Published in:

01-09-2005 | Case Report

Chronic pancreatitis with pancreaticolithiasis and pseudocyst in a 5-year-old boy with homozygous SPINK1 mutation

Authors: Axel C. Kühn, Niels Teich, Karel Caca, Anne Limbach, Wolfgang Hirsch

Published in: Pediatric Radiology | Issue 9/2005

Abstract

We report a 5-year-old boy with a 5-month history of symptoms owing to chronic pancreatitis. Abdominal imaging revealed a large pseudocyst in the pancreatic tail and concretions in the main pancreatic duct. Successful endoscopic papillotomy and stent implantation were performed. Genetic testing showed homozygous SPINK1-N34S mutation, which is an established risk factor for chronic pancreatitis.

Queizan A, Hernandez F, Rivas S (2003) Pancreatic pseudocyst caused by valproic acid: case report and review of the literature. Eur J Pediatr Surg 13:60–62CrossRefPubMed

Jeong JB, Whang JH, Ryu JK, et al (2004) Risk factors for pancreatitis in patients with anomalous union of pancreatobiliary duct. Hepatogastroenterology 51:1187–1190PubMed

Werlin SL, Kugathasan S, Frautschy BC (2003) Pancreatitis in children. J Pediatr Gastroenterol Nutr 37:591–595PubMed

Millar AJ, Rode H, Stunden RJ, et al (1988) Management of pancreatic pseudocysts in children. J Pediatr Surg 23:122–127PubMed

Asano T, Matsumoto T, Zhi CL, et al (2003) Solid and cystic tumor of the pancreas in a 12-year-old boy. Pediatr Int 45:339–341CrossRefPubMed

Illert B, Luhrs H, Timmermann W, et al (2003) Solid-pseudopapillary tumor of the pancreas as differential diagnosis in pancreatic tumors of the young. Zentralbl Chir 128:438–442CrossRefPubMed

Whitcomb DC, Gorry MC, Preston RA, et al (1996) Hereditary pancreatitis is caused by a mutation in the cationic trypsinogen gene. Nat Genet 14:141–145CrossRefPubMed

Witt H, Luck W, Becker M, et al (2001) Mutation in the SPINK1 trypsin inhibitor gene, alcohol use, and chronic pancreatitis. JAMA 285:2716–2717CrossRef

Witt H, Luck W, Hennies HC, et al (2000) Mutations in the gene encoding the serine protease inhibitor, Kazal type 1, are associated with chronic pancreatitis. Nat Genet 25:213–216CrossRefPubMed

10.

Chandak GR, Idris MM, Reddy DN, et al (2002) Mutations in the pancreatic secretory trypsin inhibitor gene (PSTI/SPINK1) rather than the cationic trypsinogen gene (PRSS1) are significantly associated with tropical calcific pancreatitis. J Med Genet 39:347–351CrossRefPubMed

11.

Chandak GR, Idris MM, Reddy DN, et al (2004) Absence of PRSS1 mutations and association of SPINK1 trypsin inhibitor mutations in hereditary and non-hereditary chronic pancreatitis. Gut 53:723–728CrossRefPubMed

12.

Haddock G, Coupar G, Youngson GG, et al (1994) Acute pancreatitis in children: a 15-year review. J Pediatr Surg 29:719–722CrossRef

13.

Kouchi K, Tanabe M, Yoshida H, et al (1999) Nonoperative management of blunt pancreatic injury in childhood. J Pediatr Surg 34:1736–1739CrossRefPubMed

14.

Burnweit C, Wesson D, Stringer D (1990) Percutaneous drainage of traumatic pancreatic pseudocysts in children. J Trauma 30:1273–1277PubMed

15.

Karaguzel G, Senocak ME, Buyukpamukcu N, et al (1995) Surgical management of the pancreatic pseudocyst in children: a long-term evaluation. J Pediatr Surg 30:777–780CrossRefPubMed

16.

Jaffe RB, Arata JA Jr, Matlak ME (1989) Percutaneous drainage of traumatic pancreatic pseudocysts in children. AJR 152:591–595PubMed

17.

Pitchumoni CS, Agarwal N (1999) Pancreatic pseudocysts. When and how should drainage be performed? gastroenterol Clin North Am 28:615–639PubMed

18.

Bridoux-Henno L, Dabadie A, Rambeau M, et al (2004) Successful endoscopic drainage of a pancreatic pseudocyst in a 17-month-old boy. Eur J Pediatr 163:482–484CrossRefPubMed

19.

Breckon V, Thomson SR, Hadley GP (2001) Internal drainage of pancreatic pseudocysts in children using an endoscopically-placed stent. Pediatr Surg Int 17:621–623CrossRefPubMed

20.

Haluszka O, Campbell A, Horvath K (2002) Endoscopic management of pancreatic pseudocyst in children. Gastrointest Endosc 55:128–131CrossRefPubMed

21.

Patty I, Kalaoui M, Al Shamali M, et al (2001) Endoscopic drainage for pancreatic pseudocyst in children. J Pediatr Surg 36:503–505CrossRefPubMed

22.

Kimble RM, Cohen R, Williams S (1999) Successful endoscopic drainage of a posttraumatic pancreatic pseudocyst in a child. J Pediatr Surg 34:1518–1520CrossRefPubMed

Title: Chronic pancreatitis with pancreaticolithiasis and pseudocyst in a 5-year-old boy with homozygous SPINK1 mutation
Authors: Axel C. Kühn
Niels Teich
Karel Caca
Anne Limbach
Wolfgang Hirsch
Publication date: 01-09-2005
Publisher: Springer-Verlag
Published in: Pediatric Radiology / Issue 9/2005
Print ISSN: 0301-0449
Electronic ISSN: 1432-1998
DOI: https://doi.org/10.1007/s00247-005-1477-8

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Chronic pancreatitis with pancreaticolithiasis and pseudocyst in a 5-year-old boy with homozygous SPINK1 mutation

Abstract

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Please log in to get access to this content

Other articles of this Issue 9/2005

Desmoplastic fibroma of the mandible in an infant

Juvenile intraosseous gout of the calcaneus

Role of MRI in the management of children with diffuse pontine tumors: a study of 15 patients and review of the literature

Hermes - September 2005

The CT appearances of delayed amniotic fluid clearance from the lungs in an infant with absent pulmonary valve and congenital lobar emphysema

CME questions - September 2005