Ağustos 2013

55-57

Gülten CAN SEZGİN¹, Eylem SEVİNÇ ² , Alper YURCİ ¹, Duran ARSLAN ² , Şebnem GÜRSOY ¹

Departments of ¹Gastroenterology and ²Pediatric Gastroenterology, Erciyes University, School of Medicine, Kayseri

Çocuklarda pankreatik kanalın, doğumsal veya edinsel darlığının bir sonucu olarak obstrüktif kronik pankreatit ortaya çıkar. Belirgin histolojik değişiklikler periduktal fibrozis ve geri kalan duktal alanda genişlemelerle karakterizedir. Tanıda genellikle endoskopik retrograd kolanjiopankreatikografi, ultrasonografi ve pankreatik fonksiyon testleri gibi yöntemler kullanılmaktadır. Eozinofili sıklıkla alerjik rinit, astım, parazitik enfeksiyonlar ve ilaç reaksiyonu ile ilişkilidir. Seyrek olarak da kronik obstrüktif pankreatik hastalıklarda tarif edilmiştir. Bu makalede; karın ağrısı ile başvuran kronik obstrüktif pankreatitli bir olguyu sunuyoruz. Olgumuzda, pankreatit semptomları ve eozinofili pankreasa stent yerleştirilmesi ile düzelmiştir.

Kronik pankreatit, eozinofili, çocuk

Chronic pancreatitis is a chronic inflammatory disease of the pancreas characterized by irreversible morphological changes that are typically associated with pain and/or loss of function. In chronic obstructive pancreatitis, the prominent histologic changes are periductal fibrosis and subsequent ductal dilatation. A variety of factors are implicated in chronic obstructive pancreatitis, including ductal obstruction due to ampullary stenosis, inflammatory or neoplastic causes, surgical ductal ligation, and fibrosis due to a pseudocyst as a complication of an episode of acute pancreatitis. Endoscopic retrograde cholangiography (ERCP) is considered by most specialists to be the first-line treatment modality for management of obstructive chronic pancreatitis (1,2).

Chronic pancreatitis is commonly defined as a continuous, inflammatory process of the pancreas, characterized by irreversible morphologic changes. This chronic inflammation can lead to chronic abdominal pain and/or impairment of endocrine and exocrine functions of the pancreas. In children, chronic pancreatitis generally originates from genetic mutations and congenital abnormalities of pancreatic and bile ducts (3). Prevalence of chronic pancreatitis is difficult to detect and varies between 0.04% and 5% of the adult population (4,5). There are only limited data concerning the frequency of eosinophilia with chronic pancreatitis (6,7). Chronic pancreatitis is characterized by parenchymal fibrosis, reduction in the number and size of asinuses, and varying sized dilatations of the pancreatic duct following triggers such as ductal obstructions, oxidative stress, genetic tendency, and metabolic abnormalities. Langerhans cells relatively reduce, and acinar loss is a stable indicator (8). Genetic, metabolic, obstructive, autoimmune, and idiopathic causes are related to chronic pancreatitis (9). Obstructive chronic pancreatitis is a process that develops under conditions such as ductal stones in the main pancreatic channel, duodenal wall cyst, and scarring or stenosis of the major/minor papilla. It is reported that the removal of the obstruction leads to the disappearance of the damage to the pancreatic tissue (10). Diagnosis of chronic pancreatitis is possible by clinical findings, tests and imaging processes that indicate exocrine function. An early diagnosis of chronic pancreatitis may prevent any further damage to the gland. Findings after the imaging of the gland and exocrine function tests may or may not be compatible (11,12). In our case, exocrine function tests, serum amylase and lipase values were within normal range, and fat and reductants in the stool were negative; however, ERCP detected a diffusely dilated pancreatic duct with local saccular expansions as well as a proximal stenotic segment. ERCP findings supported the diagnosis of chronic obstructive pancreatitis. Although there is no specific laboratory test for chronic obstructive pancreatitis, it is possible to observe some increase in amylase and lipase during the acute inflammatory attacks of the disease. After the atrophy and fibrosis of the pancreatic parenchyma, serum amylase and lipase values tend to be normal and even low. It is possible to observe moderate eosinophilia pancreatitis and autoimmune pancreatitis (6,13,14). Macrophage-derived cytokines in pancreatic duct obstruction are thought to be caused by an increase in the number of blood eosinophils (15). It is important to note that there was a moderate eosinophilia during the first visit of our patient. For the differential diagnosis of eosinophilia, it is important to rule out parasitic, allergic, and autoimmune causes. In our patient, stool parasite and serum Fasciola IFAT levels were negative, and serum IgE and IgG4 levels were within normal range. During the treatment, pain reduction is the primary goal. During this process, endoscopic and/or surgical interventions are done in order to prevent possible pancreatic deficiency. If the obstruction causes pancreatic damage, removal of the obstruction may lead to the full recovery of the pancreas (6). As indicated in the ERCP, our case had diffuse enlargement and saccular expansions, and therefore, pancreatic and biliary sphincterotomy was applied, and a 5F plastic pancreatic stent was implanted. Three weeks later, the follow-up USG indicated that the diameter of the pancreatic duct was normal, and the stent was removed. The follow-up blood count revealed that the eosinophil number had reduced to within normal range. In our case, the decrease in eosinophilia following the removal of the pancreatic duct obstruction is noteworthy. In this article, we present the case of a child, who first had the complaint of abdominal pain, and was eventually diagnosed with chronic obstructive pancreatitis accompanied by eosinophilia based on clinical, biochemical and imaging tests. The case is especially noteworthy as there are no similar cases reported in the literature. In conclusion, transient eosinophilia with chronic obstructive pancreatitis in children is rare. Chronic obstructive pancreatitis is probably an organic cause of abdominal pain and eosinophilia in children.

Chronic pancreatitis is commonly defined as a continuous, inflammatory process of the pancreas, characterized by irreversible morphologic changes. This chronic inflammation can lead to chronic abdominal pain and/or impairment of endocrine and exocrine functions of the pancreas. In children, chronic pancreatitis generally originates from genetic mutations and congenital abnormalities of pancreatic and bile ducts (3). Prevalence of chronic pancreatitis is difficult to detect and varies between 0.04% and 5% of the adult population (4,5). There are only limited data concerning the frequency of eosinophilia with chronic pancreatitis (6,7). Chronic pancreatitis is characterized by parenchymal fibrosis, reduction in the number and size of asinuses, and varying sized dilatations of the pancreatic duct following triggers such as ductal obstructions, oxidative stress, genetic tendency, and metabolic abnormalities. Langerhans cells relatively reduce, and acinar loss is a stable indicator (8). Genetic, metabolic, obstructive, autoimmune, and idiopathic causes are related to chronic pancreatitis (9). Obstructive chronic pancreatitis is a process that develops under conditions such as ductal stones in the main pancreatic channel, duodenal wall cyst, and scarring or stenosis of the major/minor papilla. It is reported that the removal of the obstruction leads to the disappearance of the damage to the pancreatic tissue (10). Diagnosis of chronic pancreatitis is possible by clinical findings, tests and imaging processes that indicate exocrine function. An early diagnosis of chronic pancreatitis may prevent any further damage to the gland. Findings after the imaging of the gland and exocrine function tests may or may not be compatible (11,12). In our case, exocrine function tests, serum amylase and lipase values were within normal range, and fat and reductants in the stool were negative; however, ERCP detected a diffusely dilated pancreatic duct with local saccular expansions as well as a proximal stenotic segment. ERCP findings supported the diagnosis of chronic obstructive pancreatitis. Although there is no specific laboratory test for chronic obstructive pancreatitis, it is possible to observe some increase in amylase and lipase during the acute inflammatory attacks of the disease. After the atrophy and fibrosis of the pancreatic parenchyma, serum amylase and lipase values tend to be normal and even low. It is possible to observe moderate eosinophilia pancreatitis and autoimmune pancreatitis (6,13,14). Macrophage-derived cytokines in pancreatic duct obstruction are thought to be caused by an increase in the number of blood eosinophils (15). It is important to note that there was a moderate eosinophilia during the first visit of our patient. For the differential diagnosis of eosinophilia, it is important to rule out parasitic, allergic, and autoimmune causes. In our patient, stool parasite and serum Fasciola IFAT levels were negative, and serum IgE and IgG4 levels were within normal range. During the treatment, pain reduction is the primary goal. During this process, endoscopic and/or surgical interventions are done in order to prevent possible pancreatic deficiency. If the obstruction causes pancreatic damage, removal of the obstruction may lead to the full recovery of the pancreas (6). As indicated in the ERCP, our case had diffuse enlargement and saccular expansions, and therefore, pancreatic and biliary sphincterotomy was applied, and a 5F plastic pancreatic stent was implanted. Three weeks later, the follow-up USG indicated that the diameter of the pancreatic duct was normal, and the stent was removed. The follow-up blood count revealed that the eosinophil number had reduced to within normal range. In our case, the decrease in eosinophilia following the removal of the pancreatic duct obstruction is noteworthy. In this article, we present the case of a child, who first had the complaint of abdominal pain, and was eventually diagnosed with chronic obstructive pancreatitis accompanied by eosinophilia based on clinical, biochemical and imaging tests. The case is especially noteworthy as there are no similar cases reported in the literature. In conclusion, transient eosinophilia with chronic obstructive pancreatitis in children is rare. Chronic obstructive pancreatitis is probably an organic cause of abdominal pain and eosinophilia in children.

1. Krishnaveni J, Devanand B, Harshavardhan TS, et al. Chronic pancreatitis presenting with pseudocyst of pancreas and pseudo-aneurysm of hepatic artery. Indian J Pediatr 2012; 79: 952–4. 2. Shyam V, Jessica TM. Review of EUS-guided pancreatic duct drainage. Gastrointest Endosc 2009; 69: 1. 3. Mehta DI. Acute and chronic pancreatitis in childhood. Indian J Pediatr 1999; 66: S81. 4. Cheng CL, Fogel EL, Sherman S, et al. Diagnostic and therapeutic endoscopic retrograde cholangiopancreatography in children: a large series report. J Pediatr Gastroenterol Nutr 2005; 41: 445-53. 5. Sultan M, Werlin S, Venkatasubramani N. Genetic prevalence and characteristics in children with recurrent pancreatitis. J Pediatr Gastroenterol Nutr 2012; 54: 645-50. 6. Wang Q, Lu CM, Guo T, et al. Eosinophilia associated with chronic pancreatitis. Pancreas 2009; 38: 149-53. 7. Fred H. Transient eosinophilia associated with pancreatitis and pseudocyst formation. Arc Intern Med. 1980; 140: 1099-100. 8. Steer ML, Waxman I, Freedman S. Chronic pancreatitis. N Engl J Med 1995; 332: 1482. 9. Etemad B, Whitcomb DC. Chronic pancreatitis: diagnosis, classification, and new genetic developments. Gastroenterology 2001; 120: 682. 10. Milovic V, Wehrmann T, Dietrich CF, et al. Extracorporeal shock wave lithotripsy with a transportable mini-lithotripter and subsequent endoscopic treatment improves clinical outcome in obstructive calcific chronic pancreatitis. Gastrointest Endosc 2011; 74: 1294-9. 11. Wang W, Liao Z, Li ZS, et al. Chronic pancreatitis in Chinese children: etiology, clinical presentation and imaging diagnosis. J Gastroenterol Hepatol 2009; 24: 1862. 12. Kahl S, Glasbrenner B, Leodolter A, et al. EUS in the diagnosis of early chronic pancreatitis: a prospective follow-up study. Gastrointest Endosc 2002; 55: 507-11. 13. Sah RP, Pannala R, Zhang L, et al. Eosinophilia and allergic disorders in autoimmune pancreatitis. Am J Gastroenterol 2010; 105: 2485-91. 14. Long SS, Ann-Christine N. Eosinophils and eosinophilia. In: Long SS, Pickering LK, editors. Long: Principles and Practice of Pediatric Infectious Diseases, Revised Reprint. 3rd ed. Philadelphia: Elsevier Inc., 2009; 1368-81. 15. Günter K. Chronic pancreatitis, pseudotumors and other-like lesions. Modern Pathol. 2007; 20: 113-31.