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ERCP: IDIOPATHIC FIBROSING PANCREATITIS скачать в хорошем качестве

ERCP: IDIOPATHIC FIBROSING PANCREATITIS 5 лет назад

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ERCP: IDIOPATHIC FIBROSING PANCREATITIS
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ERCP: IDIOPATHIC FIBROSING PANCREATITIS

Here is a case that you may not see very often (in fact less than 100 cases have been described in the world literature). A 19 year male, who was previously well, presented with a 5 week history of loose bowel (bowels opening 3-4x/day, no blood) and a 3 week history of jaundice, dark urine, nausea, vomiting, pruritus, mild lower abdominal pain and ?weight loss. There was no history of alcohol excess, new medications, recreational drugs, IVDU or new sexual contact. There was no family history of liver disease, pancreatitis or inflammatory bowel disease and no relevant travel history. Initial investigations were as follows: Hb 14.4 g/dL, WBC 8.13, platelets 316 INR 0.9 Bilirubin 125 umol/L, ALT 450, Alk Phosp 345 (NR less than130) CRP 2 Hep BsAg-ve, Hep C Ab -ve Hep A IgM -ve, Hep E IgM -ve CMV IgM-ve, EBV -ve ANA, AMA, SMA, LKM all -ve IgG 12.18 g/L (6-16), Ig A 1.39 (0.8-2.8), IgM 0.86 (0.5-1.9) IgG4 0.71 g/l (NR 0-1.3) Caeruloplasmin 0.31g/L (0.2-0.6) alpha 1 antitrypsin 1.90 g/L (0.9-1.2) Cholesterol 3.9, TG 1.2 mmol/L (normal) Ferritin 307 ng/ml (15-275) Amylase 121 and 141IU/L (NR less than 125) TTG 0.3 u/ml (0.1-6.9) TSH 0.8 mmol/L (0.4-4.9) Genetic tests: CFTR -ve, SPINK1 -ve, PRSS1 -ve AXR: normal U/S: sludge in GB, dilated IHD, CBD 11mm, PD 4mm, normal liver, pancreas, spleen CT scan: dilated IHD, CBD 11mm, bulky pancreas, ill defined, poor enhancement in HOP MRCP: no stones, sludge in GB, distal CBD stricture, dilated IHD, no pancreatic mass EUS: slightly oedematous pancreas, PD not dilated, CBD 8mm, thickened mucosal layer in CBD A diagnosis of idiopathic fibrosing pancreatitis was considered likely, even in the absence of histology. With an increasing bilirubin level (more than 250 umol/L), an ERCP was undertaken. Biliary cannulation proved difficult and various advanced techniques were necessary before we successfully entered the bile duct (see video). There was a tight lower CBD stricture (brushings showed bland cells only, no malignancy) which was successfully remodelled with a fully covered metal stent. — Idioapthic fibrosing pancreatitis is a rare form of chronic pancreatitis which affects primarily children and adolescents with a mean age of diagnosis of 10 years (4 months - 20 years). Males and females are equally affected. The condition was first described by Comfort et al in 1946 and since then less than 100 cases have been reported. Patients present with obstructive jaundice +/- abdominal pain. There is conjugated hyperbilirubinaemia, mild transaminitis and no elevation of pancreatic enzymes. Biopsies/ histology of the pancreas reveals glandular fibrosis, especially of the exocrine parenchyma with relative preservation of Islets of Langerhans. Mixed pancreatic insufficiency may occur. The pathogenesis is unknown. Theories include: autoimmune and Parvovirus. There may be an association with IBD (Crohn’s Disease) and certain genetic mutations (PRSS 1, SPINK1, CTFR-5 T, CTRC, CFTR). Most cases are diagnosed during explorative laparotomy. US, CT and MRI show dilated bile ducts and a globally enlarged pancreas. Targeted pancreatic biopsy (US/ CT guided, EUS) show fibrosis/ atrophy of exocrine pancreas with preservation of islets of Langerhans. Treatment options include: bilio-enteric bypass, pancreatic resection (few cases) +/- duodenal preservation and conservative management with temporary biliary drainage by ERCP only (as in this case) or treatment with UDCA alone or with steroids. In some cases surgery may be necessary after drainage. Spontaneous resolution occurs in some cases. However there ma be progression of pancreatic fibrosis, then atrophy leading to exocrine insufficiency and, less commonly, endocrine insufficiency. REFERENCES 1. Comfort MW, Gambill EE, Baggenstoss AH. Gastroenterology 1946;6:239-285. Chronic relapsing pancreatitis; a study of 29 cases without associated disease of the biliary or gastrointestinal tract. https://www.ncbi.nlm.nih.gov/pubmed/2... 2. Sylvestera FA et al Gut 1998;43:715-720. Management of fibrosing pancreatitis in children presenting with obstructive jaundice http://gut.bmj.com/content/43/5/715.f... 3. Mocanu SN et al Cir Esp. 2013;94:271–274. La pancreatitis idiopa´ tica fibrosante: cause infrecuente de ictericia obstructive en pacientes jo´ venes. https://www.ncbi.nlm.nih.gov/pubmed/2... 4. El-Matary W et al Eur J Pediatr. 2006 Aug;165(8):560-5. Successful conservative management of idiopathic fibrosing pancreatitis in children. https://www.ncbi.nlm.nih.gov/pubmed/1...

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