Association of N-acetylcysteine and glucagon during percutaneous cholangiography in the treatment of inspissated bile syndrome

Berrani, H.; Vasies, I.; Cron, J; Bachy, Bruno; Dosseur, P. Le; Mouterde, O.

doi:10.1016/j.arcped.2014.12.007

Cited by 7 publications

(12 citation statements)

References 7 publications

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“…IBS is characterized by obstruction of the common bile duct by biliary sludge in infants who otherwise have no anatomical abnormalities or congenital chemical bile defects. 1 3 Though no predisposing causes are found in many cases, some can occur secondary to infection, hepatocellular damage, cystic fibrosis, or hemolytic disorders. 4…”

Section: Discussionmentioning

confidence: 99%

“…1 2 3 4 With the advent and improvement in percutaneous techniques in recent years, transhepatic biliary or cholecystostomy drain placement followed by saline irrigation has become the standard of care. 1 3…”

Section: Discussionmentioning

confidence: 99%

“…Despite saline irrigation at 5 mL three times daily, subsequent upsizing to 8-French tube 3 days later with advancement of drain into duodenum to create a cholecystoduodenostomy, and increased irrigation rate to 10 mL every 3 hours, patient's bilirubin continued to rise to 15.5 mg/dL by 8 days after cholecystostomy (►Figs. 3,4). Due to liver failure, international normalized ratio increased to 5.5.…”

Section: Case Presentationmentioning

confidence: 99%

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Successful Treatment of Refractory Inspissated Bile Syndrome Using Gastrografin

Guan,

Amarneh

2023

Journal of Clinical Interventional Radiology ISVIR

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Inspissated bile syndrome (IBS) is a rare cause of obstructive jaundice in infants. Despite drainage and irrigation, obstruction may persist in some cases with limited further options. This report presents a case of persistent IBS in a 5-month-old female with worsening liver failure despite cholecystostomy and saline irrigation, and a last-ditch effort was made to irrigate with Gastrografin. Patient's bilirubin decreased from 15.5 to 8.3 mg/dL after 1 day. Her bilirubin and liver functions tests normalized by 2 weeks. Gastrografin's hyperosmolarity promotes intraluminal fluid shift and stimulates smooth muscle contraction, which may explain its effectiveness in treating refractory IBS.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Case Presentationmentioning

confidence: 99%

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Successful Treatment of Refractory Inspissated Bile Syndrome Using Gastrografin

Guan,

Amarneh

2023

Journal of Clinical Interventional Radiology ISVIR

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“…Failed medical management indicates the need for endoscopic retrograde cholangiopancreatography, percutaneous transhepatic cholangiography, or irrigation of the biliary tree with saline or a mucolytic agent through a cholecystostomy [30,31]. Co-administration of N-acetylcysteine and glucagon can effectively treat IBS [32]. Omega-three polyunsaturated fatty acids (500 mg four times per day) can be used as an alternative to surgical intervention [27].…”

Section: Inspissated Bile Syndrome (Ibs)mentioning

confidence: 99%

Neonatal cholestasis: recent insights

Ghazy

Khedr

2019

Egypt Pediatric Association Gaz

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Background: Neonatal physiological jaundice is a common benign condition that rarely extends behind the second week of life; however, it may interfere with the diagnosis of a pathological condition termed neonatal cholestasis (NC). The latter is a critical, uncommon problem characterized by conjugated hyperbilirubinaemia. This review aims to highlight the differences between physiological and pathological jaundice, identify different causes of NC, and provide a recent approach to diagnosis and management of this serious condition. Main text: NC affects 1/2500 live births, resulting in life-threatening complications due to associated hepatobiliary or metabolic abnormalities. NC is rarely benign and indicates the presence of severe underlying disease. If jaundice extends more than 14 days in full-term infants or 21 days in preterm infants, the serum bilirubin level fractionated into conjugated (direct) and unconjugated (indirect) bilirubin should be measured. A stepwise diagnostic approach starts with obtaining a complete history, and a physical examination which are valuable for the rapid diagnosis of the underlying disease. The most frequently diagnosed causes of NC are biliary atresia (BA) and idiopathic neonatal hepatitis (INH). The early diagnosis of NC ensures more accurate management and better prognosis. Despite the unavailability of any specific treatments for some causes of NC, the patient can benefit from nutritional management and early medical intervention. Future research should attempt to shed light on methods of screening for NC, especially for causes that can be effectively treated either through proper nutritional support, appropriate chemotherapeutic management, or timely surgical intervention. Conclusion: Further attention should be paid for diagnosis and treatment of NC as it may be misdiagnosed as physiological jaundice; this may delay the proper management of the underlying diseases and aggravates its complications.

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“…Daily flushing with normal saline and N-acetylcysteine via cholecystostomy and subsequent percutaneous cholangiograms were unsuccessful. Glucagon therapy was trialled following review of a case report, it was ceased due to adverse side effects [5]. Following extensive discussion between the patients’ parents, her CF team and surgeons, at 3 months of age, the patient underwent a cholecystoduodenostomy (Fig.…”

Section: Case Presentationmentioning

confidence: 99%

Case report: Cholecystoduodenostomy for cholestatic liver disease in a premature infant with cystic fibrosis and short gut syndrome

et al. 2019

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BackgroundCholecystoduodenostomy is a surgical procedure that bypasses the extrahepatic biliary tree and connects the gallbladder directly to the duodenum. This case describes the successful use of this procedure in a novel situation.Case presentationA premature (34 weeks gestation) female infant with cystic fibrosis required a laparotomy on day 1 of life due to an intrauterine small bowel perforation. Resection of small bowel and ileostomy formation resulted in short gut syndrome, with 82 cm residual small bowel and intact ileocaecal valve. Post-ileostomy reversal at 2 months old, she developed conjugated hyperbilirubinaemia. Despite conservative management including increased enteral feeding, ursodeoxycholic acid, cholecystostomy drain insertion and flushes, her cholestatic jaundice persisted. A liver biopsy revealed an “obstructive/cholestatic” picture with fibrosis. To avoid further shortening her gut with an hepatoportoenterostomy, cholecystoduodenostomy was performed at 3 months of age with subsequent post-operative improvement and eventual normalisation of her clinical jaundice and liver biochemistry.ConclusionsThis is the first reported case of a cholecystoduodenostomy being used successfully to treat an infant with persistent conjugated hyperbilirubinemia, cystic fibrosis and short gut syndrome. Cholecystoduodenostomy is a treatment option that with further study, may be considered for obstruction of the common bile duct in patients with short gut and/or where a shorter operating time with minimal intervention is preferred.

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Association of N-acetylcysteine and glucagon during percutaneous cholangiography in the treatment of inspissated bile syndrome

Cited by 7 publications

References 7 publications

Successful Treatment of Refractory Inspissated Bile Syndrome Using Gastrografin

Successful Treatment of Refractory Inspissated Bile Syndrome Using Gastrografin

Neonatal cholestasis: recent insights

Case report: Cholecystoduodenostomy for cholestatic liver disease in a premature infant with cystic fibrosis and short gut syndrome

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