NESIR case presentation

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Presentation transcript:

NESIR case presentation Rush Chewning, MD Pediatric Interventional Radiology Fellow Boston Children’s Hospital 01/09/2017

History 4-year-old female with history of: Trisomy 21 Developmental delay VSD Conductive hearing loss Marked hypotonia Multiple ophalmologic issues

Presentation Abdominal distention Elevated LFTs Pancytopenia Ultrasound ordered

Presentation Ultrasound: Liver mass Portal vein poorly seen (question thrombosis) Ascites Hepatosplenomegaly

Workup Next step? Diagnostic paracentesis Liver mass biopsy CT MRI

Workup MRI: Liver mass Diffuse lymphadenopathy (not shown) Takes up and retains Eovist FNH favored over adenoma Diffuse lymphadenopathy (not shown)

Workup MRI: Tortuous vessels, particularly left lobe Cavernous transformation portal vein? Formation of perigastric varices?

Workup summary so far Liver mass Diffuse lymphadenopathy Possible cavernous transformation portal vein Stigmata of portal hypertension Hepatosplenomegaly Ascites Esophageal varices (found on EGD)

Workup CT: Left portal vein larger than main portal vein Diminutive right portal vein Prominent variceal vessels left lobe Appeared to be contiguous with left portal vein

Workup Ultrasound: Reversal of flow in main portal vein Flow is pulsatile Mass is larger Ascites has increased

Workup Diagnosis? Venous malformation Venovenous shunt Arterioportal fistula Hypervascularity associated with malignancy

Workup Angiogram

Diagnosis Intrahepatic arterioportal fistula (IAPF) causing portal hypertension Treatment choice? Surgical excision (left lobectomy) Arterial embolization – coils Arterial embolization – particles Arterial embolization – glue Venous embolization – coils

Treatment Venous embolization

Treatment Post embolization

Early outcome Doppler US immediately post embolization Restoration of hepatopetal flow in main, right, and left portal veins Slow velocities in portal system Doppler US day 4 post embolization Continued hepatopetal flow Increased velocities compared to day 0 Ascites resolved; peritoneal drain removed Patient discharged home day 5 post embo

Portal Hypertension Portal hypertension: Cirrhosis (in West) > 90% Non-cirrhotic causes: Schistosomiasis Portal vein thrombosis Primary myelofibrosis IAPF (rare)

Intrahepatic arterioportal fistula IAPF is typically iatrogenic: Surgery Biopsy Transhepatic intervention Other causes: Trauma Rupture of hepatic artery aneurysm

Congenital IAPF Congenital IAPF as cause of PHTN is exceedingly rare As of 2015, only 39 cases reported in literature Patient presentation: Upper GI bleeding Hepatosplenomegaly Ascites Failure to thrive Protein losing enteropathy Chronic diarrhea / steatorrhea Zhang et al. World Journal of Gastroenterology 2015 Feb 21; 21(7): 2229-2235 Norton et al. Journal of Pediatric Gastroenterology and Nutrition 2006 Aug 43(2): 248-255