Adam Dobek, Katarzyna Stefańczyk-Jakubowicz, Rafał Maciąg, Przemysław Przewratil, Wojciech Szubert, Ludomir Stefańczyk
{"title":"7岁男童因先天性动脉门静脉瘘导致大量门静脉扩张,危及生命之食道静脉曲张出血。","authors":"Adam Dobek, Katarzyna Stefańczyk-Jakubowicz, Rafał Maciąg, Przemysław Przewratil, Wojciech Szubert, Ludomir Stefańczyk","doi":"10.12659/AJCR.946013","DOIUrl":null,"url":null,"abstract":"<p><p>BACKGROUND Arterioportal fistulas (APFs) are abnormal connections between the arterial and portal venous systems, leading to portal hypertension (PH) and symptoms such as gastrointestinal bleeding, splenomegaly, and hepatic pain. Symptoms typically appear by the age of 2 years in about 75% of cases. CASE REPORT A 7-year-old boy with an asymptomatic APF developed life-threatening complications following a Clostridium difficile infection. He initially had chronic diarrhea, abdominal pain, weight loss, and anorexia for 3 weeks, despite normal liver enzymes. After antibiotic and antifungal treatment, his condition worsened, resulting in severe anemia and hemorrhagic shock due to variceal bleeding. Further evaluations revealed significant PH secondary to the APF. Intensive care involved blood transfusions, fluid resuscitation, and high-frequency ventilation. Emergency embolization successfully reduced PH and controlled bleeding. After stabilization, the patient was transferred for further care. A week later, a color Doppler ultrasound (CD-US) detected a thrombus in the left portal vein, which decreased by the 19-day follow-up. Spleen size reduction indicated decreased portal pressure. The presence and reduction of the thrombus and alleviation of PH symptoms indicate therapeutic success. Ten months after embolization, the patient remained asymptomatic, with normal liver function and no thrombus on follow-up imaging. CONCLUSIONS Early diagnosis and intervention are crucial in managing congenital APF in children. Severe variceal bleeding triggered by exacerbated PH due to a Clostridium difficile infection demonstrates the complications of APF. Endovascular treatment was highly effective, resulting in significant improvement. The recommended diagnostic approach includes initial computed tomography angiogram and CD-US, followed by digital subtraction angiography with possible intervention, and monitoring with CD-US.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":"25 ","pages":"e946013"},"PeriodicalIF":1.0000,"publicationDate":"2024-12-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11660004/pdf/","citationCount":"0","resultStr":"{\"title\":\"Life-Threatening Esophageal Variceal Hemorrhage in a 7-Year-Old Boy with Massive Portal Vein Enlargement Due to Congenital Arterioportal Fistula.\",\"authors\":\"Adam Dobek, Katarzyna Stefańczyk-Jakubowicz, Rafał Maciąg, Przemysław Przewratil, Wojciech Szubert, Ludomir Stefańczyk\",\"doi\":\"10.12659/AJCR.946013\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>BACKGROUND Arterioportal fistulas (APFs) are abnormal connections between the arterial and portal venous systems, leading to portal hypertension (PH) and symptoms such as gastrointestinal bleeding, splenomegaly, and hepatic pain. Symptoms typically appear by the age of 2 years in about 75% of cases. CASE REPORT A 7-year-old boy with an asymptomatic APF developed life-threatening complications following a Clostridium difficile infection. He initially had chronic diarrhea, abdominal pain, weight loss, and anorexia for 3 weeks, despite normal liver enzymes. After antibiotic and antifungal treatment, his condition worsened, resulting in severe anemia and hemorrhagic shock due to variceal bleeding. Further evaluations revealed significant PH secondary to the APF. Intensive care involved blood transfusions, fluid resuscitation, and high-frequency ventilation. Emergency embolization successfully reduced PH and controlled bleeding. After stabilization, the patient was transferred for further care. A week later, a color Doppler ultrasound (CD-US) detected a thrombus in the left portal vein, which decreased by the 19-day follow-up. Spleen size reduction indicated decreased portal pressure. The presence and reduction of the thrombus and alleviation of PH symptoms indicate therapeutic success. Ten months after embolization, the patient remained asymptomatic, with normal liver function and no thrombus on follow-up imaging. CONCLUSIONS Early diagnosis and intervention are crucial in managing congenital APF in children. Severe variceal bleeding triggered by exacerbated PH due to a Clostridium difficile infection demonstrates the complications of APF. Endovascular treatment was highly effective, resulting in significant improvement. The recommended diagnostic approach includes initial computed tomography angiogram and CD-US, followed by digital subtraction angiography with possible intervention, and monitoring with CD-US.</p>\",\"PeriodicalId\":39064,\"journal\":{\"name\":\"American Journal of Case Reports\",\"volume\":\"25 \",\"pages\":\"e946013\"},\"PeriodicalIF\":1.0000,\"publicationDate\":\"2024-12-17\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11660004/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"American Journal of Case Reports\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.12659/AJCR.946013\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"MEDICINE, GENERAL & INTERNAL\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"American Journal of Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.12659/AJCR.946013","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"MEDICINE, GENERAL & INTERNAL","Score":null,"Total":0}
Life-Threatening Esophageal Variceal Hemorrhage in a 7-Year-Old Boy with Massive Portal Vein Enlargement Due to Congenital Arterioportal Fistula.
BACKGROUND Arterioportal fistulas (APFs) are abnormal connections between the arterial and portal venous systems, leading to portal hypertension (PH) and symptoms such as gastrointestinal bleeding, splenomegaly, and hepatic pain. Symptoms typically appear by the age of 2 years in about 75% of cases. CASE REPORT A 7-year-old boy with an asymptomatic APF developed life-threatening complications following a Clostridium difficile infection. He initially had chronic diarrhea, abdominal pain, weight loss, and anorexia for 3 weeks, despite normal liver enzymes. After antibiotic and antifungal treatment, his condition worsened, resulting in severe anemia and hemorrhagic shock due to variceal bleeding. Further evaluations revealed significant PH secondary to the APF. Intensive care involved blood transfusions, fluid resuscitation, and high-frequency ventilation. Emergency embolization successfully reduced PH and controlled bleeding. After stabilization, the patient was transferred for further care. A week later, a color Doppler ultrasound (CD-US) detected a thrombus in the left portal vein, which decreased by the 19-day follow-up. Spleen size reduction indicated decreased portal pressure. The presence and reduction of the thrombus and alleviation of PH symptoms indicate therapeutic success. Ten months after embolization, the patient remained asymptomatic, with normal liver function and no thrombus on follow-up imaging. CONCLUSIONS Early diagnosis and intervention are crucial in managing congenital APF in children. Severe variceal bleeding triggered by exacerbated PH due to a Clostridium difficile infection demonstrates the complications of APF. Endovascular treatment was highly effective, resulting in significant improvement. The recommended diagnostic approach includes initial computed tomography angiogram and CD-US, followed by digital subtraction angiography with possible intervention, and monitoring with CD-US.
期刊介绍:
American Journal of Case Reports is an international, peer-reviewed scientific journal that publishes single and series case reports in all medical fields. American Journal of Case Reports is issued on a continuous basis as a primary electronic journal. Print copies of a single article or a set of articles can be ordered on demand.