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Hepatic artery aneurysm in a child
Hepatic Artery Aneurysm in a Child By A. Navarro G6mez, C. Garcia Lorenzo, A. Ruiz Orpez, J. Azcarate L6pez, and G.A. L6pez-P~rez Malaga, Spain
9 The present report refers to a child hospitalized because of nonspecific symptomatology, who was found to have aneurysms of both hepatic arteries. A year after simple proximal ligature of both hepatic arteries, the child remains symptom-free and developing normally. Proximal ligation appears to be a simple and effective technique for treating hepatic artery aneurysm in children in preference to direct approach of the vascular pathology. 9 1987 by Grune & Stratton. Inc.
INDEX WORDS: Hepatic artery aneurysm.
ANEURYSMS of the hepatic arteries I SOLATED are extremely rare in infancy. The great majority of those described so far are part of systemic multiple arterial aneurysms such as those found in EhlersDanlos' syndrome or polyarteritis nodosa. ~3 Ligature of the hepatic arteries in children are infrequently recommended and usually only for extreme situations like uncontrolled post-traumatic hemorrhage or hemobilia, or heart failure due to intrahepatic vascular anomalies leading to severe leftto-right shunting.4-6 CASE REPORT A 2-year-old white male was admitted to the hospital because of edema of lower extremities and petechial purpura for the last 2 weeks. He had been previously found to have pyelouretheral duplication and hypospadias. On physical examination he appeared seriously ill with a painful abdomen. A diffuse mass was palpable on the right upper quadrant of the abdomen. With the original diagnosis of Henoch-SchSnlein's purpura, eorticosteroid therapy was started. However, the skin rash
Fig 1. Transverse sonogram of the left hepatic lobe showing the aneurysm.
Fig 2. Selective hepatic arteriography showing an aneurysm that includes both hepatic arteries.
became more extensive and the edema extended to the upper extremities, accompanied by generalized ache. Consequently, the original diagnosis was ruled out. Laboratory studies, including hemogram, coagulation, and biochemical tests were within normal limits. Abdominal and chest x-rays were also normal. Abdominal ultrasonography showed an abnormal vascular mass in the left hepatic lobe (Fig 1). Selective arteriography of the hepatic arteries was performed and it showed clearly an aneurysm affecting both hepatic arteries (Fig 2). Surgical treatment consisted of proximal ligature of both hepatic arteries. The cystic, gastroduodenal, and right gastric arteries were not ligated. Parenteral feedings and antibiotics were administered duirng the first six postoperative days. Recovery was uneventful and 12 months after surgery, the child remains asymptomatic. Hepatic function tests are also normal and the sonogram shows considerable decrease in the size of the vascular mass (Fig 3).
From the Pediatric Surgical Service, Hospital Materno-lnfantil de Malaga, Spain. Address reprint requests to G.A. Lbpez-Pbrez, Jefe del Servicio de Cirugia Pedihtrica, Hospital Materno-Infantil de Malaga, Malaga, Spain. 9 1987 by Grune & Stratton, Inc. 0022-3468/87/2211-0023503.00/0
Journal of Pediatric Surgery, Vo122, No 11 (November),1987: pp 1027-1028
1027
1028
NAVARRO GOMEZ ET AL
Fig 3. Postoperative ultrasonography shows a decrease of the vascular mass and a normal hepatic sonographlc structure.
DISCUSSION
Until relatively few years ago, it was thought that ligature of the main hepatic arteries would be followed by necrotic hepatic degeneration, particularly after G r a h a m ' s report of a high mortality following accidental ligature of the hepatic artery. 7 However, Madding and Kennedy suggested that in the presence of massive hemorrhage following severe hepatic injury, ligature of the hepatic arteries could be applied successfully, s Michels and Mays' reports 9'm established the basis for
modern knowledge of anatomic variations of the hepatic blood supply and for the fact that ligation of the hepatic arteries is possible without incapacitating sequelae. Angiographic studies II have shown that revascularization of the liver is possible through the intercostal, inferior phrenic, and right superior phrenic arteries. Furthermore, arteries from the falciform and round ligaments anastomose with branches of the left hepatic and middle arteries, bringing blood to the liver from the internal m a m m a r y arteries. Also the cystic and other accessory or anomalous vessels may contribute to the collateral blood flow to the liver. 9 Fasting and parenteral feeding in the immediate postoperative course are recommended because of the transient decrease of the blood supply to the liver, which is also responsible for a depletion of the hepatic glycogen. Because of this, administration of 10% glucose is also recommended during the early postoperative period. 4 Fasting also improves portal oxygenation and allows a greater contribution of oxygen to the hepatic cells, which possibly helps in preventing hepatic necrosis. It is also important to avoid hypovolemia during and after surgery. 12 Antibiotics were used because of the possibility of invasion of the portal vein by anaerobic bacteria. 6 We think that hepatic artery ligation is an easy and effective method of controlling hepatic aneurysms. Provided that there are no previous liver diseases and sepsis and shock are avoided, the procedure should be well tolerated with a low morbidity.
REFERENCES
1. Schiller M, Gordon R, Schifrin E, et al: Multiple arterial aneurysms. J Pediatr Surg 18:27-29, 1983 2. Almgrcn B, Eriksson I, Foucard T, et al: Multiple aneurysms of visceral arteries in a child with polyarteritis nodosa. J Pediatr Surg 15:347-348, 1980 3. Mirza FH, Smith PL, Line WN: Multiple aneurysms in a patient with Ehlers-Danlos syndrome: Angiographywithout sequelae. Am J Radiol 132:993-995, 1979 4. Canty TG, Aaron WS: Hepatic artery ligation for exsanguihating liver injuries in children. J Pediatr Surg 10:693-700, 1975 5. Harouchi A, Pellerin D: L'hemobilie post-traumatique. Ann Chir lnf 17:7-14, 1976 6. Mazam F, Rodgers M, Talbert JL: Hepatic artery ligation for hepatic hemangiomatosisof infancy. J Pediatr Surg 18:120-123, 1983
7. Graham RR, Cannell D: Accidental ligation of the hepatic artery. Br J Surg 20:566-579, 1933 8. Madding GF, KennedyPA: Hepatic artery ligation. Surg Clin North Am 52:719-728, 1972 9. Miehels NA: Newer anatomy of the liver and its variant blood supply and collateral circulation. Am J Surg 112:337-347, 1966 10. Mays ET: Observationsand managementafter hepatic artery ligation. Surg GynecolObstet 124:801-807, 1967 11. BengmarkS, Rosengren K: Angiographicstudy of the collateral circulation to the liver after ligation of the hepatic artery in man. Am J Surg 119:620-624, 1970 12. Mays ET, Conti S, Fallahzadeh H, et al: Hepatic artery ligation. Surgery 86:536-543, 1979
9 The present report refers to a child hospitalized because of nonspecific symptomatology, who was found to have aneurysms of both hepatic arteries. A year after simple proximal ligature of both hepatic arteries, the child remains symptom-free and developing normally. Proximal ligation appears to be a simple and effective technique for treating hepatic artery aneurysm in children in preference to direct approach of the vascular pathology. 9 1987 by Grune & Stratton. Inc.
INDEX WORDS: Hepatic artery aneurysm.
ANEURYSMS of the hepatic arteries I SOLATED are extremely rare in infancy. The great majority of those described so far are part of systemic multiple arterial aneurysms such as those found in EhlersDanlos' syndrome or polyarteritis nodosa. ~3 Ligature of the hepatic arteries in children are infrequently recommended and usually only for extreme situations like uncontrolled post-traumatic hemorrhage or hemobilia, or heart failure due to intrahepatic vascular anomalies leading to severe leftto-right shunting.4-6 CASE REPORT A 2-year-old white male was admitted to the hospital because of edema of lower extremities and petechial purpura for the last 2 weeks. He had been previously found to have pyelouretheral duplication and hypospadias. On physical examination he appeared seriously ill with a painful abdomen. A diffuse mass was palpable on the right upper quadrant of the abdomen. With the original diagnosis of Henoch-SchSnlein's purpura, eorticosteroid therapy was started. However, the skin rash
Fig 1. Transverse sonogram of the left hepatic lobe showing the aneurysm.
Fig 2. Selective hepatic arteriography showing an aneurysm that includes both hepatic arteries.
became more extensive and the edema extended to the upper extremities, accompanied by generalized ache. Consequently, the original diagnosis was ruled out. Laboratory studies, including hemogram, coagulation, and biochemical tests were within normal limits. Abdominal and chest x-rays were also normal. Abdominal ultrasonography showed an abnormal vascular mass in the left hepatic lobe (Fig 1). Selective arteriography of the hepatic arteries was performed and it showed clearly an aneurysm affecting both hepatic arteries (Fig 2). Surgical treatment consisted of proximal ligature of both hepatic arteries. The cystic, gastroduodenal, and right gastric arteries were not ligated. Parenteral feedings and antibiotics were administered duirng the first six postoperative days. Recovery was uneventful and 12 months after surgery, the child remains asymptomatic. Hepatic function tests are also normal and the sonogram shows considerable decrease in the size of the vascular mass (Fig 3).
From the Pediatric Surgical Service, Hospital Materno-lnfantil de Malaga, Spain. Address reprint requests to G.A. Lbpez-Pbrez, Jefe del Servicio de Cirugia Pedihtrica, Hospital Materno-Infantil de Malaga, Malaga, Spain. 9 1987 by Grune & Stratton, Inc. 0022-3468/87/2211-0023503.00/0
Journal of Pediatric Surgery, Vo122, No 11 (November),1987: pp 1027-1028
1027
1028
NAVARRO GOMEZ ET AL
Fig 3. Postoperative ultrasonography shows a decrease of the vascular mass and a normal hepatic sonographlc structure.
DISCUSSION
Until relatively few years ago, it was thought that ligature of the main hepatic arteries would be followed by necrotic hepatic degeneration, particularly after G r a h a m ' s report of a high mortality following accidental ligature of the hepatic artery. 7 However, Madding and Kennedy suggested that in the presence of massive hemorrhage following severe hepatic injury, ligature of the hepatic arteries could be applied successfully, s Michels and Mays' reports 9'm established the basis for
modern knowledge of anatomic variations of the hepatic blood supply and for the fact that ligation of the hepatic arteries is possible without incapacitating sequelae. Angiographic studies II have shown that revascularization of the liver is possible through the intercostal, inferior phrenic, and right superior phrenic arteries. Furthermore, arteries from the falciform and round ligaments anastomose with branches of the left hepatic and middle arteries, bringing blood to the liver from the internal m a m m a r y arteries. Also the cystic and other accessory or anomalous vessels may contribute to the collateral blood flow to the liver. 9 Fasting and parenteral feeding in the immediate postoperative course are recommended because of the transient decrease of the blood supply to the liver, which is also responsible for a depletion of the hepatic glycogen. Because of this, administration of 10% glucose is also recommended during the early postoperative period. 4 Fasting also improves portal oxygenation and allows a greater contribution of oxygen to the hepatic cells, which possibly helps in preventing hepatic necrosis. It is also important to avoid hypovolemia during and after surgery. 12 Antibiotics were used because of the possibility of invasion of the portal vein by anaerobic bacteria. 6 We think that hepatic artery ligation is an easy and effective method of controlling hepatic aneurysms. Provided that there are no previous liver diseases and sepsis and shock are avoided, the procedure should be well tolerated with a low morbidity.
REFERENCES
1. Schiller M, Gordon R, Schifrin E, et al: Multiple arterial aneurysms. J Pediatr Surg 18:27-29, 1983 2. Almgrcn B, Eriksson I, Foucard T, et al: Multiple aneurysms of visceral arteries in a child with polyarteritis nodosa. J Pediatr Surg 15:347-348, 1980 3. Mirza FH, Smith PL, Line WN: Multiple aneurysms in a patient with Ehlers-Danlos syndrome: Angiographywithout sequelae. Am J Radiol 132:993-995, 1979 4. Canty TG, Aaron WS: Hepatic artery ligation for exsanguihating liver injuries in children. J Pediatr Surg 10:693-700, 1975 5. Harouchi A, Pellerin D: L'hemobilie post-traumatique. Ann Chir lnf 17:7-14, 1976 6. Mazam F, Rodgers M, Talbert JL: Hepatic artery ligation for hepatic hemangiomatosisof infancy. J Pediatr Surg 18:120-123, 1983
7. Graham RR, Cannell D: Accidental ligation of the hepatic artery. Br J Surg 20:566-579, 1933 8. Madding GF, KennedyPA: Hepatic artery ligation. Surg Clin North Am 52:719-728, 1972 9. Miehels NA: Newer anatomy of the liver and its variant blood supply and collateral circulation. Am J Surg 112:337-347, 1966 10. Mays ET: Observationsand managementafter hepatic artery ligation. Surg GynecolObstet 124:801-807, 1967 11. BengmarkS, Rosengren K: Angiographicstudy of the collateral circulation to the liver after ligation of the hepatic artery in man. Am J Surg 119:620-624, 1970 12. Mays ET, Conti S, Fallahzadeh H, et al: Hepatic artery ligation. Surgery 86:536-543, 1979