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Fatal outcome of pylephlebitis treated with transhepatic percutaneous drainage
European Journal of Radiology Extra 70 (2009) e15–e18
Contents lists available at ScienceDirect
European Journal of Radiology Extra journal homepage: intl.elsevierhealth.com/journals/ejrex
Fatal outcome of pylephlebitis treated with transhepatic percutaneous drainage Anis Askri a,∗ , Leila Ben Farhat a , Mohamed Jarraya a , Ali Rebah a , Nadida Dali a , Wassim Said a , Rachid Ksantini b , Lotfi Hendaoui a a b
Department of Radiology, Mongi Slim Hospital, Sidi Daoud, 2046 La Marsa, Tunisia Department of Surgery, Rabta Hospital, Tunis, Tunisia
a r t i c l e
i n f o
Article history: Received 23 April 2008 Received in revised form 2 September 2008 Accepted 10 October 2008 Keywords: Pylephlebitis Liver abscess Ultrasonography Computed tomography Percutaneous drainage
a b s t r a c t Pylephlebitis, also called suppurative endophlebitis of the portal vein, is a rare potentially life-threatening acute abdominal complication of an intra-abdominal inflammatory process. Only early recognition substantially reduces the catastrophic mortality. As its clinical picture is fairly nonspecific, radiological findings, while not pathognomonic, are of great use in early diagnosis and management of these patients. We report a rare case of pylephlebitis with no identified intraperitoneal septic process in a 75-year-old man. Diagnosis was suspected on imaging features and confirmed with percutaneous transhepatic needle puncture. Thereafter, a drainage catheter was placed into the intrahepatic portal system. After a transient clinical improvement during the three following days, the drainage stopped and the patient developed a septic shock requiring transfer to intensive care but he died approximately 24 h later. © 2008 Elsevier Ireland Ltd. All rights reserved.
1. Introduction
2. Case report
Pylephlebitis is a rare but serious condition associated with a high rate of mortality [1]. It is an acute ascending infection of the portal system arising from a primary intra-abdominal septic source. Diverticulitis, appendicitis and necrotizing pancreatitis are the most common causes [1–4]. Early recognition and adequate therapy of the underlying condition are important in preventing the fatal outcome. However, diagnosis could be challenging as clinical signs may be nonspecific, while radiographic features are not pathognomonic [1]. The mainstay treatment should include broad-spectrum antibiotics as well as the eradication of underlying disease [1,5]. Transhepatic percutaneous drainage of suppurative endophlebitis of the portal vein has been proven as an alternative therapy to surgical procedures [4,6–9]. We report a rare case of fatal outcome of pylephlebitis with nonidentified abdominal septic source, despite of percutaneous drainage of the intrahepatic suppurative portal system and appropriate antibiotic therapy. We describe imaging features of pylephlebite and we also discuss the role of percutaneous drainage of the intrahepatic infected portal venous system in the management of this severe complication.
A 75-year-old man was admitted to the hospital after 5 days history of abdominal pain, fever and diarrhea. His relevant past history included an endoscopic resection of prostatic adenoma 4 years ago and chronic obstructive pulmonary disease treated with bronchodilators. He was febrile, tachypnoeic and his blood pressure was 110/70 mmHg. Physical examination was unremarkable except for moderate abdominal tenderness in the upper right quadrant. Laboratory studies showed a white blood cell count of 19,100 cells/dL, blood urea nitrogen of 86 mg/dL, serum creatine of 17.6 mg/dL with no derangement in liver function. Abdominal ultrasonographic examination showed thrombosis of the left portal vein. The right and the main portal veins were patent. There was no gallbladder thickening and the common duct was not enlarged. The patient was put under broad-spectrum antibiotic therapy because of the infectious presentation and anticoagulation therapy was started. Blood cultures obtained before antibacterial therapy were later found to be negative for bacteria. After normalization of renal function, 3 days later, an abdominal dynamic CT was performed with a helical single detector CT (SIEMENS Somatom Plus 4). Unenhanced and contrast-enhanced dynamic CT was performed. CT scan showed complete nonenhancing filling defect within the lumen of the left portal vein and partial defect in the right anterior and posterior portal veins (Fig. 1). There was transient high attenuation in the peripheral part of liver during arterial hepatic phase.
∗ Corresponding author. Tel.: +216 71 76 44 77; fax: +216 71 76 44 77. E-mail addresses: [email protected], [email protected] (A. Askri). 1571-4675/$ – see front matter © 2008 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ejrex.2008.10.005
e16
A. Askri et al. / European Journal of Radiology Extra 70 (2009) e15–e18
Fig. 1. Transverse CT scan obtained in the portal phase demonstrates partial defect in the right anterior and posterior portal veins (arrows).
The patient remained pyrexial. He presented chills and upper right quadrant pain and went in the 7th day into a progressive septic shock. Thus a follow-up CT scan looking for intraperitoneal septic process was performed after stabilisation of the hemodynamic parameters. Repeat CT scan, performed 10 days after the first one and 2 weeks after admission to the hospital, showed extension of the thrombosis to all intrahepatic branches of the portal venous system (Fig. 2). There was no evidence of gas within portal venous system. It also showed well-circumscribed hypodense cavities with peripheral rim enhancement in the right hepatic lobe which diameters ranged from 10 to 30 mm evoking multiple abscesses (Fig. 3). One of these abscesses communicated with the thrombosed right anterior portal vein (Fig. 2). The main portal vein presented normal enhancement. No other source of intra-abdominal sepsis was identified. Peritoneal and bilateral pleural effusion appeared on this repeat CT. Diagnosis of pylephlebitis was suspected according imaging findings in conjunction with patient condition. Upon fine needle aspiration of the cavity that communicated with the right anterior portal vein, purulent material was obtained confirming our diagnosis (Fig. 4). Twenty milliliters of purulent material was aspirated and sent for culture. After multidisciplinary discussion, a percutaneous drainage of the suppurative portal thrombosis was indicated. A 10-Fr multipurpose catheter was placed into the
Fig. 2. Repeat CT scan in portal phase showing complete nonenhancing filling defect within the lumen of right anterior and posterior portal veins (arrows). Note an abscess communicating with the thrombosed right anterior portal vein (arrowhead).
Fig. 3. Transverse CT scan in portal phase showing well-circumscribed hypodense cavities with peripheral rim enhancement in the right hepatic lobe (arrows). Note peritoneal and bilateral pleural effusion (arrowheads).
right anterior and left portal veins through right hepatic approach using a Seldinger technique (Fig. 5). No complications were noted after the percutaneous drainage procedure. Escherichia coli grew by culture. After percutaneous drainage, the patient defervesced quickly and had significant decrease in the amount of pain. The output from the drainage catheter was about 30 mL of purulent material a day and no appreciable blood returned with this drainage. After a transient clinical improvement during the three following days, the drainage stopped and the patient developed a rapid and profound septic shock with refractory hypotension requiring transfer to intensive care unit for mechanical ventilation and vasopressor support but he died approximately 24 h later. 3. Discussion Pylephlebitis also called suppurative thrombophlebitis of the portal vein is a rare and acute life-threatening complication of intra-abdominal infection with a high rate of mortality [1]. For many years it was associated with an almost 100% mortality, but with the advent of broad-spectrum antibiotics and early surgical intervention, incidence and mortality have decreased. Pylephlebitis
Fig. 4. Fine needle aspiration of the cavity that communicates with the right anterior portal vein.
A. Askri et al. / European Journal of Radiology Extra 70 (2009) e15–e18
Fig. 5. CT examination after percutaneous drainage of the portal suppurative thrombophlebitis showing a 10-Fr catheter placed into right anterior and left portal veins.
is usually secondary to suppuration in the region drained by the portal system and various infectious diseases affecting the gastrointestinal tract have been implicated in the development of pylephlebitis. The most common causes are acute colonic diverticulitis, acute appendicitis and necrotizing pancreatitis [1–4]. In some of the reported cases, the expected abdominal extrahepatic source of infection could not be identified [5,10,11]. Our patient had no clear intra-abdominal septic focus on physical examination, ultrasound and CT scan. As the clinical features are generally nonspecific, the diagnosis of pylephlebitis remains a challenge. Sepsis is often associated with nonspecific abdominal symptoms and clinical features depend on the site of the primary inflammatory process. Fever and abdominal pain are the most common presenting symptoms. Hepatomegaly and jaundice may be present [1,5]. The clinical presentation can lead to an extended differential diagnosis requiring laboratory and imaging studies for confirmation. Impaired liver function tests and leukocytosis are common. Bacteraemia is often present and the most common isolated pathogens are E. coli, Proteus mirabilis, Bacteroides fragilis, and other aerobic gram-negative bacilli [1,12,13]. It has been reported that negative blood cultures are obtained in approximately 20% of proved cases [12]. In our patient, blood cultures remained negative. Early diagnosis of this rare entity by means of imaging techniques is crucial due to the high morbidity and mortality. Only early detection of septic ascending pylephlebitis and adequate treatment can decrease the mortality rate. But early diagnosis is very difficult if the primary pyogenic process is not identified. The complications of pylephlebitis include formation of pyogenic hepatic abscesses and progression of the thrombus into the enteric venous system, resulting in mesenteric ischaemia [1]. Plain abdominal radiographs are generally nondiagnostic unless air is seen in the portal veins as branching radiolucency in the liver extending to within 2 cm of the capsule [14]. However, hepatic portal venous gas is not specific and it is usually related to mesenteric ischaemia [14]. Modern imaging techniques facilitate early diagnosis in cases that probably remained undiagnosed in the past. Doppler ultrasonography, contrast CT and MRI are all considered sensitive imaging techniques for diagnosis, although no comparative evidence exists. Color Doppler ultrasonography is a useful diagnostic tool identifying portal vein thrombosis or liver abscesses but it may avoid operator-dependent variability. Ultrasound shows portal vein thrombosis as an echogenic material within the lumen
e17
of the vein [15]. However, clots exhibit variable echogenicity and may, if recently formed, be hypoechoic or anechoic. Color Doppler US shows no portal signal [15]. Ultrasound may also be useful for follow-up examination to demonstrate extension of the thrombosis or recanalization of the portal vein. Abdominal CT with contrast enhancement is the preferable imaging modality in establishing the diagnosis earlier. It can not only display the portal vein thrombus but also detect small amounts of portal venous gas and show concomitant occult primary source of infection [10,16]. CT scan demonstrates portal vein thrombus as a nonenhancing, low-density thrombus within the vessel lumen. Nonhomogeneous enhancement of the hepatic parenchyma is usually present due to obstruction of the portal venous inflow and compensatory increase of the arterial inflow [16]. Liver abscesses can present variable appearances from well-circumscribed cavities with enhancing rims to heterogeneous masses indistinguishable from hepatic neoplasms [17]. MRI can help to distinguish acute portal vein thrombus from chronic portal vein thrombosis. With acute thrombosis, the portal vein appears hyperintense to the surrounding liver on a T1weighted image and becomes even brighter on T2-weighted images [15]. Imaging features are not pathognomonic and the presence of intravascular air or thrombi in portal system can be seen in various other conditions not associated with pylephlebitis. However, findings of gas and thrombus in the portal vein together support the diagnosis of septic thrombophlebitis [18]. When serial scans are available showing very rapid progression of the imaging findings with extension of portal vein thrombus and development of solitary or multiple hepatic abscesses over a brief interval, one should favor a diagnosis of infection [11]. Final diagnosis must not been made according to one-time imaging but after careful evaluation of repeated radiological examinations in conjunction with patient condition. In our case, diagnosis was suspected according to imaging features on repeat CT scan showing extension of portal thrombosis and development of hepatic abscesses. The mainstay treatment should include appropriate antibiotic therapy and removal of any primary septic source [1,5]. When possible, surgery is performed electively to eradicate the primary septic process. Percutaneous drainage of intra-abdominal abscesses has been proven to be an alternative therapy to surgical procedures in certain patients [19]. When pylephlebitis is complicated with large hepatic abscess (larger than 3 cm), a percutaneous drainage is required [10]. Surgical drainage for idiopathic suppurative pylephlebitis is an accurate but invasive treatment [20]. Minimally invasive percutaneous needle puncture of the intrahepatic portal venous system was reported as an accurate procedure for the appropriate diagnosis and treatment of suppurative pylethrombophlebitis [4,6–9]. Vivas et al. reported transhepatic direct portography under fluoroscopic guidance with aspiration of the portal vein content obtaining thrombus with pus. The authors proceeded to fragment the intraportal thrombus by inflating an angioplasty balloon catheter, intraportal infusion of urokinase and then a 8-F Sheath was inserted in the portal vein [10]. To the best of our knowledge, only five previous papers reported percutaneous transhepatic drainage under CT guidance of the infected portal venous system in six patients [4,6–9]. The first case was reported by Schwarz et al. in 1987 [6]. Five patients had pancreatitis and one had angiocholitis. All patients were not surgical candidates. As in our case, no complications were noted in all cases reported. They had significant resolution of symptoms and required no surgical intervention. In our case, diagnosis of the pylephlebitis was made 2 weeks after admission to the hospital but his course was unfavorable in spite of percutaneous drainage of the suppurative intrahepatic portal venous system. This shows that, despite the
e18
A. Askri et al. / European Journal of Radiology Extra 70 (2009) e15–e18
technically successful percutaneous drainage of the pylephlebitis, early diagnosis is necessary in order to limit disease progression and decrease the mortality rate. 4. Conclusion Pylephlebitis is an extremely rare disease with high rate of mortality if unrecognized. Percutaneous drainage of the infected intrahepatic portal system is a reasonable therapeutic option and is technically successful but our report suggests that only prompt and accurate diagnosis can improve the prognosis of these acutely ill patients. Conflict of interest None declared. References [1] Hoffman HL, Partington PF, Desanctis AL. Pylephlebitis and liver abscess. Am J Surg 1954;88(September (3)):411–6. [2] Nobili C, Uggeri F, Romano F, et al. Pylephlebitis and mesenteric thrombophlebitis in sigmoid diverticulitis: medical approach, delayed surgery. Dig Liver Dis 2007;39(December (12)):1088–90 [Epub 2007 July 23]. [3] Vanamo K, Kiekara O. Pylephlebitis after appendicitis in a child. J Pediatr Surg 2001;36(October (10)):1574–6. [4] Nouira K, Bedioui H, Azaiez O, et al. Percutaneous drainage of suppurative pylephlebitis complicating acute pancreatitis. Cardiovasc Intervent Radiol 2007;30(November–December (6)):1242–4. [5] Singh P, Yadav N, Visvalingam V, Indaram A, Bank S. Pylephlebitis-diagnosis and management. Am J Gastroenterol 2001;96(April (4)):1312–3. [6] Schwarz W, Honickman S, Ohki S, Buckman R, Warner E. Percutaneous diagnosis and drainage of pylephlebitis: a case report. Surgery 1987;101(February (2)):244–9.
[7] Dean JW, Trerotola SO, Harris VJ, Snidow JJ, Hawes D. Percutaneous management of suppurative pylephlebitis. J Vasc Interv Radiol 1995;6(July–August (4)):585–8. [8] Pelsang RE, Johlin F, Dhadha R, Bogdanowicz M, Schweiger GD. Management of suppurative pylephlebitis by percutaneous drainage: placing a drainage catheter into the portal vein. Am J Gastroenterol 2001;96(November (11)):3192–4. [9] Watanabe H, Muguruma T, Idoguchi K, Matsuoka T. Percutaneous drainage for suppurative pylethrombophlebitis developing in a patient with chronic pancreatitis. J Gastroenterol 2007;42(July (7)):589–92. [10] Vivas I, Bilbao JI, Martínez-Cuesta A, Benito A, Delgado C, Velázquez P. Combination of various percutaneous techniques in the treatment of pylephlebitis. J Vasc Interv Radiol 2000;11(June (6)):777–80. [11] Wireko M, Berry PA, Brennan J, Aga R. Unrecognized pylephlebitis causing lifethreatening septic shock: a case report. World J Gastroenterol 2005;11(January (4)):614–5. [12] Plemmons RM, Dooley DP, Longfield RN. Septic thrombophlebitis of the portal vein (pylephlebitis): diagnosis and management in the modern era. Clin Infect Dis 1995;21(November (5)):1114–20. [13] Saxena R, Adolph M, Ziegler JR, Murphy W, Rutecki GW. Pylephlebitis: a case report and review of outcome in the antibiotic era. Am J Gastroenterol 1996;91(June (6)):1251–3. [14] Liebman PR, Patten MT, Manny J, Benfield JR, Hechtman HB. Hepatic venous gas in adults: etiology, pathophysiology and clinical significance. Ann Surg 1978;187:281–7. [15] Parvey HR, Raval B, Sandler CM. Portal vein thrombosis: imaging findings. AJR Am J Roentgenol 1994;162(January (1)):77–81. [16] Balthazar EJ, Gollapudi P. Septic thrombophlebitis of the mesenteric and portal veins: CT imaging. J Comput Assist Tomogr 2000;24(September–October (5)):755–60. [17] Wildi SM, Wallace MB, Hunter B, Noone TC, Hoffman BJ. EUS diagnosis of an unusual case of pylephlebitis mimicking metastatic pancreatic cancer. Dig Dis Sci 2005;50(December (12)):2255–8. [18] Chau TN, Loke TK, Leung VK, Law ST, Lai MH, Ho YW. Hepatic portal venous gas complicating septic thrombophlebitis of the superior mesenteric vein. Hong Kong Med J 2007;13(February (1)):69–72. [19] Men S, Akhan O, Köro˘glu M. Percutaneous drainage of abdominal abscess. Eur J Radiol 2002;43(September (3)):204–18 [Review]. [20] Ohtake H, Urayama H, Nozaki Z, Harada T, Kawasuji M, Wantanabe Y. Surgical drainage for idiopathic suppurative pylephlebitis. Ann Vasc Surg 1998;12(January (1)):83–5.
Contents lists available at ScienceDirect
European Journal of Radiology Extra journal homepage: intl.elsevierhealth.com/journals/ejrex
Fatal outcome of pylephlebitis treated with transhepatic percutaneous drainage Anis Askri a,∗ , Leila Ben Farhat a , Mohamed Jarraya a , Ali Rebah a , Nadida Dali a , Wassim Said a , Rachid Ksantini b , Lotfi Hendaoui a a b
Department of Radiology, Mongi Slim Hospital, Sidi Daoud, 2046 La Marsa, Tunisia Department of Surgery, Rabta Hospital, Tunis, Tunisia
a r t i c l e
i n f o
Article history: Received 23 April 2008 Received in revised form 2 September 2008 Accepted 10 October 2008 Keywords: Pylephlebitis Liver abscess Ultrasonography Computed tomography Percutaneous drainage
a b s t r a c t Pylephlebitis, also called suppurative endophlebitis of the portal vein, is a rare potentially life-threatening acute abdominal complication of an intra-abdominal inflammatory process. Only early recognition substantially reduces the catastrophic mortality. As its clinical picture is fairly nonspecific, radiological findings, while not pathognomonic, are of great use in early diagnosis and management of these patients. We report a rare case of pylephlebitis with no identified intraperitoneal septic process in a 75-year-old man. Diagnosis was suspected on imaging features and confirmed with percutaneous transhepatic needle puncture. Thereafter, a drainage catheter was placed into the intrahepatic portal system. After a transient clinical improvement during the three following days, the drainage stopped and the patient developed a septic shock requiring transfer to intensive care but he died approximately 24 h later. © 2008 Elsevier Ireland Ltd. All rights reserved.
1. Introduction
2. Case report
Pylephlebitis is a rare but serious condition associated with a high rate of mortality [1]. It is an acute ascending infection of the portal system arising from a primary intra-abdominal septic source. Diverticulitis, appendicitis and necrotizing pancreatitis are the most common causes [1–4]. Early recognition and adequate therapy of the underlying condition are important in preventing the fatal outcome. However, diagnosis could be challenging as clinical signs may be nonspecific, while radiographic features are not pathognomonic [1]. The mainstay treatment should include broad-spectrum antibiotics as well as the eradication of underlying disease [1,5]. Transhepatic percutaneous drainage of suppurative endophlebitis of the portal vein has been proven as an alternative therapy to surgical procedures [4,6–9]. We report a rare case of fatal outcome of pylephlebitis with nonidentified abdominal septic source, despite of percutaneous drainage of the intrahepatic suppurative portal system and appropriate antibiotic therapy. We describe imaging features of pylephlebite and we also discuss the role of percutaneous drainage of the intrahepatic infected portal venous system in the management of this severe complication.
A 75-year-old man was admitted to the hospital after 5 days history of abdominal pain, fever and diarrhea. His relevant past history included an endoscopic resection of prostatic adenoma 4 years ago and chronic obstructive pulmonary disease treated with bronchodilators. He was febrile, tachypnoeic and his blood pressure was 110/70 mmHg. Physical examination was unremarkable except for moderate abdominal tenderness in the upper right quadrant. Laboratory studies showed a white blood cell count of 19,100 cells/dL, blood urea nitrogen of 86 mg/dL, serum creatine of 17.6 mg/dL with no derangement in liver function. Abdominal ultrasonographic examination showed thrombosis of the left portal vein. The right and the main portal veins were patent. There was no gallbladder thickening and the common duct was not enlarged. The patient was put under broad-spectrum antibiotic therapy because of the infectious presentation and anticoagulation therapy was started. Blood cultures obtained before antibacterial therapy were later found to be negative for bacteria. After normalization of renal function, 3 days later, an abdominal dynamic CT was performed with a helical single detector CT (SIEMENS Somatom Plus 4). Unenhanced and contrast-enhanced dynamic CT was performed. CT scan showed complete nonenhancing filling defect within the lumen of the left portal vein and partial defect in the right anterior and posterior portal veins (Fig. 1). There was transient high attenuation in the peripheral part of liver during arterial hepatic phase.
∗ Corresponding author. Tel.: +216 71 76 44 77; fax: +216 71 76 44 77. E-mail addresses: [email protected], [email protected] (A. Askri). 1571-4675/$ – see front matter © 2008 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ejrex.2008.10.005
e16
A. Askri et al. / European Journal of Radiology Extra 70 (2009) e15–e18
Fig. 1. Transverse CT scan obtained in the portal phase demonstrates partial defect in the right anterior and posterior portal veins (arrows).
The patient remained pyrexial. He presented chills and upper right quadrant pain and went in the 7th day into a progressive septic shock. Thus a follow-up CT scan looking for intraperitoneal septic process was performed after stabilisation of the hemodynamic parameters. Repeat CT scan, performed 10 days after the first one and 2 weeks after admission to the hospital, showed extension of the thrombosis to all intrahepatic branches of the portal venous system (Fig. 2). There was no evidence of gas within portal venous system. It also showed well-circumscribed hypodense cavities with peripheral rim enhancement in the right hepatic lobe which diameters ranged from 10 to 30 mm evoking multiple abscesses (Fig. 3). One of these abscesses communicated with the thrombosed right anterior portal vein (Fig. 2). The main portal vein presented normal enhancement. No other source of intra-abdominal sepsis was identified. Peritoneal and bilateral pleural effusion appeared on this repeat CT. Diagnosis of pylephlebitis was suspected according imaging findings in conjunction with patient condition. Upon fine needle aspiration of the cavity that communicated with the right anterior portal vein, purulent material was obtained confirming our diagnosis (Fig. 4). Twenty milliliters of purulent material was aspirated and sent for culture. After multidisciplinary discussion, a percutaneous drainage of the suppurative portal thrombosis was indicated. A 10-Fr multipurpose catheter was placed into the
Fig. 2. Repeat CT scan in portal phase showing complete nonenhancing filling defect within the lumen of right anterior and posterior portal veins (arrows). Note an abscess communicating with the thrombosed right anterior portal vein (arrowhead).
Fig. 3. Transverse CT scan in portal phase showing well-circumscribed hypodense cavities with peripheral rim enhancement in the right hepatic lobe (arrows). Note peritoneal and bilateral pleural effusion (arrowheads).
right anterior and left portal veins through right hepatic approach using a Seldinger technique (Fig. 5). No complications were noted after the percutaneous drainage procedure. Escherichia coli grew by culture. After percutaneous drainage, the patient defervesced quickly and had significant decrease in the amount of pain. The output from the drainage catheter was about 30 mL of purulent material a day and no appreciable blood returned with this drainage. After a transient clinical improvement during the three following days, the drainage stopped and the patient developed a rapid and profound septic shock with refractory hypotension requiring transfer to intensive care unit for mechanical ventilation and vasopressor support but he died approximately 24 h later. 3. Discussion Pylephlebitis also called suppurative thrombophlebitis of the portal vein is a rare and acute life-threatening complication of intra-abdominal infection with a high rate of mortality [1]. For many years it was associated with an almost 100% mortality, but with the advent of broad-spectrum antibiotics and early surgical intervention, incidence and mortality have decreased. Pylephlebitis
Fig. 4. Fine needle aspiration of the cavity that communicates with the right anterior portal vein.
A. Askri et al. / European Journal of Radiology Extra 70 (2009) e15–e18
Fig. 5. CT examination after percutaneous drainage of the portal suppurative thrombophlebitis showing a 10-Fr catheter placed into right anterior and left portal veins.
is usually secondary to suppuration in the region drained by the portal system and various infectious diseases affecting the gastrointestinal tract have been implicated in the development of pylephlebitis. The most common causes are acute colonic diverticulitis, acute appendicitis and necrotizing pancreatitis [1–4]. In some of the reported cases, the expected abdominal extrahepatic source of infection could not be identified [5,10,11]. Our patient had no clear intra-abdominal septic focus on physical examination, ultrasound and CT scan. As the clinical features are generally nonspecific, the diagnosis of pylephlebitis remains a challenge. Sepsis is often associated with nonspecific abdominal symptoms and clinical features depend on the site of the primary inflammatory process. Fever and abdominal pain are the most common presenting symptoms. Hepatomegaly and jaundice may be present [1,5]. The clinical presentation can lead to an extended differential diagnosis requiring laboratory and imaging studies for confirmation. Impaired liver function tests and leukocytosis are common. Bacteraemia is often present and the most common isolated pathogens are E. coli, Proteus mirabilis, Bacteroides fragilis, and other aerobic gram-negative bacilli [1,12,13]. It has been reported that negative blood cultures are obtained in approximately 20% of proved cases [12]. In our patient, blood cultures remained negative. Early diagnosis of this rare entity by means of imaging techniques is crucial due to the high morbidity and mortality. Only early detection of septic ascending pylephlebitis and adequate treatment can decrease the mortality rate. But early diagnosis is very difficult if the primary pyogenic process is not identified. The complications of pylephlebitis include formation of pyogenic hepatic abscesses and progression of the thrombus into the enteric venous system, resulting in mesenteric ischaemia [1]. Plain abdominal radiographs are generally nondiagnostic unless air is seen in the portal veins as branching radiolucency in the liver extending to within 2 cm of the capsule [14]. However, hepatic portal venous gas is not specific and it is usually related to mesenteric ischaemia [14]. Modern imaging techniques facilitate early diagnosis in cases that probably remained undiagnosed in the past. Doppler ultrasonography, contrast CT and MRI are all considered sensitive imaging techniques for diagnosis, although no comparative evidence exists. Color Doppler ultrasonography is a useful diagnostic tool identifying portal vein thrombosis or liver abscesses but it may avoid operator-dependent variability. Ultrasound shows portal vein thrombosis as an echogenic material within the lumen
e17
of the vein [15]. However, clots exhibit variable echogenicity and may, if recently formed, be hypoechoic or anechoic. Color Doppler US shows no portal signal [15]. Ultrasound may also be useful for follow-up examination to demonstrate extension of the thrombosis or recanalization of the portal vein. Abdominal CT with contrast enhancement is the preferable imaging modality in establishing the diagnosis earlier. It can not only display the portal vein thrombus but also detect small amounts of portal venous gas and show concomitant occult primary source of infection [10,16]. CT scan demonstrates portal vein thrombus as a nonenhancing, low-density thrombus within the vessel lumen. Nonhomogeneous enhancement of the hepatic parenchyma is usually present due to obstruction of the portal venous inflow and compensatory increase of the arterial inflow [16]. Liver abscesses can present variable appearances from well-circumscribed cavities with enhancing rims to heterogeneous masses indistinguishable from hepatic neoplasms [17]. MRI can help to distinguish acute portal vein thrombus from chronic portal vein thrombosis. With acute thrombosis, the portal vein appears hyperintense to the surrounding liver on a T1weighted image and becomes even brighter on T2-weighted images [15]. Imaging features are not pathognomonic and the presence of intravascular air or thrombi in portal system can be seen in various other conditions not associated with pylephlebitis. However, findings of gas and thrombus in the portal vein together support the diagnosis of septic thrombophlebitis [18]. When serial scans are available showing very rapid progression of the imaging findings with extension of portal vein thrombus and development of solitary or multiple hepatic abscesses over a brief interval, one should favor a diagnosis of infection [11]. Final diagnosis must not been made according to one-time imaging but after careful evaluation of repeated radiological examinations in conjunction with patient condition. In our case, diagnosis was suspected according to imaging features on repeat CT scan showing extension of portal thrombosis and development of hepatic abscesses. The mainstay treatment should include appropriate antibiotic therapy and removal of any primary septic source [1,5]. When possible, surgery is performed electively to eradicate the primary septic process. Percutaneous drainage of intra-abdominal abscesses has been proven to be an alternative therapy to surgical procedures in certain patients [19]. When pylephlebitis is complicated with large hepatic abscess (larger than 3 cm), a percutaneous drainage is required [10]. Surgical drainage for idiopathic suppurative pylephlebitis is an accurate but invasive treatment [20]. Minimally invasive percutaneous needle puncture of the intrahepatic portal venous system was reported as an accurate procedure for the appropriate diagnosis and treatment of suppurative pylethrombophlebitis [4,6–9]. Vivas et al. reported transhepatic direct portography under fluoroscopic guidance with aspiration of the portal vein content obtaining thrombus with pus. The authors proceeded to fragment the intraportal thrombus by inflating an angioplasty balloon catheter, intraportal infusion of urokinase and then a 8-F Sheath was inserted in the portal vein [10]. To the best of our knowledge, only five previous papers reported percutaneous transhepatic drainage under CT guidance of the infected portal venous system in six patients [4,6–9]. The first case was reported by Schwarz et al. in 1987 [6]. Five patients had pancreatitis and one had angiocholitis. All patients were not surgical candidates. As in our case, no complications were noted in all cases reported. They had significant resolution of symptoms and required no surgical intervention. In our case, diagnosis of the pylephlebitis was made 2 weeks after admission to the hospital but his course was unfavorable in spite of percutaneous drainage of the suppurative intrahepatic portal venous system. This shows that, despite the
e18
A. Askri et al. / European Journal of Radiology Extra 70 (2009) e15–e18
technically successful percutaneous drainage of the pylephlebitis, early diagnosis is necessary in order to limit disease progression and decrease the mortality rate. 4. Conclusion Pylephlebitis is an extremely rare disease with high rate of mortality if unrecognized. Percutaneous drainage of the infected intrahepatic portal system is a reasonable therapeutic option and is technically successful but our report suggests that only prompt and accurate diagnosis can improve the prognosis of these acutely ill patients. Conflict of interest None declared. References [1] Hoffman HL, Partington PF, Desanctis AL. Pylephlebitis and liver abscess. Am J Surg 1954;88(September (3)):411–6. [2] Nobili C, Uggeri F, Romano F, et al. Pylephlebitis and mesenteric thrombophlebitis in sigmoid diverticulitis: medical approach, delayed surgery. Dig Liver Dis 2007;39(December (12)):1088–90 [Epub 2007 July 23]. [3] Vanamo K, Kiekara O. Pylephlebitis after appendicitis in a child. J Pediatr Surg 2001;36(October (10)):1574–6. [4] Nouira K, Bedioui H, Azaiez O, et al. Percutaneous drainage of suppurative pylephlebitis complicating acute pancreatitis. Cardiovasc Intervent Radiol 2007;30(November–December (6)):1242–4. [5] Singh P, Yadav N, Visvalingam V, Indaram A, Bank S. Pylephlebitis-diagnosis and management. Am J Gastroenterol 2001;96(April (4)):1312–3. [6] Schwarz W, Honickman S, Ohki S, Buckman R, Warner E. Percutaneous diagnosis and drainage of pylephlebitis: a case report. Surgery 1987;101(February (2)):244–9.
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