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DIII-558; No. of Pages 4
Diagnostic and Interventional Imaging (2015) xxx, xxx—xxx
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RADIOLOGIC PATHOLOGIC CORRELATION /Gastrointestinal imaging
Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation F. Le Moigne a,∗, T. Vitry a, P. Gérôme b, N. Vinurel c, C. Ducerf d, A. Rode e, B. Bancel f a
Service de radiologie, hôpital d’instruction des armées Desgenettes, 108, boulevard Pinel, 69003 Lyon, France b Service de biologie médicale, hôpital d’instruction des armées Desgenettes, 108, boulevard Pinel, 69003 Lyon, France c Service de radiologie, hôpital Louis-Pradel, 28, avenue du Doyen-Jean-Lépine, 69500 Bron, France d Service de chirurgie digestive et de la transplantation, hôpital de la Croix-Rousse, 103, Grande Rue de la Croix-Rousse, 69004 Lyon, France e Service de radiologie, hôpital de la Croix-Rousse, 103, Grande Rue de la Croix-Rousse, 69004 Lyon, France f Service d’anatomopathologie, hôpital de la Croix-Rousse, 103, Grande Rue de la Croix-Rousse, 69004 Lyon, France
KEYWORDS Brucelloma; Liver abscess; Calcification
∗
Observation A 62-year old Algerian patient with no prior medical history was admitted to the emergency unit for upper right quadrant pain after deterioration of his general condition for 2 months. Clinical examination was normal. Biological tests showed elevated C-reactive protein without hyperleucocytosis. Liver tests showed moderate cholestasis without jaundice or associated cytolysis. Liver ultrasound showed a large heterogenous 11 cm mass in the right liver with a central macrocalcification. Abdominopelvic CT examination showed a hypodense mass with irregular borders and a calcification in the centre, strongly suggesting an infectious lesion (Fig. 1). Liver MRI (Fig. 2) showed a multilocular lesion with numerous fluid signal compartments and restricted diffusion. Microbiological agglutination tests (Wright and Rose Bengal reaction) were positive and suggested brucellosis. Fine needle ultrasound-guided biopsy of the lesion revealed pus that was shown to be due to Gram-negative bacteria that was further confirmed as Brucella melitensis after 5 days of incubation. A Brucella liver abscess was
Corresponding author. E-mail address: [email protected] (F. Le Moigne).
http://dx.doi.org/10.1016/j.diii.2014.08.007 2211-5684/© 2015 Published by Elsevier Masson SAS on behalf of the Éditions françaises de radiologie.
Please cite this article in press as: Le Moigne F, et al. Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation. Diagnostic and Interventional Imaging (2015), http://dx.doi.org/10.1016/j.diii.2014.08.007
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ARTICLE IN PRESS
2
F. Le Moigne et al. caseous necrosis surrounded by a granuloma composed of histiocyte cells arranged in palisade (Fig. 3). Several weeks later, despite continued antibiotics, there was still no improvement. The patient’s clinical condition continued to worsen and postoperative liver failure developed, then hepatorenal syndrome, so that emergency liver transplantation was decided. Histological assessment of the explanted liver showed severe sepsis on long-term cholestasis with ‘‘young’’ F2 fibrosis according to the Metavir score and diffuse nodular regenerative hyperplasia. The early post-transplantation course was associated with numerous infectious complications, such as recurrent pneumopathies. Twelve months after transplantation, the patient was in good general condition, and serology for brucellosis was negative on PCR.
Discussion Figure 1. CT scan in the axial plane during the portal phase shows hypoattenuating. Multilocular lesion with irregular borders. Round macrocalcification is visible inside the lesion.
diagnosed. Long-term oral antibiotic treatment was begun with a combination of doxycycline and rifampin associated with gentamicine, however, the size of the abscess had still not regressed after two months. Although percutanous radiological drainage was performed, a significant quantity of pus could not be evacuated. Because of the patient’s worsening clinical condition, a right hepatectomy was indicated. Pathological assessment of the surgical specimen showed an abscessed lesion with areas of focal
Brucellosis is a cosmopolitan anthropozoonosis whose incidence has markedly decreased in Northern Europe, but which is still frequent in the Mediterranean basin. It is due to the Gram-negative bacteria (B. melitensis). Human contamination is direct by cutaneo-mucosal route from animal reservoirs or indirect by contamination from unpasteurized milk products. The germ is disseminated by the lymphatic system or blood vessels to reach the cells of the reticuloendothelial system. Primary infection is characterized by a long lasting fever, multiple adenopathies and hepatosplenomegaly. The usual biological tests do not confirm the disease, and serodiagnostic tests are not specific. The
Figure 2. Liver MRI, Axial T1 (a), T2 SPAIR (b), diffusion b800 (c), ADC mapping (d), contrast-enhanced 3D T1-EG during the arterial phase (e) and delayed phase 3 min (f). Multilocular lesion with a hypointense signal of contents on T1 and hyperintense signal on T2 images, liquid type, restricted diffusion (ADC = 0.8 × 10−3 mm2 /s). Thick hyperintense walls on T2-weighted images with an increased ADC coefficient, with contrast enhancement during arterial and delayed phases. Intense enhancement of the adjacent liver during the arterial phase. Central calcification, which is seen as a low signal intensity lesion on all sequences.
Please cite this article in press as: Le Moigne F, et al. Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation. Diagnostic and Interventional Imaging (2015), http://dx.doi.org/10.1016/j.diii.2014.08.007
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ARTICLE IN PRESS
Necrotizing pseudotumoral hepatic granuloma of brucellosis origin
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Figure 3. Macroscopic features after right hepatectomy (a and b) shows a large subcapsular lesion (a) containing prurulent material (b). Histological views [c and d, HES, enlargement × 50 (c), × 100 (d)] showing pyo-necrotic material limited by an epithelioid border arranged in palisade surrounded by lymphoplasmocytes and histiocytes. In the ‘‘healthy’’ zone (E, HES enlargement × 50), regenerative non-fibrous parenchymatous nodules can be seen.
diagnosis is confirmed by Brucella culture or hemoculture during the acute phase, or organ tissue samples [1]. The development of secondary locations, in particular in the liver, are characteristic of the focal complications of brucellosis during the subacute or chronic stage. The liver is one of the prefered targets of Brucella, because of its intracellular tropism parasites Küpffer cells. Liver involvement is always granulomatous, but can have two types of anatomoclinical presentations. This most frequent is a generally asymptomatic hepatic granulomatosis that includes lymphohystiomonocytic granulomas which usually lack giant cells. The second type, which corresponds to our case study, is a pseudotumoral necrotizing granulomatosis, commonly known as brucelloma, which is very rare, with only thirty cases or so in the literature. Brucelloma is secondary to an undiagnosed liver brucellosis. It is due to the fusion of granulomatous lesions resulting in the development of caseous necrosis and leading to a true abscess. The pathological assessment shows large areas of focal caseous necrosis surrounded by an epitheloid granuloma arranged in a palisade. Reports of fibrosis of the liver parenchyma have also been described in the literature, although the pathogenic role of brucellosis in the development liver fibrosis and the progression to cirrhosis is still controversial [2]. Imaging plays a fundamental role in identifying the liver lesion usually some time after the primary infection and in a clinical context that is generally poor. The morphological features on CT scan are very specific showing a single pseudotumoral lesion, which is often found in the right liver, including compartments of thick liquid with thick walls that are enchanced after injection of a contrast medium and
containing a focal, central or marginal calcification [3—5]. This intralesional calcification was nearly always reported in the few published case studies and is considered to be specific for the diagnosis. This focal calcification is large or round. It corresponds to a calcified granuloma and is a sign of chronic disease. Liver MRI is not essential to the diagnosis. It shows elements similar to pyogenic liver abscess with a multilocular lesion including a group of cystic lesions of various sizes with a hypointense signal on T1-weighted images and a hyperintense signal on T2-weighted images [6]. Enhancement of the walls during the arterial phase, persists during the delayed phase as well as enhancement of the adjacent liver parenchyma during the arterial phase because of inflammation. Diffusion-weighted imaging can further confirm the diagnosis of an abscess rather than tumoral necrosis by showing restricted diffusion of the purulent content. The treatment of brucelloma is systematically based on double antibiotics, which are usually doxycycline combined with rifampin or an aminoglycoside for at least 3 months [1]. Percutaneous radiological drainage should be considered immediately because of the large size of the lesion at diagnosis, with a success rate that varies in the literature [7]. The semi-solid consistency of the lesion is in fact the greatest difficulty of drainage. In case of failure, surgical treatment is indicated. Liver transplantation should be considered only when the progression of the disease is extremely severe. In conclusion, brucelloma is an extremely rare liver disease. Its diagnosis is based on typical imaging features, isolation of the Brucella germ in culture and histology showing a granuloma.
Please cite this article in press as: Le Moigne F, et al. Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation. Diagnostic and Interventional Imaging (2015), http://dx.doi.org/10.1016/j.diii.2014.08.007
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ARTICLE IN PRESS
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F. Le Moigne et al.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
[4]
[5]
References [1] Maurin M, Brion J-P. Brucellose. EMC Maladies infectieuses, 6. Paris: Elsevier Masson; 2009. p. 1—12 [8-038-A-10]. [2] Akritidis N, Tzivras M, Delladetsima I, Stefanaki S, Moutsopoulos HM, Pappas G. The liver in brucellosis. Clin Gastroenterol Hepatol 2007;5:1109—12. [3] Ruiz Carazo E, Mu˜ noz Parra F, Jiménez Villares MP, del Mar Castellano García M, Moyano Calvente SL, Medina Benítez A.
[6]
[7]
Hepatosplenic brucelloma: clinical presentation and imaging features in six cases. Abdom Imaging 2005;30:291—6. Halimi C, Bringard N, Boyer N, Vilgrain V, Panis Y, Degott C, et al. Hepatic brucelloma: 2 cases and a review of the literature. Gastroenterol Clin Biol 1999;23:513—7. Sisteron O, Souci J, Chevallier P, Cua E, Bruneton JN. Hepatic abscess caused by Brucella US, CT and MRI findings: case report and review of the literature. Clin Imaging 2002;26: 414—7. Chourmouzi D, Boulogianni G, Kalomenopoulou M, Kanellos I, Drevelegas A. Brucella liver abscess; imaging approach, differential diagnosis, and therapeutic management: a case report. Cases J 2009;2:7143. Pramateftakis M-G, Kanellos D, Kanellos I. Percutaneous drainage of hepatic brucelloma. Am Surg 2009;75:1143—4.
Please cite this article in press as: Le Moigne F, et al. Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation. Diagnostic and Interventional Imaging (2015), http://dx.doi.org/10.1016/j.diii.2014.08.007
ARTICLE IN PRESS
DIII-558; No. of Pages 4
Diagnostic and Interventional Imaging (2015) xxx, xxx—xxx
Disponible en ligne sur
ScienceDirect www.sciencedirect.com
RADIOLOGIC PATHOLOGIC CORRELATION /Gastrointestinal imaging
Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation F. Le Moigne a,∗, T. Vitry a, P. Gérôme b, N. Vinurel c, C. Ducerf d, A. Rode e, B. Bancel f a
Service de radiologie, hôpital d’instruction des armées Desgenettes, 108, boulevard Pinel, 69003 Lyon, France b Service de biologie médicale, hôpital d’instruction des armées Desgenettes, 108, boulevard Pinel, 69003 Lyon, France c Service de radiologie, hôpital Louis-Pradel, 28, avenue du Doyen-Jean-Lépine, 69500 Bron, France d Service de chirurgie digestive et de la transplantation, hôpital de la Croix-Rousse, 103, Grande Rue de la Croix-Rousse, 69004 Lyon, France e Service de radiologie, hôpital de la Croix-Rousse, 103, Grande Rue de la Croix-Rousse, 69004 Lyon, France f Service d’anatomopathologie, hôpital de la Croix-Rousse, 103, Grande Rue de la Croix-Rousse, 69004 Lyon, France
KEYWORDS Brucelloma; Liver abscess; Calcification
∗
Observation A 62-year old Algerian patient with no prior medical history was admitted to the emergency unit for upper right quadrant pain after deterioration of his general condition for 2 months. Clinical examination was normal. Biological tests showed elevated C-reactive protein without hyperleucocytosis. Liver tests showed moderate cholestasis without jaundice or associated cytolysis. Liver ultrasound showed a large heterogenous 11 cm mass in the right liver with a central macrocalcification. Abdominopelvic CT examination showed a hypodense mass with irregular borders and a calcification in the centre, strongly suggesting an infectious lesion (Fig. 1). Liver MRI (Fig. 2) showed a multilocular lesion with numerous fluid signal compartments and restricted diffusion. Microbiological agglutination tests (Wright and Rose Bengal reaction) were positive and suggested brucellosis. Fine needle ultrasound-guided biopsy of the lesion revealed pus that was shown to be due to Gram-negative bacteria that was further confirmed as Brucella melitensis after 5 days of incubation. A Brucella liver abscess was
Corresponding author. E-mail address: [email protected] (F. Le Moigne).
http://dx.doi.org/10.1016/j.diii.2014.08.007 2211-5684/© 2015 Published by Elsevier Masson SAS on behalf of the Éditions françaises de radiologie.
Please cite this article in press as: Le Moigne F, et al. Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation. Diagnostic and Interventional Imaging (2015), http://dx.doi.org/10.1016/j.diii.2014.08.007
+Model DIII-558; No. of Pages 4
ARTICLE IN PRESS
2
F. Le Moigne et al. caseous necrosis surrounded by a granuloma composed of histiocyte cells arranged in palisade (Fig. 3). Several weeks later, despite continued antibiotics, there was still no improvement. The patient’s clinical condition continued to worsen and postoperative liver failure developed, then hepatorenal syndrome, so that emergency liver transplantation was decided. Histological assessment of the explanted liver showed severe sepsis on long-term cholestasis with ‘‘young’’ F2 fibrosis according to the Metavir score and diffuse nodular regenerative hyperplasia. The early post-transplantation course was associated with numerous infectious complications, such as recurrent pneumopathies. Twelve months after transplantation, the patient was in good general condition, and serology for brucellosis was negative on PCR.
Discussion Figure 1. CT scan in the axial plane during the portal phase shows hypoattenuating. Multilocular lesion with irregular borders. Round macrocalcification is visible inside the lesion.
diagnosed. Long-term oral antibiotic treatment was begun with a combination of doxycycline and rifampin associated with gentamicine, however, the size of the abscess had still not regressed after two months. Although percutanous radiological drainage was performed, a significant quantity of pus could not be evacuated. Because of the patient’s worsening clinical condition, a right hepatectomy was indicated. Pathological assessment of the surgical specimen showed an abscessed lesion with areas of focal
Brucellosis is a cosmopolitan anthropozoonosis whose incidence has markedly decreased in Northern Europe, but which is still frequent in the Mediterranean basin. It is due to the Gram-negative bacteria (B. melitensis). Human contamination is direct by cutaneo-mucosal route from animal reservoirs or indirect by contamination from unpasteurized milk products. The germ is disseminated by the lymphatic system or blood vessels to reach the cells of the reticuloendothelial system. Primary infection is characterized by a long lasting fever, multiple adenopathies and hepatosplenomegaly. The usual biological tests do not confirm the disease, and serodiagnostic tests are not specific. The
Figure 2. Liver MRI, Axial T1 (a), T2 SPAIR (b), diffusion b800 (c), ADC mapping (d), contrast-enhanced 3D T1-EG during the arterial phase (e) and delayed phase 3 min (f). Multilocular lesion with a hypointense signal of contents on T1 and hyperintense signal on T2 images, liquid type, restricted diffusion (ADC = 0.8 × 10−3 mm2 /s). Thick hyperintense walls on T2-weighted images with an increased ADC coefficient, with contrast enhancement during arterial and delayed phases. Intense enhancement of the adjacent liver during the arterial phase. Central calcification, which is seen as a low signal intensity lesion on all sequences.
Please cite this article in press as: Le Moigne F, et al. Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation. Diagnostic and Interventional Imaging (2015), http://dx.doi.org/10.1016/j.diii.2014.08.007
+Model DIII-558; No. of Pages 4
ARTICLE IN PRESS
Necrotizing pseudotumoral hepatic granuloma of brucellosis origin
3
Figure 3. Macroscopic features after right hepatectomy (a and b) shows a large subcapsular lesion (a) containing prurulent material (b). Histological views [c and d, HES, enlargement × 50 (c), × 100 (d)] showing pyo-necrotic material limited by an epithelioid border arranged in palisade surrounded by lymphoplasmocytes and histiocytes. In the ‘‘healthy’’ zone (E, HES enlargement × 50), regenerative non-fibrous parenchymatous nodules can be seen.
diagnosis is confirmed by Brucella culture or hemoculture during the acute phase, or organ tissue samples [1]. The development of secondary locations, in particular in the liver, are characteristic of the focal complications of brucellosis during the subacute or chronic stage. The liver is one of the prefered targets of Brucella, because of its intracellular tropism parasites Küpffer cells. Liver involvement is always granulomatous, but can have two types of anatomoclinical presentations. This most frequent is a generally asymptomatic hepatic granulomatosis that includes lymphohystiomonocytic granulomas which usually lack giant cells. The second type, which corresponds to our case study, is a pseudotumoral necrotizing granulomatosis, commonly known as brucelloma, which is very rare, with only thirty cases or so in the literature. Brucelloma is secondary to an undiagnosed liver brucellosis. It is due to the fusion of granulomatous lesions resulting in the development of caseous necrosis and leading to a true abscess. The pathological assessment shows large areas of focal caseous necrosis surrounded by an epitheloid granuloma arranged in a palisade. Reports of fibrosis of the liver parenchyma have also been described in the literature, although the pathogenic role of brucellosis in the development liver fibrosis and the progression to cirrhosis is still controversial [2]. Imaging plays a fundamental role in identifying the liver lesion usually some time after the primary infection and in a clinical context that is generally poor. The morphological features on CT scan are very specific showing a single pseudotumoral lesion, which is often found in the right liver, including compartments of thick liquid with thick walls that are enchanced after injection of a contrast medium and
containing a focal, central or marginal calcification [3—5]. This intralesional calcification was nearly always reported in the few published case studies and is considered to be specific for the diagnosis. This focal calcification is large or round. It corresponds to a calcified granuloma and is a sign of chronic disease. Liver MRI is not essential to the diagnosis. It shows elements similar to pyogenic liver abscess with a multilocular lesion including a group of cystic lesions of various sizes with a hypointense signal on T1-weighted images and a hyperintense signal on T2-weighted images [6]. Enhancement of the walls during the arterial phase, persists during the delayed phase as well as enhancement of the adjacent liver parenchyma during the arterial phase because of inflammation. Diffusion-weighted imaging can further confirm the diagnosis of an abscess rather than tumoral necrosis by showing restricted diffusion of the purulent content. The treatment of brucelloma is systematically based on double antibiotics, which are usually doxycycline combined with rifampin or an aminoglycoside for at least 3 months [1]. Percutaneous radiological drainage should be considered immediately because of the large size of the lesion at diagnosis, with a success rate that varies in the literature [7]. The semi-solid consistency of the lesion is in fact the greatest difficulty of drainage. In case of failure, surgical treatment is indicated. Liver transplantation should be considered only when the progression of the disease is extremely severe. In conclusion, brucelloma is an extremely rare liver disease. Its diagnosis is based on typical imaging features, isolation of the Brucella germ in culture and histology showing a granuloma.
Please cite this article in press as: Le Moigne F, et al. Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation. Diagnostic and Interventional Imaging (2015), http://dx.doi.org/10.1016/j.diii.2014.08.007
+Model DIII-558; No. of Pages 4
ARTICLE IN PRESS
4
F. Le Moigne et al.
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
[4]
[5]
References [1] Maurin M, Brion J-P. Brucellose. EMC Maladies infectieuses, 6. Paris: Elsevier Masson; 2009. p. 1—12 [8-038-A-10]. [2] Akritidis N, Tzivras M, Delladetsima I, Stefanaki S, Moutsopoulos HM, Pappas G. The liver in brucellosis. Clin Gastroenterol Hepatol 2007;5:1109—12. [3] Ruiz Carazo E, Mu˜ noz Parra F, Jiménez Villares MP, del Mar Castellano García M, Moyano Calvente SL, Medina Benítez A.
[6]
[7]
Hepatosplenic brucelloma: clinical presentation and imaging features in six cases. Abdom Imaging 2005;30:291—6. Halimi C, Bringard N, Boyer N, Vilgrain V, Panis Y, Degott C, et al. Hepatic brucelloma: 2 cases and a review of the literature. Gastroenterol Clin Biol 1999;23:513—7. Sisteron O, Souci J, Chevallier P, Cua E, Bruneton JN. Hepatic abscess caused by Brucella US, CT and MRI findings: case report and review of the literature. Clin Imaging 2002;26: 414—7. Chourmouzi D, Boulogianni G, Kalomenopoulou M, Kanellos I, Drevelegas A. Brucella liver abscess; imaging approach, differential diagnosis, and therapeutic management: a case report. Cases J 2009;2:7143. Pramateftakis M-G, Kanellos D, Kanellos I. Percutaneous drainage of hepatic brucelloma. Am Surg 2009;75:1143—4.
Please cite this article in press as: Le Moigne F, et al. Necrotizing pseudotumoral hepatic brucelloma: Imaging-pathologic correlation. Diagnostic and Interventional Imaging (2015), http://dx.doi.org/10.1016/j.diii.2014.08.007
