Histological characterization of liver involvement in systemic mastocytosis

Background and Aims Systemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been marginally described and accurate histological classification is critical, given the consequences of aggressive SM diagnosis. We aimed to describ...

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Published inLiver international Vol. 44; no. 7; pp. 1680 - 1688
Main Authors Rossignol, Julien, Canioni, Danielle, Aouba, Achille, Bulai‐Livideanu, Cristina, Barete, Stéphane, Lancesseur, Charles, Polivka, Laura, Madrange, Marine, Ballul, Thomas, Neuraz, Antoine, Greco, Celine, Agopian, Julie, Brenet, Fabienne, Dubreuil, Patrice, Lemal, Richard, Tournilhac, Olivier, Terriou, Louis, Launay, David, Bouillet, Laurence, Gourguechon, Clément, Frenzel, Laurent, Meni, Cécile, Gaudy‐Marqueste, Caroline, Gousseff, Marie, Le Mouel, Edwige, Hamidou, Mohamed, Neel, Antoine, Ranta, Dana, Jaussaud, Roland, Guilpain, Philippe, Molina, Thierry J, Bruneau, Julie, Lhermitte, Ludovic, Garcelon, Nicolas, Javier, Rose‐Marie, Pelletier, Fabien, Castelain, Florence, Retornaz, Frederique, Cabrera, Quentin, Zunic, Patricia, Gourin, Marie Pierre, Wierzbicka‐Hainaut, Ewa, Viallard, Jean François, Lavigne, Christian, Hoarau, Cyrille, Durieu, Isabelle, Heiblig, Maël, Dimicoli‐Salazar, Sophie, Torregrosa‐Diaz, Jose M, Soria, Angèle, Arock, Michel, Lortholary, Olivier, Bodemer, Christine, Pol, Stanislas, Mallet, Vincent, Hermine, Olivier, Damaj, Ghandi
Format Journal Article
LanguageEnglish
Published United States Wiley Subscription Services, Inc 01.07.2024
Wiley-Blackwell
SeriesLiver International
Subjects
advanced systemic mastocytosis
Alkaline phosphatase
Biopsy
Cirrhosis
Diagnosis
Fibrosis
Histology
Hyperplasia
Hypertension
Life Sciences
Liver
Mast cells
Mastocytosis
Medical prognosis
nodular regenerative hyperplasia
Patients
portal hypertension
Subgroups
nodular regenerative hyperplasia
fibrosis
portal hypertension
mast cells
liver
advanced systemic mastocytosis
Online AccessGet full text
ISSN1478-3223
1478-3231
1478-3231
DOI10.1111/liv.15913

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Abstract Background and Aims Systemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been marginally described and accurate histological classification is critical, given the consequences of aggressive SM diagnosis. We aimed to describe the histological features associated with liver SM using updated tools. Methods Using the database of the French Reference Centre for Mastocytosis, we retrospectively identified patients with a liver biopsy (LB) and a diagnosis of SM. All LB procedures were performed according to the local physician in charge and centrally reviewed by an expert pathologist. Results A total of 28 patients were included: 6 had indolent SM, 9 had aggressive SM, and 13 had SM with an associated hematologic neoplasm. Twenty‐five (89%) patients presented hepatomegaly, and 19 (68%) had portal hypertension. The LB frequently showed slight sinusoid dilatation (82%). Fibrosis was observed in 3/6 indolent SM and in almost all advanced SM cases (21/22), but none of them showed cirrhosis. A high MC burden (>50 MCs/high‐power field) was correlated with elevated blood alkaline phosphatase levels (p = .030). The presence of portal hypertension was associated with a higher mean fibrosis grade (1.6 vs. 0.8 in its absence; p = .026). In advanced SM, the presence of nodular regenerative hyperplasia (NRH) was associated with decreased overall survival (9.5 vs. 46.3 months, p = .002). Conclusions MC infiltration induced polymorphic hepatic lesions and the degree of fibrosis is associated with portal hypertension. NRH identifies a poor prognosis subgroup of patients with advanced SM. Assessing liver histology can aid in SM prognostic evaluation.
AbstractList Background and AimsSystemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been marginally described and accurate histological classification is critical, given the consequences of aggressive SM diagnosis. We aimed to describe the histological features associated with liver SM using updated tools.MethodsUsing the database of the French Reference Centre for Mastocytosis, we retrospectively identified patients with a liver biopsy (LB) and a diagnosis of SM. All LB procedures were performed according to the local physician in charge and centrally reviewed by an expert pathologist.ResultsA total of 28 patients were included: 6 had indolent SM, 9 had aggressive SM, and 13 had SM with an associated hematologic neoplasm. Twenty-five (89%) patients presented hepatomegaly, and 19 (68%) had portal hypertension. The LB frequently showed slight sinusoid dilatation (82%). Fibrosis was observed in 3/6 indolent SM and in almost all advanced SM cases (21/22), but none of them showed cirrhosis. A high MC burden (>50 MCs/high-power field) was correlated with elevated blood alkaline phosphatase levels (p = .030). The presence of portal hypertension was associated with a higher mean fibrosis grade (1.6 vs. 0.8 in its absence; p = .026). In advanced SM, the presence of nodular regenerative hyperplasia (NRH) was associated with decreased overall survival (9.5 vs. 46.3 months, p = .002).ConclusionsMC infiltration induced polymorphic hepatic lesions and the degree of fibrosis is associated with portal hypertension. NRH identifies a poor prognosis subgroup of patients with advanced SM. Assessing liver histology can aid in SM prognostic evaluation.
Systemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been marginally described and accurate histological classification is critical, given the consequences of aggressive SM diagnosis. We aimed to describe the histological features associated with liver SM using updated tools. Using the database of the French Reference Centre for Mastocytosis, we retrospectively identified patients with a liver biopsy (LB) and a diagnosis of SM. All LB procedures were performed according to the local physician in charge and centrally reviewed by an expert pathologist. A total of 28 patients were included: 6 had indolent SM, 9 had aggressive SM, and 13 had SM with an associated hematologic neoplasm. Twenty-five (89%) patients presented hepatomegaly, and 19 (68%) had portal hypertension. The LB frequently showed slight sinusoid dilatation (82%). Fibrosis was observed in 3/6 indolent SM and in almost all advanced SM cases (21/22), but none of them showed cirrhosis. A high MC burden (>50 MCs/high-power field) was correlated with elevated blood alkaline phosphatase levels (p = .030). The presence of portal hypertension was associated with a higher mean fibrosis grade (1.6 vs. 0.8 in its absence; p = .026). In advanced SM, the presence of nodular regenerative hyperplasia (NRH) was associated with decreased overall survival (9.5 vs. 46.3 months, p = .002). MC infiltration induced polymorphic hepatic lesions and the degree of fibrosis is associated with portal hypertension. NRH identifies a poor prognosis subgroup of patients with advanced SM. Assessing liver histology can aid in SM prognostic evaluation.
Systemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been marginally described and accurate histological classification is critical, given the consequences of aggressive SM diagnosis. We aimed to describe the histological features associated with liver SM using updated tools.BACKGROUND AND AIMSSystemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been marginally described and accurate histological classification is critical, given the consequences of aggressive SM diagnosis. We aimed to describe the histological features associated with liver SM using updated tools.Using the database of the French Reference Centre for Mastocytosis, we retrospectively identified patients with a liver biopsy (LB) and a diagnosis of SM. All LB procedures were performed according to the local physician in charge and centrally reviewed by an expert pathologist.METHODSUsing the database of the French Reference Centre for Mastocytosis, we retrospectively identified patients with a liver biopsy (LB) and a diagnosis of SM. All LB procedures were performed according to the local physician in charge and centrally reviewed by an expert pathologist.A total of 28 patients were included: 6 had indolent SM, 9 had aggressive SM, and 13 had SM with an associated hematologic neoplasm. Twenty-five (89%) patients presented hepatomegaly, and 19 (68%) had portal hypertension. The LB frequently showed slight sinusoid dilatation (82%). Fibrosis was observed in 3/6 indolent SM and in almost all advanced SM cases (21/22), but none of them showed cirrhosis. A high MC burden (>50 MCs/high-power field) was correlated with elevated blood alkaline phosphatase levels (p = .030). The presence of portal hypertension was associated with a higher mean fibrosis grade (1.6 vs. 0.8 in its absence; p = .026). In advanced SM, the presence of nodular regenerative hyperplasia (NRH) was associated with decreased overall survival (9.5 vs. 46.3 months, p = .002).RESULTSA total of 28 patients were included: 6 had indolent SM, 9 had aggressive SM, and 13 had SM with an associated hematologic neoplasm. Twenty-five (89%) patients presented hepatomegaly, and 19 (68%) had portal hypertension. The LB frequently showed slight sinusoid dilatation (82%). Fibrosis was observed in 3/6 indolent SM and in almost all advanced SM cases (21/22), but none of them showed cirrhosis. A high MC burden (>50 MCs/high-power field) was correlated with elevated blood alkaline phosphatase levels (p = .030). The presence of portal hypertension was associated with a higher mean fibrosis grade (1.6 vs. 0.8 in its absence; p = .026). In advanced SM, the presence of nodular regenerative hyperplasia (NRH) was associated with decreased overall survival (9.5 vs. 46.3 months, p = .002).MC infiltration induced polymorphic hepatic lesions and the degree of fibrosis is associated with portal hypertension. NRH identifies a poor prognosis subgroup of patients with advanced SM. Assessing liver histology can aid in SM prognostic evaluation.CONCLUSIONSMC infiltration induced polymorphic hepatic lesions and the degree of fibrosis is associated with portal hypertension. NRH identifies a poor prognosis subgroup of patients with advanced SM. Assessing liver histology can aid in SM prognostic evaluation.
Background and Aims Systemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been marginally described and accurate histological classification is critical, given the consequences of aggressive SM diagnosis. We aimed to describe the histological features associated with liver SM using updated tools. Methods Using the database of the French Reference Centre for Mastocytosis, we retrospectively identified patients with a liver biopsy (LB) and a diagnosis of SM. All LB procedures were performed according to the local physician in charge and centrally reviewed by an expert pathologist. Results A total of 28 patients were included: 6 had indolent SM, 9 had aggressive SM, and 13 had SM with an associated hematologic neoplasm. Twenty‐five (89%) patients presented hepatomegaly, and 19 (68%) had portal hypertension. The LB frequently showed slight sinusoid dilatation (82%). Fibrosis was observed in 3/6 indolent SM and in almost all advanced SM cases (21/22), but none of them showed cirrhosis. A high MC burden (>50 MCs/high‐power field) was correlated with elevated blood alkaline phosphatase levels (p = .030). The presence of portal hypertension was associated with a higher mean fibrosis grade (1.6 vs. 0.8 in its absence; p = .026). In advanced SM, the presence of nodular regenerative hyperplasia (NRH) was associated with decreased overall survival (9.5 vs. 46.3 months, p = .002). Conclusions MC infiltration induced polymorphic hepatic lesions and the degree of fibrosis is associated with portal hypertension. NRH identifies a poor prognosis subgroup of patients with advanced SM. Assessing liver histology can aid in SM prognostic evaluation.
Author Neel, Antoine
Ranta, Dana
Viallard, Jean François
Wierzbicka‐Hainaut, Ewa
Hermine, Olivier
Lortholary, Olivier
Gourin, Marie Pierre
Durieu, Isabelle
Gourguechon, Clément
Molina, Thierry J
Rossignol, Julien
Castelain, Florence
Bodemer, Christine
Barete, Stéphane
Jaussaud, Roland
Damaj, Ghandi
Dimicoli‐Salazar, Sophie
Tournilhac, Olivier
Pelletier, Fabien
Neuraz, Antoine
Mallet, Vincent
Aouba, Achille
Greco, Celine
Arock, Michel
Launay, David
Hamidou, Mohamed
Le Mouel, Edwige
Zunic, Patricia
Hoarau, Cyrille
Gaudy‐Marqueste, Caroline
Gousseff, Marie
Javier, Rose‐Marie
Meni, Cécile
Bruneau, Julie
Dubreuil, Patrice
Soria, Angèle
Polivka, Laura
Torregrosa‐Diaz, Jose M
Lancesseur, Charles
Garcelon, Nicolas
Frenzel, Laurent
Retornaz, Frederique
Lavigne, Christian
Canioni, Danielle
Lemal, Richard
Heiblig, Maël
Madrange, Marine
Cabrera, Quentin
Pol, Stanislas
Agopian, Julie
Bulai‐Livideanu, Cristina
Terriou, Louis
Bouillet, Laurence
Brenet, Fabienne
Guilpain, Philippe
Ballul, Thomas
Lhermitte, Ludovic
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Issue 7
Keywords nodular regenerative hyperplasia
fibrosis
portal hypertension
mast cells
liver
advanced systemic mastocytosis
Language English
License Attribution
2024 The Authors. Liver International published by John Wiley & Sons Ltd.
Attribution: http://creativecommons.org/licenses/by
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Notes Julien Rossignol, Danielle Canioni Achille Aouba, Olivier Hermine and Ghandi Damaj are contributed equally.
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Snippet Background and Aims Systemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been...
Systemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been marginally described and...
Background and AimsSystemic mastocytosis (SM) is characterized by the accumulation of atypical mast cells (MCs) in organs. Liver histology of SM has been...
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SubjectTerms advanced systemic mastocytosis
Alkaline phosphatase
Biopsy
Cirrhosis
Diagnosis
Fibrosis
Histology
Hyperplasia
Hypertension
Life Sciences
Liver
Mast cells
Mastocytosis
Medical prognosis
nodular regenerative hyperplasia
Patients
portal hypertension
Subgroups
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Title Histological characterization of liver involvement in systemic mastocytosis
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