RÉSUMÉ
INTRODUCTION: Aetiology of protein-losing enteropathy in single-ventricle type CHD is multi-factorial. REPORT: We describe two Fontan patients with protein-losing enteropathy who presented with cytomegalovirus-associated colitis. DISCUSSION: Fontan patients display risk factors for cytomegalovirus-induced gastroenteropathy that may affect lymph angiogenesis, disease development, and progression. CONCLUSION: Cytomegalovirus enteropathy may be common among Fontan patients who suffer from protein-losing enteropathy. Polymerase chain reaction is important for detection.
Sujet(s)
Colite/étiologie , Infections à cytomégalovirus/diagnostic , Procédure de Fontan/effets indésirables , Entéropathie exsudative/étiologie , Enfant , Enfant d'âge préscolaire , Colite/virologie , Infections à cytomégalovirus/complications , Cardiopathies congénitales/chirurgie , Humains , Mâle , Complications postopératoires , Entéropathie exsudative/virologie , Facteurs de risqueSujet(s)
Infections à cytomégalovirus/complications , Cytomegalovirus/isolement et purification , Entéropathie exsudative/étiologie , Adulte , Anticorps antiviraux/sang , Anticorps antiviraux/immunologie , Infections à cytomégalovirus/diagnostic , Diagnostic différentiel , Duodénite/étiologie , Urgences , Infections à virus Epstein-Barr/complications , Herpèsvirus humain de type 4/isolement et purification , Herpèsvirus humain de type 4/physiologie , Humains , Hypoalbuminémie/étiologie , Immunocompétence , Immunoglobuline M/sang , Immunoglobuline M/immunologie , Hyperlymphocytose/étiologie , Mâle , Épanchement péricardique/étiologie , Péritonite/diagnostic , Entéropathie exsudative/virologie , Latence viraleSujet(s)
Adénocarcinome/thérapie , Infections à cytomégalovirus/complications , Entérite/virologie , Maladies du jéjunum/virologie , Complications postopératoires/virologie , Entéropathie exsudative/virologie , Tumeurs de l'estomac/thérapie , Chimioradiothérapie adjuvante , Infections à cytomégalovirus/traitement médicamenteux , Entérite/anatomopathologie , Gastrectomie , Humains , Sujet immunodéprimé , Maladies du jéjunum/anatomopathologie , Mâle , Adulte d'âge moyen , Complications postopératoires/anatomopathologie , Entéropathie exsudative/anatomopathologieRÉSUMÉ
Ménétrier's disease is a childhood protein-losing gastroenteropathy characterized by hypertrophy of the gastric mucosa, of unknown etiology, although most of reported cases have been associated with viral infections. Clinical manifestation is edema and biochemically there are hypoproteinemia and hypoalbuminemia. This disease is very rare in children and they have a benign and self-limiting course in contrast to adults where tend to be chronic and occasionally to become malignant. We present a child with Ménétrier disease with edema and ascites possibly associated with a cytomegalovirus infection.
Sujet(s)
Infections à cytomégalovirus/complications , Gastrite hypertrophique/virologie , Entéropathie exsudative/virologie , Enfant d'âge préscolaire , Humains , MâleRÉSUMÉ
La enfermedad de Ménétrier infantil es una gastroenteropatía perdedora de proteínas caracterizada por hipertrofa de la mucosa gástrica, de causa desconocida, aunque la mayoría de los casos informados se han asociado a infecciones virales. Se manifesta clínicamente por edema y bioquímicamente por hipoproteinemia e hipoalbuminemia. Es poco común en los niños, en quienes tiene un carácter benigno y autolimitado, a diferencia de los adultos, en los que tiende a cronifcarse y, en ocasiones, a malignizarse. Se presenta un caso de enfermedad de Ménétrier infantil con síndrome ascítico edematoso posiblemente asociado a una infección por citomegalovirus.
Ménétrier's disease is a childhood protein-losing gastroenteropathy characterized by hypertrophy of the gastric mucosa, of unknown etiology, although most of reported cases have been associated with viral infections. Clinical manifestation is edema and biochemically there are hypoproteinemia and hypoalbuminemia. This disease is very rare in children and they have a benign and self-limiting course in contrast to adults where tend to be chronic and occasionally to become malignant. We present a child with Ménétrier disease with edema and ascites possibly associated with a cytomegalovirus infection.
Sujet(s)
Enfant d'âge préscolaire , Humains , Mâle , Infections à cytomégalovirus/complications , Gastrite hypertrophique/virologie , Entéropathie exsudative/virologieRÉSUMÉ
La enfermedad de Ménétrier infantil es una gastroenteropatía perdedora de proteínas caracterizada por hipertrofa de la mucosa gástrica, de causa desconocida, aunque la mayoría de los casos informados se han asociado a infecciones virales. Se manifesta clínicamente por edema y bioquímicamente por hipoproteinemia e hipoalbuminemia. Es poco común en los niños, en quienes tiene un carácter benigno y autolimitado, a diferencia de los adultos, en los que tiende a cronifcarse y, en ocasiones, a malignizarse. Se presenta un caso de enfermedad de Ménétrier infantil con síndrome ascítico edematoso posiblemente asociado a una infección por citomegalovirus.(AU)
Ménétriers disease is a childhood protein-losing gastroenteropathy characterized by hypertrophy of the gastric mucosa, of unknown etiology, although most of reported cases have been associated with viral infections. Clinical manifestation is edema and biochemically there are hypoproteinemia and hypoalbuminemia. This disease is very rare in children and they have a benign and self-limiting course in contrast to adults where tend to be chronic and occasionally to become malignant. We present a child with Ménétrier disease with edema and ascites possibly associated with a cytomegalovirus infection.(AU)
Sujet(s)
Enfant d'âge préscolaire , Humains , Mâle , Infections à cytomégalovirus/complications , Gastrite hypertrophique/virologie , Entéropathie exsudative/virologieRÉSUMÉ
Ménétriers disease is a childhood protein-losing gastroenteropathy characterized by hypertrophy of the gastric mucosa, of unknown etiology, although most of reported cases have been associated with viral infections. Clinical manifestation is edema and biochemically there are hypoproteinemia and hypoalbuminemia. This disease is very rare in children and they have a benign and self-limiting course in contrast to adults where tend to be chronic and occasionally to become malignant. We present a child with Ménétrier disease with edema and ascites possibly associated with a cytomegalovirus infection.
Sujet(s)
Infections à cytomégalovirus/complications , Gastrite hypertrophique/virologie , Entéropathie exsudative/virologie , Enfant d'âge préscolaire , Humains , MâleRÉSUMÉ
A 14 year old common variable immunodeficiency patient developed severe protein-losing enteropathy. A chronic enteral infection with human parechovirus type 1 and norovirus was diagnosed. Treatment strategies aimed at virus eradication and providing supportive care were ineffective. The antipicornavirus agent pleconaril did not have any effect on viral replication. Symptoms improved on immunosuppressive therapy, suggesting infection-related immune dysregulation in an immunocompromised host.
Sujet(s)
Agammaglobulinémie/complications , Antiviraux/pharmacologie , Résistance virale aux médicaments , Oxadiazoles/pharmacologie , Parechovirus/effets des médicaments et des substances chimiques , Infections à Picornaviridae/virologie , Entéropathie exsudative/virologie , Adolescent , Infections à Caliciviridae/complications , Infections à Caliciviridae/virologie , Maladie chronique , Fèces/virologie , Humains , Sujet immunodéprimé , Mâle , Norovirus/isolement et purification , Oxazoles , Parechovirus/isolement et purificationRÉSUMÉ
An immunocompetent 8-year-old boy with cytomegalovirus (CMV)-associated transient protein-losing enteropathy (PLE) is described. Colonoscopic examination revealed lymphoid hyperplasia of the terminal ileum. Histological examination of the biopsied specimens showed marked dilation of the lymphatic vessels. Primary CMV infection was demonstrated by serological test and polymerase chain reaction. The child had complete resolution of the disease without antiviral treatment. The present case suggests the etiologic role of CMV infection in PLE resulting from intestinal lymphangiectasia in childhood.
Sujet(s)
Infections à cytomégalovirus/diagnostic , Cytomegalovirus/isolement et purification , Lymphangiectasie intestinale/diagnostic , Entéropathie exsudative/diagnostic , Anticorps antiviraux/sang , Enfant , Cytomegalovirus/génétique , Infections à cytomégalovirus/complications , Infections à cytomégalovirus/virologie , ADN viral/sang , Oedème , Humains , Iléum/anatomopathologie , Iléum/virologie , Immunocompétence , Muqueuse intestinale/anatomopathologie , Muqueuse intestinale/virologie , Lymphangiectasie intestinale/complications , Mâle , Réaction de polymérisation en chaîne , Entéropathie exsudative/étiologie , Entéropathie exsudative/virologieRÉSUMÉ
Cytomegalovirus infection of the gastro-intestinal tract is frequent and may be serious in the immunocompromised patient. We report a case of cytomegalovirus infection in an immunocompetent young man who presented total food intolerance, pleural effusion and oedema as the result of severe protein losing hypertrophic gastropathy. Hypertrophic gastropathy with severe mucosal protein loss has been described in Menetrier's disease, a condition of unknown cause which involves foveolar hyperplasia of the gastric mucosa. Related hypoalbuminemia is responsible for a clinical picture of diffuse edema. In adults, the natural course of the disease is marked by a chronic course and carries a bad prognosis. In our case, the disease ran a protracted disabling course, likely shortened by ganciclovir therapy, followed by slow clinical, endoscopic and biochemical resolution after several' months course.
Sujet(s)
Infections à cytomégalovirus/diagnostic , Gastrite hypertrophique/virologie , Immunocompétence/physiologie , Adulte , Antiviraux/usage thérapeutique , Oedème/virologie , Études de suivi , Ganciclovir/usage thérapeutique , Muqueuse gastrique/virologie , Humains , Mâle , Épanchement pleural/virologie , Entéropathie exsudative/virologieRÉSUMÉ
A 3-year-old boy developed transient protein-losing gastroenteropathy associated with cytomegalovirus (CMV) infection. Both IgG and IgM antibodies to CMV were positive in a serologic blood test. Upper gastrointestinal endoscopy showed multiple erosions throughout the body of the stomach, without enlarged gastric folds. Histological examination of the biopsy specimens indicated eosinophilic gastroenteritis and CMV infection. The patient had complete resolution without specific therapy for CMV in four weeks. An allergic reaction as well as CMV infection played important roles in the pathogenesis of this case.
Sujet(s)
Anticorps antiviraux/sang , Infections à cytomégalovirus/complications , Cytomegalovirus/immunologie , Éosinophilie/complications , Hypersensibilité alimentaire/complications , Gastroentérite/complications , Immunocompétence , Entéropathie exsudative/virologie , Enfant d'âge préscolaire , Infections à cytomégalovirus/immunologie , Infections à cytomégalovirus/anatomopathologie , Éosinophilie/immunologie , Éosinophilie/anatomopathologie , Éosinophilie/virologie , Hypersensibilité alimentaire/immunologie , Hypersensibilité alimentaire/anatomopathologie , Gastroentérite/immunologie , Gastroentérite/anatomopathologie , Gastroentérite/virologie , Humains , Immunoglobuline G/sang , Immunoglobuline M/sang , Mâle , Entéropathie exsudative/immunologie , Entéropathie exsudative/anatomopathologie , Estomac/immunologie , Estomac/anatomopathologie , Estomac/virologieRÉSUMÉ
Protein-losing gastropathy is an uncommon disease of uncertain etiology, known also as Menetrier's disease. In medical literature only 50 pediatric cases have been described. These childhood forms, in contrast to classic adult Menetrier's disease, have a typical benign and transient course, and require only supportive therapy. The role of Cytomegalovirus (CMV) in the pathogenesis has been demonstrated by gastric biopsy in one third of the cases. Also other infectious, allergic and immunological factors have been hypothesized. We describe a case of hypertrophic gastropathy with important protein-loss, admitted to our Pediatric Department for evaluation because of vomit, weight loss, abdominal pain and hypoalbuminemia. Gastric mucosal biopsy revealed a morphological evidence of CMV infection.
Sujet(s)
Infections à cytomégalovirus/complications , Cytomegalovirus/pathogénicité , Gastrite hypertrophique/virologie , Entéropathie exsudative/virologie , Hormones corticosurrénaliennes/usage thérapeutique , Biopsie , Enfant d'âge préscolaire , Infections à cytomégalovirus/diagnostic , Infections à cytomégalovirus/traitement médicamenteux , Diagnostic différentiel , Femelle , Muqueuse gastrique/anatomopathologie , Gastrite hypertrophique/diagnostic , Gastrite hypertrophique/traitement médicamenteux , Humains , Entéropathie exsudative/diagnostic , Entéropathie exsudative/traitement médicamenteux , Résultat thérapeutiqueRÉSUMÉ
Transient protein-losing hypertrophic gastropathy with similarity to Ménétrier's disease is described. Acute infection with cytomegalovirus (CMV) could be shown to play a causative role. Immunodeficiency was ruled out. The 34-year-old patient had complete resolution of the disease without antiviral treatment. To our knowledge the present report is the first case of CMV-associated protein-losing hypertrophic gastropathy in an immunocompetent adult. To date, a similar disorder has only been described in children. CMV infection should be considered in patients with acute and symptomatic protein loss of gastrointestinal origin.