Reovirus type-2–triggered autoimmune cholangitis in extrahepatic bile ducts of weanling DBA/1J mice

Nakashima, Tomomi; Hayashi, Toshiharu; Tomoeda, Saki; Yoshino, Midori; Mizuno, Takuya

doi:10.1038/pr.2013.170

Cited by 13 publications

(10 citation statements)

References 32 publications

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“…On the other hand, Nakashima et al experimen tally found that reovirustypeII infection in newborn mice triggers an autoimmune inflammation induced by Thelper lymphocytes, which they claimed are seen in the bile ducts in BA [8].…”

Section: Discussionmentioning

confidence: 99%

Whole exome sequencing analysis for mutations in isolated type III biliary atresia patients

Gürünlüoğlu¹,

Koç²,

Durmuş³

et al. 2020

ceh

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Aim of the study: Biliary atresia is an idiopathic, destructive disease that affects both extrahepatic and intrahepatic bile ducts with severe inflammation and manifests as progressive jaundice within the first few months of life. In this study, we aimed to investigate the significance of genetic mutations in the onset of biliary atresia disease. Material and methods: With the approval of the ethics committee and parental consent, blood was taken from patients to obtain their DNA, and the study commenced. In this prospective study, we examined the DNA of 10 patients with no disease other than biliary atresia, and an exome sequence analysis was performed with the new-generation DNA sequencing method. The genetic structure of biliary atresia disease was examined by statistical analysis of the mutations, which were determined according to the reference DNA sequencing. Results: In the exome sequence analysis, the number of mutations detected among the patients changed significantly; the lowest number was 12,591, and the maximum was 19,863. By examining these mutations, we identified the mutated genes that were common to all patients. Conclusions: In this study, the highest mutation rates were detected in the PRIM2 and MAP2K3 genes. These genes have not previously been associated with biliary atresia.

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Section: Discussionmentioning

confidence: 99%

Whole exome sequencing analysis for mutations in isolated type III biliary atresia patients

Gürünlüoğlu¹,

Koç²,

Durmuş³

et al. 2020

ceh

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“…In murine models, pre- or perinatal infections with rotavirus (16, 17), cytomegalovirus (15), and reovirus (18) were shown to infect and damage bile duct epithelia giving support to the hypothesis of a primary cholangiotropic viral infection as the initiating event of BA. In humans, however, studies focusing on the detection of viral infections at the time of diagnosis have produced conflicting results (19).…”

Section: Introductionmentioning

confidence: 90%

Neonatal Cholestasis â€“ Differential Diagnoses, Current Diagnostic Procedures, and Treatment

et al. 2015

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Cholestatic jaundice in early infancy is a complex diagnostic problem. Misdiagnosis of cholestasis as physiologic jaundice delays the identification of severe liver diseases. In the majority of infants, prolonged physiologic jaundice represent benign cases of breast milk jaundice, but few among them are masked and caused by neonatal cholestasis (NC) that requires a prompt diagnosis and treatment. Therefore, a prolonged neonatal jaundice, longer than 2 weeks after birth, must always be investigated because an early diagnosis is essential for appropriate management. To rapidly identify the cases with cholestatic jaundice, the conjugated bilirubin needs to be determined in any infant presenting with prolonged jaundice at 14 days of age with or without depigmented stool. Once NC is confirmed, a systematic approach is the key to reliably achieve the diagnosis in order to promptly initiate the specific, and in many cases, life-saving therapy. This strategy is most important to promptly identify and treat infants with biliary atresia, the most common cause of NC, as this requires a hepatoportoenterostomy as soon as possible. Here, we provide a detailed work-up approach including initial treatment recommendations and a clinically oriented overview of possible differential diagnoses in order to facilitate the early recognition and a timely diagnosis of cholestasis. This approach warrants a broad spectrum of diagnostic procedures and investigations including new methods that are described in this review.

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“…The authors further demonstrated that antibody is protective against RRV-induced BA and that early viral clearance is likely a critical determinant of disease. (14, 15) Nakashima et al reported that hepatobiliary pathology with inoculation of DBA/1J mouse pups with Reovirus-2 mimicks that of human BA as well as that of RRV-induced BA. (14) Past studies demonstrated the role of T-, B- and natural killer (NK) cells in the immunologic destruction of the extrahepatic bile duct in part mediated by Interferon-γ, IL-2, Tumor Necrosis Factor-α and IL-12.…”

Section: Pathogenesis Of Biliary Atresiamentioning

confidence: 99%

“…(14, 15) Nakashima et al reported that hepatobiliary pathology with inoculation of DBA/1J mouse pups with Reovirus-2 mimicks that of human BA as well as that of RRV-induced BA. (14) Past studies demonstrated the role of T-, B- and natural killer (NK) cells in the immunologic destruction of the extrahepatic bile duct in part mediated by Interferon-γ, IL-2, Tumor Necrosis Factor-α and IL-12. (16) Brindley et al showed a robust hepatic T-cell memory with significant increase in Interferon-γ in response to Cytomegalovirus (CMV) exposure, suggesting perinatal CMV exposure may be a possible initiator to BA.…”

Section: Pathogenesis Of Biliary Atresiamentioning

confidence: 99%

Recent advances in the pathogenesis and management of biliary atresia

Zagory

Nguyen

Wang

2015

Current Opinion in Pediatrics

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Purpose of review The purpose of this study is to review advances in both the pathogenesis and clinical management of biliary atresia (BA). Recent Findings Immunologic studies have further characterized roles of helper T-cells, B-cells, and natural killer cells in the immune dysregulation following viral replication within and damage of biliary epithelium. PROMININ-1 expressing portal fibroblasts may play an integral role in the biliary fibrosis associated with BA. A number of genetic polymorphisms have been characterized as leading to susceptibility for BA. Postoperative corticosteroid therapy is not associated with greater transplant-free survival. Newborn screening may improve outcomes of infants with BA and may also provide a long-term cost benefit. Summary Although recent advances have enhanced our understanding of pathogenesis and clinical management, BA remains a significant challenge requiring further investigation.

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Reovirus type-2–triggered autoimmune cholangitis in extrahepatic bile ducts of weanling DBA/1J mice

Cited by 13 publications

References 32 publications

Whole exome sequencing analysis for mutations in isolated type III biliary atresia patients

Whole exome sequencing analysis for mutations in isolated type III biliary atresia patients

Neonatal Cholestasis â€“ Differential Diagnoses, Current Diagnostic Procedures, and Treatment

Recent advances in the pathogenesis and management of biliary atresia

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