Absence of Maternal Microchimerism in Regional Lymph Nodes of Children With Biliary Atresia

Engelmann, C; Maelzer, Madeleine; Kreyenberg, Hermann

doi:10.1097/mpg.0000000000001093

Cited by 4 publications

(5 citation statements)

References 38 publications

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“…Our data are consistent with the results of qPCR-based analyses of other tissues; Nelson et al identified maternal microchimerism in four of 24 blood samples of healthy children with the estimated number of maternal cells per 10 6 total cells ranging from 0 to 79 (11). In this regard, while Engelmann et al (7) did not detect maternal cells in any of the five liver lymph node samples obtained from patients with BA, this can be explained by the relatively high limit-of-detection of that study (0.1% maternal cells in total cells).…”

Section: Discussionsupporting

confidence: 91%

“…The frequency and quantitative levels of maternal microchimerism were comparable between the BA and control groups. These results, in conjunction with those of the previous study by Engelmann et al using liver lymph node samples (7), argue against a major role of maternal microchimerism in the etiology of BA. In this regard, recent studies have cast doubt on the etiological relationship between maternal microchimerism and various autoimmune disorders (15–17).…”

Section: Discussionsupporting

confidence: 81%

“…Considering the limited sensitivity of FISH and other methods used in the previous studies (2,(4)(5)(6)(7), the aforementioned results need to be validated. In this regard, Nelson et al (11) have developed a quantitative PCR (qPCR)-based method to quantify maternal noninherited HLA alleles in the genomic DNA of children.…”

Section: What Is Newmentioning

confidence: 99%

“…These results indicate that maternal microchimerism in the liver may cause immune reactions leading to BA. However, Engelmann et al (7) questioned this hypothesis, because their short tandem repeat and single nucleotide polymorphism analyses detected no maternal cells in liver lymph nodes obtained from five patients with BA.…”

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confidence: 99%

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Quantification of Maternal Microchimeric Cells in the Liver of Children With Biliary Atresia

Tamaoka¹,

Fukuda

Katoh‐Fukui³

et al. 2022

J. pediatr. gastroenterol. nutr.

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Biliary atresia (BA) is a rare disorder of unknown etiology. There is a debate as to whether maternal microchimerism plays a significant role in the development of BA or in graft tolerance after liver transplantation. Here, we performed quantitative-PCR-based assays for liver tissues of children with BA and other diseases. Maternal cells were detected in 4/13 and 1/3 of the BA and control groups, respectively. The estimated number of maternal cells ranged between 0 and 34.7 per 10 6 total cells. The frequency and severity of maternal microchimerism were similar between the BA and control groups, and between patients with and without acute rejection of maternal grafts. These results highlight the high frequency of maternal microchimerism in the liver. This study provides no evidence for roles of microchimerism in the etiology of BA or in graft tolerance. Thus, the biological consequences of maternal microchimerism need to be clarified in future studies.

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Section: Discussionsupporting

confidence: 91%

Section: Discussionsupporting

confidence: 81%

Section: What Is Newmentioning

confidence: 99%

mentioning

confidence: 99%

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Quantification of Maternal Microchimeric Cells in the Liver of Children With Biliary Atresia

Tamaoka¹,

Fukuda

Katoh‐Fukui³

et al. 2022

J. pediatr. gastroenterol. nutr.

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“…The lack of an identifiable genetic cause of BA has led to the hypothesis that maternal microchimerism (postzygotic somatic mutation) may be part of the etiology 17, 18 . Although a recent study has shown no evidence of maternal microchimerism in lymph nodes 19 , additional studies would be required to both demonstrate the presence of microchimerism in BA and show that it has a causal role in the disease.…”

Section: Genetic Susceptibility In Biliary Atresiamentioning

confidence: 95%

Recent advances in understanding biliary atresia

2019

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Biliary atresia (BA) is a neonatal liver disease characterized by progressive obstruction and fibrosis of the extrahepatic biliary tree as well as fibrosis and inflammation of the liver parenchyma. Recent studies found that infants who will go on to develop BA have elevated direct bilirubin levels in the first few days of life, suggesting that the disease starts in utero. The etiology and pathogenesis of BA, however, remain unknown. Here, we discuss recent studies examining potential pathogenetic mechanisms of BA, including genetic susceptibility, involvement of the immune system, and environmental insults such as viruses and toxins, although it is possible that there is not a single etiological agent but rather a large group of injurious insults that result in a final common pathway of extrahepatic bile duct obstruction and liver fibrosis. The management and diagnosis of BA have not advanced significantly in the past decade, but given recent advances in understanding the timing and potential pathogenesis of BA, we are hopeful that the next decade will bring early diagnostics and novel therapeutics.

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Unique manifestations of biliary atresia provide new immunological insight into its etiopathogenesis

2017

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Biliary atresia (BA) is a unique cholestatic disease of newborns with a background of exaggerated immune response in the liver of unknown mechanism. Three hypotheses have been proposed; autoimmune type of cholangiopathy triggered by virus infection, graft-versus-host disease type of immune-mediated disease associated with maternal microchimerism and ductal plate malformation theory. Researchers on virus infection theory have experimentally explored immune process causing cholangiopathy on murine models of this disease, while in maternal microchimerism hypothesis were detected maternal cells in the BA patients' liver, of which roles are yet to be determined. Ductal plate malformation theory is an intriguing hypothesis in the sense that it suggests the onset of this disease is in the first trimester. This theory can be secondary to either one of these two immune-related insults. In this review, four unique points are focused; (1) the timing of onset, (2) hepatitis-like pathological picture, (3) heterogenous atrophy of the liver segments when advanced, and (4) swollen lymph nodes at the porta hepatis. These unique clinicopahtological aspects of this disease should be well explained by these hypotheses.

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Absence of Maternal Microchimerism in Regional Lymph Nodes of Children With Biliary Atresia

Abstract: This result challenges the hypothesis that maternal cells play a role in hilar lymphadenopathy of children with BA.

Cited by 4 publications

References 38 publications

Quantification of Maternal Microchimeric Cells in the Liver of Children With Biliary Atresia

Quantification of Maternal Microchimeric Cells in the Liver of Children With Biliary Atresia

Recent advances in understanding biliary atresia

Unique manifestations of biliary atresia provide new immunological insight into its etiopathogenesis

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