Transient Neonatal Liver Disease After Maternal Antenatal Intravenous Ig Infusions in Gestational Alloimmune Liver Disease Associated With Neonatal Haemochromatosis

Baruteau, Julien; Heissat, Sophie; Broué, Pierre; Collardeau‐Frachon, Sophie; Bouvier, Raymonde; Fabrè, Monique; Dębiec, Hanna; Ronco, Pierre; Uzan, M; Narcy, P; Cordier, Marie‐Pierre; Lachaux, Alain; Lamireau, T.; Elleau, Christophe; Filet, J P; Mitanchez, Delphine; Dupuy, Marie-Pierre; Salaün, Jean François; Odent, Sylvie; Davison, James; Debray, Dominique; Guigonis, Vincent

doi:10.1097/mpg.0000000000000514

Cited by 16 publications

(8 citation statements)

References 48 publications

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“…So, in addition to the several possible mechanisms of action cited above for alloimmune thrombocytopenia, inhibition of activation of the terminal complement cascade may be important in the action of antenatal IVIG therapy in GALD. Whatever the mechanism, wide experience suggests that it is effective in limiting the effect of GALD in pregnancies at risk [11,15,[21][22][23][24][25][26][27]. Although the extent of protection from fetal liver injury in GALD cannot be ascertained to the same degree as in alloimmune thrombocytopenia, where the platelet count is a direct measure of effectiveness, the results in the present study clearly demonstrate improved outcomes with IVIG treatment.…”

Section: Discussionmentioning

confidence: 54%

Antenatal Treatment with Intravenous Immunoglobulin to Prevent Gestational Alloimmune Liver Disease: Comparative Effectiveness of 14-Week versus 18-Week Initiation

et al. 2017

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Background: Antenatal therapy with high-dose intravenous immunoglobulin (IVIG) may prevent gestational alloimmune liver disease (GALD). Objective: The objective of this study was to determine the effectiveness of this approach in a large cohort of women at risk for poor pregnancy outcome due to GALD. Methods: Women with a history of affected offspring were provided antenatal IVIG treatment and data were acquired prospectively from 1997 to 2015. The outcomes of treated pregnancies were compared to those of untreated pregnancies, and the effectiveness of starting at 14 weeks was compared to that of starting at 18 weeks. Results: A total of 188 treated pregnancies in 151 women were analyzed. Only 30% (n = 105) of untreated gestations resulted in healthy offspring as compared to 94% (n = 177) of treated pregnancies (p < 0.0001). Treated gestations of both the 14-week (n = 108) and the 18-week (n = 80) start cohort showed a decreased rate of fetal loss relative to untreated gestations (p < 0.0001). Equivalent outcomes were recorded in the 18-week versus the 14-week start cohort (p > 0.05). Few adverse events or complications of antenatal therapy were recorded. Conclusion: Antenatal therapy with high-dose IVIG initiated at either 18 or 14 gestational weeks effectively prevents poor outcome of pregnancies at risk for GALD.

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Section: Discussionmentioning

confidence: 54%

Antenatal Treatment with Intravenous Immunoglobulin to Prevent Gestational Alloimmune Liver Disease: Comparative Effectiveness of 14-Week versus 18-Week Initiation

et al. 2017

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“…As such, preventing the recurrence of NH is very important. It has been reported that high‐dose Ig for recurrent NH is safe and effective, but the present case has highlighted two issues regarding this treatment that had been unclear.…”

Section: Discussionmentioning

confidence: 81%

Antenatal immunoglobulin for prevention of neonatal hemochromatosis

Okada

Ihara

Urahashi

et al. 2016

Pediatrics International

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Neonatal hemochromatosis (NH) is a rare disease with a poor prognosis, particularly prior to 2008. Antenatal maternal high-dose immunoglobulin (Ig) is effective in preventing NH recurrence, but the adverse effects of this treatment have not been documented as yet. Here, we report on a patient who underwent high-dose Ig treatment to prevent NH recurrence. The patient was a 31-year-old pregnant Japanese woman. Her first child died of NH after receiving living donor liver transplantation. The patient received high-dose Ig treatment to prevent recurrence of NH from gestational weeks 16 to 35. During the treatment, platelet count gradually decreased, and cesarean section was required at 35 gestational weeks. The child did not develop liver failure. High-dose Ig prevented the recurrence of NH. Caution should be exercised due to possible adverse effects of this treatment.

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“…Neonatal hemochromatosis is a clinical syndrome in which liver disease in the newborn is accompanied by siderosis of hepatic and extrahepatic tissues in the pattern seen in hereditary hemochromatosis. 1,5,8 Studies suggested a genetic prevalence of 0.03-0.038 or a heterozygosity prevalence of 6-7%. 10 Gestational alloimmune liver disease is a materno-fetal alloimmune disorder directed at the fetal liver, often producing neonatal liver failure.…”

Section: Discussionmentioning

confidence: 99%

“…

Neonatal hemochromatosis (NH) is a rare fatal liver disease accompanied by hepatic and extrahepatic iron overload. [1][2][3] Gestational alloimmune liver disease (GALD) is a materno-fetal alloimmune disorder and leading cause of NH. 2,4,5 This condition allows an interplay between the maternal adaptive immune system and the fetus, resulting in an allograft to the mother.

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mentioning

confidence: 99%

Neonatal hemochromatosis attributed to gestational alloimmune liver disease treated with intravenous immunoglobulin and exchange transfusion therapy: an evidence-based case report

Iskandar

Jamarin

Mulyana³

2021

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Neonatal hemochromatosis (NH) is a rare fatal liver disease accompanied by hepatic and extrahepatic iron overload.1-3 Gestational alloimmune liver disease (GALD) is a materno-fetal alloimmune disorder and leading cause of NH.2,4,5 This condition allows an interplay between the maternal adaptive immune system and the fetus, resulting in an allograft to the mother. The mother becomes sensitized to an alloantigen expressed by the fetus and forms specific reactive antibodies. Immunoglobulin G (IgG) is transported through the placenta and attacks the fetal hepatocytes, resulting in severe loss of hepatocytes and fetal iron overload.3,6 Liver transplantation has been the only definitive treatment for NH for many years, with a survival rate of ±35%. Conventional therapy containing antioxidants and chelation agents reportedly have very poor success, with survival rate of only 10-20%. A new treatment paradigm involving intravenous immunoglobulin (IVIG) and exchange transfusion (ET) therapy has shown significant success in survival rate in NH, decreasing the need for liver transplantation.3,7,8 Here we present a case of NH caused by GALD and treated successfully with a combination of IVIG therapy and ET. We also aimed to evaluate the efficacy of IVIG and ET therapy for NH.

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Transient Neonatal Liver Disease After Maternal Antenatal Intravenous Ig Infusions in Gestational Alloimmune Liver Disease Associated With Neonatal Haemochromatosis

Abstract: Antenatal treatment with IVIG infusion in women at risk for gestational alloimmune disease recurrence improves the outcome of pregnancies despite mild signs of transient neonatal liver disease.

Cited by 16 publications

References 48 publications

Antenatal Treatment with Intravenous Immunoglobulin to Prevent Gestational Alloimmune Liver Disease: Comparative Effectiveness of 14-Week versus 18-Week Initiation

Antenatal Treatment with Intravenous Immunoglobulin to Prevent Gestational Alloimmune Liver Disease: Comparative Effectiveness of 14-Week versus 18-Week Initiation

Antenatal immunoglobulin for prevention of neonatal hemochromatosis

Neonatal hemochromatosis attributed to gestational alloimmune liver disease treated with intravenous immunoglobulin and exchange transfusion therapy: an evidence-based case report

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