Use of oral vancomycin in children with autoimmune liver disease: A single centre experience
Angelo Di Giorgio1, Anna Tulone2, Emanuele Nicastro2
1Pediatric Hepatology, Gastroenterology and Transplantation, Hospital Papa Giovanni XXIII , Bergamo 24127, Italy. adigiorgio@asst-pg23.it.
Insights
Oral vancomycin treatment (OVT) effectively improved biochemical remission rates in children with autoimmune liver diseases (AILD) unresponsive to standard immunosuppressive therapy (IS). This suggests OVT is a valuable option for achieving remission in difficult AILD cases.
Area of Science:
- Pediatric Gastroenterology
- Hepatology
- Autoimmune Liver Diseases
Background:
- Limited data exists on oral vancomycin treatment (OVT) for autoimmune liver diseases (AILD) beyond primary sclerosing cholangitis.
- Experience with OVT in autoimmune hepatitis (AIH) and autoimmune sclerosing cholangitis (ASC) in children is particularly scarce.
Purpose of the Study:
- To evaluate the effectiveness of immunosuppressive treatment (IS) and OVT in pediatric patients diagnosed with AILD.
- To assess treatment response in children with AIH and ASC.
Main Methods:
- Retrospective analysis of pediatric AIH and ASC cases over 10 years.
- Patients received standard IS; non-responders were treated with OVT.
- Biochemical and immunological remission rates, and SCOPE index were assessed.
Main Results:
- Of 75 children (54 AIH, 21 ASC), 84% initially received IS, with 61 achieving biochemical remission.
- 16% not responding to IS, predominantly with ASC, received OVT, achieving biochemical remission in 8 patients.
- OVT increased overall biochemical remission from 81% to 92% and significantly reduced liver enzymes.
Conclusions:
- Pediatric AIH and ASC patients respond well to standard IS.
- OVT offers a promising therapeutic strategy for achieving biochemical remission in IS-refractory AILD.
- Further prospective studies are warranted to confirm OVT efficacy in AILD.
Background:
Previous reports showed some beneficial effect of oral vancomycin treatment (OVT) in children with primary sclerosing cholangitis; conversely, the experience in patients with other autoimmune liver diseases (AILD), including autoimmune hepatitis (AIH) and autoimmune sclerosing cholangitis (ASC), is scant.
Aim:
To assess the response to immunosuppressive treatment (IS) and to OVT in children diagnosed with AILD.
Methods:
Retrospective study of children diagnosed with AIH (normal biliary tree at cholangiography) and ASC (abnormal biliary tree at cholangiography) in the last 10 years. All underwent standard immunosuppressive therapy (IS), but non-responders received also OVT. Biochemical remission [normal aspartate aminotransferase (AST)] and immunological remission (normal IgG and negative autoantibodies) rates and Sclerosing Cholangitis Outcomes in Pediatrics (SCOPE) index were assessed and compared during the follow up.
Results:
75 children were included [69% female, median age 10.5 years (5.6-13.4 years), AIH = 54, ASC= 21]. Sixty-three patients (84%, AIH = 52, ASC = 11) were treated with standard IS and 61 achieved biochemical remission, whereas 12 not responding to IS [16%, F = 75%, median age 13.5 years, (12.2-15.7), 10 with ASC] required OVT and 8 achieved biochemical remission. Overall OVT increased the biochemical remission rate of the whole group of AILD patients from 81% (61/75) to 92% (69/75). Median values of AST, alanine aminotransferase (ALT) and gamma-glutamyl transferase (GGT) decreased significantly after OVT start (P < 0.05). Complete normalization of livers enzymes (AST, ALT and GGT) was observed in 6/12 patients (50%). Decrease in SCOPE index score was reported in 5/12 patients (42%). At last follow up (median of 4.4 years, range 0.6-13.8 years) all 75 patients are alive, 6 (8%, 1 with ASC) successfully discontinued medications, 1 (with ASC) required liver transplantation.
Conclusion:
Children with AIH and ASC respond well to IS treatment. OVT may represent a valuable treatment option to achieve biochemical remission in patients not responding to standard IS. These promising preliminary results suggest that a prospective study is indicated to define the efficacy of OVT in AILD.
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