Sambon, Pauline
[UCL]
Sharat, Varma
[UCL]
Komuta, Mina
[UCL]
Clapuyt, Philippe
[UCL]
Sokal, Etienne
[UCL]
Background and Aims: Concomitant presence of autoimmune hepatitis (AIH) and primary sclerosing cholangitis (PSC) is labelled as AIH-PSC overlap syndrome or autoimmune sclerosing cholangitis (ASC). Treatment of AIH with corticosteroids and azathioprine; and of the PSC component with ursodeoxycholic acid (UDCA) is the standard practice. Antibiotics are increasingly being shown to have benefit in PSC but their role in paediatric ASC is not well evaluated. We investigated the response to oral antibiotics as initial or subsequent therapy in children with ASC. Methods: Patients diagnosed with ASC on basis of biochemical, liver biopsy and radiology findings were included. They received metronidazole or vancomycin for 14 days [10-220] either at diagnosis (i.e. initial therapy) or during their maintenance period. When antibiotics were administered as initial therapy, steroid free induction regime was adopted. In children during the maintenance phase antibiotics were administered if they had not achieved biochemical remission with their standard treatment of steroids, azathioprine and UDCA. The outcome parameters to assess the efficacy of antibiotics were achievement of biochemical remission and additionally steroid avoidance when given in the initial therapy. Results: Ten children with ASC were included, of which 6 received oral antibiotics (4 metronidazole, 2 vancomycin) at diagnosis and 4 received metronidazole during the maintenance period. All patients showed a significant decrease in their AST (-55%, p=0,005), ALT (-84%, p=0,003) and GGT (-53%, p=0,003), without significant difference across the two groups. All six children in the initial therapy group did not need corticosteroids and continued to be in remission until last follow up duration of 400 days [216-888]. Among the four children administered antibiotics in the maintenance phase, two showed biochemical remission and steroids could be tapered; while two did not show any benefit. There was transient biochemical relapse after stopping antibiotics in one responder, for which they were restarted and continued until last follow up while continuing to be in remission. Conclusions: We demonstrate the benefit of antibiotics in ASC by achieving steroid free treatment when given at diagnosis as induction regime. When given in the maintenance phase they assist in achieving long term biochemical remission in an otherwise uncontrolled ASC


Bibliographic reference |
Sambon, Pauline ; Sharat, Varma ; Komuta, Mina ; Clapuyt, Philippe ; Sokal, Etienne. Antibiotics induce remission in pediatric PSC-AIH overlap syndrome allowing corticosteroid-free therapy .50th Annual Meeting ESPGHAN (Prague, Czech Republic, du 10/05/2017 au 13/05/2017). |
Permanent URL |
http://hdl.handle.net/2078.1/182542 |