Literature DB >> 23278674

Refractory childhood pemphigoid successfully treated with rituximab.

Irene Fuertes1, Jesús Luelmo, Lorena Leal, Jorge Romaní, Silvia Sánchez, José M Mascaró.   

Abstract

Bullous pemphigoid is an acquired autoimmune blistering disorder affecting mostly elderly people that is rare in children. There have been fewer than 100 cases of childhood bullous pemphigoid reported. Childhood bullous pemphigoid (CBP) generally has a good prognosis. Remission is usually achieved within several weeks to a few months, and relapses are rare, although refractory cases can occur, and the disease may be life threatening, particularly when lesions generalize. Herein we describe the case of an infant with severe CBP refractory to multiple treatments. In our case, therapy with azathioprine and intravenous immunoglobulin resulted in a slight clinical response, but only combined with high doses of prednisolone. Administration of rituximab led to a complete clinical response and allowed almost complete steroid tapering. There are no current guidelines indicating what doses and frequency of rituximab are safest or most effective in children with blistering diseases.
© 2012 Wiley Periodicals, Inc.

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Year:  2012        PMID: 23278674     DOI: 10.1111/pde.12057

Source DB:  PubMed          Journal:  Pediatr Dermatol        ISSN: 0736-8046            Impact factor:   1.588


  5 in total

1.  Bullous pemphigoid in infant post vaccination: Myth or reality?

Authors:  Iria Neri; Antonella Greco; Andrea Bassi; Jacinto Orgaz-Molina; Riccardo Balestri; Teresa Oranges; Annalisa Patrizi; Maurizio de Martino
Journal:  Int J Immunopathol Pharmacol       Date:  2015-12-18       Impact factor: 3.219

2.  [Bullous pemphigoid: a new look at a well-known disease].

Authors:  G Balakirski; H F Merk; M Megahed
Journal:  Hautarzt       Date:  2014-12       Impact factor: 0.751

Review 3.  Bullous Diseases in Children: A Review of Clinical Features and Treatment Options.

Authors:  Brittney Schultz; Kristen Hook
Journal:  Paediatr Drugs       Date:  2019-10       Impact factor: 3.022

4.  Rituximab, Omalizumab, and Dupilumab Treatment Outcomes in Bullous Pemphigoid: A Systematic Review.

Authors:  Peng Cao; Wenjing Xu; Litao Zhang
Journal:  Front Immunol       Date:  2022-06-13       Impact factor: 8.786

5.  Bullous pemphigoid in infants: characteristics, diagnosis and treatment.

Authors:  Agnes Schwieger-Briel; Cornelia Moellmann; Birgit Mattulat; Franziska Schauer; Dimitra Kiritsi; Enno Schmidt; Cassian Sitaru; Hagen Ott; Johannes S Kern
Journal:  Orphanet J Rare Dis       Date:  2014-12-10       Impact factor: 4.123

  5 in total

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