Literature DB >> 25685271

Rare case of dysphagia, skin blistering, missing nails in a young boy.

Jasbir Makker1, Bharat Bajantri1, Prospere Remy1.   

Abstract

Epidermolysis bullosa is a group of genetic disorders with an autosomal dominant or an autosomal recessive mode of inheritance and more than 300 mutations. The disorder is characterized by blistering mucocutaneous lesions and has several varying phenotypes due to anchoring defect between the epidermis and dermis. The variation in phenotypic expression depends on the involved structural protein that mediates cell adherence between different layers of the skin. Epidermolysis bullosa can also involve extra-cutaneous sites including eye, nose, ear, upper airway, genitourinary tract and gastrointestinal tract. The most prominent feature of the gastrointestinal tract involvement is development of esophageal stricture. The stricture results from recurrent esophageal mucosal blistering with consequent scarring and most commonly involves the upper esophagus. Here we present a case of a young boy with dominant subtype of dystrophic epidermolysis bullosa who presented with dysphagia, extensive skin blistering and missing nails. Management of an esophageal stricture eventually requires dilatation of the stricture or placement of a gastrostomy tube to keep up with the nutritional requirements. Gastrostomy tube also provides access for esophageal stricture dilatation in cases where antegrade approach through the mouth has failed.

Entities:  

Keywords:  Blistering; Dysphagia; Epidermolysis bullosa; Esophageal stenosis; Gastrostomy

Year:  2015        PMID: 25685271      PMCID: PMC4325311          DOI: 10.4253/wjge.v7.i2.154

Source DB:  PubMed          Journal:  World J Gastrointest Endosc


  15 in total

Review 1.  Epidermolysis bullosa and cancer.

Authors:  R Mallipeddi
Journal:  Clin Exp Dermatol       Date:  2002-11       Impact factor: 3.470

2.  Epidermolysis bullosa in Northern Ireland.

Authors:  K E McKenna; M Y Walsh; E A Bingham
Journal:  Br J Dermatol       Date:  1992-10       Impact factor: 9.302

Review 3.  Epidermolysis bullosa in Australia and New Zealand.

Authors:  Dédée F Murrell
Journal:  Dermatol Clin       Date:  2010-04       Impact factor: 3.478

Review 4.  Extracutaneous manifestations and complications of inherited epidermolysis bullosa: part II. Other organs.

Authors:  Jo-David Fine; Jemima E Mellerio
Journal:  J Am Acad Dermatol       Date:  2009-09       Impact factor: 11.527

Review 5.  Epidermolysis bullosa: management of esophageal strictures and enteric access by gastrostomy.

Authors:  Alan E Mortell; Richard G Azizkhan
Journal:  Dermatol Clin       Date:  2010-04       Impact factor: 3.478

Review 6.  Human skin basement membrane in health and in autoimmune diseases.

Authors:  L S Chan
Journal:  Front Biosci       Date:  1997-07-15

7.  The prevalence of epidermolysis bullosa in Scotland.

Authors:  H M Horn; G C Priestley; R A Eady; M J Tidman
Journal:  Br J Dermatol       Date:  1997-04       Impact factor: 9.302

Review 8.  Inherited epidermolysis bullosa.

Authors:  Jo-David Fine
Journal:  Orphanet J Rare Dis       Date:  2010-05-28       Impact factor: 4.123

9.  Gastrointestinal complications of inherited epidermolysis bullosa: cumulative experience of the National Epidermolysis Bullosa Registry.

Authors:  Jo-David Fine; Lorraine B Johnson; Madeline Weiner; Chirayath Suchindran
Journal:  J Pediatr Gastroenterol Nutr       Date:  2008-02       Impact factor: 2.839

10.  Correction of junctional epidermolysis bullosa by transplantation of genetically modified epidermal stem cells.

Authors:  Fulvio Mavilio; Graziella Pellegrini; Stefano Ferrari; Francesca Di Nunzio; Enzo Di Iorio; Alessandra Recchia; Giulietta Maruggi; Giuliana Ferrari; Elena Provasi; Chiara Bonini; Sergio Capurro; Andrea Conti; Cristina Magnoni; Alberto Giannetti; Michele De Luca
Journal:  Nat Med       Date:  2006-11-19       Impact factor: 53.440

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