Literature DB >> 27072857

A Difficult and Rare Diagnosis of Autoimmune Enteropathy in a Patient Affected by Down Syndrome.

Anne Depince-Berger1, Clara Cremilieux2, Melanie Rinaudo-Gaujous1, Christian Genin1, Benedicte de Freminville3, Claude Lambert1, J Bruneau4, Stephane Paul5.   

Abstract

Patients with Down syndrome are more susceptible to autoimmune pathologies, in particular endocrine or digestive diseases such as celiac disease. Autoimmune enteropathy is another form of digestive autoimmune disease, non-gluten-dependant, more often diagnosed in male neonates with immunodysregulation and polyendocrinopathy such as the Immunodysregulation, Polyendocrinopathy, Enteropathy, X-linked syndrome. It also exists in the adult, but this pathology is less known and therefore frequently under-diagnosed. Clinical manifestations are similar to celiac disease, but not improved after a gluten-free diet. Autoimmune enteropathy is frequently associated with other autoimmune diseases, such as thyroiditis, myasthenia gravis, lupus or immune deficiencies, as Common Variable Immunodeficiency. Pathological analysis of intestinal biopsies can frequently distinguish autoimmune enteropathy and celiac disease. Autoimmune enteropathy usually has an important lymphoplasmacytic infiltration of the mucosa and a lack of intraepithelial lymphocytes in the gastrointestinal mucosal surface, while celiac disease usually has a polymorph infiltration of the mucosa and an important intraepithelial lymphocytes infiltration. Nevertheless, the two pathological patterns may overlap. Here we report the first case of a patient with Down syndrome associated to autoimmune enteropathy (initially diagnosed as celiac disease), chronic pancreatitis and cutaneous lupus erythematosus. Even if autoimmune pathologies are much more common in patients with Down syndrome, we would like to report on this rare and original association found in our patient.

Entities:  

Keywords:  Down syndrome; autoimmune enteropathy; celiac disease; immunity; immunodeficiency

Mesh:

Year:  2016        PMID: 27072857     DOI: 10.1007/s10875-016-0280-7

Source DB:  PubMed          Journal:  J Clin Immunol        ISSN: 0271-9142            Impact factor:   8.317


  36 in total

1.  Common variable immunodeficiency: clinical and immunological features of 248 patients.

Authors:  C Cunningham-Rundles; C Bodian
Journal:  Clin Immunol       Date:  1999-07       Impact factor: 3.969

2.  A case of Down's syndrome complicated by retinoblastoma and celiac disease.

Authors:  D Bentley
Journal:  Pediatrics       Date:  1975-07       Impact factor: 7.124

Review 3.  Coeliac disease.

Authors:  G R Corazza; V Villanacci
Journal:  J Clin Pathol       Date:  2005-06       Impact factor: 3.411

Review 4.  Autoimmune enteropathy: a review and update of clinical management.

Authors:  Nicole M Gentile; Joseph A Murray; Darrell S Pardi
Journal:  Curr Gastroenterol Rep       Date:  2012-10

Review 5.  Infections and immunodeficiency in Down syndrome.

Authors:  G Ram; J Chinen
Journal:  Clin Exp Immunol       Date:  2011-02-24       Impact factor: 4.330

6.  HLA-DQ typing in the diagnosis of celiac disease.

Authors:  Katri Kaukinen; Jukka Partanen; Markku Mäki; Pekka Collin
Journal:  Am J Gastroenterol       Date:  2002-03       Impact factor: 10.864

7.  The enteropathy associated with common variable immunodeficiency: the delineated frontiers with celiac disease.

Authors:  Georgia Malamut; Virginie Verkarre; Felipe Suarez; Jean-François Viallard; Anne-Sophie Lascaux; Jacques Cosnes; Yoram Bouhnik; Olivier Lambotte; Dominique Béchade; Marianne Ziol; Anne Lavergne; Olivier Hermine; Nadine Cerf-Bensussan; Christophe Cellier
Journal:  Am J Gastroenterol       Date:  2010-06-15       Impact factor: 10.864

8.  A 75-kD autoantigen recognized by sera from patients with X-linked autoimmune enteropathy associated with nephropathy.

Authors:  I Kobayashi; K Imamura; M Yamada; M Okano; A Yara; S Ikema; N Ishikawa
Journal:  Clin Exp Immunol       Date:  1998-03       Impact factor: 4.330

9.  Adult autoimmune enteropathy: Mayo Clinic Rochester experience.

Authors:  Salma Akram; Joseph A Murray; Darrell S Pardi; Glenn L Alexander; John A Schaffner; Pierre A Russo; Susan C Abraham
Journal:  Clin Gastroenterol Hepatol       Date:  2007-08-01       Impact factor: 11.382

Review 10.  Intrinsic defect of the immune system in children with Down syndrome: a review.

Authors:  M A A Kusters; R H J Verstegen; E F A Gemen; E de Vries
Journal:  Clin Exp Immunol       Date:  2009-01-22       Impact factor: 4.330

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