Literature DB >> 35737255

Protein-losing Enteropathy as a Complication and/or Differential Diagnosis of Common Variable Immunodeficiency.

Sébastien Sanges1,2,3,4, Nicolas Germain1,5, Stéphane Vignes6, David Séguy7, Sarah Stabler1,8, Nicolas Etienne3, Louis Terriou3, David Launay1,2,3,4, Éric Hachulla1,2,3,4, Damien Huglo1,9, Sylvain Dubucquoi1,2,5, Myriam Labalette1,2,5, Guillaume Lefèvre10,11,12.   

Abstract

As protein-losing enteropathy (PLE) can lead to hypogammaglobulinemia and lymphopenia, and since common variable immunodeficiency (CVID) is associated with digestive complications, we wondered if (1) PLE could occur during CVID and (2) specific features could help determine whether a patient with antibody deficiency has CVID, PLE, or both. Eligible patients were thus classified in 3 groups: CVID + PLE (n = 8), CVID-only (= 19), and PLE-only (n = 13). PLE was diagnosed using fecal clearance of α1-antitrypsin or 111In-labeled albumin. Immunoglobulin (Ig) A, G, and M, naive/memory B and T cell subsets were compared between each group. CVID + PLE patients had multiple causes of PLE: duodenal villous atrophy (5/8), nodular follicular hyperplasia (4/8), inflammatory bowel disease-like (4/8), portal hypertension (4/8), giardiasis (3/8), and pernicious anemia (1/8). Compared to the CVID-only group, CVID + PLE patients had similar serum Ig levels, B cell subset counts, but lower naive T cell proportion and IgG replacement efficiency index. Compared to the CVID-only group, PLE-only patients did not develop infections but had higher serum levels of IgG (p = 0.03), IgA (p < 0.0001), and switched memory B cells (p = 0.001); and decreased naive T cells (CD4+: p = 0.005; CD8+: p < 0.0001). Compared to the PLE-only group, CVID + PLE patients had higher infection rates (p = 0.0003), and lower serum Ig (especially IgA: p < 0.001) and switched memory B cells levels. In conclusion, PLE can occur during CVID and requires higher IgG replacement therapy dosage. PLE can also mimic CVID and is associated with milder immunological abnormalities, notably mildly decreased to normal serum IgA and switched memory B cell levels.
© 2022. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  Antibody deficiency; Common variable immunodeficiency; Hypogammaglobulinemia; Lymphopenia; Primary intestinal lymphangiectasia; Protein-losing enteropathy

Year:  2022        PMID: 35737255     DOI: 10.1007/s10875-022-01299-1

Source DB:  PubMed          Journal:  J Clin Immunol        ISSN: 0271-9142            Impact factor:   8.542


  44 in total

1.  [A colourful villous atrophy].

Authors:  Jérémy Augustin; Nadia Hoyeau; Harry Sokol; Jean-François Fléjou; Magali Svrcek
Journal:  Ann Pathol       Date:  2013-11-21       Impact factor: 0.407

2.  Identical twins with XLA requiring differing amounts of 20% subcutaneous immunoglobulin secondary to protein-losing enteropathy.

Authors:  Jennifer Lan; John Eshun; Jay Adam Lieberman
Journal:  J Allergy Clin Immunol Pract       Date:  2018-01-12

3.  Giardia infection: protein-losing enteropathy in an adult with immunodeficiency.

Authors:  Alexandre Khodr Furtado; Virginia Lucia Ribeiro Cabral; Thiago Nunes Santos; Eli Mansour; Cristiane Kibune Nagasako; Sonia Leticia Lorena; Rogerio Antunes Pereira-Filho
Journal:  World J Gastroenterol       Date:  2012-05-21       Impact factor: 5.742

4.  Clinical picture and treatment of 2212 patients with common variable immunodeficiency.

Authors:  Benjamin Gathmann; Nizar Mahlaoui; Laurence Gérard; Eric Oksenhendler; Klaus Warnatz; Ilka Schulze; Gerhard Kindle; Taco W Kuijpers; Rachel T van Beem; David Guzman; Sarita Workman; Pere Soler-Palacín; Javier De Gracia; Torsten Witte; Reinhold E Schmidt; Jiri Litzman; Eva Hlavackova; Vojtech Thon; Michael Borte; Stephan Borte; Dinakantha Kumararatne; Conleth Feighery; Hilary Longhurst; Matthew Helbert; Anna Szaflarska; Anna Sediva; Bernd H Belohradsky; Alison Jones; Ulrich Baumann; Isabelle Meyts; Necil Kutukculer; Per Wågström; Nermeen Mouftah Galal; Joachim Roesler; Evangelia Farmaki; Natalia Zinovieva; Peter Ciznar; Efimia Papadopoulou-Alataki; Kirsten Bienemann; Sirje Velbri; Zoya Panahloo; Bodo Grimbacher
Journal:  J Allergy Clin Immunol       Date:  2014-02-28       Impact factor: 10.793

5.  Efficacy of subcutaneous immunoglobulins in primary immunodeficiency with Crohn's-like phenotype: report of a case.

Authors:  M Sanges; G Spadaro; M Miniero; D Mattera; R Sollazzo; F P D'Armiento; G D De Palma; A Pecoraro; F Borrelli; A Genovese; A D'Arienzo
Journal:  Eur Rev Med Pharmacol Sci       Date:  2015       Impact factor: 3.507

6.  Gastrointestinal tract pathology in patients with common variable immunodeficiency (CVID): a clinicopathologic study and review.

Authors:  Jason A Daniels; Howard M Lederman; Anirban Maitra; Elizabeth A Montgomery
Journal:  Am J Surg Pathol       Date:  2007-12       Impact factor: 6.394

7.  Improved outcomes on subcutaneous IgG in patients with humoral immunodeficiency and co-morbid bowel disease.

Authors:  Shaili N Shah; Krista Todoric; Teresa K Tarrant
Journal:  Clin Case Rep Rev       Date:  2015-07-28

8.  A High Prevalence of Gastrointestinal Manifestations in Common Variable Immunodeficiency.

Authors:  Sampsa Pikkarainen; Timi Martelius; Ari Ristimäki; Sanna Siitonen; Mikko R J Seppänen; Martti Färkkilä
Journal:  Am J Gastroenterol       Date:  2019-04       Impact factor: 10.864

Review 9.  Protein losing enteropathy: comprehensive review of the mechanistic association with clinical and subclinical disease states.

Authors:  David G Levitt; Michael D Levitt
Journal:  Clin Exp Gastroenterol       Date:  2017-07-17

10.  Human Inborn Errors of Immunity: 2019 Update on the Classification from the International Union of Immunological Societies Expert Committee.

Authors:  Stuart G Tangye; Waleed Al-Herz; Aziz Bousfiha; Talal Chatila; Charlotte Cunningham-Rundles; Amos Etzioni; Jose Luis Franco; Steven M Holland; Christoph Klein; Tomohiro Morio; Hans D Ochs; Eric Oksenhendler; Capucine Picard; Jennifer Puck; Troy R Torgerson; Jean-Laurent Casanova; Kathleen E Sullivan
Journal:  J Clin Immunol       Date:  2020-01-17       Impact factor: 8.317

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.