Literature DB >> 15316525

A 55-year-old man with hypogammaglobulinemia, lymphopenia, and unrelenting cutaneous warts.

Jerry Lynn1, Adina K Knight, Malek Kamoun, Arnold I Levinson.   

Abstract

A 55-year-old white man with a history of hypertension, fibromyalgia, and colonic polyps presented with unrelenting plantar warts on his hands and feet for the past 4 years. He was otherwise healthy and without a history of recurrent infections. Physical examination was unremarkable except for extensive warts on his hands and feet. Pertinent laboratory findings included hypoalbuminemia, hypogammaglobulinemia, and lymphopenia most severely affecting CD4(+) T cells. Testing for HIV infection was negative. This clinical and laboratory presentation suggested a combined humoral and cellular immunodeficiency syndrome that could be best explained by loss of lymphocytes, immunoglobulins, and other serum proteins. Additional immunologic testing revealed a marked reduction in peripheral blood naive (CD4(+)CD45RA(+)) T cells. A 24-hour stool collection showed a markedly elevated alpha(1)-antitrypsin level. These findings were most consistent with the diagnosis of intestinal lymphangiectasia, a type of protein-losing enteropathy associated with hypoalbuminemia, hypogammaglobulinemia, and lymphopenia, characterized by a preferential loss of naive CD4(+) T cells into the gastrointestinal tract. This case illustrates the importance of considering intestinal loss of immunoglobulins and lymphocytes in the differential diagnosis of the adult patient who presents with laboratory evidence of a combined humoral and cellular immunodeficiency. It also underscores the diagnostic utility of the clinical immunology laboratory and how flow cytometry, in particular, can contribute to an understanding of pathogenic mechanisms.

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Year:  2004        PMID: 15316525     DOI: 10.1016/j.jaci.2004.02.033

Source DB:  PubMed          Journal:  J Allergy Clin Immunol        ISSN: 0091-6749            Impact factor:   10.793


  10 in total

Review 1.  Disseminated Cutaneous Warts in X-Linked Hyper IgM Syndrome.

Authors:  Hsi-En Ho; Minji Byun; Charlotte Cunningham-Rundles
Journal:  J Clin Immunol       Date:  2018-05-05       Impact factor: 8.317

2.  Curcumin for the prevention of progression in monoclonal gammopathy of undetermined significance: A word of caution.

Authors:  A J M Vermorken; J Zhu; W J M VAN DE Ven; Y Cui; J P Fryns
Journal:  Exp Ther Med       Date:  2010-03-01       Impact factor: 2.447

3.  Clinical outcome of nutrition-oriented intervention for primary intestinal lymphangiectasia.

Authors:  Qing-Ya Tang; Jie Wen; Jiang Wu; Ying Wang; Wei Cai
Journal:  World J Pediatr       Date:  2010-12-30       Impact factor: 2.764

Review 4.  Primary intestinal lymphangiectasia: four case reports and a review of the literature.

Authors:  Jie Wen; Qingya Tang; Jiang Wu; Ying Wang; Wei Cai
Journal:  Dig Dis Sci       Date:  2010-03-03       Impact factor: 3.199

Review 5.  Warts and all: human papillomavirus in primary immunodeficiencies.

Authors:  Jennifer W Leiding; Steven M Holland
Journal:  J Allergy Clin Immunol       Date:  2012-10-01       Impact factor: 10.793

6.  Primary intestinal lymphangiectasia with generalized warts.

Authors:  Soon Jae Lee; Hyun Joo Song; Sun-Jin Boo; Soo-Young Na; Heung Up Kim; Chang Lim Hyun
Journal:  World J Gastroenterol       Date:  2015-07-21       Impact factor: 5.742

7.  Hypogammaglobulinemia with facial edema.

Authors:  Adina Kay Knight; Lloyd Mayer; Andrew G Franks; Charlotte Cunningham-Rundles
Journal:  PLoS Med       Date:  2006-12       Impact factor: 11.069

8.  Case report of primary intestinal lymphangiectasia diagnosed in an octogenarian by ileal intubation and by push enteroscopy after missed diagnosis by standard colonoscopy and EGD.

Authors:  Mitchell S Cappell; Ahmed Edhi; Mitual Amin
Journal:  Medicine (Baltimore)       Date:  2018-01       Impact factor: 1.889

Review 9.  Primary intestinal lymphangiectasia in an adult patient: A case report and review of literature.

Authors:  Rudolf Huber; Georg Semmler; Alexander Mayr; Felix Offner; Christian Datz
Journal:  World J Gastroenterol       Date:  2020-12-08       Impact factor: 5.742

10.  The Heart of the Matter: Secondary Hypogammaglobulinemia and Constrictive Pericarditis.

Authors:  Erica G Schmitt; Aarti S Dalal; Alok Kothari; Maleewan Kitcharoensakkul
Journal:  Pediatrics       Date:  2021-06       Impact factor: 9.703

  10 in total

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