Amoruso, Annacinzia

DI LEO, Alfredo

Ierardi, Enzo

Licinio, Raffaele

Piscitelli, Domenico

Principi, Mariabeatrice

PRINCIPI, MARIABEATRICE

AMORUSO, Annacinzia

PISCITELLI, Domenico

Archivio istituzionale della ricerca - Università di Bari

Letters to the Editor 3J Gastrointestin Liver Dis, December 2013 Vol. 22 No 4: 1) Gastroenterology Service, ASL BAT; 2) Department of Pathology, “Lorenzo Bonomo” Hospital”, Andria (BT) - ItalyConflicts of interest: None to declareCorresponding author: Antonio Tursi, antotursi@tiscali.itREFERENCES    1. Bultink IE, Vis M, van der Horst-Bruinsma IE, Lems WF. Inflammatory rheumatic disorders and bone. Curr Rheumatol Rep 2012;14:224-230. 2. Yüksel I, Ataseven H, Başar O, et al. Peripheral arthritis in the course of inflammatory bowel diseases. Dig Dis Sci 2011;56:183-187. 3. De Vos M, Mielants H, Cuvelier C, Elewaut A, Veys E. Long-term evolution of gut inflammation in patients with spondyloarthropathy. Gastroenterology 1996;110:1696-1703. 4. Faustini F, Zoli A, Ferraccioli GF. Immunologic and genetic links between spondylarthropathies and inflammatory bowel diseases. Eur Rev Med Pharmacol Sci 2009;13 Suppl 1:1-9.   Celiac disease and common variable immunodeficiency: a familial inheritance?To the Editor,Celiac disease (CD) is associated with autoimmune disorders and immune deficits, such as common variable immunodeficiency (CVID) [1]. CVID is a heterogeneous group of deficient antibody production diagnosed by well-stated criteria [2]. It may display infectious diarrhea [3] and its association with CD has been controversial, since villous atrophy may not always be improved by a gluten-free diet (GFD). This aspect has been clarified by Biagi et al who pointed out the possibility that the only suitable tool is the response to GFD [4]. Both CD and CVID show a family inheritance and inducible co-stimulator (ICOS, a genetic polymorphisms invoked for CD) has a crucial role in altered lymphocyte maturation characterizing CVID [5]. Nevertheless, the presence of CD in first-degree relatives of patients affected by CVID has never been detected. Therefore, we believe it is of interest to report the case of a 30 year young male patient with CD whose sister suffered from CVID.Admitted to our Unit for acute diarrhea and fever, his family history revealed a sister affected by CVID, treated with periodic immunoglobulin replacement. Empiric rehydration and antibiotic therapy (ciprofloxacin 250mg x 2/day) induced rapid disappearance of symptoms. Laboratory tests excluded humoral immune deficiency, but showed folate, B12 vitamin, iron and transferrin saturation deficiency. So, we performed the anti-tissue transglutaminase and anti-endomysium assay (positive) and upper endoscopy with distal duodenum histology (Marsh grade 3A CD). A GFD was  initiated and six months later laboratory and duodenal histology became normal.Accordingly, we believe that this case could stimulate a better knowledge of the topic. The  principal point is the possibility that CD and CVID association may be observed not only in the same subject, but even in two different first-degree relatives. Moreover, it emphasizes some other points. Firstly, patients with CVID could be screened for infectious, immunological causes of gastrointestinal symptoms and CD. In the presence of malabsorption and absence of intestinal infections, a distal duodenal biopsy should be performed. On the other hand, in first-degree relatives of patients with CVID, in the presence of either atypical clinical signs of CD and features of malabsorption, the  search for anti-transglutaminase antibodies could be useful. This approach may be interesting to  detect inheritance patterns between CD and CVID in a large series of patients.Raffaele Licinio, Mariabeatrice Principi, Annacinzia Amoruso, Domenico Piscitelli, Enzo Ierardi, Alfredo Di LeoDepartment of Emergency and Organ Transplantation, University of Bari, ItalyConflicts of interest: None to declareCorresponding author: Prof. A. Di Leo; alfredo.dileo@uniba.itREFERENCES  1. Malamut G, Verkarre V, Suarez F, et al. The enteropathy associated with common variable immunodeficiency: the delineated frontiers with celiac disease. Am J Gastroenterol 2010;105:2262-2275.  2. Wehr C, Kivioja T, Schmitt C, et al. The EUROclass trial: defining subgroups in common variable immunodeficiency. Blood 2008;111:77-85.  3. Abolhassani H, Sagvand BT, Shokuhfar T, Mirminachi B, Rezaei N, Aghamohammadi A. A review on guidelines for management and treatment of common variable immunodeficiency. Expert Rev Clin Immunol 2013;9:561-574. 4. Biagi F, Bianchi PI, Zilli A, et al. The significance of duodenal mucosal atrophy in patients with common variable immunodeficiency: a clinical and histopathologic study. Am J Clin Pathol 2012;138:185-189. 5. Haimila K, Einarsdottir E, de Kauwe A, et al. The shared CTLA4-ICOS risk locus in celiac disease, IgA deficiency and common variable immunodeficiency. Genes Immun 2009;10:151-161.Renal amyloidosis after a short period of diagnosis with ileocolonic Crohn‘s diseaseTo the Editor,We read the study of Pukitis et al. with great interest. Pukitis and colleagues described a patient with Crohn’s disease (CD) complicated by AA amyloidosis, which was treated with infliximab [1]. On this occasion, we would like to share our experiences with a case of AA amyloidosis due to ileocolonic CD. A 44 year-old man presented with peripheral edema of both lower extremities. The patient had a history of ileocolonic CD for six months and had been on corticosteroid (60 mg/day) and azathioprine (200 mg/day) treatment for three months owing to activation of CD. After the active disease was controlled, remission was continued with azathioprine treatment. He had 1-2 stools per day with normal form. On physical examination, 

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Celiac disease and common variable immunodeficiency: a familial inheritance?

Celiac disease and common variable immunodeficiency: a familial inheritance?

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Archivio istituzionale della ricerca - Università di Bari