Diffuse Nodular Lymphoid Hyperplasia of the Intestine Caused by Common Variable Immunodeficiency and Refractory Giardiasis

Choi, Jung Hye; Han, Dong Soo; Kim, Jieun; Yi, Kijong; Oh, Yu Whan; Kim, Yongsoo

doi:10.2169/internalmedicine.56.7305

Cited by 12 publications

(8 citation statements)

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“…Giardia lamblia is a common infective cause for acute and chronic diarrhea in CVID patients [ 8 , 38 ] and can be challenging to eradicate [ 39 ]. It has often been found in association with nodular lymphoid hyperplasia [ 20 , 40 ] and can also cause villous atrophy in the small bowel. Detection of Giardia lamblia in the stool is more sensitive via PCR than through microscopy [ 41 ].…”

Section: Introductionmentioning

confidence: 99%

Bowel Histology of CVID Patients Reveals Distinct Patterns of Mucosal Inflammation

et al. 2021

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Diarrhea is the commonest gastrointestinal symptom in patients with common variable immunodeficiency (CVID). Different pathologies in patients’ bowel biopsies have been described and links with infections have been demonstrated. The aim of this study was to analyze the bowel histology of CVID patients in the Royal-Free-Hospital (RFH) London CVID cohort. Ninety-five bowel histology samples from 44 adult CVID patients were reviewed and grouped by histological patterns. Reasons for endoscopy and possible causative infections were recorded. Lymphocyte phenotyping results were compared between patients with different histological features. There was no distinctive feature that occurred in most diarrhea patients. Out of 44 patients (95 biopsies), 38 lacked plasma cells. In 14 of 21 patients with nodular lymphoid hyperplasia (NLH), this was the only visible pathology. In two patients, an infection with Giardia lamblia was associated with NLH. An IBD-like picture was seen in two patients. A coeliac-like picture was found in six patients, four of these had norovirus. NLH as well as inflammation often occurred as single features. There was no difference in blood lymphocyte phenotyping results comparing groups of histological features. We suggest that bowel histology in CVID patients with abdominal symptoms falls into three major histological patterns: (i) a coeliac-like histology, (ii) IBD-like changes, and (iii) NLH. Most patients, but remarkably not all, lacked plasma cells. CVID patients with diarrhea may have an altered bowel histology due to poorly understood and likely diverse immune-mediated mechanisms, occasionally driven by infections.

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Section: Introductionmentioning

confidence: 99%

Bowel Histology of CVID Patients Reveals Distinct Patterns of Mucosal Inflammation

et al. 2021

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“…We consider the treatment successful because stool examination has remained negative to date. So far, only six refractory giardiasis cases in CVID patients have been published, and different curative treatments have been applied [ 20 , 22 , 26 , 30 , 31 , 33 ]. Currently, there is no standard therapy for refractory giardiasis, and multiple drugs have been tried [ 15 ].…”

Section: Discussionmentioning

confidence: 99%

Incidence, Management Experience and Characteristics of Patients with Giardiasis and Common Variable Immunodeficiency

Díaz-Alberola

Gutiérrez‐Bautista

Espuch-Oliver

et al. 2022

JCM

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Common variable immunodeficiency (CVID) is an antibody immunodeficiency with a wide variety of clinical and immunological manifestations, and whose genetic cause is found in about 25% of diagnosed cases. Giardia lamblia is one of the main causes of gastrointestinal infections in CVID. 5-Nitroimidazoles are the most used first-line treatment, but nitroimidazole-refractory giardiasis is increasing. Nevertheless, only a few cases of refractory giardiasis in CVID have been reported. This study aimed to determine the incidence of Giardia infection in our CVID cohort, shows our management experience and describes patients’ phenotypic features. Clinical data collection, immunological, immunogenetics and microbiology assays were performed, and previous cases of giardiasis in CVID were reviewed. The incidence of symptomatic giardiasis was 12.9%. The main immunological features were undetectable or decreased IgA levels and reduced switched memory B cells. A probable PTEN pathogenic variant was detected in one. Three patients responded to metronidazole but suffered reinfections, and one was a refractory giardiasis eradicated with innovative quinacrine plus paromomycin combination. This work could contribute to the decision-making and therapeutic management of future patients with CVID and giardiasis, highlighting the importance of the early detection and treatment of infections in patients with CVID to ensure a good quality of life.

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“…Similarly, the finding of opportunistic or unusual pathogens, such as Microsporidia or Cryptosporidia, is rare and might suggest investigating for a combined immunodeficiency, characterized by greater degrees of T-cell dysfunction (70). Liver is a possible extra-intestinal localization of Giardia lamblia, which is a common cause of chronic enteritis in CVID (70,71). Therefore, liver involvement should always be ruled out in case of Giardia detection from stool or duodenal samples.…”

Section: Infectionsmentioning

confidence: 99%

Heterogeneity of Liver Disease in Common Variable Immunodeficiency Disorders

et al. 2020

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Diffuse Nodular Lymphoid Hyperplasia of the Intestine Caused by Common Variable Immunodeficiency and Refractory Giardiasis

Cited by 12 publications

References 10 publications

Bowel Histology of CVID Patients Reveals Distinct Patterns of Mucosal Inflammation

Bowel Histology of CVID Patients Reveals Distinct Patterns of Mucosal Inflammation

Incidence, Management Experience and Characteristics of Patients with Giardiasis and Common Variable Immunodeficiency

Heterogeneity of Liver Disease in Common Variable Immunodeficiency Disorders

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