Short article: Mortality and differential diagnoses of villous atrophy without coeliac antibodies

Schiepatti, Annalisa; Biagi, Federico; Fraternale, Giacomo; Vattiato, C.; Balduzzi, Davide; Agazzi, Simona; Alpini, Claudia; Klersy, Catherine; Corazza, Gino Roberto

doi:10.1097/meg.0000000000000836

Cited by 44 publications

(22 citation statements)

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“…In Giardiasis, the villous architecture is usually normal (96% of investigated patients) [ 5 ] , mainly affecting the lamina propria, in which lymphoid hyperplasia and increased numbers of chronic inflammatory cells and eosinophils are seen. Sometimes, Giardia lamblia can induce increase of duodenal intraepithelial lymphocytes associated or not to crypt hyperplasia and different grades of villous atrophy [ 10 – 12 ]. These alterations lead to a significant range of symptoms going from abdominal chronic pain to diarrhea and signs of malabsorption [ 13 ].…”

Section: Discussionmentioning

confidence: 99%

“…Recent case series and systematic reviews have shown that a wrong CD diagnosis can be reached in a consistent percentage of seronegative patients [ 3 , 12 ]. In fact, a recent study on 200 seronegative patients showed a percentage of seronegative non-coeliac disease patients of 69% [ 14 ].…”

Section: Discussionmentioning

confidence: 99%

“…In fact, a recent study on 200 seronegative patients showed a percentage of seronegative non-coeliac disease patients of 69% [ 14 ]. These patients had different causes of villous atrophy, such as giardiasis, drugs mucosal damage, Crohn disease and autoimmune enteropathy; even if some cases remained unknown [ 12 , 14 ]. Until 10 years ago, prevalence of seronegative CD was reported to be around 10 to 20% [ 15 , 16 ].…”

Section: Discussionmentioning

confidence: 99%

“…Until 10 years ago, prevalence of seronegative CD was reported to be around 10 to 20% [ 15 , 16 ]. Nowadays, due to the increased sensitivity of serological assays (anti-tTG and Ema detection) and to the acknowledgement of different alternative diagnosis of villous atrophy and their specific diagnostic work-up, this percentage decreased to 3–5% of the total number of patients with a real diagnosis of CD [ 12 , 17 ].…”

Section: Discussionmentioning

confidence: 99%

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Improving basic skills in celiac-like disease diagnosis: a case report

et al. 2018

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BackgroundThe diagnosis of Coeliac disease (CD) requires a combination of sign/symptoms, positivity of specific antibodies and duodenal histological evidence of villous atrophy. Duodenal villous atrophy, despite representing the CD landmark, is not specific since it is found in many gastrointestinal disorders. Giardiasis is one of the most common human intestinal protozoan infestations in industrialized countries whose histological duodenal mucosa damage could mimic that of CD. The present report shows how a wise clinical and laboratory assessment led us shortly to a correct diagnosis.Case presentationA 42-year-old outpatient woman without previous significant gastrointestinal diseases, was referred with dyspeptic symptoms, fatigue and mild diarrhea from 4 months. Her first investigations including immunoglobulin A (IgA) anti-tissue transglutaminase antibodies (anti-tTG) and stool parasitological and cultural analysis were negative. An esophagogastroduodenoscopy (EGDS) showed no mucosal alteration. But histology demonstrated a Helicobacter Pylori (HP) pan-gastritis while duodenal mucosa showed villous atrophy consistent with a diagnosis of CD Marsh type 3b. While on gluten-free diet (GFD) the patient didn’t experience any improvement of symptoms. Duodenal biopsies were then reviewed showing the presence of trophozoites of Giardia on the luminal surface of the duodenal wall and at the same time, a second stool examination revealed the presence of trophozoites and cysts of Giardia. Treated with metronidazole, 500 mg twice daily for 6 days the patient reduced diarrhea after few days. After about 2 months of GFD she was invited to discontinue it. At the same time stool examination was repeated with negative results. She subsequently performed eradication for Hp with triple therapy (Pylera®). Around 6 months later, the patient did not complain any gastrointestinal symptoms. Serological tests were normal and at a follow-up EGDS, duodenal mucosa had normal histology with normal finger-like villi and absence of Giardia trophozoites.ConclusionThis case report shows how CD diagnosis can sometimes be manifold. Intestinal villous atrophy alone may not automatically establish a diagnosis of CD. In the present case the clinical scenario could be fully explained by giardiasis. Indeed, different diagnostic tools and a multi-step approaches have been used to determine the final correct diagnosis.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

See 2 more Smart Citations

Improving basic skills in celiac-like disease diagnosis: a case report

et al. 2018

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“…Our patient was seronegative but carried the coeliac risk-serotype HLA-DQ2. Seronegative coeliac disease is rare among patients with coeliac disease but is a common cause of seronegative villous atrophy and is associated with high mortality 2 3. Enteropathy-associated T cell lymphoma is a rare form of extranodal T cell lymphoma that is usually, but not always, associated with untreated or undiagnosed coeliac disease 4.…”

Section: Answermentioning

confidence: 99%

Jejunal perforation and central retinal vein occlusion in a 55-year-old European man

Thomas

Summers

Goodhand

et al. 2019

Gut

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Association Between Celiac Disease and Mortality Risk in a Swedish Population

Lebwohl

Green

Söderling

et al. 2020

JAMA

116

113

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IMPORTANCE Celiac disease may be associated with a modest but persistent increased long-term mortality risk. It is uncertain whether this risk has changed in the era of wider diagnosis rates, less severe clinical disease, and more widespread availability of gluten-free food. OBJECTIVE To evaluate the association between celiac disease and mortality risk in a population-based cohort in Sweden. DESIGN, SETTING, AND PARTICIPANTS All individuals in Sweden with celiac disease diagnosed between 1969 and 2017 were identified through the Epidemiology Strengthened by histoPathology Reports in Sweden (ESPRESSO) cohort. Participants (n = 49 829) were observed starting on the day of the biopsy. The final date of follow-up was December 31, 2017. EXPOSURES Celiac disease was defined by the presence of small intestinal villus atrophy on histopathology specimens during the years 1969-2017 from Sweden's 28 pathology departments. Each individual was matched with as many as 5 control participants in the general population by age, sex, county, and calendar period. MAIN OUTCOMES AND MEASURES The primary outcome was all-cause mortality, and the secondary outcome was cause-specific mortality. Patients with celiac disease were compared with controls using stratified Cox proportional modeling, stratifying by year of diagnosis. RESULTS There were 49 829 patients with celiac disease, including 24% who were diagnosed between the years 2010 and 2017. The mean (SD) age at diagnosis was 32.2 (25.2) years and 62.4% were women. During a median follow-up time of 12.5 years, 13.2% (n = 6596) died. Compared with controls (n = 246 426), overall mortality was increased in those with celiac disease (9.7 vs 8.6 deaths per 1000 person-years; absolute difference, 1.2 per 1000 person-years; hazard ratio [HR], 1.21 [95% CI, 1.17-1.25]). The relative increase in mortality risk was present in all age groups and was greatest in those diagnosed in the age range of 18 to 39 years (1.9 vs 1.1 per 1000 person-years; HR, 1.69 [95% CI, 1.47-1.94]; P values for heterogeneity comparing 18-39 years with 40-59 years and with Ն60 years were both <.001). Individuals with celiac disease were at increased risk of death from cardiovascular disease (3.5 vs 3.4 per 1000 person-years; HR, 1.08 [95% CI, 1.02-1.13]), cancer (2.7 vs 2.2 per 1000 person-years; HR, 1.29 [95% CI, 1.22-1.36]), and respiratory disease (0.6 vs 0.5 per 1000 person-years; HR, 1.21 [95% CI, 1.08-1.37]). When compared with controls, the overall mortality risk was greatest in the first year after diagnosis (15.3 vs 6.5 per 1000 person-years; HR, 2.34 [95% CI, 2.14-2.55]) but persisted beyond 10 years after diagnosis (10.5 vs 10.1 per 1000 person-years; HR, 1.15 [95% CI, 1.10-1.20]). The mortality risk was likewise present for patients diagnosed during the years 2010-2017 (7.5 vs 5.5 per 1000 person-years; HR, 1.35 [95% CI, 1.21-1.51]). CONCLUSIONS AND RELEVANCE In a Swedish population studied between 1969 and 2017, a diagnosis of celiac disease compared with the general population was associated with a smal...

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Short article: Mortality and differential diagnoses of villous atrophy without coeliac antibodies

Abstract: Patients with VA and negative endomysial antibodies are rare. However, these forms of VA identify specific causes that can be diagnosed. These patients are affected by a very high mortality.

Cited by 44 publications

References 20 publications

Improving basic skills in celiac-like disease diagnosis: a case report

Improving basic skills in celiac-like disease diagnosis: a case report

Jejunal perforation and central retinal vein occlusion in a 55-year-old European man

Association Between Celiac Disease and Mortality Risk in a Swedish Population

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