Progression of pediatric celiac disease from potential celiac disease to celiac disease: a retrospective cohort study

Sakhuja, Shruti; Holtz, Lori R.

doi:10.1186/s12887-021-02625-z

Cited by 7 publications

(7 citation statements)

References 23 publications

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“…Intestinal deposits of tTG2 identify children at risk for VA [ 30 ]. Similar results were reported by Sakhuja et al in a small retrospective study where 4% of children who initially had a negative biopsy later developed VA [ 31 ]. Lionetti et al reported in a recent study the results from the follow-up CELIPREV study that assessed children with CD-predisposing HLA genotypes from birth.…”

Section: Introductionsupporting

confidence: 88%

Is There a Time and a Place for the Gluten-Free Diet in Potential Celiac Disease?

Nemteanu,

Clim,

Hincu

et al. 2023

Nutrients

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Potential celiac disease (PCD) is characterized by the absence of villous atrophy on duodenal biopsies (Marsh 0 or 1) despite positive celiac serology and HLA DQ2 or DQ8 heterodimers. Recent epidemiological studies report that PCD represents one fifth of the total CD diagnoses. Compared to patients with CD, the majority of adult patients with PCD show lower rates of nutrient deficiencies and extraintestinal symptoms at diagnosis. Recommending a gluten-free diet (GFD) to PCD patients depends on whether they have symptoms or not. A significant clinical improvement is reported by symptomatic patients, but for asymptomatic PCD, diet implementation is still a matter of debate. Some questions remain to be answered: does PCD serve as an intermediary phase leading to the progression of true CD? Is it reasonable to hypothesize that PCD and active CD represent different manifestations of the same condition? Is there a potential for both underdiagnosis and overdiagnosis of CD in those who may have the condition? Additional research is required to address these inquiries and ascertain the specific subset of people with potential progression to overt CD, as well as to determine the potential advantages of early implementation of a GFD for these individuals. The investigation of risk factors in CD warrants examination of variables such as the timing of diagnosis, the genetic profile, the extent of gluten exposure, and the composition of the microbiome.

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Section: Introductionsupporting

confidence: 88%

Is There a Time and a Place for the Gluten-Free Diet in Potential Celiac Disease?

Nemteanu,

Clim,

Hincu

et al. 2023

Nutrients

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“…This is more likely to be a problem near the lower bounds of abnormality, but half of the cases in whom the Marsh score was 0/1 had antibody values to tTG at least triple the upper limit of normal. Finally, we cannot exclude the possibility that the patients with elevated antibodies to tTG and Marsh scores of 0/1 will eventually develop histologic findings typical of CD ( 22 , 23 ).…”

Section: Discussionmentioning

confidence: 99%

Rhamnose Is Superior to Mannitol as a Monosaccharide in the Dual Sugar Absorption Test: A Prospective Randomized Study in Children With Treatment-Naïve Celiac Disease

et al. 2022

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Background and AimWe sought to correlate two different measures of gut permeability [lactulose:mannitol (L:M) and lactulose:rhamnose (L:R)] to the severity of duodenal histopathology in children with and without elevated antibodies to tissue transglutaminase (tTG). A secondary objective was to correlate gut permeability with celiac disease (CD) serology and indices of inflammation and bacterial product translocation.MethodsWe prospectively randomized children undergoing endoscopy with abnormal (n = 54) and normal (n = 10) concentrations of circulating antibodies to tTG, to either L:M or L:R. Biopsies underwent modified Marsh scoring to measure mucosal injury. Circulating anticore Escherichia coli lipopolysaccharide (LPS) IgG, α-1 acid glycoprotein, LPS-binding protein, and C-reactive protein concentrations were measured by enzyme immunoassays.ResultsOf the 54 cases with positive celiac serology, 31 and 69% had modified Marsh 0/1 scores or ≥3a, respectively. Circulating tTG IgA correlated with the modified Marsh score (p = 0.03). L:R, but not L:M or percent L excreted, differed according to modified Marsh scores (p = 0.01). There was no significant association between any systemic marker of inflammation or gut injury, and modified Marsh scores. Concerningly, most participants had evidence of urinary M before the challenge sugar was administered.ConclusionsL:R, but not L:M, is associated with modified Marsh scores in children undergoing small bowel biopsy for suspected CD. Despite increased intestinal permeability, we see scant evidence of systemic exposure to gut microbes in these children. Gut permeability testing with L:R may predict which patients with abnormal celiac serology will have biopsy evidence for celiac disease and reduce the proportion of such patients undergoing endoscopy whose Marsh scores are ≤1. M should not be used as a monosaccharide for permeability testing in children.

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“…69 Febrile illness may be accompanied by TTG IgA elevation in young children without CD. 70 A spectrum model of CD is also compelling because it affords the flexibility to incorporate the seroconversion-reversion phenomenon and the associated notions of progression and, perhaps, regression, 71 as well as the tantalising possibility of therapeutic disease interception.…”

Section: P Otential Coeliac D Is E a S Ementioning

confidence: 99%

“…Progression from PCD to CD may be more common among symptomatic individuals, 79,81 first degree relatives, and high-risk individuals who undergo asymptomatic screening (eg, T1DM, hypothyroidism, trisomy 21). 71 Younger patients with PCD may also be more likely to progress to CD, highlighting the need for ongoing monitoring. 75 In contrast to CD, PCD is more common among those with DQ8 than those with DQ2.…”

Section: Natural History Of Pcdmentioning

confidence: 99%

“…are spontaneous reversion to seronegative, remaining persistently seropositive with normal villous architecture, or fluctuating between sreonegative and seropositive (Figure 1). Progression from PCD to CD may be more common among symptomatic individuals, 79,81 first degree relatives, and high‐risk individuals who undergo asymptomatic screening (eg, T1DM, hypothyroidism, trisomy 21) 71 . Younger patients with PCD may also be more likely to progress to CD, highlighting the need for ongoing monitoring 75 .…”

Section: Potential Coeliac Diseasementioning

confidence: 99%

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Review article: Becoming and being coeliac—special considerations for childhood, adolescence and beyond

Chang

O'Shea

Therrien

et al. 2022

Aliment Pharmacol Ther

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Classically considered a disease of early childhood characterised by malabsorption and failure to thrive, coeliac disease is now recognised to arise in genetically susceptible individuals at any age. Although permissive HLA genotypes are the strongest predictor of coeliac disease, they are not sufficient. Several prospective cohort studies enrolling genetically at-risk infants have investigated the role of potential triggers of coeliac disease autoimmunity, such as timing of gluten introduction, viral infections and dietary patterns. Much less is known about triggers of coeliac disease in adulthood. Better understanding of factors leading to coeliac disease may be helpful in the management of those with potential coeliac disease (elevated serum celiac antibodies without villous atrophy in the small intestine), many of whom initiate a gluten-free diet without demonstration of villous atrophy. There are a range of clinical presentations of celiac disease in childhood and patterns of coeliac serology, including fluctuation and spontaneous reversion on a gluten-containing diet, vary.There is a current debate over best strategies to manage adults and children with potential coeliac disease to avoid over-treatment and under-treatment. Childhood and adolescence carry unique issues pertaining to the diagnosis and management of coeliac disease, and include nutrition and growth, rescreening, repeat biopsy, dietary adherence concerns and transition to adult care. In conclusion, while coeliac disease has similar pathogenesis and general clinical manifestations in paediatric and adult populations, diagnostic and management approaches need to adapt to the developmental stages.

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Progression of pediatric celiac disease from potential celiac disease to celiac disease: a retrospective cohort study

Cited by 7 publications

References 23 publications

Is There a Time and a Place for the Gluten-Free Diet in Potential Celiac Disease?

Is There a Time and a Place for the Gluten-Free Diet in Potential Celiac Disease?

Rhamnose Is Superior to Mannitol as a Monosaccharide in the Dual Sugar Absorption Test: A Prospective Randomized Study in Children With Treatment-Naïve Celiac Disease

Review article: Becoming and being coeliac—special considerations for childhood, adolescence and beyond

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