Celiac crisis as the life-threatening onset of celiac disease in children: a case report

Mauro, Angela; Casini, Francesca; Talenti, Antonella; Mari, C. Di; Benincaso, Anna Rita; Nardo, Giovanni Di; Bernardo, Luca

doi:10.3389/fped.2023.1163765

Front. Pediatr.

2023

DOI: 10.3389/fped.2023.1163765

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Celiac crisis as the life-threatening onset of celiac disease in children: a case report

Angela Mauro

Francesca Casini

Antonella Talenti

et al.

Abstract: Celiac disease (CD) is an immune-mediated enteropathy caused by a permanent sensitivity to gluten in genetically susceptible individuals. In rare cases, CD may occur with a severe potential life-threatening manifestation known as a celiac crisis (CC). This may be a consequence of a delayed diagnosis and expose patients to possible fatal complications. We report the case of a 22-month-old child admitted to our hospital for a CC characterized by weight loss, vomiting, and diarrhea associated with a malnutrition … Show more

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2024

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Cited by 3 publications

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Wheat-Triggered Food Protein-Induced Enterocolitis Syndrome in Celiac Children on Gluten-Free Diet: A New Clinical Association

Giraldo-Tugores,

Camarero,

Roy

et al. 2024

Int Arch Allergy Immunol

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Introduction: The association between food protein-induced enterocolitis syndrome (FPIES) and wheat ingestion in children with celiac disease is unknown at this time. Methods: We present seven cases of children with celiac disease who presented with symptoms of wheat-triggered acute FPIES (a-FPIES). An oral food challenge (OFC) with wheat allergen followed by 4 h of observation was performed. Activation of innate system cells was measured at baseline (T0), during symptoms (Ts), and 4 h after symptom onset (Ts + 4). A panel of human inflammatory cytokines was also performed. Results: All patients reacted to the first allergen dose. Three patients experienced a decrease of 30 mm Hg in systolic blood pressure and tachycardia and required hemodynamic resuscitation. Neutrophilia and a decrease in eosinophil count were evident at 4 h after symptom onset. At 4 h after symptom onset, cytokines (IL-6 and IL-8, and to a lesser degree, IL-10) were elevated. Conclusion: In a small sample of celiac patients with wheat exposure in an OFC, symptoms and acute immunological changes in serum inflammatory cytokine profile were consistent with a-FPIES.

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Wheat-Triggered Food Protein-Induced Enterocolitis Syndrome in Celiac Children on Gluten-Free Diet: A New Clinical Association

Giraldo-Tugores,

Camarero,

Roy

et al. 2024

Int Arch Allergy Immunol

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Celiac Crisis: A Life-Threatening Complication of Celiac Disease

Soni,

Gogia,

Kundavaram

2024

Middle East J Dig Dis

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Celiac disease (CD) is an immune-mediated enteropathy with varied systemic involvement and association with increased morbidity and mortality. Strong clinical suspicion is the key, and diagnosis is made using histopathology and serology. Though the consumption of a strict gluten-free diet can improve symptoms and limit mucosal damage, curative therapy is still lacking. Significant clinical improvement can be seen after treatment with immunosuppressive therapy; however, there is no definitive role of immunosuppression in preventing complications. Celiac crisis (CC), a serious and life-threatening complication of CD, is characterized by acute onset and rapid progression of gastrointestinal manifestations associated with metabolic and electrolyte disturbances and neurological and renal dysfunction. Management comprises urgent hospitalization, fluid resuscitation, correction of electrolyte imbalance, and albumin infusion. Early identification and diagnosis of CD and timely initiation of a gluten-free diet with proper compliance are of paramount importance in preventing complications, including CC. Regular follow-up after diagnosis is a good approach to assessing adherence to the gluten-free diet, disease activity, and screening for complications. With the advent of improved diagnostic facilities and access to the health care system, timely diagnosis, and efficient management, prognosis has improved significantly in recent years.

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Crisi celiaca: una complicanza rara e insidiosa della celiachia

Barca,

Boccia,

Improta

et al. 2024

Medico E Bambino Pagine Elettroniche

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Coeliac Crisis is an urgent and potentially fatal complication of Coeliac Disease (CeD), which is most frequent in early childhood. The crisis may occur at onset or develop in CeD patients with poor compliance to the gluten-free diet (GFD). Stress, surgery and infections can act as triggers. The diagnosis is based on the acute onset of rapidly progressive diarrhoea associated with at least two conditions among severe dehydration, metabolic acidosis, dyselectrolytemia, renal dysfunction, neurological alterations, hypoproteinemia and weight loss. Haematological manifestations, such as thrombocytopenia, increased INR, aplastic anaemia and leukopenia, are rare. The cornerstones of therapy are intravenous rehydration, correction of hypoalbuminemia, acidosis and dyshionemia along with a strict GFD. Steroids may be also used in refractory cases. The paper describes a recent case of a patient presenting with a severe life-threatening coeliac crisis characterized by inability to walk due to lower limb weakness, severe abdominal distension, diarrhoea and peripheral oedema.

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Celiac crisis as the life-threatening onset of celiac disease in children: a case report

Cited by 3 publications

References 42 publications

Wheat-Triggered Food Protein-Induced Enterocolitis Syndrome in Celiac Children on Gluten-Free Diet: A New Clinical Association

Wheat-Triggered Food Protein-Induced Enterocolitis Syndrome in Celiac Children on Gluten-Free Diet: A New Clinical Association

Celiac Crisis: A Life-Threatening Complication of Celiac Disease

Crisi celiaca: una complicanza rara e insidiosa della celiachia

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