Coeliac disease presenting as chronic cough in an 8-year-old child

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Patients with the 'classical' gastrointestinal symptoms of coeliac disease (CD) are usually readily diagnosed and treated with a gluten exclusion diet. However, the advent of sensitive serological investigations has revealed that over half of childhood CD remains asymptomatic or presents with more subtle non-gastrointestinal symptoms, with a significant risk to the health of undiagnosed children. In view of its changing presentation there is an increasing need for clinicians to have a low threshold to investigate patients for this disease. Rare cases of CD associated with respiratory symptoms have been reported. The authors report an interesting case presenting as chronic cough in an otherwise asymptomatic 8-year-old girl. As a result of having a low threshold of suspicion for underlying CD, a diagnosis has been made and the long-term health risks of undiagnosed disease have been improved in both the patient and her immediate family.

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Objectives: Celiac disease is an autoimmune disorder that develops because of sensitivity to gluten-containing grains in genetically disposed individuals. Nasal mucociliary clearance is the most important protective factor that protects the upper and lower airways from foreign particulates. This study aimed to investigate the effect of celiac disease on nasal mucociliary clearance. Methods: The study included patients with celiac disease and healthy children. Nasal mucociliary clearance time was measured using the saccharin test. The children's saccharin taste time was recorded in seconds. Results: Overall, 65 children were included: 43 patients with celiac disease (66.2%) and 22 healthy children (33.8%). Of all the children, 42 (64.6%) were female, and the average age was 11.8 ± 4 years. Nasal mucociliary clearance time of patients with celiac disease (531 ± 155 s) was significantly prolonged in comparison to that of healthy children (448 ± 80 s) (p = 0.006). No relationships were found between the diagnosis age, celiac type, and histopathological phase and compliance with the gluten-free diet and nasal mucociliary clearance time of patients with celiac disease. Conclusions: This study showed that nasal mucociliary clearance was prolonged in patients with celiac disease. A defect in nasal mucociliary clearance increases the risk of infection and inflammation in small airways. Studies reported a high prevalence of respiratory tract infection in patients with celiac disease, which was associated with malnutrition, vitamin deficiency, and hyposplenism. The findings of the present study indicated that impairment of nasal mucociliary clearance could play a role in the development of frequent lung infections in patients with celiac disease.
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There has been a growing recognition that coeliac disease is much more common than previously recognised, and this has coincided with the increasingly widespread use of serological testing. To determine whether the age at presentation and the clinical presentation of coeliac disease have changed with the advent of serological testing. A 21-year review of prospectively recorded data on the mode of presentation of biopsy confirmed coeliac disease in a single regional centre. Presenting features over the past 5 years were compared with those of the previous 16 years. Between 1983 and 1989 (inclusive), no serological testing was undertaken; between 1990 and 1998, antigliadin antibody was used with occasional use of antiendomysial antibody and antireticulin antibody. From 1999 onwards, anti-tissue transglutaminase was used. 86 patients were diagnosed over the 21-year period: 50 children between 1999 and 2004 compared with 25 children between 1990 and 1998 and 11 children between 1983 and 1989. The median age at presentation has risen over the years. Gastrointestinal manifestations as presenting features have decreased dramatically. In the past 5 years, almost one in four children with coeliac disease was diagnosed by targeted screening. This study reports considerable changes in the presentation of coeliac disease-namely, a decreased proportion presenting with gastrointestinal manifestations and a rise in the number of patients without symptoms picked up by targeted screening. Almost one in four children with coeliac disease is now diagnosed by targeted screening. Most children with coeliac disease remain undiagnosed. Paediatricians and primary care physicians should keep the possibility of coeliac disease in mind and have a low threshold for testing, so that the potential long-term problems associated with untreated coeliac disease can be prevented.
The clinical, radiological and pathological features of 7 patients with coeliac disease (CD) who developed lung abscesses or cavities are described. These patients were seen during a 20-year period during which time approximately 600 coeliacs were seen and 50 died. Six of the coeliac patients with lung abscess died. The patients were middle aged. Staphylococcal infection, Klebsiella pneumoniae, bronchial carcinoma and previous pulmonary tuberculosis accounted for the cavities in 4 patients. In the 3 other patients a definite cause could not be identified. Hyposplenism and malnutrition were common. Next to malignancy pulmonary abscess was the commonest cause of death in the coeliac population. The development of respiratory symptoms should be regarded as a potentially serious and a life-threatening event in the middle-aged coeliac patients. Lung abscess should be added to the list of respiratory diseases associated with coeliac disease.
Respiratory disease and subclinical pulmonary abnormalities are recognised complications of both coeliac disease (CD) and inflammatory bowel disease (IBD) but the pathogenesis of the lung disease remains uncertain. We have studied lung function, including permeability measured by clearance of inhaled technetium-99m diethylene triamine pentaacetic acid in 25 patients with IBD, 18 patients with CD on a gluten-free diet, and in 20 normal controls, all without respiratory symptoms. In IBD there was evidence of obstruction to airflow (mean forced expiratory volume in 1 s/forced vital capacity equals 75.8%, control 81%; p less than 0.05) but no change in pulmonary permeability (half-time clearance equals 70.3 vs. 69.2 min). In CD airflow was not significantly different from control (forced expiratory volume in 1 s/forced vital capacity equals 80%) but there was an increase in pulmonary permeability (half-time clearance equals 48.9 min; p less than 0.01). These findings suggest that the mechanisms of lung disease in CD differs from that in IBD and supports the hypothesis of a common mucosal defect in lung and small intestine in CD allowing increased permeability.
Chronic cough is a common reason for presentation to a respiratory clinic. In up to 20% of cases the cause remains unclear after investigations. We report one such case where there was bronchoscopic evidence of lymphocytic airway inflammation in association with newly diagnosed coeliac disease. All features improved markedly on a gluten free diet, suggesting a causal relationship between coeliac disease, cough, and lymphocytic bronchoalveolitis.
Idiopathic pulmonary haemosiderosis (IPH) is a rare and serious disorder in children of unknown aetiolopathogeny. Association of IPH and coeliac disease (CD) is even rarer. Immunological origin of IPH is now well accepted. We report the case of an 11-year-old female admitted for evaluation of recurrent streaky haemoptysis that had been evolving over the previous 9 months. Physical examination revealed weight loss with normal weight, but there was cutaneous and mucosal pallor due to severe anaemia (haemoglobin 4.6g/dl). The chest X-rays showed unilateral alveolo-intertitial infiltrate. Broncho-alveolar lavage revealed 70% haemosiderin-laden macrophages. The diagnosis of IPH was made. Since severe anaemia is disproportionate to radiologic findings, searching associated CD was performed and then confirmed by biological and histological examinations. A gluten-free diet was initiated. Evolution was favourable. Looking for especially CD in IPH should be systematic, even in the absence of gastrointestinal symptoms.