Pulmonary haemosiderosis in a 13-year-old girl with coeliac disease after 3 months on a gluten-free diet: case report and review of the literature

Najada, Abdelhamid S.; Dahabreh, Muna M.

doi:10.1179/146532810x12786388978841

Cited by 4 publications

(16 citation statements)

References 14 publications

(49 reference statements)

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“…Increased mucosal vascularity was noted in patients, 32,35 whereas, one patient 30 had pale airway mucosa. 3/17 patients also underwent transbronchial lung biopsy during bronchoscopy 24,28,30 . The lung biopsy reveled hemosiderin laden macrophages in the alveolar space.…”

Section: Diagnosis Of Iphmentioning

confidence: 85%

“…However, due to the inherently invasive nature of the biopsy and risks associated with it, many pediatric patients are diagnosed without histopathologic proof. Only 3 patients underwent transbronchial lung biopsy among all identified patients in this manuscript 24,28,30 . Although there is a possibility of missing AAV or isolated pauci‐immune pulmonary capillaritis (despite a negative autoantibody screening) without a lung biopsy, fortunately, these diseases are extremely rare in children 73 …”

Section: Discussionmentioning

confidence: 99%

“…The authors noticed evidence of old bleeding in the airways. One patient had clear BAL return but hemosiderin laden macrophages were seen on microscopic examination 30 . Increased mucosal vascularity was noted in patients, 32,35 whereas, one patient 30 had pale airway mucosa.…”

Section: Diagnosis Of Iphmentioning

confidence: 90%

“…Only 3 patients underwent transbronchial lung biopsy among all identified patients in this manuscript. 24,28,30 Although there is a possibility of missing AAV or isolated pauci-immune pulmonary capillaritis (despite a negative autoantibody screening) without a lung biopsy, fortunately, these diseases are extremely rare in children. 73 Iron deposition in nonpulmonary tissue has never been demonstrated in IPH.…”

Section: Discussionmentioning

confidence: 99%

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Association between idiopathic pulmonary hemosiderosis and celiac disease in pediatric patients: A scoping review of the literature over the past 50 years

Saha

Bonnier

et al. 2022

Pediatric Pulmonology

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Introduction: Idiopathic pulmonary hemosiderosis (IPH) is a rare cause of diffuse alveolar hemorrhage, the mechanism of which is currently unknown. Nearly onethird of pediatric patients with IPH test positive for Celiac disease (CD) serology.Several hypothetical mechanisms have been proposed to unify the coexistence of these two entities, also referred to as Lane-Hamilton syndrome (LHS).Method: This manuscript is a scoping review of the medical literature. Medline, Embase, and PubMed Central databases were searched between 1971 and 2021 with appropriate search words to identify all cases of pediatric LHS.Results: A total of 20 manuscripts with 23 pediatric patients with LHS were identified. The mean age was 11 years, and 13/23 (56.5%) of the children were boys.Hemoptysis was present in 57% of patients during diagnosis. Bronchoscopy with bronchoalveolar lavage demonstrating hemosiderin laden macrophages was the primary mode of diagnostic confirmation. Only three patients underwent lung biopsy. Any significant GI symptom was reported in a minority of patients (22%). Iron deficiency anemia on presentation was described in 83% of children. The majority of patients were malnourished. Serology for CD was positive in all patients, as was the histopathologic analysis of the small bowel biopsy. No patients had any other autoantibody positivity. The introduction of gluten free diet (GFD) was associated with a positive response in 20/23 patients. Conclusion:All pediatric patients with IPH should undergo screening for CD. Low serum ferritin in patients with IPH could be suggestive of coexisting CD. Strict GFD should be tried as the initial therapy.

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Section: Diagnosis Of Iphmentioning

confidence: 85%

Section: Discussionmentioning

confidence: 99%

Section: Diagnosis Of Iphmentioning

confidence: 90%

Section: Discussionmentioning

confidence: 99%

See 2 more Smart Citations

Association between idiopathic pulmonary hemosiderosis and celiac disease in pediatric patients: A scoping review of the literature over the past 50 years

Saha

Bonnier

et al. 2022

Pediatric Pulmonology

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“…Beside these studies there are only paediatric case reports or series that contain mostly adult patients [44][45][46][47][48][49][50]; no major complications were reported. TBB was not helpful in making a diagnosis of pulmonary cryptococcosis [51].…”

Section: Transbronchial and Endobronchial Biopsymentioning

confidence: 99%

ERS statement: interventional bronchoscopy in children

et al. 2017

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Paediatric airway endoscopy is accepted as a diagnostic and therapeutic procedure, with an expanding number of indications and applications in children. The aim of this European Respiratory Society task force was to produce a statement on interventional bronchoscopy in children, describing the evidence available at present and current clinical practice, and identifying areas deserving further investigation. The multidisciplinary task force panel performed a systematic review of the literature, focusing on whole lung lavage, transbronchial and endobronchial biopsy, transbronchial needle aspiration with endobronchial ultrasound, foreign body extraction, balloon dilation and occlusion, laser-assisted procedures, usage of airway stents, microdebriders, cryotherapy, endoscopic intubation, application of drugs and other liquids, and caregiver perspectives. There is a scarcity of published evidence in this field, and in many cases the task force had to resort to the collective clinical experience of the committee to develop this statement. The highlighted gaps in knowledge underline the need for further research and serve as a call to paediatric bronchoscopists to work together in multicentre collaborations, for the benefit of children with airway disorders.

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Celiac disease and idiopathic pulmonary hemosiderosis: a literature review of the Lane–Hamilton syndrome

Τryfon

Papadopoulou

Psarros³

et al. 2022

Postgraduate Medicine

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Pulmonary haemosiderosis in a 13-year-old girl with coeliac disease after 3 months on a gluten-free diet: case report and review of the literature

Cited by 4 publications

References 14 publications

Association between idiopathic pulmonary hemosiderosis and celiac disease in pediatric patients: A scoping review of the literature over the past 50 years

Association between idiopathic pulmonary hemosiderosis and celiac disease in pediatric patients: A scoping review of the literature over the past 50 years

ERS statement: interventional bronchoscopy in children

Celiac disease and idiopathic pulmonary hemosiderosis: a literature review of the Lane–Hamilton syndrome

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