An interesting co-existence of celiac disease and idiopathic pulmonary hemosiderosis: Lane Hamilton syndrome

Reis, Gökçe Pınar; Fettah, Ali; Volkan, Burcu; Özmen, Sevilay; Çalık, İlknur; Certel, Alev Cansu

doi:10.18621/eurj.406178

Cited by 3 publications

(11 citation statements)

References 14 publications

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“…Ferritin level was measured in 7/23 (30%) patients. 16,18,21,22,30 The ferritin was normal in four 16,21,28,30 and reduced in three. 18,22 Two studies were reported before assessment of antiendomysial or anti-tissue transglutaminase (anti-TTG) antibodies became routine practice for evaluation of CD.…”

Section: Diagnosis Of Celiac Diseasementioning

confidence: 96%

“…These patients were underweight and had short stature. 16,18,19,22,24,25,28,29,34,36 12 | DIAGNOSIS OF IPH…”

Section: Study Characteristicsmentioning

confidence: 99%

“…Ferritin level was measured in 7/23 (30%) patients 16,18,21,22,30 . The ferritin was normal in four 16,21,28,30 and reduced in three 18,22 …”

Section: Diagnosis Of Celiac Diseasementioning

confidence: 99%

“…Of the 14 patients who were treated only with GFD, no recurrence of DAH was specified in 11 patients. 16,18,21,22,23,24,27,29,[33][34][35] In two patients, the future absence of respiratory symptoms were not specified. 31,36 One patient was accidentally exposed to gluten and suffered from recurrence of hemoptysis.…”

Section: Treatment and Outcomesmentioning

confidence: 99%

“…14/23 (61%) patients were malnourished at the time of IPH diagnosis. These patients were underweight and had short stature 16,18,19,22,24,25,28,29,34,36 …”

Section: Subject Demographicsmentioning

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 Celiac Diseasementioning

confidence: 96%

“…These patients were underweight and had short stature. 16,18,19,22,24,25,28,29,34,36 12 | DIAGNOSIS OF IPH…”

Section: Study Characteristicsmentioning

confidence: 99%

“…Ferritin level was measured in 7/23 (30%) patients 16,18,21,22,30 . The ferritin was normal in four 16,21,28,30 and reduced in three 18,22 …”

Section: Diagnosis Of Celiac Diseasementioning

confidence: 99%

Section: Treatment and Outcomesmentioning

confidence: 99%

“…14/23 (61%) patients were malnourished at the time of IPH diagnosis. These patients were underweight and had short stature 16,18,19,22,24,25,28,29,34,36 …”

Section: Subject Demographicsmentioning

confidence: 99%

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