Lane-Hamilton Syndrome: An Illustration of Extraintestinal Manifestations of Celiac Disease

Reed, Anna W.; Dabrowski, Ania K.; Joseph, Shelly; Paranjape, Shruti; Karwowski, Christine

doi:10.1177/0009922820941210

Cited by 3 publications

(14 citation statements)

References 23 publications

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“…Although microcytic anemia was reported in more than eighty percent of patients (83%) in our cohort, a serum ferritin level was reported in only seven patients. Four of these patients had a normal ferritin level 16,21,28,30 . A reduced ferritin level in a patient with IPH could suggest iron deficiency, and underlying CD should be sought after.…”

Section: Discussionmentioning

confidence: 95%

“…14/23 (61%) patients were treated with GFD alone. 16,18,21,22,23,24,25,27,29,31,[33][34][35][36] Six patients required systemic corticosteroid, 18,19,26,28,30,32 while two was on inhaled corticosteroid 19 in addition to a GFD. One patient was on corticosteroid and azathioprine and was never started on GFD.…”

Section: Treatment and Outcomesmentioning

confidence: 99%

“…The response to GFD was largely positive. Of the 14 patients who were treated only with GFD, no recurrence of DAH was specified in 11 patients 16,18,21,22,23,24,27,29,33–35 . In two patients, the future absence of respiratory symptoms were not specified 31,36 .…”

Section: Treatment and Outcomesmentioning

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%

“…Bilateral diffuse alveolar opacity (ground glass opacity on computed tomography) predominantly involving mid and lower lung was present in the majority of patients. However, interstitial opacity, in the form of reticular, nodular, reticulonodular and miliary pattern was also noted 16,19,21,34,36,37 …”

Section: Diagnosis Of Iphmentioning

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: Discussionmentioning

confidence: 95%

Section: Treatment and Outcomesmentioning

confidence: 99%

Section: Treatment and Outcomesmentioning

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: Diagnosis Of Iphmentioning

confidence: 99%

See 3 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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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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Lane-Hamilton syndrome: a case report

Curval,

Vieira,

da Silva-Cardoso

et al. 2024

PJP

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Idiopathic pulmonary hemosiderosis is an uncommon disease of childhood, manifesting with the triad of recurrent hemoptysis, diffuse parenchymal infiltrates on chest radiographs and iron deficiency anemia. Celiac disease is an enteropathy characterized by life-long intolerance to ingested gluten in genetically susceptible people. In 1971 Lane and Hamilton first described the association of idiopathic pulmonary hemosiderosis with celiac disease and since then a few isolated cases have been reported in literature. We report a child with pulmonary hemosiderosis presenting with hemoptysis, severe anemia, and diffuse alveolar infiltrates in whom we made a diagnosis of celiac disease. In our case, severe anemia and alveolar infiltrates markedly improved with gluten-free diet.

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Lane-Hamilton Syndrome: An Illustration of Extraintestinal Manifestations of Celiac Disease

Cited by 3 publications

References 23 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

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

Lane-Hamilton syndrome: a case report

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