Lane Hamilton Syndrome

Panda, Prasan Kumar; Sriranga, R; Kaur, Kavneet; Sood, Rita

doi:10.1007/s12098-017-2548-1

Cited by 6 publications

(21 citation statements)

References 3 publications

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“…The treatment for this patient cohort with LHS consisted primarily of gluten free diet (GFD) with or without immunosuppressive medications (Table 2). 14/23 (61%) patients were treated with GFD alone 16,18,21,22,23,24,25,27,29,31,33‐36 . Six patients required systemic corticosteroid, 18,19,26,28,30,32 while two was on inhaled corticosteroid 19 in addition to a GFD.…”

Section: Treatment and Outcomesmentioning

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%

“…Only 5/23 (22%) patients had any significant gastrointestinal symptoms at the time of diagnosis. 16,28,30,35,24 Frequent diarrhea was the most common symptom. Concurrent iron deficiency anemia was reported in 19/23 (83%) of patients.…”

Section: Diagnosis Of Celiac Diseasementioning

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: Treatment and Outcomesmentioning

confidence: 99%

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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“…6,7 Since this initial report, there have been 20 pediatric case reports of CD in conjunction with IPH or LHS. 2-4,7-16…”

Section: Discussionmentioning

confidence: 99%

“…6,7 Since this initial report, there have been 20 pediatric case reports of CD in conjunction with IPH or LHS. [2][3][4][7][8][9][10][11][12][13][14][15][16] Celiac disease is an autoimmune disorder with an immune reaction to gluten protein manifested in the small intestine and extraintestinal sites. 17,18 About 1% of the population has CD, with notable geographic variation.…”

Section: Discussionmentioning

confidence: 99%

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

Reed

Dabrowski

Joseph

et al. 2020

Clin Pediatr (Phila)

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Diffuse alveolar hemorrhage in children with interstitial lung disease: Determine etiologies!

Knoflach

Rapp

Schwerk

et al. 2023

Pediatric Pulmonology

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Objective Diffuse alveolar hemorrhage (DAH) in children is a rare condition resulting from different underlying diseases. This study aimed at describing characteristics and diagnostic measures in children with ILD (children's interstitial lung disease, chILD) and DAH to improve the diagnostic approach by increasing clinician's awareness of diagnostic shortcomings. Patients and Methods A retrospective data analysis of patients with ILD and DAH treated in our own or collaborating centers between 01/07/1997 and 31/12/2020 was performed. Data on clinical courses and diagnostic measures were systematically retrieved as case‐vignettes and investigated. To assess suitability of diagnostic software‐algorithms, the Human Phenotype Ontology (HPO) was revised and expanded to optimize conditions of its associated tool the “Phenomizer.” Results For 97 (74%) of 131 patients, etiology of pulmonary hemorrhage was clarified. For 34 patients (26%), no underlying condition was found (termed as idiopathic pulmonary hemorrhage, IPH). Based on laboratory findings or clinical phenotype/comorbidities, 20 of these patients were assigned to descriptive clusters: IPH associated with autoimmune features (9), eosinophilia (5), renal disease (3) or multiorgan involvement (3). For 14 patients, no further differentiation was possible. Conclusion Complete and sometimes repeated diagnostics are essential for establishing the correct diagnosis in children with DAH. We suggest assignment of patients with IPH to descriptive clusters, which may also guide further research. Digital tools such as the Phenomizer/HPO are promising, but need to be extended to increase diagnostic accuracy.

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Lane Hamilton Syndrome

Cited by 6 publications

References 3 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

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

Diffuse alveolar hemorrhage in children with interstitial lung disease: Determine etiologies!

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