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

Τryfon, Stavros; Papadopoulou, Erofili; Psarros, Georgios; Agrafiotis, Michael; Saroglou, Maria

doi:10.1080/00325481.2022.2109121

Cited by 6 publications

(19 citation statements)

References 86 publications

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“…The follow-up period was noted only in 16 cases, whereas seven cases required further treatment with steroids or azathioprine due to relapse or refratory disease. Of note, two patients with poor compliance with GFD relapsed even under azathioprine or high dosage of prolonged steroid therapy, which supports the importance of GFD [ 7 ].…”

Section: Discussionmentioning

confidence: 94%

“…First- and second-degree relatives of patients with CD are at increased risk [ 5 ] as well as individuals with other diseases such as autoimmune disorders (eg. type 1 diabetes mellitus, autoimmune thyroiditis), Down and Turner syndromes [ 6 ] and idiopathic pulmonary hemosiderosis (IPH) [ 7 ].…”

Section: Introductionmentioning

confidence: 99%

“…The association between IPH and CD was first described in 1971 by Lane and Hamilton. This combination, known as Lane-Hamilton syndrome (LHS), is extremely rare and, to date, no more than 80 cases have been reported [ 7 ].…”

Section: Introductionmentioning

confidence: 99%

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Lane-Hamilton Syndrome in an Adult With Down Syndrome: A Case Report

Fontes¹,

Sousa²,

Moreira³

et al. 2023

Cureus

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The Lane-Hamilton syndrome (LHS) is an extremely rare association between celiac disease (CD) and idiopathic pulmonary hemosiderosis (IPH), with only a few cases reported in the literature.The authors report a case of a 32-year-old man with Down syndrome who presented to the emergency department with a history of hemoptysis and chronic diarrhea. The complementary investigation revealed iron deficiency anemia and features suggestive of diffuse alveolar hemorrhage on computed tomography (CT) scan of the chest. After excluding all competing diagnosis, the IPH diagnosis was made. The biopsy of the small intestine confirmed CD and the diagnosis of LHS was established. A gluten-free diet and steroids were given to the patient with a very good clinical response.Since there is an established association between IPH and CD, if the diagnosis of IPH is made, it's recommended to screen for CD even in patients without gastrointestinal symptoms.

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Section: Discussionmentioning

confidence: 94%

Section: Introductionmentioning

confidence: 99%

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Lane-Hamilton Syndrome in an Adult With Down Syndrome: A Case Report

Fontes¹,

Sousa²,

Moreira³

et al. 2023

Cureus

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“…Nearly 80% of adult patients with LHS were found to be males in one study 42 . However, another review reported a higher incidence of LHS in adult women 44 . These discrepancies are likely due to the methodological differences between the studies.…”

Section: Clinical Aspects In Iphmentioning

confidence: 97%

“…42 However, another review reported a higher incidence of LHS in adult women. 44 These discrepancies are likely due to the methodological differences between the studies. The Adult patients in whom non-CD autoantibodies were identified tended to be older and predominantly males.…”

Section: Demographicsmentioning

confidence: 99%

Updates in idiopathic pulmonary hemosiderosis in 2022: A state of the art review

Saha

Aiman

Chong

et al. 2022

Pediatric Pulmonology

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This manuscript reports the recent advances in idiopathic pulmonary hemosiderosis (IPH), a rare cause of diffuse alveolar hemorrhage in children and adults. This narrative review of the literature summarizes different aspects of IPH, including proposed pathogenesis, patient demographics, clinical and radiological characteristics, treatment, and prognosis. Additionally, the association between Celiac Disease (CD) and IPH is carefully evaluated. IPH is a frequently misdiagnosed disease. The delay in the diagnosis of IPH is often significant but fortunately, appears to have decreased in recent years. IPH in adults and children have distinct demographic preferences. The autoantibodies are common in IPH but with a definite difference between the adult and pediatric populations. The definitive diagnosis of IPH requires lung biopsy and careful exclusion of all competing diagnoses, even with lung biopsy showing bland pulmonary hemorrhage. The presence of nonspecific inflammatory cells or lymphoid aggregates may suggest a secondary immunologic phenomenon and needs careful evaluation and follow-up. A substantial number of patients suffer from coexisting CD, also known as Lane−Hamilton syndrome (LHS), and all patients with IPH need to be evaluated for LHS by serology. Although strict gluten free diet can manage the majority of patients with LHS, other patients generally require immunosuppressive therapy. The corticosteroids are the backbone of IPH therapy. Recently utilized experimental treatment options include mesenchymal stem cell transplant, liposteroid and bronchial artery embolization. The immunosuppression should be adjusted to achieve optimal disease control. Patients may progress to endstage lung disease despite all measures, and lung transplantation may be the only viable option.

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

Cited by 6 publications

References 86 publications

Lane-Hamilton Syndrome in an Adult With Down Syndrome: A Case Report

Lane-Hamilton Syndrome in an Adult With Down Syndrome: A Case Report

Updates in idiopathic pulmonary hemosiderosis in 2022: A state of the art review

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

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