Prevalence of autoantibodies in pediatric patients with idiopathic pulmonary hemosiderosis: a scoping review of the literature in the period 1980–2021

Saha, Biplab K.; Bonnier, Alyssa; Chenna, Praveen; Milman, Nils

doi:10.1007/s10067-021-06029-3

Cited by 11 publications

(16 citation statements)

References 61 publications

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“…Several retrospective observational studies have reported the prevalence of celiac antibodies in pediatric patients with IPH 11,42,48,49 . The prevalence has varied between 17.6% and 40% 50 . Although only a small number of patients were tested and two of the studies 42,49 reported patients from the same French database, these studies suggest fairly high seropositivity for CD among patients with IPH.…”

Section: Discussionmentioning

confidence: 99%

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: 99%

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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“…The exclusion criteria were as follows: 1) articles where the patient was diagnosed with IPH as a child, even if the patient was reported as an adult; 2) articles that reported autoantibodies, demonstrated signs and symptoms of vasculitis, and had lung or kidney biopsies consistent with vasculitis during the initial diagnosis of IPH (these patients were considered to be suffering from primary vasculitides); 3) patients with bland pulmonary hemorrhage with clinical and laboratory findings diagnostic of a connective tissue disease during initial diagnosis (pulmonary hemorrhage in these patients were determined to be secondary to the rheumatologic disease and the patient was not labeled with IPH), however, if a patient with biopsy-proven IPH developed a connective tissue disease years later, the patient was included, as the causation could not be proven definitively; 4) pediatric patients with IPH and autoantibodies; these have been reported in a recent paper [ 13 ]; 5) patients reported as meeting abstracts; 6) patients reported in a non-English language.…”

Section: Reviewmentioning

confidence: 99%

“…Although LHS has been a known entity for half a century, the prevalence of LHS among IPH patients has been unknown. In children with IPH, the prevalence of anti-CD antibodies was 25.9% [ 13 ]. Based on this review, the prevalence among adults is also 25%.…”

Section: Reviewmentioning

confidence: 99%

Comparative Analysis of Adult Patients With Idiopathic Pulmonary Hemosiderosis and Lane-Hamilton Syndrome: A Systematic Review of the Literature in the Period 1971-2022

Saha¹,

Datar²,

Aiman³

et al. 2022

Cureus

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Idiopathic pulmonary hemosiderosis (IPH) causes diffuse alveolar hemorrhage (DAH) by a yet unknown mechanism. The coexistence of IPH and celiac disease (CD), also known as Lane-Hamilton syndrome (LHS), has been reported in both pediatric and adult patients. The objective of this study was to compare demographics, clinical and radiologic findings, treatment, and outcomes between adult patients with IPH and LHS. This is a systematic review of the literature. Multiple databases were searched using appropriate formulas to identify relevant articles. A total of 60 studies reporting 65 patients were included in the review. Forty-nine of these patients had IPH and 16 had LHS. The prevalence of anti-CD antibodies among tested patients was 13/22 (59%). The symptom onset and diagnosis of IPH occurred earlier in patients with LHS. The median delay in diagnosis was the same between the two groups (52 weeks). The classic triad was more likely to be present in patients with LHS. Only 20% of patients in the LHS cohort had any significant gastrointestinal (GI) symptoms at the time of IPH diagnosis. A gluten-free diet alone was effective in the majority of patients. Fewer patients in the LHS cohort received systemic corticosteroid than the IPH cohort. The recurrence and mortality in patients with LHS appear to be less than in the IPH cohort. The prevalence of CD is 25% in adult patients with IPH. Patients with LHS may have a milder course than patients without CD. Serologic testing for CD should be performed in all patients diagnosed with IPH.

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“…One of the critical aspects of such considerations is the presence of autoantibodies in patients with IPH. Although autoantibodies have been reported in pediatric IPH patients consistently [ 12 ], the true prevalence of autoantibodies in adults has been largely unknown until recently. We have recently discussed the prevalence of autoantibodies in a cohort of adult IPH patients as reported in the literature [ 13 ].…”

Section: Introductionmentioning

confidence: 99%

“…We have recently discussed the prevalence of autoantibodies in a cohort of adult IPH patients as reported in the literature [ 13 ]. Similar to pediatric patients, a majority of these patients had antibodies suggestive of celiac disease (CD), a combination known as Lane Hamilton syndrome (LHS) [ 12 , 14 , 15 ].…”

Section: Introductionmentioning

confidence: 99%

The Spectrum of Autoantibodies in Adult Patients With Idiopathic Pulmonary Hemosiderosis: A Brief Review of the Literature

Saha¹,

Bonnier²,

Saha³

et al. 2022

Cureus

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While autoimmune antibodies or autoantibodies have been reported sporadically in adult patients with idiopathic pulmonary hemosiderosis (IPH), their true prevalence is unknown. The question as to whether any difference exists between antibody-positive and negative patients has not been explored. The primary objective of this paper was to assess the spectrum of autoantibody testing and its positivity rate. The other objectives included a comparative analysis of demographics, symptom onset, clinical manifestations, and differences in clinical outcomes between antibody-positive (cohort A) and negative (cohort B) patients. To that end, we conducted a retrospective review of the relevant published literature. Multiple databases were searched to retrieve studies published between 1990 and 2022. A total of 35 studies, involving 38 patients, were identified. Five of these patients had a positive autoantibody. Patients in cohort A were older and more likely to be male. The frequencies of testing for these antibodies were as follows: antineutrophil cytoplasmic antibody (ANCA): 37/38 (97.4%), antinuclear antibody (ANA): 31/38 (81.6%), and anti-glomerular basement membrane antibody (anti-GBM): 30/38 (78.9%); 5/38 (13.2%) patients tested positive for an autoantibody, and two of these patients were positive for ANA, two for antithyroid antibody, and one patient tested positive for ANCA, rheumatoid factor (RF), and granulocyte monocyte-colony stimulating factor (GM-CSF) antibody. There was no difference between the cohorts regarding their clinical presentations, recurrence risks, and survival. The occurrence of autoantibodies is uncommon in adult IPH patients. This is in contrast with the pediatric IPH patient population, where the prevalence is much higher (26.4% vs. 13.2%), and the antibodies are more diverse. Unlike pediatric patients, adult patients with autoantibodies do not necessarily have worse outcomes.

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Prevalence of autoantibodies in pediatric patients with idiopathic pulmonary hemosiderosis: a scoping review of the literature in the period 1980–2021

Cited by 11 publications

References 61 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

Comparative Analysis of Adult Patients With Idiopathic Pulmonary Hemosiderosis and Lane-Hamilton Syndrome: A Systematic Review of the Literature in the Period 1971-2022

The Spectrum of Autoantibodies in Adult Patients With Idiopathic Pulmonary Hemosiderosis: A Brief Review of the Literature

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