Coincidence or connection? A patient with concurrent Lane Hamilton Syndrome and idiopathic membranous nephropathy

Austin, Scarlett; Kobrin, Dale; Villgran, Vipin; Nestasie, Michael; Chen, Feifan; Hardman, Brent; Malik, Khalid

doi:10.1016/j.rmcr.2021.101446

Cited by 3 publications

(10 citation statements)

References 18 publications

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“…Cohort A included patients with IPH without CD (49 patients). Cohort B consisted of patients with LHS and consisted of 16 patients [ 11 , 22 , 26 - 27 , 31 , 37 , 41 , 45 , 52 - 53 , 55 - 56 , 58 - 59 , 67 , 72 ].…”

Section: Reviewmentioning

confidence: 99%

“…The radiologic findings in cohorts A and B were as follows: bilateral infiltrate (91.5% vs.100%), emphysema or cystic changes (19.1% vs. 18.8%), and interstitial changes (27.7% vs. 25%). Pleural effusion was present in only one patient with LHS [ 67 ]. The classic triad was present in 74.5% vs. 93.8% of patients.…”

Section: Reviewmentioning

confidence: 99%

“…All but one study specified histopathologic analysis of small bowel biopsy to confirm CD [ 45 ]. Three out of 16 patients were known to have CD before the diagnosis of IPH [ 37 , 45 , 67 ]. Two patients were diagnosed with CD after IPH was identified [ 22 , 52 ].…”

Section: Reviewmentioning

confidence: 99%

“…The biopsy techniques employed between cohorts A and B are as follows: surgical lung biopsy (46.3% vs. 27.2%), video-assisted thoracoscopic surgery (21.95% vs. 18.18%), and transbronchial lung biopsy (TBLB; 24.39% vs. 54.54%). One of the TBLB was a cryobiopsy [ 67 ]. In cohort A, the histopathologic analysis was performed post-mortem in three (7.31%).…”

Section: Reviewmentioning

confidence: 99%

“…Three patients were initially treated with GFD and CS, and successfully tapered off steroids [ 52 , 55 , 59 ]. One patient, in addition to GFD, required long-term CS [ 67 ]. However, the compliance to GFD in this patient was questionable.…”

Section: Reviewmentioning

confidence: 99%

See 4 more Smart Citations

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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Section: Reviewmentioning

confidence: 99%

Section: Reviewmentioning

confidence: 99%

Section: Reviewmentioning

confidence: 99%

Section: Reviewmentioning

confidence: 99%

Section: Reviewmentioning

confidence: 99%

See 3 more Smart Citations

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

View full text Add to dashboard Cite

show abstract

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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Adult patients with idiopathic pulmonary hemosiderosis: a comprehensive review of the literature

Saha

Bonnier

Saha³

et al. 2022

Clin Rheumatol

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Coincidence or connection? A patient with concurrent Lane Hamilton Syndrome and idiopathic membranous nephropathy

Cited by 3 publications

References 18 publications

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

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

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

Adult patients with idiopathic pulmonary hemosiderosis: a comprehensive review of the literature

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