Short Review of Liposteroid: A Novel Targeted Glucocorticoid Preparation for Treatment of Autoimmune and Inflammatory Diseases

Saha, Biplab K.; Milman, Nils

doi:10.14712/23362936.2021.23

Cited by 12 publications

(8 citation statements)

References 36 publications

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“…11,24,42,49,53,64 The dismal survival in the past has improved with early introduction of immunosuppressive therapy. 11,53,63,82 Intriguingly, the patients in this cohort demonstrated a remarkable response to GFD. The majority of patients were managed with GFD without any recurrence of their hemorrhage.…”

Section: Discussionmentioning

confidence: 74%

“…Previous experience suggests that the treatment is often complicated by frequent recurrences and sometimes development of end‐stage lung disease requiring lung transplantation 11,24,42,49,53,64 . The dismal survival in the past has improved with early introduction of immunosuppressive therapy 11,53,63,82 . Intriguingly, the patients in this cohort demonstrated a remarkable response to GFD.…”

Section: Discussionmentioning

confidence: 90%

See 1 more Smart Citation

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: 74%

Section: Discussionmentioning

confidence: 90%

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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“…Approximately one-third of reported adult patients have required a second-line immunosuppressive agent to control their disease [ 86 ]. Unconventional therapies, such as liposteroid, leflunomide, and mesenchymal stem cell transplant, have also been tried [ 100 - 102 ]. In contrast, GFD alone was effective in controlling DAH in the majority of patients with LHS.…”

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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“…Liposteroid is a liposomal preparation of dexamethasone 21‐palmitate, which is more lipophilic than dexamethasone sodium phosphate (DSP). Liposteroid achieves a higher concentration in the inflamed tissue and demonstrates more potent anti‐inflammatory properties than DSP 75 . Several case reports and a retrospective study have reported the use of liposteroid in children with IPH 48,76,77 .…”

Section: Clinical Aspects In Iphmentioning

confidence: 99%

“…Liposteroid achieves a higher concentration in the inflamed tissue and demonstrates more potent anti-inflammatory properties than DSP. 75 Several case reports and a retrospective study have reported the use of liposteroid in children with IPH. 48,76,77 The liposteroid appears to be efficacious and associated with a better side-effect profile than conventional steroid therapy.…”

Section: Treatmentmentioning

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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Short Review of Liposteroid: A Novel Targeted Glucocorticoid Preparation for Treatment of Autoimmune and Inflammatory Diseases

Cited by 12 publications

References 36 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

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

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