Diffuse lung disease in infants less than 1 year of age: Histopathological diagnoses and clinical outcome

O'Reilly, Ruth; Kilner, David; Ashworth, Michael; Aurora, Paul

doi:10.1002/ppul.23124

Cited by 12 publications

(12 citation statements)

References 30 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…The benefit of surgical biopsy remains controversial, particularly in well patients and in those with persistent tachypnoea of infancy [25]. However histological diagnosis may guide treatment decisions, especially withdrawal of care [24]. It was reassuring that in our study none of our patients underwent a transbronchial biopsy which is not recommended.…”

Section: Discussionmentioning

confidence: 86%

“…VATS has rapidly developed in the last two decades with lower rate of complications and better post-operative clinical course compared to open lung biopsy, and is the recommended approach by the US chILD committee [5, 23]. In contrast, a retrospective review of outcomes in infants with suspected chILD who had all undergone open lung biopsies at a single centre in London, found the procedure to be safe, with few adverse effects directly related to the procedure [24]. The European guidelines do not make recommendations on the type of surgical approach [20].…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Childhood interstitial lung diseases in immunocompetent children in Australia and New Zealand: a decade’s experience

Saddi

Beggs

Bennetts

et al. 2017

Orphanet J Rare Dis

View full text Add to dashboard Cite

BackgroundChildhood interstitial lung disease (chILD) represents a rare heterogeneous group of respiratory disorders. In the absence of randomized controlled clinical trials, global collaborations have utilized case series with an aim to standardising approaches to diagnosis and management. Australasian data are lacking. The aim of this study was to calculate prevalence and report the experience of chILD in Australasia over a decade.MethodsPaediatric pulmonologists in Australia and New Zealand involved in the care of patients aged 0–18 years with chILD completed a questionnaire on demographics, clinical features and outcomes, over a 10 year period. These data, together with data from the 2 reference genetics laboratories, were used to calculate prevalence.ResultsOne hundred fifteen cases were identified equating to a period prevalence (range) of 1.5 (0.8–2.1) cases/million for children aged 0–18years. Clinical data were provided on 106 patients: the <2 year group comprised 66 children, median age (range) 0.50 years (0.01–1.92); the ≥2 year group comprised 40 children, median age 8.2 years (2.0–18.0). Management approach was heterogeneous. Overall, 79% of patients had a good clinical outcome. Mortality rate was 7% in the study population.ConclusionchILD is rare in Australasia. This study demonstrates variation in the investigations and management of chILD cases across Australasia, however the general outcome is favorable. Further international collaboration will help finesse the understanding of these disorders.

show abstract

Section: Discussionmentioning

confidence: 86%

Section: Discussionmentioning

confidence: 99%

Childhood interstitial lung diseases in immunocompetent children in Australia and New Zealand: a decade’s experience

Saddi

Beggs

Bennetts

et al. 2017

Orphanet J Rare Dis

View full text Add to dashboard Cite

show abstract

“…prior to the current manuscript is 5.5 months. 20 Once patients are older than 12 months, continued respiratory symptoms should be attributed to other comorbidities and treated accordingly. Although there are typical CT findings of GGO and cystic regions, 21 biopsy is required for diagnosis of P.I.G.…”

Section: Discussionmentioning

confidence: 99%

Pulmonary interstitial glycogenosis: Diagnostic evaluation and clinical course

Liptzin

Baker

Darst

et al. 2018

Pediatric Pulmonology

View full text Add to dashboard Cite

The P.I.G. phenotype has not been comprehensively described, and poor recognition and misconceptions about P.I.G. persist. P.I.G. is a disease that presents in early infancy, requires significant medical intervention, and frequently is seen in association with alveolar simplification and/or cardiovascular disease. CT findings include ground glass opacities and cysts. Patients should be monitored for pulmonary hypertension. Without life-threatening comorbidities, many patients do well over time, although respiratory symptoms may persist into adolescence.

show abstract

“…Histological examinations show oval, glycogen-rich mesenchymal cells expanding the interstitium. No fibrosis or inflammatory reaction is seen (11)(12)(13)(14)(15)(16)(17)(18)(19)(20). Of interest, NECs are also hyperplastic (10).…”

mentioning

confidence: 99%

Persistent Tachypnea of Infancy. Usual and Aberrant

Rauch¹,

Wetzke

Reu

et al. 2016

Am J Respir Crit Care Med

View full text Add to dashboard Cite

RATIONALE: Persistent tachypnea of infancy (PTI) is a specific clinical entity of undefined etiology comprising the two diseases neuroendocrine cell hyperplasia of infancy (NEHI) and pulmonary interstitial glycogenosis. The outcome of typical NEHI is favorable. The outcome may be different for patients without a typical NEHI presentation, and thus a lung biopsy to differentiate the diseases is indicated. OBJECTIVES: To determine whether infants with the characteristic clinical presentation and computed tomographic (CT) imaging of NEHI (referred to as "usual PTI") have long-term outcome and biopsy findings similar to those of infants with an aberrant presentation and/or with additional localized minor CT findings (referred to as "aberrant PTI"). METHODS: In a retrospective cohort study, 89 infants with PTI were diagnosed on the basis of clinical symptoms and, if available, CT scans and lung biopsies. Long-term outcome in childhood was measured on the basis of current status. MEASUREMENTS AND MAIN RESULTS: Infants with usual PTI had the same respiratory and overall outcomes during follow-up of up to 12 years (mean, 3.8 yr) as infants who had some additional localized minor findings (aberrant PTI) visualized on CT images. Both usual and aberrant PTI had a relatively favorable prognosis, with 50% of the subjects fully recovered by age 2.6 years. None of the infants died during the study period. This was independent of the presence or absence of histological examination. CONCLUSIONS: PTI can be diagnosed on the basis of typical history taking, clinical findings, and a high-quality CT scan. Further diagnostic measures, including lung biopsies, may be limited to rare, complicated cases, reducing the need for an invasive and potentially harmful procedure. AbstractRationale: Persistent tachypnea of infancy (PTI) is a specific clinical entity of undefined etiology comprising the two diseases neuroendocrine cell hyperplasia of infancy (NEHI) and pulmonary interstitial glycogenosis. The outcome of typical NEHI is favorable. The outcome may be different for patients without a typical NEHI presentation, and thus a lung biopsy to differentiate the diseases is indicated.

show abstract

Diffuse lung disease in infants less than 1 year of age: Histopathological diagnoses and clinical outcome

Abstract: Lung biopsy in infants with suspected ILD is safe, and histopathological diagnosis frequently assists treatment decisions, particularly with regard to withdrawal of care.

Cited by 12 publications

References 30 publications

Childhood interstitial lung diseases in immunocompetent children in Australia and New Zealand: a decade’s experience

Childhood interstitial lung diseases in immunocompetent children in Australia and New Zealand: a decade’s experience

Pulmonary interstitial glycogenosis: Diagnostic evaluation and clinical course

Persistent Tachypnea of Infancy. Usual and Aberrant

Contact Info

Product

Resources

About