Incidence of alveolar capillary dysplasia in severe idiopathic persistent pulmonary hypertension of the newborn

Tibballs, James; Chow, C. W.

doi:10.1046/j.1440-1754.2002.00014.x

Cited by 45 publications

(34 citation statements)

References 34 publications

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“…Increased incidence of persistent pulmonary hypertension of the newborn with DS suggests that histopathological changes may be present in Ts65Dn lungs [89,90]. Histopathological analysis of lung samples from both euploid and Ts65Dn mouse littermates were evaluated in a blind manner.…”

Section: Resultsmentioning

confidence: 99%

Abnormal MicroRNA Expression in Ts65Dn Hippocampus and Whole Blood: Contributions to Down Syndrome Phenotypes

et al. 2011

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Down syndrome (DS; trisomy 21) is one of the most common genetic causes of intellectual disability, which is attributed to triplication of genes located on chromosome 21. Elevated levels of several microRNAs (miRNAs) located on chromosome 21 have been reported in human DS heart and brain tissues. The Ts65Dn mouse model is the most investigated DS model with a triplicated segment of mouse chromosome 16 harboring genes orthologous to those on human chromosome 21. Using ABI TaqMan miRNA arrays, we found a set of miRNAs that were significantly up- or downregulated in the Ts65Dn hippocampus compared to euploid controls. Furthermore, miR-155 and miR-802 showed significant overexpression in the Ts65Dn hippocampus, thereby confirming results of previous studies. Interestingly, miR-155 and miR-802 were also overexpressed in the Ts65Dn whole blood but not in lung tissue. We also found overexpression of the miR-155 precursors, pri- and pre-miR-155 derived from the miR-155 host gene, known as B cell integration cluster, suggesting enhanced biogenesis of miR-155. Bioinformatic analysis revealed that neurodevelopment, differentiation of neuroglia, apoptosis, cell cycle, and signaling pathways including ERK/MAPK, protein kinase C, phosphatidylinositol 3-kinase, m-TOR and calcium signaling are likely targets of these miRNAs. We selected some of these potential gene targets and found downregulation of mRNA encoding Ship1, Mecp2 and Ezh2 in Ts65Dn hippocampus. Interestingly, the miR-155 target gene Ship1 (inositol phosphatase) was also downregulated in Ts65Dn whole blood but not in lung tissue. Our findings provide insights into miRNA-mediated gene regulation in Ts65Dn mice and their potential contribution to impaired hippocampal synaptic plasticity and neurogenesis, as well as hemopoietic abnormalities observed in DS.

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Section: Resultsmentioning

confidence: 99%

Abnormal MicroRNA Expression in Ts65Dn Hippocampus and Whole Blood: Contributions to Down Syndrome Phenotypes

et al. 2011

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“…Furthermore, it seems likely that some cases originally classified as idiopathic persistent pulmonary hypertension of the newborn (PPHN) may actually have been ACD/MPV (14,20). A slight male predominance (60%) has appeared in reported cases (21,22).…”

Section: Epidemiologymentioning

confidence: 99%

“…Lung recruitment strategies may induce respiratory alkalosis and a transient improvement in oxygenation, but may also contribute to rapid deterioration due to lung overdistension and associated compromise of right atrial filling. Recent case series have reported high rates of unilateral or bilateral pneumothorax, which could be another consequence of excessive airway pressures (20,54).…”

Section: Treatmentmentioning

confidence: 99%

“…At least one additional report showed that ACD/MPV was found in the majority of infants with idiopathic PPHN who did not respond to ECMO (six of seven infants) (20). The registry maintained by the Extracorporeal Life Support Organization indicates that more than 20% of infants with idiopathic PPHN do not respond to ECMO support, which should prompt investigation for ACD/ MPV (20,57).…”

Section: Treatmentmentioning

confidence: 99%

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Alveolar Capillary Dysplasia

Bishop

Stankiewicz

Steinhorn

2011

Am J Respir Crit Care Med

206

220

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“…However, the availability of lung biopsy is not widespread. Other conditions like congenital pulmonary lymphangiectasis and alveolar capillary dysplasia may also produce the same conundrum (22,23). Though rare, these entities present in the neonatal period and make the day-to-day diagnosis and management of ARDS challenging.…”

mentioning

confidence: 99%