Prediction of postnatal outcome in fetuses with congenital lung malformation: 2‐year follow‐up study

Peters, N.; Hijkoop, Annelieke; Hermelijn, Sergei M.; Schoonhoven, M.M. van; Eggink, A.J.; Rosmalen, Joost van; Otter, Suzan C M Cochius-den; Tibboel, Dick; IJsselstijn, Hanneke; Schnater, J. Marco; Cohen–Overbeek, Titia E.

doi:10.1002/uog.23542

Cited by 13 publications

(5 citation statements)

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“…At this point, the prenatal ultrasound images will be retrospectively evaluated according to a structured report (see online supplemental file 7 ). 46 In case of randomisation to the surgical arm, resected specimen will be sent to the local pathology department for analysis and the pathological results will be discussed with parents during the hospital admission or the postoperative visit at the outpatient clinic some 2–6 weeks after surgery.…”

Section: Methods and Analysismentioning

confidence: 99%

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COllaborative Neonatal Network for the first European CPAM Trial (CONNECT): a study protocol for a randomised controlled trial

et al. 2023

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IntroductionConsensus is lacking on the optimal management of asymptomatic congenital pulmonary airway malformation (CPAM). For future studies, the CONNECT consortium (the COllaborative Neonatal Network for the first European CPAM Trial)—an international collaboration of specialised caregivers—has established consensus on a core outcome set of outcome parameters concerning respiratory insufficiency, surgical complications, mass effect and multifocal disease. These outcome parameters have been incorporated in the CONNECT trial, a randomised controlled trial which, in order to develop evidence-based practice, aims to compare conservative and surgical management of patients with an asymptomatic CPAM.Methods and analysisChildren are eligible for inclusion after the CPAM diagnosis has been confirmed on postnatal chest CT scan and they remain asymptomatic. On inclusion, children are randomised to receive either conservative or surgical management. Subsequently, children in both groups are enrolled into a standardised, 5-year follow-up programme with three visits, including a repeat chest CT scan at 2.5 years and a standardised exercise tolerance test at 5 years.The primary outcome is exercise tolerance at age 5 years, measured according to the Bruce treadmill protocol. Secondary outcome measures are molecular genetic diagnostics, validated questionnaires—on parental anxiety, quality of life and healthcare consumption—, repeated imaging and pulmonary morbidity during follow-up, as well as surgical complications and histopathology. This trial aims to end the continuous debate surrounding the optimal management of asymptomatic CPAM.Ethics and disseminationThis study is being conducted in accordance with the Declaration of Helsinki. The Medical Ethics Review Board of Erasmus University Medical Centre Rotterdam, The Netherlands, has approved this protocol (MEC-2022–0441). Results will be disseminated through peer-reviewed scientific journals and conference presentations.Trial registration numberNCT05701514.

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Section: Methods and Analysismentioning

confidence: 99%

“…CPAM characteristics on prenatal ultrasound images, reported according to structured report (see online supplemental file 7 ). 46 If multiple ultrasounds were undertaken, those images closest to the gestational age of 30 weeks will be used for analysis.…”

Section: Methods and Analysismentioning

confidence: 99%

COllaborative Neonatal Network for the first European CPAM Trial (CONNECT): a study protocol for a randomised controlled trial

et al. 2023

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“…According to the Stocker classification, CPAM is classified into five types, namely, type 0 (3%), type I (60-70%), type II (15-20%), type III (5-10%), and type IV (10-15%) [3,4]. Imaging and histopathological analysis of biopsies are the most commonly used tools for the clinical diagnosis for CPAM; however, CPAM can be asymptomatic, easily missed, or misdiagnosed, leading to increased risk of tumor development [5][6][7]. The pathogenesis of CPAM is complex, calling for a better understanding of the molecular mechanisms underlying its development and progression.…”

Section: Introductionmentioning

confidence: 99%

The Role of the miR-548au-3p/CA12 Axis in Tracheal Chondrogenesis in Congenital Pulmonary Airway Malformations

Zeng

Zhang

Yang

et al. 2023

Oxidative Medicine and Cellular Longevity

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Background. Literature has identified differentially expressed miRNAs in congenital pulmonary airway malformation (CPAM). However, the functional role of these miRNAs in CPAM remains unclear. Methods. We obtained diseased lung tissues as well as adjacent normal lung tissue from CPAM patients attending the centre. Hematoxylin and eosin (H&E) and Alcian blue staining were performed. Differentially expressed mRNA expression profile was CPAM tissue, and matched normal tissue specimens were examined by high-throughput RNA sequencing. CCK-8 assay, EdU staining, TUNEL staining, flow cytometry, and the Transwell assay were performed to investigate the effect of miR-548au-3p/CA12 axis on proliferation, apoptosis, and chondrogenic differentiation in rat tracheal chondrocytes. mRNA and protein expression levels were determined using reverse transcription-quantitative PCR and western blot analysis, respectively. The relationship between miR-548au-3p and CA12 was evaluated using the luciferase reporter assay. Results. The expression level of miR-548au-3p was significantly increased in diseased tissues compared with normal adjacent tissues from patients with CPAM. Our results indicate that miR-548au-3p functions as a positive regulator in rat tracheal chondrocyte proliferation and chondrogenic differentiation. At molecular level, miR-548au-3p promoted N-cadherin, MMP13, and ADAMTS4 expressions and reduced E-cadherin, aggrecan, and Col2A1 expressions. CA12 has been previously reported as a predicted target of miR-548au-3p, and here, we show that overexpression of CA12 in rat tracheal chondrocyte mimics the effects of inhibition of miR-548au-3p. On the other hand, CA12 knockdown reversed the effects of miR-548au-3p on cell proliferation, apoptosis, and chondrogenic differentiation. Conclusions. In conclusion, the miR-548au-3p/CA12 axis plays a role in the pathogenesis of CPAM and may lead to identification of new approaches for CPAM treatment.

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“…The incidence of CLA has increased in recent years to ∼4 in 10,000 births, presumably because the performance of antenatal ultrasound-guided diagnostics has improved [ 3 ]. If an infant remains asymptomatic after birth, which is true for the majority of cases, the choice between surgical intervention and conservative wait-and-see management is not standardized and highly depends on local clinical guidelines [ 4–8 ]. However, if symptoms occur in the first years of life (i.e.…”

Section: Introductionmentioning

confidence: 99%

Preoperative visualization of congenital lung abnormalities: hybridizing artificial intelligence and virtual reality

Bakhuis

Kersten

Sadeghi

et al. 2022

European Journal of Cardio-Thoracic Surgery

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OBJECTIVES When surgical resection is indicated for a congenital lung abnormality, lobectomy is often preferred over segmentectomy, mostly because the latter is associated with more residual disease. Presumably, this occurs in children because sublobar surgery often does not adhere to anatomical borders (wedge resection instead of segmentectomy), thus increasing the risk of residual disease. This study investigated the feasibility of identifying eligible cases for anatomical segmentectomy by combining virtual reality and artificial intelligence. METHODS Semi-automated segmentation of bronchovascular structures and lesions were visualized with virtual reality and artificial intelligence technology. Two specialists independently evaluated via a questionnaire the informative value of regular computed tomography versus three-dimensional-virtual reality images. RESULTS Five asymptomatic, non-operated cases were selected. Bronchovascular segmentation, volume calculation, and image visualization in the VR environment was successful in all cases. Based on the computed tomography images, assignment of the congenital lung abnormality lesion to specific lung segments matched between the consulted specialists in only one out of the cases. Based on the three three-dimensional-virtual reality images, however, the localization matched in three of the five cases. If the patients would have been operated, adding the three-dimensional-virtual reality tool to the pre-operative workup would have resulted in changing the surgical strategy (i.e., lobectomy versus segmentectomy) in four cases. CONCLUSIONS This study demonstrated the technical feasibility of a hybridized artificial intelligence –virtual reality visualization of segment-level lung anatomy in patients with congenital lung abnormalities. Further exploration of the value of three-dimensional-virtual reality in identifying eligible cases for anatomical segmentectomy is therefore warranted.

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Prediction of postnatal outcome in fetuses with congenital lung malformation: 2‐year follow‐up study

Abstract: This article has been accepted for publication and undergone full peer review but has not been through the copyediting, typesetting, pagination and proofreading process which may lead to differences between this version and the Version of Record. Please cite this article as

Cited by 13 publications

References 30 publications

COllaborative Neonatal Network for the first European CPAM Trial (CONNECT): a study protocol for a randomised controlled trial

COllaborative Neonatal Network for the first European CPAM Trial (CONNECT): a study protocol for a randomised controlled trial

The Role of the miR-548au-3p/CA12 Axis in Tracheal Chondrogenesis in Congenital Pulmonary Airway Malformations

Preoperative visualization of congenital lung abnormalities: hybridizing artificial intelligence and virtual reality

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