Proinflammatory macrophage migratory inhibition factor and interleukin‐6 are concentrated in pleural effusion of human fetuses with prenatal chylothorax

Chen, Ming; Hsieh, Chang‐Yao; Shih, Jin-Chung; Chou, Chia-Hung; Ma, Gwo‐Chin; Chen, Tze-Ho; Lee, Tsung-Hsien; Tsai, Horng‐Der; Cameron, Alan; Chen, Chih‐Ping

doi:10.1002/pd.1704

Cited by 15 publications

(14 citation statements)

References 44 publications

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“…Familial occurrence in an autosomal recessive inheritance pattern has been observed in four families where chylothorax has affected 2 or more siblings [20,32]. In our cohort, 1 fetus (case 9) that did not have resolution with OK-432 and had NND had 2 previous siblings affected by PFHT with hydrops; one pregnancy resulting in FDIU at 23 weeks and another resulted in a LB at 32 weeks after in utero treatment with TAS and postnatal chest tube.…”

Section: Discussionmentioning

confidence: 88%

“…The mean GA at insertion of shunt was 29 weeks, with a GA delivery of 34 weeks. Three case series involving 61 fetuses with PFHT were treated with OK-432 [20,21,22]. The largest, most recent series of 45 fetuses with PFHTs reported neonatal survival of only 36% and survival of 15 and 67% in hydropic and non-hydropic fetuses, respectively.…”

Section: Resultsmentioning

confidence: 99%

“…Potential therapeutic interventions include thoracocentesis (either single and multiple procedures), thoraco-amniotic shunting (TAS) [8,9,10,11,12,13] and, more recently, pleurodesis using OK-432 (Picibanil) [14,15,16,17,18,19,20,21,22]. The primary aim of all these interventions is to remove fluid from the chest and reduce the adverse effects of the space-occupying collection on pulmonary development, cardiovascular function and fetal swallowing.…”

Section: Introductionmentioning

confidence: 99%

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Treatment of Primary Fetal Hydrothorax with OK-432 (Picibanil): Outcome in 14 Fetuses and a Review of the Literature

O'Brien¹,

Kesby²,

Ogle³

et al. 2015

Fetal Diagn Ther

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Background: Primary fetal hydrothorax (PFHT) is an uncommon condition with an estimated prevalence of 1 in 10,000/15,000 pregnancies. Therapeutic interventions include thoracocentesis, thoraco-amniotic shunting (TAS), and pleurodesis using OK-432. Methods: A review of the literature was performed to identify all cases of PFHT treated with TAS and OK-432. All cases of PFHT referred to the Fetal Maternal Unit at Royal Prince Alfred Hospital between 2002 and 2012 were retrospectively reviewed. In the cohort of fetuses treated with OK-432, the main perinatal outcomes evaluated were termination of pregnancy, live birth, neonatal death, and fetal death in utero. Secondary outcomes included gestational age (GA) at diagnosis, GA at treatment, GA at resolution, birth weight, and GA at birth. The development of the children was screened using the Ages and Stages Questionnaires, Version 3 (ASQ-3, 2009). Results: Primary hydrothorax was diagnosed in 31 fetuses, of which 14 had treatment with OK-432. One pregnancy terminated after treatment with OK-432. Survival was 85% (11/13): 100% in fetuses treated with OK-432 without hydrops, and 78% in those treated with hydrops. This compares well to the cases of TAS in the literature with an average survival of 63%: 85% in fetuses without hydrops and 55% with hydrops. The mean GA at birth was 36⁺⁴ weeks and mean birth weight 3,007 g. Eight of the 9 children screened with ASQ-3 scored well within the normal range. Conclusion: OK-432 appears to be a valid treatment option in fetuses with PFHT, particularly in those diagnosed at early GAs.

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Section: Discussionmentioning

confidence: 88%

Section: Resultsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Treatment of Primary Fetal Hydrothorax with OK-432 (Picibanil): Outcome in 14 Fetuses and a Review of the Literature

O'Brien¹,

Kesby²,

Ogle³

et al. 2015

Fetal Diagn Ther

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“…IL-6 also seems to play an important anti-inflammatory role in pleural effusions, breaking with the traditional idea of IL-6 being a proinflammatory acute phase reaction cytokine only [20]. IL-6, IL-8, and VEGF also play a role in the pathogenesis of prenatal chylothoraces in human fetuses [21]. The diagnosis of tuberculous pleural effusions by determination of CRP, IL-1α, IL-6, tumour necrosis factor (TNF)-α, and interferon (IFN)-γ has been the focus of numerous studies [2,22,23].…”

Section: Discussionmentioning

confidence: 92%

Mediators in pleural effusions of different origin: a two-step diagnostic study

Stephan

Hoheisel

Geßner

et al. 2014

LaboratoriumsMedizin

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Background: Many mediators in pleural effusions give diagnostic information. This study therefore aimed to find out whether the repeated determination of interleukin (IL)-6, IL-15, vascular endothelial growth factor (VEGF), and tissue inhibitors of metalloproteinase (TIMP)-2 would confirm previous results and, furthermore, whether a combination of these parameters could achieve a higher diagnostic yield. Methods: Two consecutive groups of patients with pleural effusions were included. The underlying disease entities in series I vs. series II were: 12 vs. 0 tuberculosis (TB), 19 vs. 30 primary lung cancer, 7 vs. 14 secondaries to the lung, 5 vs. 13 congestive heart failure, and 2 vs. 7 parapneumonic effusion.

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“…Congenital chylothorax (CC), or fetal chylothorax (FC), is a rare condition (estimated incidence, 1 in 12 000), characterized by accumulation of lymphocyte-rich fluid in the pleural cavity [1]. Its pathogenesis remains elusive.…”

Section: Introductionmentioning

confidence: 99%

Genome-Wide Gene Expression Analysis Implicates the Immune Response and Lymphangiogenesis in the Pathogenesis of Fetal Chylothorax

Yeang

Shih

et al. 2012

PLoS ONE

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Fetal chylothorax (FC) is a rare condition characterized by lymphocyte-rich pleural effusion. Although its pathogenesis remains elusive, it may involve inflammation, since there are increased concentrations of proinflammatory mediators in pleural fluids. Only a few hereditary lymphedema-associated gene loci, e.g. VEGFR3, ITGA9 and PTPN11, were detected in human fetuses with this condition; these cases had a poorer prognosis, due to defective lymphangiogenesis. In the present study, genome-wide gene expression analysis was conducted, comparing pleural and ascitic fluids in three hydropic fetuses, one with and two without the ITGA9 mutation. One fetus (the index case), from a dizygotic pregnancy (the cotwin was unaffected), received antenatal OK-432 pleurodesis and survived beyond the neonatal stage, despite having the ITGA9 mutation. Genes and pathways involved in the immune response were universally up-regulated in fetal pleural fluids compared to those in ascitic fluids. Furthermore, genes involved in the lymphangiogenesis pathway were down-regulated in fetal pleural fluids (compared to ascitic fluid), but following OK-432 pleurodesis, they were up-regulated. Expression of ITGA9 was concordant with overall trends of lymphangiogenesis. In conclusion, we inferred that both the immune response and lymphangiogenesis were implicated in the pathogenesis of fetal chylothorax. Furthermore, genome-wide gene expression microarray analysis may facilitate personalized medicine by selecting the most appropriate treatment, according to the specific circumstances of the patient, for this rare, but heterogeneous disease.

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Proinflammatory macrophage migratory inhibition factor and interleukin‐6 are concentrated in pleural effusion of human fetuses with prenatal chylothorax

Cited by 15 publications

References 44 publications

Treatment of Primary Fetal Hydrothorax with OK-432 (Picibanil): Outcome in 14 Fetuses and a Review of the Literature

Treatment of Primary Fetal Hydrothorax with OK-432 (Picibanil): Outcome in 14 Fetuses and a Review of the Literature

Mediators in pleural effusions of different origin: a two-step diagnostic study

Genome-Wide Gene Expression Analysis Implicates the Immune Response and Lymphangiogenesis in the Pathogenesis of Fetal Chylothorax

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