Congenital cystic adenomatoid malformation of the lung and fetal hydrops – a case with favourable outcome

Entezami, Michael; Halis, G; Waldschmidt, J.; Opri, F; Runkel, Sanyukta

doi:10.1016/s0301-2115(98)00035-9

Cited by 21 publications

(10 citation statements)

References 7 publications

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“…However, if the fetus does not fall within these gestational age restrictions or fetal resection is not an option secondary to maternal choice or due to co-morbidities, no proven options exist beyond observation or pregnancy termination. Recently, a limited number of case reports have supported the resolution of hydrops and decreased mortality in fetuses with CCAMs associated with hydrops following maternal betamethasone administration [16][17][18][19] . In the current retrospective study, the experience at our institution with fetuses diagnosed with CCAM or hybrid lesions in utero whose mothers were given a two-dose course of betamethasone was reviewed.…”

Section: Discussionmentioning

confidence: 99%

“…Recent case reports in the literature have highlighted prenatal steroid administration as a potential treatment for microcystic CCAMs associated with hydrops [16][17][18][19] . The present study is a retrospective review of patients prenatally diagnosed with CCAMs who received two prenatal betamethasone injections and were not amenable to therapeutic options such as cyst decompression by thoracoamniotic shunting or needle aspiration.…”

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confidence: 99%

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Effect of Maternal Betamethasone Administration on Prenatal Congenital Cystic Adenomatoid Malformation Growth and Fetal Survival

et al. 2007

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Objective: To evaluate the effect of prenatal steroid treatment on the growth of congenital cystic adenomatoid malformations (CCAM) and survival in affected fetuses not amenable to other percutaneous ultrasound-guided prenatal interventions. Methods: A retrospective review of patients with a CCAM or hybrid lesion treated with two maternal prenatal betamethasone injections was performed. Patients receiving cyst aspiration or thoracoamniotic shunting at the time of or after steroid administration were excluded. Growth rates and survival data were compared to historical non-steroid treated controls. Results: Eleven patients were treated with prenatal steroids (10 microcystic and 1 macrocystic). Survival was 100% in fetuses with hydrops (5/5) or a CCAM volume ratio (CVR) >1.6 (7/7) at the time of steroid administration. This compares to a mortality of 100 and 56.2% respectively in historical non-treated controls. Resolution of hydrops was seen in 80% (4/5) of steroid-treated patients. CCAM growth rates were variable after steroid administration. However, when compared to historical data where CVR and CCAM volume have been documented to increase until 28 weeks’ gestation, the CVR and CCAM volume growth rates decreased in 72.73% and 50% of patients respectively from the time of steroid administration to 28 weeks’ gestation. Conclusions: In the fetus with a CCAM, the presence of hydrops fetalis or a CVR >1.6 is indicative of poor fetal outcome without prenatal intervention. The observed effect of antenatal steroid treatment on CCAM growth is variable, but its potential to improve survival in these high-risk groups is encouraging and warrants further controlled evaluations.

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Section: Discussionmentioning

confidence: 99%

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confidence: 99%

Effect of Maternal Betamethasone Administration on Prenatal Congenital Cystic Adenomatoid Malformation Growth and Fetal Survival

et al. 2007

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“…Although resolution of hydrops is extremely rare, it has been reported either with [14][15][16][17] or without [7][8][9] medical therapy. Therefore, performing the operation in hydropic cases late in gestation (later than 28 weeks) may be unwarranted.…”

Section: Discussionmentioning

confidence: 99%

Residual congenital cystic adenomatoid malformation and thoracic scar deformation after fetal surgery: a case report

Phaloprakarn

Bartsch

Harrison

2006

Journal of Pediatric Surgery

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“…Digitalization was also useful in a case of severe aortic stenosis with endocardial fi broelastosis at 27 weeks and resulted in the resolution of hydrops and a live-birth delivery at term [ 231 ]. Digoxin has also been useful in the management of cystic adenomatoid malformations of the lung (CCAM) in conjunction with neonatal surgery [ 232 ]. In cases of fetal tachycardia (reentrant supraventricular, junctional ectopic, or ventricular) refractory to digoxin, the addition of maternal oral amiodarone can be effective in fetal hydrops and/or ventricular dysfunction [ 233 ].…”

Section: Prenatal Therapy For Fetal Hydropsmentioning

confidence: 99%

Fetal Hydrops

Nagy¹,

Malcomson²

2015

Keeling’s Fetal and Neonatal Pathology

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Hydrops fetalis, meaning "edema of the fetus," is a clinicopathological term describing an end-stage status of fetal fl uid imbalance, and the diagnostic criteria include generalized skin edema with one or more serous cavity effusions. The underlying causes include various maternal, fetal, and placental conditions. Even if the underlying forces to edema formation are the same as has been described in adults (Starling forces), the unique features of fetal physiology, including characteristics of fetal microcirculation and immaturity of the developing organs, predispose to fetal edema formation. This chapter aims to provide some insight into the underlying pathophysiological mechanisms and should help to rationalize and categories the myriad conditions that may give rise to fetal hydrops. A better understanding of the various processes at work in the generation of hydrops provides more information for parental counseling and for more targeted antepartum/perinatal treatments. The importance of postmortem examination, including ancillary investigations, cannot be emphasized enough. Recent publications highlight the usefulness of immunohistochemical assessment of lymphatic vessels in idiopathic cases. It has been also shown that, when postmortem examinations are completed along with clinical assessments, the underlying causes can be found in very high percentage of the cases (>90 %). Even if the underlying forces to edema formation are the same as has been described in adults (Starling forces), the unique features of fetal physiology, including characteristics of fetal microcirculation and immaturity of the developing organs, predispose to fetal edema formation. This chapter aims to provide some insight into the underlying pathophysiological mechanisms and should help to rationalize and categories the myriad conditions that may give rise to fetal hydrops. A better understanding of the various processes at work in the generation of hydrops provides more information

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Congenital cystic adenomatoid malformation of the lung and fetal hydrops – a case with favourable outcome

Cited by 21 publications

References 7 publications

Effect of Maternal Betamethasone Administration on Prenatal Congenital Cystic Adenomatoid Malformation Growth and Fetal Survival

Effect of Maternal Betamethasone Administration on Prenatal Congenital Cystic Adenomatoid Malformation Growth and Fetal Survival

Residual congenital cystic adenomatoid malformation and thoracic scar deformation after fetal surgery: a case report

Fetal Hydrops

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