Congenital adenomatoid malformation of the lung: When is active fetal therapy indicated?

Dommergues, Marc; Louis-Sylvestre, Christine; Mandelbrot, Laurent; Aubry, Marie Cécile; Révillon, Y.; Jarreau, Pierre Henri

doi:10.1016/s0002-9378(97)70300-4

Cited by 91 publications

(52 citation statements)

References 10 publications

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“…Thoracoamniotic shunting decreased the CCAM mass volume by 50%, with improved survival (table 4). The neonatal survival was similar for both treatment groups: 67 and 70% for PE and CCAM cases, respectively, which exceeds that of previously reported series (table 6) [19][20][21][22][23][24].…”

Section: Discussionsupporting

confidence: 42%

“…The prenatal treatment of the fetal populations with PE and CCAM are summarized in tables 6 [8,[18][19][20][21][22] and 7 [21,[23][24][25][26][27][28]. Fetal hydrops impacts survival for CCAM, as shown in table 7, for the liveborn continuing group at 8/117 (7%) while in the perinatal loss group at 22/42 (52%).…”

Section: Discussionmentioning

confidence: 99%

“…There was no definition of factors used to define prolonged severe lung compression. Dommergues et al [24] recommended conservative management in cases of CCAM without acute polyhydramnios or hydrops. This was a prospective cohort study of 33 cases, in whom thoracoamniotic shunting was offered in only 9 macrocystic CCAM cases with acute polyhydramnios or hydrops.…”

Section: Discussionmentioning

confidence: 99%

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Thoracoamniotic Shunts: Fetal Treatment of Pleural Effusions and Congenital Cystic Adenomatoid Malformations

et al. 2004

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Objective: To determine whether fetuses that underwent thoracoamniotic shunt placement for treatment of pleural effusion (PE) or macrocystic congenital cystic adenomatoid malformation (CCAM) have an improved outcome as compared with an untreated population. Methods: A retrospective review from a single tertiary center was performed using thoracoamniotic shunt placement to treat PE or macrocystic CCAM between 1998 and 2001. Thoracoamniotic shunts were used on 26 occasions in 19 pregnancies. Results: The average gestational age at the diagnosis of PE and CCAM was 22 + 4 and 20 + 0 weeks, respectively. Shunts were offered in pregnancies complicated by hydrops or at significant risk for pulmonary hypoplasia. Shunts were placed at 26 + 2 weeks (average) and 23 + 1 weeks (average) in the PE and CCAM groups, respectively. In CCAM patients, the mean pre- and postshunting mass volumes were 50.5 and 25.7 cm³, representing a 51% reduction in mass volume following shunt placement. In the PE group, the average delivery age was 33 + 5 weeks, with an average shunt placement to delivery time of 7 + 3 weeks. In the CCAM group, the average delivery was 33 + 3 weeks, with an average shunt placement to delivery time of 10 + 2 weeks. The postnatal survival rates were 67% (6/9) and 70% (7/10) in the PE and CCAM groups, respectively. Conclusions: (1) Thoracoamniotic shunts should be considered as a treatment option for selected PE or macrocystic CCAM fetuses with hydrops or a significant risk for pulmonary hypoplasia; (2) the neonatal survival with shunting was similar for PE and CCAM groups and was improved as compared with literature reports, and (3) fetuses with CCAM presented earlier with hydrops than those with PE. Successful shunting resulted in a prolongation of pregnancy into the 3rd trimester in both groups.

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Section: Discussionsupporting

confidence: 42%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Thoracoamniotic Shunts: Fetal Treatment of Pleural Effusions and Congenital Cystic Adenomatoid Malformations

et al. 2004

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“…Thoracoamniotic shunting is effective in cases with a large predominant cyst as long as there is not a large solid component to the CCAM ( fig. 1) [3,7]. During the past 3 years, we have managed 9 CCAM pregnancies with thoracoamniotic shunting [8].…”

Section: Fetal Lung Lesionsmentioning

confidence: 99%

History of Fetal Diagnosis and Therapy: Children’s Hospital of Philadelphia Experience

et al. 2003

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Since its inception in 1995, the mission of the Center for Fetal Diagnosis and Treatment at the Children’s Hospital of Philadelphia (CHOP) has been to provide comprehensive, multidisciplinary expertise in all facets of prenatal diagnosis, reproductive genetics, and prenatal, perinatal, and postnatal treatment for abnormal fetuses. Most of the authors were trained and/or served on faculty at centers in San Francisco (N.S.A., A.W.F., T.M.C., L.J.H.) or Detroit (A.W.F, M.P.J.). Accurate prenatal detection by serial sonographic study and the development of ultrafast magnetic resonance imaging (MRI) scanners has permitted delineation of the natural history of anatomic malformations. Definition of the pathophysiological features that affect clinical outcome and formulation of management based on prognosis has allowed families to make informed decisions. Outcomes have been improved through carefully planned and coordinated perinatal management. In some cases of life-threatening or severely debilitating defects, prenatal surgery has been offered.

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“…Serial sonographic study of fetuses with thoracic lesions has helped define the natural history of these lesions, determine the pathophysiologic features that affect clinical outcome, and formulate management based on prognosis [1,2,3,4]. A large mass may cause mediastinal shift, hypoplasia of normal lung tissue, polyhydramnios, and cardiovascular compromise leading to fetal hydrops and death.…”

Section: Introductionmentioning

confidence: 99%

Early Neurodevelopmental Outcome of Infants with High-Risk Fetal Lung Lesions

Danzer

Siegle²,

D’Agostino³

et al. 2012

Fetal Diagn Ther

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Objective: To evaluate the neurodevelopmental outcome of infants with high-risk fetal lung lesions defined as (1) requiring fetal intervention and/or ex utero intrapartum therapy (EXIT), or (2) acute respiratory decompensation postnatally necessitating emergent resection within 48 h of life. Methods: We reviewed the medical records of 13 consecutive patients with high-risk fetal lung lesions who were enrolled in our prospective interdisciplinary follow-up program. Neurodevelopmental status was evaluated using the Bayley Scales of Infant Development-III (children ≤3 years, n = 12), or the Wechsler Preschool and Primary Scale of Intelligence-III (children ≥4 years, n = 1). Results: Eight children (62%) underwent prenatal intervention (EXIT, n = 6; fetal resection, n = 1; intrauterine shunt placement, n = 1), and 5 (38%) required emergent resection postnatally. Median age at evaluation was 25 months (range: 5–80). Average scores for cognitive development were found in all children assessed under 3 years of age. The one child who was tested for cognitive ability at 6 years of age scored in the borderline range of intellectual functioning. For language outcome, 15% scored above average, 54% scored within the average range, and 31% had mild deficits. Overall, 77% scored within the average range for neuromotor outcome, while 23% scored within the mildly delayed range. None of the children had severe delays. Cognitive, language, and psychomotor scores were similar between both groups. Hypotonicity was found in 23%. Autism was suspected in one child who underwent an EXIT procedure and was postnatally diagnosed with mosaic trisomy 18. Conclusion: The majority of children with high-risk fetal lung lesions have age-appropriate neurodevelopmental scores.

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Congenital adenomatoid malformation of the lung: When is active fetal therapy indicated?

Cited by 91 publications

References 10 publications

Thoracoamniotic Shunts: Fetal Treatment of Pleural Effusions and Congenital Cystic Adenomatoid Malformations

Thoracoamniotic Shunts: Fetal Treatment of Pleural Effusions and Congenital Cystic Adenomatoid Malformations

History of Fetal Diagnosis and Therapy: Children’s Hospital of Philadelphia Experience

Early Neurodevelopmental Outcome of Infants with High-Risk Fetal Lung Lesions

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