Combined Pre- and Postnatal Minimally Invasive Approach to a Complex Symptomatic Congenital Pulmonary Airway Malformation
Congenital pulmonary airway malformation (CPAM) is a rare congenital lung lesion that usually remains asymptomatic during the fetal and neonatal period. However, it can occasionally cause prenatal cardiocirculatory failure and fetal hydrops, requiring a thoraco-amniotic shunt (TAS) placement. In oth...
| Main Authors: | , , , , , |
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| Format: | Article |
| Language: | English |
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Georg Thieme Verlag KG
2023-01-01
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| Series: | European Journal of Pediatric Surgery Reports |
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| Online Access: | http://www.thieme-connect.de/DOI/DOI?10.1055/a-2107-0409 |
| _version_ | 1797771767978131456 |
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| author | Macchini Macchini Mazzoleni Mazzoleni Cavallaro Cavallaro Persico Persico Borzani Borzani Leva Leva |
| author_facet | Macchini Macchini Mazzoleni Mazzoleni Cavallaro Cavallaro Persico Persico Borzani Borzani Leva Leva |
| author_sort | Macchini Macchini |
| collection | DOAJ |
| description | Congenital pulmonary airway malformation (CPAM) is a rare congenital lung lesion that usually remains asymptomatic during the fetal and neonatal period. However, it can occasionally cause prenatal cardiocirculatory failure and fetal hydrops, requiring a thoraco-amniotic shunt (TAS) placement. In other cases, it can also cause symptoms at birth (such as respiratory distress) and may require urgent surgical intervention. Thoracoscopic lobectomy for neonates is rarely reported. Here, we report a case of right macrocystic CPAM causing fetal hydrops at 27 weeks of gestation. The fetus was treated with a TAS placement that successfully resolved the hydrops. At 39 weeks of gestation, a male neonate was born (weight 2,850 g). The TAS spontaneously displaced during delivery, causing an open pneumothorax (PNX), initially treated with a drainage. His condition gradually worsened, requiring ventilatory support. Computed tomography (CT) scan showed different giant cysts in the context of the right lower lobe, left mediastinal shift, and compression of the rest of the lung. An urgent surgical management was required. A thoracoscopic right lower lobectomy was performed at 10 days of life (weight 2,840 g). The postoperative course was uneventful; the child remained totally asymptomatic and showed a good recovery. To the best of our knowledge, this is the first reported case of open iatrogenic PNX following TAS positioning and the second of neonatal thoracoscopic lobectomy in a newborn weighting less than 3 kg. The purpose of this report is to indicate that minimally invasive surgery is feasible, safe, and effective for the resection of CPAM, even in small newborns. |
| first_indexed | 2024-03-12T21:41:29Z |
| format | Article |
| id | doaj.art-7f9ba84f4528492197b620b2e6a7e599 |
| institution | Directory Open Access Journal |
| issn | 2194-7619 2194-7627 |
| language | English |
| last_indexed | 2024-03-12T21:41:29Z |
| publishDate | 2023-01-01 |
| publisher | Georg Thieme Verlag KG |
| record_format | Article |
| series | European Journal of Pediatric Surgery Reports |
| spelling | doaj.art-7f9ba84f4528492197b620b2e6a7e5992023-07-26T17:33:25ZengGeorg Thieme Verlag KGEuropean Journal of Pediatric Surgery Reports2194-76192194-76272023-01-011101e36e3910.1055/a-2107-0409Combined Pre- and Postnatal Minimally Invasive Approach to a Complex Symptomatic Congenital Pulmonary Airway MalformationMacchini Macchini0Mazzoleni Mazzoleni1https://orcid.org/0000-0003-0132-8609Cavallaro Cavallaro2https://orcid.org/0000-0002-4921-1437Persico Persico3Borzani Borzani4Leva Leva5Department of Pediatric Surgery, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Lombardia, ItalyDepartment of Pediatric Surgery, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Lombardia, ItalyDepartment of Neonatal Intensive Care Unit, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Lombardia, ItalyDepartment of Obstetrics and Gynecology, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Lombardia, ItalyDepartment of Pediatric Radiology Unit, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Lombardia, ItalyDepartment of Pediatric Surgery, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Lombardia, ItalyCongenital pulmonary airway malformation (CPAM) is a rare congenital lung lesion that usually remains asymptomatic during the fetal and neonatal period. However, it can occasionally cause prenatal cardiocirculatory failure and fetal hydrops, requiring a thoraco-amniotic shunt (TAS) placement. In other cases, it can also cause symptoms at birth (such as respiratory distress) and may require urgent surgical intervention. Thoracoscopic lobectomy for neonates is rarely reported. Here, we report a case of right macrocystic CPAM causing fetal hydrops at 27 weeks of gestation. The fetus was treated with a TAS placement that successfully resolved the hydrops. At 39 weeks of gestation, a male neonate was born (weight 2,850 g). The TAS spontaneously displaced during delivery, causing an open pneumothorax (PNX), initially treated with a drainage. His condition gradually worsened, requiring ventilatory support. Computed tomography (CT) scan showed different giant cysts in the context of the right lower lobe, left mediastinal shift, and compression of the rest of the lung. An urgent surgical management was required. A thoracoscopic right lower lobectomy was performed at 10 days of life (weight 2,840 g). The postoperative course was uneventful; the child remained totally asymptomatic and showed a good recovery. To the best of our knowledge, this is the first reported case of open iatrogenic PNX following TAS positioning and the second of neonatal thoracoscopic lobectomy in a newborn weighting less than 3 kg. The purpose of this report is to indicate that minimally invasive surgery is feasible, safe, and effective for the resection of CPAM, even in small newborns.http://www.thieme-connect.de/DOI/DOI?10.1055/a-2107-0409congenital pulmonary airway malformationthoraco-amniotic shuntthoracoscopic lobectomyneonatecase report |
| spellingShingle | Macchini Macchini Mazzoleni Mazzoleni Cavallaro Cavallaro Persico Persico Borzani Borzani Leva Leva Combined Pre- and Postnatal Minimally Invasive Approach to a Complex Symptomatic Congenital Pulmonary Airway Malformation European Journal of Pediatric Surgery Reports congenital pulmonary airway malformation thoraco-amniotic shunt thoracoscopic lobectomy neonate case report |
| title | Combined Pre- and Postnatal Minimally Invasive Approach to a Complex Symptomatic Congenital Pulmonary Airway Malformation |
| title_full | Combined Pre- and Postnatal Minimally Invasive Approach to a Complex Symptomatic Congenital Pulmonary Airway Malformation |
| title_fullStr | Combined Pre- and Postnatal Minimally Invasive Approach to a Complex Symptomatic Congenital Pulmonary Airway Malformation |
| title_full_unstemmed | Combined Pre- and Postnatal Minimally Invasive Approach to a Complex Symptomatic Congenital Pulmonary Airway Malformation |
| title_short | Combined Pre- and Postnatal Minimally Invasive Approach to a Complex Symptomatic Congenital Pulmonary Airway Malformation |
| title_sort | combined pre and postnatal minimally invasive approach to a complex symptomatic congenital pulmonary airway malformation |
| topic | congenital pulmonary airway malformation thoraco-amniotic shunt thoracoscopic lobectomy neonate case report |
| url | http://www.thieme-connect.de/DOI/DOI?10.1055/a-2107-0409 |
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