Macchini Francesco, Mazzoleni Stefano, Cavallaro Giacomo, Persico Nicola, Borzani Irene, Leva Ernesto
Department of Pediatric Surgery, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Lombardia, Italy.
Department of Neonatal Intensive Care Unit, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Lombardia, Italy.
European J Pediatr Surg Rep. 2023 Jul 17;11(1):e36-e39. doi: 10.1055/a-2107-0409. eCollection 2023 Jan.
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.
先天性肺气道畸形（CPAM）是一种罕见的先天性肺部病变，在胎儿期和新生儿期通常无症状。然而，它偶尔会导致产前心循环衰竭和胎儿水肿，需要放置胸羊膜分流管（TAS）。在其他情况下，它也可能在出生时引起症状（如呼吸窘迫），可能需要紧急手术干预。关于新生儿胸腔镜肺叶切除术的报道很少。在此，我们报告一例妊娠27周时因右大囊型CPAM导致胎儿水肿的病例。胎儿接受了TAS放置治疗，成功解决了水肿问题。妊娠39周时，一名男婴出生（体重2850克）。TAS在分娩过程中自行移位，导致开放性气胸（PNX），最初进行了引流治疗。他的病情逐渐恶化，需要通气支持。计算机断层扫描（CT）显示右下叶有不同的巨大囊肿，左纵隔移位，其余肺组织受压。需要进行紧急手术处理。在出生后10天（体重2840克）进行了胸腔镜右下叶切除术。术后过程顺利；患儿完全无症状，恢复良好。据我们所知，这是首例报道的TAS定位后发生的医源性开放性PNX病例，也是第二例体重小于3千克新生儿的胸腔镜肺叶切除术病例。本报告的目的是表明，即使对于小新生儿，微创手术对于CPAM的切除也是可行、安全且有效的。
