Neonatal Surgery for Congenital Lung Malformations: Indications, Outcomes and Association With Malignancy

IF 2.4 2区医学 Q1 PEDIATRICS

Journal of pediatric surgery Pub Date : 2025-02-21 DOI:10.1016/j.jpedsurg.2025.162253

Ancuta Muntean , Laura Marsland , Oishi Sikdar , Christopher Harris , Niyi Ade-Ajayi , Shailesh B. Patel , James Cook , Maria Sellars , Anne Greenough , Kypros Nicolaides , Mark Davenport

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引用次数: 0

Abstract

Aim of the study

Congenital lung malformations (CLM) are a heterogeneous group of anomalies diagnosed with increasing frequency antenatally. Postnatal management is dictated by the clinical status in the early neonatal period. We report our centre's experience in managing symptomatic neonates with CLM.

Method

Single centre retrospective review (January 1995–December 2023) of all infants with CLM that underwent thoracotomy for resectional surgery during the neonatal period. Data are quoted as median (IQR), unless stated otherwise. SPSS version 28 (IBM) and GraphPad Prism were used for analysis of data. A P value of ≤0.05 was regarded as significant.

Results

During the study period 228 patients underwent surgery for CLM. Forty-nine infants (21.5 %) (33 males) required surgery during the first 30 days of life. Surgery in those infants was performed at 5 (1–10) days (emergency surgery, n = 24; expedited, n = 25). The indications included respiratory distress (n = 29; mechanical ventilation n = 16) and size of lesion (n = 20). Antenatal diagnosis was established in 45 (91.8 %) infants and 18 had undergone antenatal intervention with 7 fetuses having more than one procedure. The CLM was left sided in 29 (59 %). Postoperative stay for the surviving infants was 14 (7–21) days with extubation being achieved after 2 (1–4) days. The overall survival was 91.8 % (45/49).

Infants that underwent antenatal intervention had a higher rate of postnatal pneumothorax (P = 0.01) and required higher FiO₂ delivery (P = 0.01). Infants that underwent surgery in an emergency setting were found to have a higher need for antenatal intervention (P = 0.01), postnatally required intubation (P < 0.001) with higher FiO₂ delivery (p < 0.001) and developed pneumothorax (P = 0.002).

Histopathology findings showed the entire spectrum of CLM, with 6 cases of CPAM Type 1 associated with mucinous adenocarcinoma. Cross sectional radiological imaging was available for 31 patients and in 4 patients with malignancy, a solid component of the lesion was appreciable on CT. Median follow-up period was 6 (2.1–11) years. Sixteen (35 %) children developed a degree of chest wall deformity through none have required surgical intervention.

Conclusions

This is one of the largest series documenting the postnatal management of symptomatic neonates with CLM. One fifth of the cohort undergoing surgery for CLM required neonatal intervention. Histology at surgery was heterogeneous with 12 % of cases being associated with mucinous adenocarcinoma.

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研究目的先天性肺畸形（CLM）是一类异质性畸形，在产前诊断的频率越来越高。产后管理取决于新生儿早期的临床状况。我们报告了本中心在处理有症状的 CLM 新生儿方面的经验。方法单中心回顾性研究（1995 年 1 月至 2023 年 12 月）所有在新生儿期接受开胸切除手术的 CLM 婴儿。除非另有说明，否则数据均以中位数（IQR）表示。数据分析采用 SPSS 28 版（IBM）和 GraphPad Prism。研究期间，228 名患者接受了 CLM 手术。49名婴儿（21.5%）（33名男性）在出生后30天内需要接受手术。这些婴儿的手术时间为 5 (1-10) 天（急诊手术，24 例；快速手术，25 例）。手术指征包括呼吸窘迫（29 例；机械通气 16 例）和病变大小（20 例）。45名（91.8%）婴儿的产前诊断已经确定，18名接受过产前干预，其中7名胎儿接受过一次以上的产前干预。29例（59%）的CLM为左侧。存活婴儿的术后住院时间为 14 天（7-21 天），2 天（1-4 天）后即可拔管。接受产前干预的婴儿产后气胸发生率更高（P = 0.01），所需的 FiO2 分娩率更高（P = 0.01）。在急诊环境下接受手术的婴儿需要更多的产前干预（P = 0.01），产后需要插管（P < 0.001），FiO2分娩率更高（P < 0.001），并出现气胸（P = 0.002）。组织病理学结果显示了CLM的整个谱系，其中6例CPAM 1型与粘液腺癌有关。有31例患者获得了横断面放射影像学检查结果，其中4例患者为恶性肿瘤，CT检查结果显示病灶中可见实性成分。中位随访时间为 6（2.1-11）年。16名患儿（35%）出现了一定程度的胸壁畸形，但无一例需要手术干预。五分之一因 CLM 而接受手术的患儿需要新生儿干预。手术组织学表现不一，12%的病例伴有粘液腺癌。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Journal of pediatric surgery 医学-外科

CiteScore

1.10

自引率

12.50%

发文量

569

审稿时长

38 days

期刊介绍： The journal presents original contributions as well as a complete international abstracts section and other special departments to provide the most current source of information and references in pediatric surgery. The journal is based on the need to improve the surgical care of infants and children, not only through advances in physiology, pathology and surgical techniques, but also by attention to the unique emotional and physical needs of the young patient.