Prenatal ve Postnatal Dönemde Tanı Alan Konjenital Akciğer Malformasyonu Olan Çocukların Değerlendirilmesi

Year 2022, Volume: 16 Issue: 4, 336 - 342, 07.07.2022

Tuğba Ramaslı Gürsoy Tuğba Şişmanlar Eyüboğlu , Ayşe Tana Aslan , Ramazan Karabulut , Abdullah Taştepe

https://doi.org/10.12956/tchd.1034487

Abstract

Amaç: Çalışmamızda prenatal ve postnatal dönemde tanı alan konjenital akciğer malformasyonu olan hastaların klinik özelliklerini
karşılaştırmayı amaçladık.

Gereç ve Yöntemler: 2007-2021 yılları arasında Çocuk Göğüs Hastalıkları Bölümü’nde izlenen konjenital akciğer malformasyonu olan
hastalar cinsiyetleri, yaşları, başvuru yakınmaları, ilk semptom zamanı, tanı yaşları, tanı yöntemleri, doğum haftaları, doğum ağırlıkları,
ebeveyn akrabalığı, operasyon varlığı, operasyon yaşı ve endikasyonları, uzun dönem komplikasyonları açısından değerlendirildi. Prenatal
dönemde tanı alan hastalar ile postnatal dönemde tanı alanlar klinik özellikleri açısından karşılaştırıldı.

Bulgular: Konjenital akciğer malformasyonu nedeniyle izlenen 37 hastanın ortalama yaşları 6.7±5.8 yıldı ve 17’si (%45.9) kızdı. Prenatal
dönemde tanı alan hastalar (n:18) yakınması olmadan başvururken diğer hastaların en sık başvuru nedenleri öksürük ve tekrarlayan akciğer
enfeksiyonuydu. Postnatal dönemde tanı alan hastaların (n:19) ortanca tanı yaşları 30 (en küçük:10; en büyük:1080) gündü. Hastaların
18’i (%48.6) prenatal ultrasonografi, 14’ü (%37.8) bilgisayarlı tomografi, beşi (%13.6) akciğer grafisi ile tanı aldı. İzlemde iki hastanın
malformasyonu kendiliğinden geriledi. Oniki hasta opere olurken diğer hastalar mevcut anomalileri ile takip edilmektedir. Prenatal dönemde
tanı alan hastaların asemptomatik izlem süresi, postnatal tanı alan hastalardan istatistiksel olarak anlamlı farklıydı (36.5±4.7 ve 24.0±12.7
ay) (p:0.004).

Sonuç: Çalışmamızda prenatal dönemde tanı alan konjenital akciğer malformasyonu olan hastaların daha uzun süre

Keywords

Çocuk, konjenital akciğer malformasyonu, prenatal tanı

Evaluation of Children with Congenital Lung Malformations Who Were Diagnosed in The Prenatal and Postnatal Period

Abstract

Objective: We aimed to compare clinical features of children with congenital lung malformations (CLM) who were
diagnosed in prenatal and postnatal period.

Material and Methods: Children with CLM followed in our pediatric pulmonology department between 2007-2021
were evaluated in terms of sex, age, complaints at presentation, time of onset of symptoms, age at diagnosis, diagnostic
methods, gestational ages, birth weights, parental consanguinity, presence of any operations, age and indications of
operations and long-term complications. Children who were diagnosed in prenatal period and those in postnatal period
were compared in terms of their clinical features.

Results: The mean age of 37 children with CLM was 6.7±5.8 years, and seventeen (45.9%) of the children were girls.
Children who were diagnosed during the prenatal period (n:18) had no complaints, whereas cough and recurrent
pneumonia were the most common reasons at admission in others. Median age at diagnosis of children who
were postnatally diagnosed (n:19) was 30 days (10-1080). Eighteen (48.6%) children were diagnosed by prenatal
ultrasonography, 14 (37.8%) by computed tomography, and five (13.6%) by chest X-ray. During follow-up, malformations
of two children regressed spontaneously. Twelve children were operated while others were followed up with their
anomalies. Asymptomatic follow-up duration of children who were prenatally diagnosed was significantly different than
the children who were diagnosed in the postnatal period (36.5±4.7 vs 24.0±12.7 months) (p:0.004).

Conclusion: Children with CLM who were diagnosed in the prenatal period were found to remain asymptomatic for
longer. Prenatal diagnosis enables them to live longer without symptoms with appropriate surgical timing.

Keywords

Child, congenital lung malformations, prenatal diagnosis

References

• Leblanc C, Baron M, Desselas E, Phan MH, Rybak A, Thouvenin G, et al. Congenital pulmonary airway malformations: state-of-the-art review for pediatrician's use. Eur J Pediatr 2017;176(12):1559-1571.
• Stocker LJ, Wellesley DG, Stanton MP, Parasuraman R, Howe DT. The increasing incidence of foetal echogenic congenital lung malformations: an observational study. Prenat Diagn. 2015;35(2):148-153.
• Thacker PG, Rao AG, Hill JG, Lee EY. Congenital lung anomalies in children and adults: current concepts and imaging findings. Radiol Clin N Am. 2014;52(1):155-81.
• Bush A. Congenital lung disease: a plea for clear thinking and clear nomenclature. Pediatr Pulmonol. 2001;32(4):328-337.
• Baez JC, Ciet P, Mulkern R, Seethamraju RT, Lee EY. Pediatric Chest MR Imaging: Lung and Airways. Magn Reson Imaging Clin N Am. 2015;23(2):337-349.
• Alamo L, Gudinchet F, Reinberg O, Vial Y, Francini K, Osterheld MC, et al. Prenatal diagnosis of congenital lung malformations. Pediatr Radiol. 2012;42(3):273-283.
• Thompson AJ, Sidebotham EL, Chetcuti PA, Crabbe DC. Prenatally diagnosed congenital lung malformations—A long-term outcome study. Pediatric Pulmonology. 2018;1–5.
• Mon RA, Johnson KN, Ladino-Torres M, Heider A, Mychaliska GB, Treadwell MC, et al. Diagnostic accuracy of imaging studies in congenital lung malformations. Arch Dis Child Fetal Neonatal Ed. 2019;104(4):F372-F377.
• Graham BL, Steenbruggen I, Miller MR, Barjaktarevic IZ, Cooper BG, Hall GL, et al. Standardization of Spirometry 2019 Update. An Official American Thoracic Society and European Respiratory Society Technical Statement. Am J Respir Crit Care Med. 2019;200(8):e70-e88.
• WHO Multicentre Growth Reference Study Group: WHO Child Growth Standards: Length/heightfor-age, weight-for-age, weight-for-length, weight-for-height and body mass index-for-age: Methods and development. Geneva, World Health Organization, 2006. http://www.who.int/childgrowth/standards/technical_report/en/index.html. Ulaşım tarihi 20 Temmuz 2021.
• Torres A, Cilloniz C, Niederman MS, Menéndez R, Chalmers JD, Wunderink RG, et al. Pneumonia. Nat Rev Dis Primers. 2021;7(1):25.
• Montella S, Corcione A, Santamaria F. Recurrent Pneumonia in Children: A Reasoned Diagnostic Approach and a Single Centre Experience. Int J Mol Sci. 2017;18(2):296.
• Fred Mo, ME Cunningham. Pediatric scoliosis. Curr Rev Musculoskelet Med (2011) 4:175–182.
• Zeeshan S, Hussain SNF, Mughal Z, Anwar SSM, Naeem SN. Bifurcated rib with vertebral defects - A rare anatomical variant: Case report with literature review. Int J Surg Case Rep. 2020;67:203-206.
• Blanco FC, Elliott ST, Sandler AD. Management of congenital chest wall deformities. Semin Plast Surg. 2011;25(1):107-116.
• David M, Lamas-Pinheiro R, Henriques-Coelho T. Prenatal and Postnatal Management of Congenital Pulmonary Airway Malformation. Neonatology. 2016;110(2):101-115.
• Shanti CM, Klein MD. Cystic lung disease. Semin Pediatr Surg. 2008;17(1):2-8.
• Chakraborty RK, Modi P, Sharma S. Pulmonary Sequestration. In: StatPearls. Treasure Island (FL): StatPearls Publishing; July 26, 2021.
• Limaiem F, Mlika M. Bronchogenic Cyst. In: StatPearls. Treasure Island (FL): StatPearls Publishing; July 20, 2021.
• Stanton M, Njere I, Ade-Ajayi N, Patel S, Davenport M. Systematic review and meta-analysis of the postnatal management of congenital cystic lung lesions. J Pediatr Surg. 2009;44(5):1027–1033.
• Kuroda T, Nishijima E, Maeda K, Fuchimoto Y, Hirobe S, Tazuke Y, et al. Clinical Features of Congenital Cystic Lung Diseases: A Report on a Nationwide Multicenter Study in Japan. Eur J Pediatr Surg. 2016;26(1):91-95.
• Thompson A, Morley H, Chetcuti PA, Crabbe DC. Congenital lung malformations: the case for conservative management. British Association of Paediatric Surgeons Annual Congress, Edinburgh, 2014.
• Marshall KW, Blane CE, Teitelbaum DH, van Leeuwen K. Congenital cystic adenomatoid malformation: impact of prenatal diagnosis and changing strategies in the treatment of the asymptomatic patient. AJR Am J Roentgenol. 2000;175(6):1551-1554.
• Wong KKY, Flake AW, Tibboel D, Rottier RJ, Tam PKH. Congenital pulmonary airway malformation: advances and controversies. Lancet Child Adolesc Health. 2018;2(4):290‐297.
• Hijkoop A, van Schoonhoven MM, van Rosmalen J, Tibboel D, van der Cammen-van Zijp MHM, Pijnenburg MW, et al. Lung function, exercise tolerance, and physical growth of children with congenital lung malformations at 8 years of age. Pediatr Pulmonol. 2019;54(8):1326-1334.
• Annunziata F, Bush A, Borgia F, Raimondi F, Montella S, Poeta M, et al. Congenital Lung Malformations: Unresolved Issues and Unanswered Questions. Front Pediatr. 2019;7:239.
• Nakajima C, Kijimoto C, Yokoyama Y, Miyakawa T, Tsuchiya Y, Kuroda T, et al. Longitudinal follow-up of pulmonary function after lobectomy in childhood—factors affecting lung growth. Pediatr Surg Int. 1998;13(5-6):341–345.
• Keijzer R, Chiu PP, Ratjen F, Langer JC. Pulmonary function after early vs late lobectomy during childhood: a preliminary study. J Pediatr Surg. 2009;44(5): 893–895.
• Naito Y, Beres A, Lapidus-Krol E, Ratjen F, Langer JC. Does earlier lobectomy result in better long-term pulmonary function in children with congenital lung anomalies? A prospective study. J Pediatr Surg. 2012;47(5):852–856.
• Babu R, Kyle P, Spicer RD. Prenatal sonographic features of congenital lobar emphysema. Fetal Diagn Ther 2001; 16:200.
• Liu YP, Shih SL. Congenital lobar emphysema: appearance on fetal MRI. Pediatr Radiol 2008; 38:1264.
• Quinton AE, Smoleniec JS. Congenital lobar emphysema--the disappearing chest mass: prenatal ultrasound appearance. Ultrasound Obstet Gynecol 2001; 17:169.
• Cook J, Chitty LS, De Coppi P, Ashworth M, Wallis C. The natural history of prenatally diagnosed congenital cystic lung lesions: long-term follow-up of 119 cases. Arch Dis Child. 2017;102(9):798-803.
• Vu LT, Farmer DL, Nobuhara KK, Miniati D, Lee H. Thoracoscopic versus open resection for congenital cystic adenomatoid malformations of the lung. J Pediatr Surg. 2008;43(1):35–39.
• Makita S, Kaneko K, Ono Y, Uchida H. Risk factors for thoracic and spinal deformities following lung resection in neonates, infants, and children. Surg Today. 2017;47(7):810-814.