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Category: Volume 11 Issue 04 April 2023
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Title: Clinical-Diagnostic and Morphopathological Correlations in Congenital Pulmonary Malformation of the Respiratory Tract Type I in Newborn with Progressive Respiratory Distress

Clinical Case Presentation and Literature Review

Authors: Babuci S., Petrovici V., Revenco I., Haidarlî D., Maniuc A.

 DOI: https://dx.doi.org/10.18535/jmscr/v11i4.01

Abstract

Summary

The author’s reported experiencing a case of progressive acute respiratory distress caused by MCCA type I in a newborn with successful surgical resection. The presented case reflects characteristic clinical symptomatology, the preoperative image of diagnosis being of vital importance in the management of the malformation.

The histological examination made it possible to establish some characteristic structural aspects for MPCCR type 1 which presents as a vicious bronchopulmonary abnormality, which, simultaneously with cystic lung lesions, are present signs of immaturity and hypoplasia, cystic bronchiectasis that mimic perifocal adenomatoid aspects associated with primary atelectasis, focal distelectasis concurrent with arterial hypertrophic-stenotic angiopathy.

The authors conclude that early surgical intervention with economical resection of the congenital cystic formation and preservation of the remaining adjacent lung parenchyma in cases of acute respiratory distress in the neonate with adenomatoid cystic malformation type 1 can be considered a potentially effective option, which can be performed in safe conditions with acceptable results.

Congenital cystic malformations of the lungs in children are serious lesions with similar embryological and clinical features that result from compromised interaction between mesodermal and ectodermal lung elements during embryological development. Included in this group were: bronchogenic cysts, pulmonary sequestration, congenital cystic adenomatoid malformation, and congenital lobar emphysema[30,46]. Currently, the terminology is being revised, the spectrum of these congenital lesions includes: bronchogenic cysts, congenital airway malformation, and peripheral cystic alveolar growth anomalies. The differential diagnosis of pleuropulmonary blastoma, which is a malignant neoplasm characteristic of the child's age, should also be considered[21].

References

  1. Aziz D., Langer J.C., Tuuha S.E., Ryan G., Ein S.H., Kim P.C. Perinatally diagnosed symptomatic congenital cystic adenomatoid malformation: to resect or not? J. Pediatr. Surg. 2004;39:329-34.
  2. Bain G.O. Congenital adenomatoid malformation of the lung. Chest J. 1959; 36(4):430-3.
  3. Belanger R., La Fleche, Picard J.L. Congenital cystic adenomatoid malformation of the lung. Thorax. 1964; 19:1-11.
  4. Brcic L.,Fakler F., Eidenhammer S., Thueringer A., Kashofer K. et al. Pleuropulmonaryblastoma type I might arise in congenital pulmonary airway malformation type 4 by acquiring a Dicer 1 mutation. Virchows Arch. 2020; doi: 10.1007/s00428-020-02789-6.
  5. Casagrande A., Pederiva F. Association between congenital lung malformations and lung tumors in children and adults: A systematic review. J. Thor. Oncol. 2016; 11(11):1837-45.
  6. Chen H.W., Hsu W.M., Lu F.L., Chen P.C., Jeng S.F., et al. Management of congenital adenomatoid malformation and bronchopulmonary sequestration in newborns. Pediatr. Neonatol. 2020; 51(3):172-7.
  7. Conforti A., Aloi I., Trucchi A., Morini F., Nahom A. et al. Asymptomatic congenital cystic adenomatoid malformation of the lung: Is it time to operate? J. Thor. Cardiovasc. Surg. 2009; 138(4):826-30.
  8. David M., Lamas-Pinheiro R., Henriques-Coelho T. Prenatal and postnatal management of congenital pulmonary airway malformation. Neonatol. 2016; 110:101-15.
  9. De Leon-Urena Z.A., Sadowinski-Pine S., Jamaica-Balderas L., Penchyna-Grub J. Pulmonary sequestration associated with congenital pulmonary airway malformation. Med. Hosp. Infant Mex. 2018;75:105-12.
  10. Doladzas T., Arvelakis A., Karavokyros I.G., Gougoudi E., Pikoulis E., Patsouris E. Primary rhabdomyosarcoma of the lung arising over cystic pulmonary adenomatoid malformation. Pediatr. Hematol. Oncol. 2005; 22(6):525-9.
  11. Durell J.,Lakhoo K. Congenital cystic lesions of the lung. Early Human Develop. 2014; 90: 935-39.
  12. El Amraoui W.,Bentalha A., Hamri H., El Kettani S.E., El Koraichi A. Congenital cystic adenomatoid malformation – dangers of misdiagnosis: a case report. J. med. Case Rep. 2017; 11:212. DOI 10.1186/s13256-017-1349-5.
  13. Fan D., Wu S., Wang R., Huang Y., Fu Y. et al. Successfully treated congenital cystic adenomatoid malformation by open fetal suegery. Medicine. 2017; 96:2.
  14. Feng A., Cai H., Sun Q., Zhang Y., Chen L., Meng F. Congenital cystic adenomatoid malformation of lung in adults: 2 rare cases report and review of the literature. Diagn. Pathol. 2012; 7:37. Doi:10.1186/1746-1596-7-37.
  15. Gallegos S.A., Sibaja M.J.R., Gaona J.M.G., Calvo J.A.R., Torrez B.V., et al. Congenital adenomatoid cystic malformation: expectant management outcomes. J. Preg. Child Health. 2018; 5: 387. doi:4172/2376-127X.1000387.
  16. Goyal J.P., Jindal S., Mishra M., Bhakhiri B.K. Congenital cyst adenoid malformation masquerading as bronchial asthma. Int. J. App. Basic Med. Res. 2020; 7:199-201.
  17. Holder T.M., Christy M.G. Cystic adenomatoid malformation of the lung. J. Thor. Cardiovasc. Surg. 1964; 47(5):590-7.
  18. Ishida M., Igarashi T., Teramoto T., Hanaoka J., Iwai M. et al. Mucinous bronchioloalveolar carcinoma with K-ras mutation arising in type 1 congenital cystic adenomatoid malformation: a case report with review of the literature. Int. J. Clin. Exp. Pathol. 2013; 6(11):2597-2602.
  19. Jain A.,Anand K., Singla S., Kumar A. Congenital Cystic Lung Diseases. J. Clin. Imaging Sci. 2013;3:5. http://www.clinicalimagingscience.org/text.asp?2013/3/1/5/106620.
  20. Jiang Y., Luo Y., Tang Y., Moats R., Warburton D. et al. Alteration of cystic airway mesenchyme in congenital pulmonary airway malformation. Scientific Rep. 2019; 9:5296. https://doi.org/10.1038/s41598-019-41777-y.
  21. Jones K.D. Pulmonary cystic disease and its mimics. Surg. Pathol. 2020; 13:141-63.
  22. Kanavi J.V., Thangaraj J., Thomas A. Congenital cystic adenomatoid malformation (CCAM): antenatal and postnatal management. Int. J. reprod. Contracept. Obstet. Gynecol. 2017; 6(6):2447-51.
  23. Kim M.Y., Kang C.H., Park S.H. Multifocal synchronous mucinous adenocarcinomas arising in congenital pulmonary airway malformation: a case report with molecular study. Histopathology. 2014;65: 926-32.
  24. Kim Y.T., Kim J.S., Park J.D., Kang C.H., Sung S.W., Kim J.H. Treatment of congenital cystic adenomatoid malformation – does resection in the early postnatal period increase surgical risk? J. Cardio-thor. Surg. 2005; 27:658-61.
  25. Koh J., Jung E., Jang S.J., Kim D.K., Lee B.S. et al. Case of mucinous adenocarcinoma of the lung associated with congenital pulmonary airway malformation in a neonate. Korean J. Pediatr. 2018; 61(1):30-4/
  26. Komori K.,Kamagata S., Hirobe S., Toma M., Okumura K et al. Radionuclide imaging study of long-term pulmonary function after lobectomy in children with congenital cystic lung disease. J.Pediatr. Surg. 2009;44:2096-2100.
  27. Kotecha S.,Barbato A., Bush A., Claus F., Davenport M. et al. Antenatal and postnatal management of congenital cystic adenomatoid malformation. Paediatr. Respir. Rev. 2012;13:162-71.
  28. Kwittken J., Reiner L. Congenital cystic adenomatoid malformation of the lung. Pediatrics. 1962; 30:759-68.
  29. LakhooManagement of congenital cystic adenomatousmalformations of the lung. Arch Dis. Child Fetal Neonatal Ed. 2009;94:F73-F76. doi:10.1136/adc.2007.130542.
  30. Lima M., Gargano T., Ruggeri G., Manuele R., Gentili A. et al. Clinical spectrum and management of congenital pulmonary cystic lesions. Pediatr. Chir. 2008; 30(2):79-88.
  31. Mathai A.M.,Kini H., Pai M.R., Shetty A.B., Murali N. Congenital cystic adenomatoid malformation of lung type 1. J. Pediatr. Surg. 2010; 45:E25-E28.
  32. Moncriefe M.W., Cameron A.H., Astley R., Roberts K.D., Abrams L.D., Mann J.R. Congenital cystic adenomatoid malformation of the lung. Thorax. 1969; 24:476-87.
  33. Morini F., Zani A., Conforti A., van Heurn E., Eaton S. et al. Current management of congenital pulmonary airway malformations: A “European Pediatric Surgeon s Association” Survey. Eur. J. Pediatr. Surg. 2018; 28(1):1-5.
  34. Oh M.H., Jung E.A., Lee J.H., Cho H.D., Seo K.H., et al. Coexistence of intrapulmonary bronchogenic cyst and congenital cystic adenomatoid malformation – A case report. Korean J. Pathol. 2011; 45:92-5.
  35. Pardo L.,Viveros J.M., Carrillo A., Polo A., Lopez A. et al. Radiological manifestations of congenital lung malformations. Experience of three hospitals in Bogota. Rev. Colomb. Radiol. 2019; 30(2):5117-25.
  36. Pogoriler J., Swarr D., Kreiger P., Adzick N.S., Peranteau W. Congenital cystic lesions. Redefining the natural distribution of subtypes and assessing the risk of malignancy. Am. J. Surg. Pathol. 2019; 43(1):47-55.
  37. Raman V.S.,Agarwala S., Bhatnagar V., Panda S.S., Gupta A.K. Congenital cystic lesions of the lungs: The perils of misdiagnosis - A single-center experience. Lung India. 2015; 32:116-8.
  38. Ramos S.G., Barbosa G.H., Tavora F.R., Jeudy J., Torres L.A., et al. Bronchioloalveolar carcinoma arising in a congenital pulmonary airway malformation in a child: case report with an update of this association. J.Pediatr. Surg. 2007;42:E1-4.
  39. Scampolino A., Ambrosini A., Turini E., Morandi U., Stefani A. Congenital cystic adenomatoid malformation in adults, presenting as a single cyst. Asian Cardiovasc. Thorac. Ann. 2018; 26(5):407-9.
  40. Sfakianaki A.K., Copel J.A. Congenital cystic lesions of the lung: congenital cystic adenomatoid malformation and bronchopulmonary sequestration. Rev.Obstet. 2012;5(2):85-93.
  41. Spector R.G., Claireaux A.E., Williams E.R. Congenital adenomatoid malformation of lung with pneumothorax. Arch. Dis. Child. 1960; 35(183):475-80.
  42. Stocker J.T. Congenital pulmonary airway malformation: a new name and expanded classification of congenital cystic adenomatoid malformation of the lung. Histopathology. 2002; 41(Suppl):424-
  43. Stocker J.T.,Madewell J.E., Drake R.M. Congenital cystic adenomatoid malformation of the lung: classification and morphologic spectrum. Hum. Pathol. 1977; 8:155-71.
  44. Style C.C., Cass Darell L., Verla M.A., Cruz S.M., Lau P.E. et al. Early vs late resection of asymptomatic congenital lung malformation. J. Pediatr. Surg. 2019; 54(1):70-4.
  45. Sudou M.,Sugi K., Murakami T. Bronchioloalveolar carcinoma arising from a congenital cystic adenomatoid malformation in an adolescent: The first case report from the Orient. J. Thorac. Cardiovasc. Surg. 2003; 126:902-3.
  46. Takeda S., Miyoshi S., Inoue M., Omori K., Okumura M. et al. Clinical spectrum of congenital cystic disease of the lung in children. Eur. J. Cardio-thor. Surg. 1999; 15:11-7.
  47. Vladareanu R., Pelinescu-Onciul D., Zvanca M., Mihailescu D. Congenital cystic adenomatoid malformation. Perinatol. 2007;16(2):68-72.
  48. Yoo J., Lee S.M., Jung J.H., Ahn M.I., Cho D.G. et al. Adenocarcinoma arising in type 1 congenital cystic adenomatoid malformation: A case report and review of the literature. Korean J. Pathol. 2008; 42:396-400.

Corresponding Author

Babuci S.

IMSP Mother and Child Institute

"Natalia Gheorghiu" National Scientific-Practical Center for Pediatric Surgery

"Nicolae Testemițanu" State University of Medicine and Pharmacy