Clinical and Experimental Pediatrics

The Korean Pediatric Society (대한소아청소년과학회)
권호 표지

A Case of Pulmonary Sequestration with Pleural Effusion

산전 진찰에서 발견된 흉막액을 동반한 폐격리증 1례

  • Ku, Hye Jin (Department of Pediatrics, Pusan National University College of Medicine) ;
  • Park, Ji Ae (Department of Pediatrics, Pusan National University College of Medicine) ;
  • Park, Su Eun (Department of Pediatrics, Pusan National University College of Medicine) ;
  • Kim, Young Dae (Department of Cardiovascular Surgery, Pusan National University College of Medicine)
  • 구혜진 (부산대학교 의과대학 소아과학교실) ;
  • 박지애 (부산대학교 의과대학 소아과학교실) ;
  • 박수은 (부산대학교 의과대학 소아과학교실) ;
  • 김영대 (부산대학교 의과대학 흉부외과학교실)
  • Received : 2004.09.06
  • Accepted : 2004.10.11
  • Published : 2005.02.15
Download PDF
⟨ Previous Next ⟩

Abstract

Congenital pleural effusions are uncommon. The majority of cases are due to chylothorax, hydrops fetalis, and infection. Effusions of this nature are, for the most part, self-limited. We experienced a rare case of a congenital unilateral pleural effusion due to an extralobar sequestraion with pulmonary lymphangiectasia. Pleural effusion was found by antenatal ultrasonography and confirmed by CT scans and CT angiography of the chest in the neonatal period. The patient underwent an open thoracotomy where extralobar sequestraion located between the diaphragm and the left lower lobe was removed. His postoperative course was uncomplicated and there was complete resolution of the pleural effusion.

선천성 흉막액을 동반한 외엽형 폐격리증은 매우 드문 선천성 폐기형으로 아직까지 국내에서의 보고는 없다. 저자들은 재태 기간 27주에 시행한 산전 초음파상 좌측 흉막액이 발견되었고, 출생 후 시행한 흉부 전산화 단층 촬영 및 전산화 단층 혈관 촬영술상 선천성 흉막액을 동반한 외엽형 폐격리증으로 진단하였으며, 생후 80일경 수술 후 특별한 문제없이 퇴원하여 현재 외래 추적 관찰 중인 1례를 경험하였기에 문헌 고찰과 함께 보고하는 바이다.

Keywords

References

  1. Horowitz RN. Extralobar sequestation of lung in newborn infant. Am J Dis Child 1965;110:195-8
  2. Vade A, Kramer L. Extralobar pulmonary sequestration presenting as intractable pleural effusion. Pediatr Radiol 1989;19:333-4 https://doi.org/10.1007/BF02467308
  3. Lucaya J, Garcia-Conesa JA, Bernado L. Pulmonary sequestration associated with unilateral pulmonary hypoplasia and massive pleural effusion. Pediatr Radiol 1984;14:228-9. https://doi.org/10.1007/BF01042247
  4. Bliek AJ, Mulholland DJ. Extralobar lung sequestration associated with fatal neonatal respiratory distress. Thorax 1971;26:125-30 https://doi.org/10.1136/thx.26.1.125
  5. Dresler S. Massive pleural effusion and hypoplasia of the lung accompanying extralobar pulmonary sequestration. Hum Pathol 1981;12:862-4 https://doi.org/10.1016/S0046-8177(81)80094-9
  6. Boyer J, Dozor A, Brudnicki A, Slim M, Paliotta M, Kwark HE. Extralobar pulmonary sequestration masquerading as a congenital pleural effusion. Pediatrics 1996;97: 115-7
  7. Pryce DM. Lower accessory pulmonary artery with intralobar sequestration of lung; A report of seven cases. J Pathol 1946;58:457 https://doi.org/10.1002/path.1700580316
  8. Lager DJ, Kuper KA, Haake GK. Subdiaphragmatic extralobar pulmonary sequestration. Arch Pathol Lab Med 1991; 115:536-8
  9. Weinbaum PJ, Bors-Koefoed R, Green KW, Prenatt L. Antenatal sonographic findings in a case of intra-abdominal pulmonary sequestration. Obstet Gynecol 1989;73:860-2
  10. Savic B, Birtel FJ, Tholen W, Funke HD, Knoche R. Lung sequestration : report of seven cases and review of 540 published cases. Thorax 1979;34:96-101 https://doi.org/10.1136/thx.34.1.96
  11. Collin PP, Desjardins JG, Khan AH. Pulmonary sequestration. J Pediatr Surg 1987;22:750-3 https://doi.org/10.1016/S0022-3468(87)80620-6
  12. White J, Chan YF, Neuberger S, Wilson T. Prenatal sonographic detection of intra-abdominal extralobar pulmonary sequestration : report of three cases and literature review. Prenat Diagn 1994;14:653-8 https://doi.org/10.1002/pd.1970140802
  13. Black MD, Bass J, Martin DJ, Carpenter BF. Intraabdominal pulmonary sequestration. J Pediatr Surg 1991;26: 1381-3 https://doi.org/10.1016/0022-3468(91)91038-Z
  14. Levine MM, Nudel DB, Gootman N, Wolpowitz A, Wisoff BG. Pulmonary sequestration causing congestive heart failure in infancy : a report of two cases and review of the literature. Ann Thorac Surg 1982;34:581-5 https://doi.org/10.1016/S0003-4975(10)63007-X
  15. Louie HW, Martin SM, Mulder DG. Pulmonary sequestration: 7-year experience at UCLA. Am Surg 1993;59:801-5
  16. Barret J, Chitayat D, Sermer M, Amankwah K, Morrow R, Toi A, et al. The prognostic factors in the prenatal diagnosis of the echogenic fetal lung. Prenat Diagn 1995;15:849-53 https://doi.org/10.1002/pd.1970150910