Pulmonary sequestration in children: a clinical analysis of 48 cases.

BACKGROUND This study aimed to explore clinical features, diagnosis, treatment, and outcomes of children's pulmonary sequestration (PS) to reduce misdiagnosis. METHODS Clinical records of 48 children with PS in Children's Hospital of Chongqing Medical University between April 1994 and April 2013 were retrieved, and the literature was reviewed. RESULTS 48 cases were collected, 30 cases confirmed (Group A) and 18 suspicious cases (Group B). In Group A, 16 cases were confirmed before operation by 64-row enhanced CT (4 cases), enhanced CT combined with three-dimensional reconstruction (9 cases), and digital subtraction angiography (3 cases). Misdiagnosis rate was 36.7%, while missed diagnosis rate 10%. 26 cases received surgery and were confirmed finally. Aberrant arterial supply mainly originated from thoracic aorta (22 cases) and abdominal aorta (5 cases). Hypoplasia and chronic inflammation were shown by postoperative histopathological examinations in all children with surgery. There was no operative mortality. Encapsulated pleural effusion occurred in one patient as only post-operation complication. All were discharged after successful treatment. CONCLUSION Chest X-ray and color Doppler ultrasound can be used for routine screening for PS. Technique of choice for confirmation is three-dimensional chest CT. Identifying anomalous systemic artery is key for confirmed diagnosis. Surgery is recommended as early as possible. X-ray plus ultrasound as routine screening combined with three-dimensional CT for definitive diagnosis and video-assisted thoracoscopic surgery might be best choice for PS in future.

[1]  M. Pezer,et al.  Sensitization profile in differential diagnosis: Allergic asthma vs. chronic (nonspecific) cough syndrome , 2013, Medical science monitor : international medical journal of experimental and clinical research.

[2]  Yifeng Zhou,et al.  A functional copy number variation in the WWOX gene is associated with lung cancer risk in Chinese. , 2013, Human molecular genetics.

[3]  C. Kang,et al.  Video-assisted thoracoscopic lobectomy in children: safety, efficacy, and risk factors for conversion to thoracotomy. , 2013, The Annals of thoracic surgery.

[4]  Ryan E. Mills,et al.  Copy number variation prevalence in known asthma genes and their impact on asthma susceptibility , 2013, Clinical and experimental allergy : journal of the British Society for Allergy and Clinical Immunology.

[5]  M. Masuda,et al.  Intralobar pulmonary sequestration supplied by an anomalous aneurysmal artery. , 2012, The Annals of thoracic surgery.

[6]  Fan Li,et al.  Pulmonary sequestration: a retrospective analysis of 2625 cases in China. , 2011, European journal of cardio-thoracic surgery : official journal of the European Association for Cardio-thoracic Surgery.

[7]  N. Buchs,et al.  Intralobar pulmonary sequestration with an aberrant artery arising from the celiac trunk. , 2011, European journal of cardio-thoracic surgery : official journal of the European Association for Cardio-thoracic Surgery.

[8]  Heebal Kim,et al.  Genome-wide association study of copy number variations associated with pulmonary function measures in Korea Associated Resource (KARE) cohorts. , 2011, Genomics.

[9]  M. Peycelon,et al.  Thoracoscopy in the management of congenital lung diseases in infancy , 2011, Surgical Endoscopy.

[10]  C. F. Andrade,et al.  [Surgical treatment of congenital lung malformations in pediatric patients]. , 2010, Jornal brasileiro de pneumologia : publicacao oficial da Sociedade Brasileira de Pneumologia e Tisilogia.

[11]  K. Weng,et al.  Early and late outcomes of coil embolization of pulmonary sequestration in children. , 2009, Circulation journal : official journal of the Japanese Circulation Society.

[12]  Shih-Huang Lee,et al.  Angiographic and clinical manifestations of coronary fistulas in Chinese people: 15-year experience. , 2008, Circulation journal : official journal of the Japanese Circulation Society.

[13]  K. Sung,et al.  Neonatal pulmonary sequestration: Clinical experience with transumbilical arterial embolization , 2008, Pediatric pulmonology.

[14]  D. Tibboel,et al.  Congenital lung malformations , 2008 .

[15]  C. Barnewolt,et al.  Bronchial Atresia is Common to Extralobar Sequestration, Intralobar Sequestration, Congenital Cystic Adenomatoid Malformation, and Lobar Emphysema , 2006, Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society.

[16]  A. Gözübüyük,et al.  Pulmonary Sequestration and Surgical Treatment , 2006, Asian cardiovascular & thoracic annals.

[17]  V. De Maertelaer,et al.  Should determination of the karyotype be systematic for all malformations detected by obstetrical ultrasound? , 2005, Prenatal diagnosis.

[18]  K. Gow,et al.  Video-assisted thoracoscopic surgical excision of cystic lung disease in children. , 2005, Journal of pediatric surgery.

[19]  R. Hammad,et al.  Lower lobe segments and pulmonary sequestrations , 2004 .

[20]  E. Crushell,et al.  Occlusion of an aberrant artery to a pulmonary sequestration using a duct occluder. , 2002, Journal of interventional cardiology.

[21]  J. Laberge,et al.  The multiple facets of pulmonary sequestration. , 2001, Journal of pediatric surgery.

[22]  K. Blakemore,et al.  Non‐mosaic trisomy 20 presenting at 21 weeks' gestation as a thoraco‐abdominal mass , 2001, Prenatal diagnosis.

[23]  P. Moerman,et al.  Pulmonary sequestration: a comparison between pediatric and adult patients. , 2001, European journal of cardio-thoracic surgery : official journal of the European Association for Cardio-thoracic Surgery.

[24]  F. Boyvat,et al.  Coil embolization of pulmonary sequestration in two infants: a safe alternative to surgery. , 2000, AJR. American journal of roentgenology.

[25]  R. Aliaga,et al.  Commentary , 1998, Thorax.

[26]  K. Kontani,et al.  [A case report of anomalous systemic arterial supply to the left basal lung]. , 1997, [Zasshi] [Journal]. Nihon Kyobu Geka Gakkai.

[27]  D. Mulder,et al.  Pulmonary sequestration: 17-year experience at UCLA. , 1993, The American surgeon.

[28]  J. Kim,et al.  Coexistent intralobar and extralobar pulmonary sequestration: imaging findings. , 1993, AJR. American journal of roentgenology.

[29]  I. Tonkin,et al.  Imaging of pulmonary sequestration. , 1990, AJR. American journal of roentgenology.

[30]  H. Hoefler,et al.  Bronchial carcinoid arising in intralobar bronchopulmonary sequestration with vascular supply from the left gastric artery. Case report. , 1985, The Journal of thoracic and cardiovascular surgery.

[31]  H. Funke,et al.  Lung sequestration: report of seven cases and review of 540 published cases , 1979, Thorax.

[32]  K. Heithoff,et al.  Bronchopulmonary foregut malformations. A unifying etiological concept. , 1976, AJR. American journal of roentgenology.

[33]  D. Pryce.  Lower accessory pulmonary artery with intralobar sequestration of lung; a report of seven cases. , 1946, The Journal of pathology and bacteriology.