Shaun M Kunisaki1, Dave R Lal2, Jacqueline M Saito3, Mary E Fallat4, Shawn D St Peter5, Zachary D Fox6, Amer Heider6, Sherwin S Chan5, Kevin P Boyd2, R Cartland Burns7, Katherine J Deans8, Samir K Gadepalli6, Ronald B Hirschl6, Rashmi Kabre9, Matthew P Landman7, Charles M Leys10, Grace Z Mak11, Peter C Minneci8, Tiffany N Wright4, Michael A Helmrath12. 1. Division of General Pediatric Surgery, Department of Surgery, Johns Hopkins University School of Medicine, Johns Hopkins Children's Center, Baltimore, Maryland; skunisa1@jhmi.edu. 2. Children's Hospital of Wisconsin, Medical College of Wisconsin, Milwaukee, Wisconsin. 3. Division of Pediatric Surgery, Department of Surgery, Washington University School of Medicine, St Louis, Missouri. 4. Division of Pediatric Surgery, Department of Surgery, University of Louisville, Louisville, Kentucky. 5. Department of Surgery, Children's Mercy Hospital, University of Kansas School of Medicine, Kansas City, Missouri. 6. Section of Pediatric Surgery, University of Michigan and Michigan Medicine, C.S. Mott Children's Hospital, Ann Arbor, Michigan. 7. Division of Pediatric Surgery, Department of Surgery, Indiana University School of Medicine, Indiana University, Indianapolis, Indiana. 8. Center for Surgical Outcomes Research, the Research Institute and Department of Surgery, Nationwide Children's Hospital, Ohio State University College of Medicine, Columbus, Ohio. 9. Division of Pediatric Surgery, Department of Surgery, Ann & Robert H. Lurie Children's Hospital of Chicago, Northwestern University Feinberg School of Medicine, Chicago, Illinois. 10. Division of Pediatric Surgery, Department of Surgery, University of Wisconsin-Madison, Madison, Wisconsin. 11. Section of Pediatric Surgery, Department of Surgery, Comer Children's Hospital, University of Chicago Medicine and Biological Sciences, Comer Children's Hospital, Chicago, Illinois; and. 12. Division of Pediatric Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
Abstract
BACKGROUND: Pediatric lung lesions are a group of mostly benign pulmonary anomalies with a broad spectrum of clinical disease and histopathology. Our objective was to evaluate the characteristics of children undergoing resection of a primary lung lesion and to identify preoperative risk factors for malignancy. METHODS: A retrospective cohort study was conducted by using an operative database of 521 primary lung lesions managed at 11 children's hospitals in the United States. Multivariable logistic regression was used to examine the relationship between preoperative characteristics and risk of malignancy, including pleuropulmonary blastoma (PPB). RESULTS: None of the 344 prenatally diagnosed lesions had malignant pathology (P < .0001). Among 177 children without a history of prenatal detection, 15 (8.7%) were classified as having a malignant tumor (type 1 PPB, n = 11; other PPB, n = 3; adenocarcinoma, n = 1) at a median age of 20.7 months (interquartile range, 7.9-58.1). Malignancy was associated with the DICER1 mutation in 8 (57%) PPB cases. No malignant lesion had a systemic feeding vessel (P = .0427). The sensitivity of preoperative chest computed tomography (CT) for detecting malignant pathology was 33.3% (95% confidence interval [CI]: 15.2-58.3). Multivariable logistic regression revealed that increased suspicion of malignancy by CT and bilateral disease were significant predictors of malignant pathology (odds ratios of 42.15 [95% CI, 7.43-340.3; P < .0001] and 42.03 [95% CI, 3.51-995.6; P = .0041], respectively). CONCLUSIONS: In pediatric lung masses initially diagnosed after birth, the risk of PPB approached 10%. These results strongly caution against routine nonoperative management in this patient population. DICER1 testing may be helpful given the poor sensitivity of CT for identifying malignant pathology.
BACKGROUND: Pediatric lung lesions are a group of mostly benign pulmonary anomalies with a broad spectrum of clinical disease and histopathology. Our objective was to evaluate the characteristics of children undergoing resection of a primary lung lesion and to identify preoperative risk factors for malignancy. METHODS: A retrospective cohort study was conducted by using an operative database of 521 primary lung lesions managed at 11 children's hospitals in the United States. Multivariable logistic regression was used to examine the relationship between preoperative characteristics and risk of malignancy, including pleuropulmonary blastoma (PPB). RESULTS: None of the 344 prenatally diagnosed lesions had malignant pathology (P < .0001). Among 177 children without a history of prenatal detection, 15 (8.7%) were classified as having a malignant tumor (type 1 PPB, n = 11; other PPB, n = 3; adenocarcinoma, n = 1) at a median age of 20.7 months (interquartile range, 7.9-58.1). Malignancy was associated with the DICER1 mutation in 8 (57%) PPB cases. No malignant lesion had a systemic feeding vessel (P = .0427). The sensitivity of preoperative chest computed tomography (CT) for detecting malignant pathology was 33.3% (95% confidence interval [CI]: 15.2-58.3). Multivariable logistic regression revealed that increased suspicion of malignancy by CT and bilateral disease were significant predictors of malignant pathology (odds ratios of 42.15 [95% CI, 7.43-340.3; P < .0001] and 42.03 [95% CI, 3.51-995.6; P = .0041], respectively). CONCLUSIONS: In pediatric lung masses initially diagnosed after birth, the risk of PPB approached 10%. These results strongly caution against routine nonoperative management in this patient population. DICER1 testing may be helpful given the poor sensitivity of CT for identifying malignant pathology.
Authors: Tasnim Muhamad; Khairil Amir Sayuti; Noor Hasnita Ismail Mokhtar; Nor Hayati Yunus; Noor Syafawati Ismail Journal: Qatar Med J Date: 2022-03-01
Authors: Abigail J Engwall-Gill; Sherwin S Chan; Kevin P Boyd; Jacqueline M Saito; Mary E Fallat; Shawn D St Peter; Stephanie Bolger-Theut; Eric J Crotty; Jared R Green; Rebecca L Hulett Bowling; Sachin S Kumbhar; Mantosh S Rattan; Cody M Young; Joseph K Canner; Katherine J Deans; Samir K Gadepalli; Michael A Helmrath; Ronald B Hirschl; Rashmi Kabre; Dave R Lal; Matthew P Landman; Charles M Leys; Grace Z Mak; Peter C Minneci; Tiffany N Wright; Shaun M Kunisaki Journal: JAMA Netw Open Date: 2022-06-01
Authors: Casper M Kersten; Sergei M Hermelijn; Dhanya Mullassery; Nagarajan Muthialu; Nazan Cobanoglu; Silvia Gartner; Pietro Bagolan; Carmen Mesas Burgos; Alberto Sgrò; Stijn Heyman; Holger Till; Janne Suominen; Maarten Schurink; Liesbeth Desender; Paul Losty; Henri Steyaert; Suzanne Terheggen-Lagro; Martin Metzelder; Arnaud Bonnard; Rony Sfeir; Michael Singh; Iain Yardley; Noor R V M Rikkers-Mutsaerts; Cornelis K van der Ent; Niels Qvist; Des W Cox; Robert Peters; Michiel A G E Bannier; Lucas Wessel; Marijke Proesmans; Michael Stanton; Edward Hannon; Marco Zampoli; Francesco Morini; Harm A W M Tiddens; René M H Wijnen; Johannes M Schnater Journal: Children (Basel) Date: 2022-07-30