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Primary lung carcinoma in children and adolescents: An analysis of the European Cooperative Study Group on Paediatric Rare Tumours (EXPeRT)

M. Abele, V. Bajčiová, F. Wright, S. Behjati, S. Voggel, DT. Schneider, C. Mallebranche, M. Česen Mazič, G. Guillén, M. Krawczyk, E. Bień, J. Roganovic, G. Bisogno, S. Chiaravalli, A. Ferrari, IB. Brecht, D. Orbach, Y. Reguerre, C. Virgone

. 2022 ; 175 (-) : 19-30. [pub] 20220907

Jazyk angličtina Země Anglie, Velká Británie

Typ dokumentu časopisecké články, práce podpořená grantem

Perzistentní odkaz   https://www.medvik.cz/link/bmc22032850

BACKGROUND: Primary lung carcinoma is an exceptionally rare childhood tumour, as per definition of the European Cooperative Study Group on Paediatric Rare Tumours (EXPeRT), with an incidence of 0.1-0.2/1,000,000 per year. Little is known about the clinical characteristics of children with primary lung carcinoma, a gap which this joint analysis of the EXPeRT group aimed to fill. PATIENTS AND METHODS: We performed a retrospective case series of children (aged 0-18 years) with primary lung carcinoma, as collected through the EXPeRT databases between 2000 and 2021. We recorded relevant clinical characteristics including treatment and outcome. RESULTS: Thirty-eight patients were identified with a median age of 12.8 years at diagnosis (range: 0-17). Mucoepidermoid carcinoma (MEC) was the most frequent entity (n = 20), followed by adenocarcinoma (n = 12), squamous cell carcinoma (n = 4), adenosquamous carcinoma (n = 1) and small-cell lung cancer (n = 1). Patients with MEC presented rarely with lymph node metastases (2/20 cases). Overall, 19/20 patients achieved long-lasting remission by surgical resection only. Patients with other histologies often presented in advanced stages (14/18 TNM stage IV). With multimodal treatment, 3-year overall survival was 52% ± 13%. While all patients with squamous cell carcinoma died, the 12 patients with adenocarcinoma had a 3-year overall survival of 64% ± 15%. CONCLUSIONS: Primary lung carcinomas rarely occur in children. While the outcome of children with MEC is favourable with surgery alone, patients with other histotypes have a poor prognosis, despite aggressive treatment, highlighting the need to develop new strategies for these children, such as mutation-guided treatment.

Citace poskytuje Crossref.org

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$a BACKGROUND: Primary lung carcinoma is an exceptionally rare childhood tumour, as per definition of the European Cooperative Study Group on Paediatric Rare Tumours (EXPeRT), with an incidence of 0.1-0.2/1,000,000 per year. Little is known about the clinical characteristics of children with primary lung carcinoma, a gap which this joint analysis of the EXPeRT group aimed to fill. PATIENTS AND METHODS: We performed a retrospective case series of children (aged 0-18 years) with primary lung carcinoma, as collected through the EXPeRT databases between 2000 and 2021. We recorded relevant clinical characteristics including treatment and outcome. RESULTS: Thirty-eight patients were identified with a median age of 12.8 years at diagnosis (range: 0-17). Mucoepidermoid carcinoma (MEC) was the most frequent entity (n = 20), followed by adenocarcinoma (n = 12), squamous cell carcinoma (n = 4), adenosquamous carcinoma (n = 1) and small-cell lung cancer (n = 1). Patients with MEC presented rarely with lymph node metastases (2/20 cases). Overall, 19/20 patients achieved long-lasting remission by surgical resection only. Patients with other histologies often presented in advanced stages (14/18 TNM stage IV). With multimodal treatment, 3-year overall survival was 52% ± 13%. While all patients with squamous cell carcinoma died, the 12 patients with adenocarcinoma had a 3-year overall survival of 64% ± 15%. CONCLUSIONS: Primary lung carcinomas rarely occur in children. While the outcome of children with MEC is favourable with surgery alone, patients with other histotypes have a poor prognosis, despite aggressive treatment, highlighting the need to develop new strategies for these children, such as mutation-guided treatment.
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$a Bajčiová, Viera $u Department of Pediatric Oncology, Childrens University Hospital, Brno, Czech Republic
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$a Wright, Fiona $u Department of Pediatric Hematology and Oncology, Cambridge Univeristy Hospital NHS Foundation Trust, Cambridge, United Kingdom
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$a Behjati, Sam $u Department of Pediatric Hematology and Oncology, Cambridge Univeristy Hospital NHS Foundation Trust, Cambridge, United Kingdom
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$a Voggel, Sarah $u Pediatric Hematology/Oncology, Department of Pediatrics, University Hospital Tuebingen, Tuebingen, Germany
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$a Česen Mazič, Maja $u University Medical Center Ljubljana, Ljubljana, Slovenia
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$a Guillén, Gabriela $u Surgical Oncology and Neonatal Surgery, Pediatric Surgery Department, Hospital Infantil Universitari Vall d'Hebron, Barcelona, Spain
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$a Krawczyk, Malgorzata $u Department of Pediatrics, Hematology and Oncology, Medical University of Gdansk, Gdansk, Poland
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$a Bień, Ewa $u Department of Pediatrics, Hematology and Oncology, Medical University of Gdansk, Gdansk, Poland
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$a Roganovic, Jelena $u Hematology-Oncology Division, Department of Pediatrics, Clinical Hospital Center Rijeka, University of Rijeka, Rijeka, Croatia
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$a Bisogno, Gianni $u Hematology-Oncology Division, Department of Pediatrics, Padova University Hospital, Padua, Italy
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$a Chiaravalli, Stefano $u Pediatric Oncology Unit, Fondazione IRCCS Istituto Nazionale dei Tumori di Milano, Milan, Italy
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$a Ferrari, Andrea $u Pediatric Oncology Unit, Fondazione IRCCS Istituto Nazionale dei Tumori di Milano, Milan, Italy
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$a Brecht, Ines B $u Pediatric Hematology/Oncology, Department of Pediatrics, University Hospital Tuebingen, Tuebingen, Germany
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$a Reguerre, Yves $u Department of Pediatric Hematology and Oncology, Félix Guyon University Hospital, St Denis, Réunion Island, France
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