Malignant transformation and tumour recurrence in sacrococcygeal teratoma: a global, retrospective cohort study

Introduction: Sacrococcygeal teratoma (SCT) is a rare congenital tumour. The risk of malignancy and recurrence is not well defined. Previous studies are small and report differing conclusions about the timing of surgery and the duration of follow-up. The authors studied the risk of malignant transformation and SCT recurrence after surgery to address these gaps. Methods: This was a global retrospective cohort study. Data of consecutive SCT patients was obtained from 145 institutes in 62 countries. Malignant transformation, defined as malignancy at initial resection, malignant recurrence or death due to malignancy, and its risk factors were analysed. Results: Of the 3612 included patients, 3407 entered analysis. The risk of malignant transformation of the initial tumour was 3.3, 5.1, 10.1, and 32.9% at age 3 months, 6 months, 1 year, and 2 years, respectively. After 6 years, the censored risk of malignancy (64%) did not further increase. Recurrent SCT was diagnosed in 349 (10.2%) children with 126 (36.1%) malignant recurrences. Risk factors for recurrence were Altman type II [odds ratio (OR): 1.6, 95% confidence interval (CI): 1.2–2.2], Altman type III (OR: 1.6, 95% CI: 1.2–2.3), initial immature histology (OR: 1.9, 95% CI: 1.4–2.6), and initial malignant histology (OR: 4.0, 95% CI: 2.9–5.4). Conclusion: The risk of malignancy at initial resection in SCT increases with age reaching a plateau at 6 years of age. Recurrence after resection occurred in 10% of patients and 36% of these were malignant at that time. Altman type II or type III, and immature or malignant histology were associated with recurrence. © © 2024 The Author(s).

Авторы
Van Heurn Lieke J. 1 , Derikx Joep P.M. 1 , Hall Nigel J. 10 , Aldrink Jennifer Hall 3 , Bailez María Marcela 9 , Chirdan Lohfa B. 5 , Fumino Shigehisa 6 , Hesse Afua Adwo Jectey 7 , Soyer Tuktu 8 , Stpeter Shawn 4 , Twisk Jos W.R. 2 , Yang Tianyou 11 , Van Heurn Ernest L.W. 1 , Ignacio Siffredi Juan 12 , Lobos Pablo Andrés 13 , Till Holger Karl H. 14 , Rahman Ashrarur Rahman 15 , Govorukhina Olga A. 16 , Prokopenya Natalya S. 16 , De Backer Antoine 17 , Reusens Helena 18 , De Campos Vieira Abib Simone 19 , Stefanova Penka Peneva 20 , Tolekova Nadezhda 21 , Faustin Mouafo Tambo 22 , Laberge Jean Martin 23 , Zani Augusto 24 , Walker Richard J.B. 24, 25 , Pérez Maricarmen Olivos 26 , Valenzuela Marco Andrés 26 , Shen Yuanchao 29 , Zou Yan 29 , Ćavar Stanko 30 , Pogorelić Zenon 27, 31 , Pos Lucie 32 , Škába Richard 33 , Jørgensen Peter Hjorth 34 , Abouzeid Amr Abdelhamid 35 , Elfiky M.A. 36 , Taher H.M.A. 37 , Märtson Matis 38 , Derbew Miliard 39 , Tigabie Workye Molla 40 , Koivusalo Antti Ilmari 41 , Pakarinen Mikko Petteri 41, 42 , Abbo Olivier 43 , Arnaud Alexis P. 44 , Ballouhey Quentin 45 , Bastard François 46 , Dariel Anne Laure 47 , Irtan Sabine 48 , Lecompte Jean François 49 , Levard Guillaume 50 , Sarnacki Sabine 51 , Sfeir Rony 52 , Vinit Nicolas 53 , Holler Anne Sophie Bernadette 54 , Jank Marietta 55 , Lacher Martin 56 , Muensterer Oliver J. 54 , Rothe Karin 57 , Rolle Udo 58 , Lamprinou Zoi 59 , Jakab Zsuzsanna 60 , Vajda Péter L. 61 , Vojcek Ágnes 62 , Wong Kenneth Kak Yuen 63 , Abraham Mohan K. 64 , Rathod Kirtikumar Jagdish 65 , Sharma Shilpa H. 66 , Gunadi 67 , Sarafi Mehdi 68 , Khaleghnejad Tabari Ahmad 68 , Al Ani Muataz 69 , Kane Gavin Alan 70 , Sukhotnik Igor 71 , Avanzini Stefano 72 , Bagolan Pietro 73, 74 , Gamba Piergiorgio 75 , Guanà Riccardo 76 , Inserra Alessandro 77 , Lima Mario 78 , Morabito Antonino 79 , Raffaele Alessandro 80 , Riccipetitoni Giovanna 81 , Virgone Calogero 75 , Aké Yapi Landry 82 , Hirohata Yoshiaki 6 , Inoue Maho 83 , Kanamori Yutaka 84 , Kawakubo Naonori 85 , Sasaki Takashi 86 , Taguchi Tomoaki 85 , Tajiri Tatsurou 6 , Tomita Hirofumi 87 , Usui Noriaki 88 , Kakar Mohit 89 , Barauskas Vidmantas 90 , Risteski Toni 91 , Abdul Aziz Dayang Anita 92
Journal
International Journal of Surgery
Номер выпуска
11
Язык
English
Страницы
7177-7186
Статус
Published
Ссылка
Внешняя ссылка
DOI
10.1097/JS9.0000000000002045
Том
110
Год
2024
Организации
  • 1 Department of Pediatric Surgery, Universiteit van Amsterdam, Amsterdam, Noord-Holland, Netherlands
  • 2 Department of Epidemiology and Data Science, Vrije Universiteit Amsterdam, Amsterdam, Noord-Holland, Netherlands
  • 3 Department of Surgery, The Ohio State University College of Medicine, Columbus, OH, United States
  • 4 Department of Surgery, Children's Mercy Kansas City, Kansas City, MO, United States
  • 5 Department of Surgery, University of Jos, Jos, Plateau, Nigeria
  • 6 Department of Pediatric Surgery, Kyoto Prefectural University of Medicine, Kyoto, Kyoto, Japan
  • 7 Paediatric Surgery and Anatomy, Accra College of Medicine, Accra, Ghana
  • 8 Department of Pediatric Surgery, Hacettepe Üniversitesi, Ankara, Turkey
  • 9 Department of Surgery, Hospital de Pediatria SAMIC Prof. Dr. Juan P. Garrahan, Buenos Aires, Argentina
  • 10 University of Southampton, Faculty of Medicine, Southampton, Hampshire, United Kingdom
  • 11 Department of Surgical Oncology, Guangzhou Medical University, Guangzhou, Guangdong, China
  • 12 Department of Pediatric Surgery, Hospital de Pediatria SAMIC Prof. Dr. Juan P. Garrahan, Buenos Aires, Argentina
  • 13 Hospital Italiano de Buenos Aires, Buenos Aires, Argentina
  • 14 Department of Paediatrics and Adolescent Medicine, Medizinische Universität Graz, Graz, Styria, Austria
  • 15 Dhaka Shishu Hospital, Dhaka, Bangladesh
  • 16 Center of Pediatric Surgery, Belarus
  • 17 Universitair Ziekenhuis Brussel, Jette, Belgium
  • 18 Department of Pediatric Surgery, Université Libre de Bruxelles, Brussels, BRU, Belgium
  • 19 Pediatric Oncology, Universidade Federal de São Paulo, Sao Paulo, SP, Brazil
  • 20 Department of Pediatric Surgery, St.George University Hospital, Plovdiv, Bulgaria
  • 21 Clinic of Internal Diseases UMHATEM “N. I. Pirogov”, Sofia, Bulgaria
  • 22 Faculty of Medicine and Biomedical Sciences, Université de Yaoundé I, Yaounde, Cameroon
  • 23 Centre Universitaire de Santé McGill, Hôpital de Montreal Pour Enfants, Montreal, QC, Canada
  • 24 Division of General and Thoracic Surgery, The Hospital for Sick Children, Toronto, ON, Canada
  • 25 Department of Surgery, University of Toronto, Toronto, ON, Canada
  • 26 Hospital de Niños Roberto del Río, Santiago, RM, Chile
  • 27 Department of Surgery, School of Medicine, University of Split, Split, Split-Dalmatia, Croatia
  • 28 Department of Pediatric Surgery, University Teaching Hospital Lusaka, Lusaka, Zambia
  • 29 Department of Pediatric Surgery, Guangzhou Medical University, Guangzhou, Guangdong, China
  • 30 Division of Pediatric Surgery, KBC Zagreb, Zagreb, Croatia
  • 31 Department of Pediatric Surgery, Klinički Bolnički Centar Split, Split, Split-Dalmatia, Croatia
  • 32 Department of Pediatric Surgery, Fakultní Nemocnice v Motole, Prague, Czech Republic
  • 33 Department of Pediatric Surgery, Charles University, Prague, Czech Republic
  • 34 Department of Pediatric Surgery, Rigshospitalet, Copenhagen, Hovedstaden, Denmark
  • 35 Faculty of Medicine, Ain Shams University, Cairo, Cairo, Egypt
  • 36 Faculty of Medicine, Cairo, Cairo, Egypt
  • 37 Cairo University, Giza, Giza, Egypt
  • 38 Surgical Clinic, Tallinn Children's Hospital, Tallinn, Harjumaa, Estonia
  • 39 Medical Education Partnership Initiative Junior Faculty Project, School of Medicine, Addis Ababa, Ethiopia
  • 40 St. Peter Specialized Hospital, Addis Ababa, Addis Ababa, Ethiopia
  • 41 Section of Pediatric Surgery, Helsingin Yliopisto, Helsinki, Uusimaa, Finland
  • 42 Department of Women's and Children's Health, Karolinska Institutet, Stockholm, Stockholms, Sweden
  • 43 Department of Surgery, Hopital des Enfants, Toulouse, Occitanie, France
  • 44 Department of Pediatric Surgery, Université de Rennes, Rennes, Brittany, France
  • 45 Department of Pediatric Surgery, Université de Limoges, Limoges, Nouvelle-Aquitaine, France
  • 46 CHU Angers, Angers, Pays-de-la-Loire, France
  • 47 Department of Pediatric Surgery, Hopital La Timone, Marseille, France
  • 48 Hôpital Armand-Trousseau, Paris, Ile-de-France, France
  • 49 Department of Pediatric Surgery, Centre Hospitalier Universitaire de Nice, Nice, Provence-Alpes-Cote d'Azur, France
  • 50 Department of Pediatric Surgery, Centre Hospitalier Universitaire de Poitiers, Poitiers, France
  • 51 Department of Pediatric Surgery, Université Paris Cité, Paris, Ile-de-France, France
  • 52 Pediatric Unit, CHU Lille, Lille, Hauts-de-France, France
  • 53 Department of Pediatric Surgery and Urology, Université Paris Cité, Paris, Ile-de-France, France
  • 54 Department of Pediatric Surgery, Klinikum der Universität München, Munich, Bayern, Germany
  • 55 Department of Pediatric Surgery, Universitätsklinikum Mannheim, Mannheim, Baden-Wurttemberg, Germany
  • 56 Department of Pediatric Surgery, Universität Leipzig, Leipzig, Sachsen, Germany
  • 57 Department of Pediatric Surgery, Charité – Universitätsmedizin Berlin, Berlin, Berlin, Germany
  • 58 Department of Paediatric Surgery and Paediatric Urology, Universitätsklinikum Frankfurt, Frankfurt am Main, Hessen, Germany
  • 59 P & A Kyriakou Children's Hospital, Athens, Athens, Greece
  • 60 Hungarian Childhood Cancer Registry, II. No. Pediatric Clinic, Budapest, Hungary
  • 61 Division of Paediatric Surgery, University of Pécs Medical School, Pecs, Baranya, Hungary
  • 62 Department of Pediatrics, University of Pécs Medical School, Pecs, Baranya, Hungary
  • 63 Department of Surgery, The University of Hong Kong Li Ka Shing Faculty of Medicine, Hong Kong, Hong Kong
  • 64 Department of Pediatric Surgery and Pediatric Urology, Amrita Institute of Medical Sciences India, Kochi, KL, India
  • 65 Department of Pediatric Surgery, All India Institute of Medical Sciences, Jodhpur, Jodhpur, RJ, India
  • 66 Department of Pediatric Surgery, All India Institute of Medical Sciences, New Delhi, New Delhi, DL, India
  • 67 Department of Surgery, Universitas Gadjah Mada, Yogyakarta, Yogyakarta, Indonesia
  • 68 Research Institute for Children's Health, SBUMS Pediatric Surgery Research Center, Tehran, Tehran, Iran
  • 69 Ninevah University, Mosul, Nineveh, Iraq
  • 70 Department of Pediatric Surgery, Children's Health Ireland at Crumlin, Dublin, Ireland
  • 71 Department of Pediatric Surgery, Tel Aviv University, Tel Aviv-Yafo, Tel Aviv District, Israel
  • 72 Department of Pediatric Surgery, Istituto Giannina Gaslini, Genoa, Italy
  • 73 IRCCS Ospedale Pediatrico Bambino Gesù, Rome, RM, Italy
  • 74 Department of Systems Medicine, Università degli Studi di Roma "Tor Vergata", Rome, RM, Italy
  • 75 Department of Women's and Children's Health, Università degli Studi di Padova, Padua, PD, Italy
  • 76 Pediatric and Neonatal Surgery Unit, Ospedale Infantile Regina Margherita, Turin, TO, Italy
  • 77 Graduate School in Pediatric Surgery, Università degli Studi di Roma "Tor Vergata", Rome, RM, Italy
  • 78 IRCCS Azienda Ospedaliero-Universitaria di Bologna, Bologna, BO, Italy
  • 79 Università degli Studi di Firenze, Florence, FI, Italy
  • 80 Department of Pediatric Surgery, Fondazione IRCCS Policlinico San Matteo, Pavia, PV, Italy
  • 81 Fondazione IRCCS Policlinico San Matteo, Pavia, PV, Italy
  • 82 Department of Mother and Child Health, University Teaching Hospital of Cocody, Abidjan, Abidjan, Cote d'Ivoire
  • 83 Department of Surgery, Saitama Children's Medical Center, Iwatsuki, Saitama, Japan
  • 84 Department of General Surgery, National Center for Child Health and Development, Tokyo, Japan
  • 85 Department of Pediatric Surgery, Graduate School of Medical Sciences, Fukuoka, Fukuoka, Japan
  • 86 Department of Pediatric Surgery, Osaka City General Hospital, Osaka, Osaka, Japan
  • 87 Department of Surgery, Tokyo Metropolitan Children's Medical Center, Fuchu, Tokyo, Japan
  • 88 Department of Pediatric Surgery, Osaka Women's and Children's Hospital, Izumi, Osaka, Japan
  • 89 Department of Pediatric Surgery, Riga Stradins University, Riga, RIX, Latvia
  • 90 Vaiku Chirurgijos Klinika, Lietuvos Sveikatos Mokslų Universitetas, Kaunas, Kaunas, Lithuania
  • 91 Faculty of Medicine, University of Skopje, Skopje, North Macedonia
  • 92 Department of Surgery, Universiti Kebangsaan Malaysia, Bangi, Selangor, Malaysia
  • 93 Kuala Lumpur Women and Children Hospital, Kuala Lumpur, Kuala Lumpur, Malaysia
  • 94 Servicio de Cirugía Oncológica, Instituto Nacional de Pediatría, Mexico, DF, Mexico
  • 95 Institute for Children's Disease, University of Montenegro, Podgorica, Montenegro
  • 96 Department of General Surgery, CHU Mohammed-VI, Marakech, Marrakesh-Safi, Morocco
  • 97 Department of Pediatric Surgery, Universitair Medisch Centrum Groningen, Groningen, Groningen, Netherlands
  • 98 Radboud University Medical Center, Nijmegen, Gelderland, Netherlands
  • 99 Department of Pediatric Surgery, Maastricht Universitair Medisch Centrum+, Maastricht, Limburg, Netherlands
  • 100 Universiteit van Amsterdam, Amsterdam, Noord-Holland, Netherlands
  • 101 Department of Pediatric Surgery, Erasmus MC Sophia Children’s Hospital, Rotterdam, Zuid-Holland, Netherlands
  • 102 Maxima Medical Center, Veldhoven, Veldhoven, Noord-Brabant, Netherlands
  • 103 Intensive Care and Department of Pediatric Surgery, Erasmus MC Sophia Children’s Hospital, Rotterdam, Zuid-Holland, Netherlands
  • 104 Department of Pediatric Surgery, Wilhelmina Kinderziekenhuis, Utrecht, Netherlands
  • 105 Department of Surgery, College of Medicine, University of Lagos, Lagos, Lagos, Nigeria
  • 106 Department of Surgery, Lagos University Teaching Hospital, Lagos, Lagos, Nigeria
  • 107 Nnamdi Azikiwe University, Awka, Anambra, Nigeria
  • 108 Department of Surgery, The University of Chicago, Chicago, IL, United States
  • 109 Abubakar Tafawa Balewa University, Bauchi, Bauchi, Nigeria
  • 110 Department of Pediatric Surgery, Oslo Universitetssykehus, Oslo, Oslo, Norway
  • 111 The Aga Khan University, Karachi, Sindh, Pakistan
  • 112 Department of Pediatric Surgery, Shaheed Zulfiqar Ali Bhutto Medical University, Islamabad, Pakistan
  • 113 University of Child Health Sciences, Lahore, Pakistan
  • 114 Department of Pediatric Surgery, Philippine Children's Medical Center, Quezon City, Philippines
  • 115 Department of Surgery, Philippine General Hospital, Manila, Philippines
  • 116 University of the Philippines College of Medicine, Manila, Philippines
  • 117 Department of Pathology, Instytut "Pomnik - Centrum Zdrowia Dziecka", Warsaw, MA, Poland
  • 118 Department of Pediatric Surgery and Organ Transplantation, Instytut "Pomnik - Centrum Zdrowia Dziecka", Warsaw, MA, Poland
  • 119 Department of Pediatric Surgery and Urology, Gdanski Uniwersytet Medyczny, Gdansk, Pomeranian, Poland
  • 120 Department of Pediatric Surgery, Hospital de Dona Estefânia, Lisbon, Portugal
  • 121 Central Lisbon University Hospital Center, EPE (CHULC), Lisbon, Lisboa, Portugal
  • 122 Department of Pediatric Surgery and Orthopedics, Universitatea de Medicina si Farmacie Victor Babes din Timisoara, Timisoara, Timis, Romania
  • 123 FSAI "NMRC of Children Health" MHRF, Moscow, Russian Federation
  • 124 RUDN University, Moscow, Moscow Oblast, Russian Federation
  • 125 Volgogradskij Gosudarstvennyj Medicinskij Universitet, Volgograd, Volgograd Oblast, Russian Federation
  • 126 Regional Center for Pediatric Minimally Invasive Surgery and Pediatric Robotics, Irkutsk Regional Children’s Hospital, Irkutsk, Russian Federation
  • 127 Department of Pediatric Surgery, Irkutsk State Medical University, Irkutsk, Irkutsk Oblast, Russian Federation
  • 128 Department of Pediatric Surgery, Stavropol State Medical University, Stavropol, Stavropol Krai, Russian Federation
  • 129 Center for Pediatric Surgery, University Children's Hospital, Belgrade, Belgrade, Serbia
  • 130 Department of Pediatric Surgery, KK Women's And Children's Hospital, Singapore City, Singapore
  • 131 Duke-NUS Medical School, Singapore City, Singapore
  • 132 National Institute of Children's Diseases, Bratislava, Slovakia
  • 133 Department of Pediatric Surgery, Univerzitetni Klinični Center Ljubljana, Ljubljana, Slovenia
  • 134 Department of Pediatric Surgery, Seoul National University Hospital, Seoul, South Korea
  • 135 Department of Pediatric Surgery, Seoul National University College of Medicine, Seoul, South Korea
  • 136 Department of Pediatric Surgery, University of Ulsan College of Medicine, Seoul, South Korea
  • 137 Department of Surgery, Inje University Paik Hospital, Seoul, Nowon-gu, South Korea
  • 138 Department of Pediatric Surgery, Hospital Universitari Vall d'Hebron, Barcelona, Barcelona, Spain
  • 139 General-Oncologic Pediatric Surgery, Children's Hospital la Paz, Spain
  • 140 Department of Pediatric Surgery, Hospital Universitario Virgen del Rocío, Sevilla, Seville, Spain
  • 141 Surgery Service, Hospital Universitario Reina Sofia, Cordoba, Cordoba, Spain
  • 142 Department of Pediatric Surgery, Hospital Clínico Universitario Virgen de la Arrixaca, Murcia, Murcia, Spain
  • 143 Department of Pediatric Surgery, Hospital Universitari i Politècnic La Fe, Valencia, Valencia, Spain
  • 144 Department of Surgery, University of Colombo, Colombo, Sri Lanka
  • 145 Department of Pediatric Surgery, Skånes Universitetssjukhus, Lund, Skane, Sweden
  • 146 Department of Women's and Children's Health, Karolinska Universitetssjukhuset, Stockholm, Stockholms, Sweden
  • 147 Department of Pediatric Surgery, Karolinska Universitetssjukhuset, Stockholm, Stockholms, Sweden
  • 148 Karolinska Universitetssjukhuset, Stockholm, Stockholms, Sweden
  • 149 Department of Pediatric Surgery, Children's University Hospital, Damascus, Damascus, Syrian Arab Republic
  • 150 Division of Hematology and Oncology, Chang Gung University College of Medicine, Taoyuan, Taiwan
  • 151 Department of Surgery, Queen Sirikit National Institute of Child Health, Bangkok, Thailand
  • 152 Department of Pediatric Surgery, CHU Hedi-Chaker, Sfax, Tunisia
  • 153 Department of Pediatric Surgery, CHU Fattouma-Bourguiba, Monastir, Monastir, Tunisia
  • 154 Department of Pediatric Surgery, Ege Üniversitesi, Izmir, Turkey
  • 155 Department of Pediatric Surgery, Istanbul Medeniyet University, Istanbul, Turkey
  • 156 Department of Pediatric Surgery, University of Health Sciences, Istanbul, Turkey
  • 157 Department of Pediatric Oncology, Hacettepe Üniversitesi, Ankara, Turkey
  • 158 Department of Pediatric Surgery, Bogomolets National Medical University, Kyiv, Ukraine
  • 159 Department of Pediatric Surgery, Leeds Teaching Hospitals NHS Trust, Leeds, West Yorkshire, United Kingdom
  • 160 Department of Pediatric Surgery, Cambridge University Hospitals NHS Foundation Trust, Cambridge, Cambridgeshire, United Kingdom
  • 161 Department of Pediatric Surgery, Great North Children's Hospital, Newcastle Upon Tyne, Tyne and Wear, United Kingdom
  • 162 MS Paediatric Surgery, Birmingham Children's Hospital, Birmingham, West Midlands, United Kingdom
  • 163 Department of Pediatric Surgery, Royal Manchester Children's Hospital, Manchester, Greater Manchester, United Kingdom
  • 164 Department of Pediatric Surgery, The Royal London Hospital, London, United Kingdom
  • 165 Department of Pediatric Surgery, Universitätsklinikum Frankfurt, Frankfurt am Main, Hessen, Germany
  • 166 Bristol Royal Hospital for Children, Bristol, United Kingdom
  • 167 Developmental Biology and Cancer Programme, UCL Great Ormond Street Institute of Child Health, London, United Kingdom
  • 168 Department of Specialist Neonatal and Paediatric Surgery, Great Ormond Street Hospital for Children NHS Foundation Trust, London, United Kingdom
  • 169 Institute of Life Course and Medical Sciences, University of Liverpool, Liverpool, Merseyside, United Kingdom
  • 170 Department of Pediatric Surgery, University Hospital Southampton NHS Foundation Trust, Southampton, Hampshire, United Kingdom
  • 171 Royal Manchester Children's Hospital, Manchester, Greater Manchester, United Kingdom
  • 172 Department of Paediatric Urology, Bristol Royal Hospital for Children, Bristol, United Kingdom
  • 173 Department of Pediatric Surgery, Royal Hospital for Sick Children, Edinburgh, Scotland, United Kingdom
  • 174 Birmingham Children's Hospital, Birmingham, West Midlands, United Kingdom
  • 175 Department of Surgery, University of Utah School of Medicine, Salt Lake City, UT, United States
  • 176 Department of Surgery, University of Tennessee, San Jose, CA, United States
Ключевые слова
malignant transformation; recurrence; sacrococcygeal teratoma
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