Evidence of a dual histogenetic pathway of sacrococcygeal teratomas

Research output: Contribution to journalArticle

2 Citations (Scopus)

Abstract

Aims: Sacrococcygeal teratomas are rare tumours that occur most frequently in neonates, although adult cases also occur. The molecular pathogenesis of these tumours and their long-term prognosis is uncertain. We investigated the i(12p) status of a large number of primary sacrococcygeal teratomas in both children and adults, including cases with malignant germ cell tumour elements. Methods and results: Fifty-four sacrococcygeal teratoma specimens from 52 patients were identified, and available follow-up information was obtained. Fluorescence in-situ hybridization analysis was performed to identify isochromosome 12p [i(12p)] abnormalities on paraffin blocks of the tumours. Among the 48 paediatric patients, there were 44 teratomas and four tumours with combined teratoma and yolk sac tumour (one of whom also had primitive neuroectodermal tumour). The teratomas included 37 mature teratomas and 11 immature teratomas (four grade 1, two grade 2, and five grade 3). The 44 teratomas lacking a yolk sac tumour component were all negative for i(12p). The four tumours with a yolk sac tumour component were all positive for i(12p). The four adult cases all lacked non-teratomatous germ cell tumour components, immature elements, and i(12p). Follow-up information was available for 32 patients. Two patients with teratoma had recurrence, but were alive with no evidence of disease after long-term follow-up. One patient with combined teratoma and yolk sac tumour had recurrence 7 months after resection. The other patients were alive with no evidence of disease at last follow-up. Conclusions: Our data suggest that paediatric sacrococcygeal teratomas should be considered as two distinct groups with divergent histogenetic pathways. The prognosis of these tumours is excellent, despite rare recurrence.

Original languageEnglish (US)
JournalHistopathology
DOIs
StateAccepted/In press - 2016

Fingerprint

Teratoma
Isochromosomes
Endodermal Sinus Tumor
Neoplasms
Germ Cell and Embryonal Neoplasms
Recurrence
Pediatrics
Primitive Neuroectodermal Tumors
Cellular Structures
Fluorescence In Situ Hybridization
Paraffin
Newborn Infant

Keywords

  • Germ cell tumour
  • Histogenesis
  • Isochromosome 12p
  • Molecular genetics
  • Sacrococcygeal teratoma
  • Testis

ASJC Scopus subject areas

  • Pathology and Forensic Medicine
  • Histology

Cite this

@article{cd13f3863095418d9105efbb3f1d114d,
title = "Evidence of a dual histogenetic pathway of sacrococcygeal teratomas",
abstract = "Aims: Sacrococcygeal teratomas are rare tumours that occur most frequently in neonates, although adult cases also occur. The molecular pathogenesis of these tumours and their long-term prognosis is uncertain. We investigated the i(12p) status of a large number of primary sacrococcygeal teratomas in both children and adults, including cases with malignant germ cell tumour elements. Methods and results: Fifty-four sacrococcygeal teratoma specimens from 52 patients were identified, and available follow-up information was obtained. Fluorescence in-situ hybridization analysis was performed to identify isochromosome 12p [i(12p)] abnormalities on paraffin blocks of the tumours. Among the 48 paediatric patients, there were 44 teratomas and four tumours with combined teratoma and yolk sac tumour (one of whom also had primitive neuroectodermal tumour). The teratomas included 37 mature teratomas and 11 immature teratomas (four grade 1, two grade 2, and five grade 3). The 44 teratomas lacking a yolk sac tumour component were all negative for i(12p). The four tumours with a yolk sac tumour component were all positive for i(12p). The four adult cases all lacked non-teratomatous germ cell tumour components, immature elements, and i(12p). Follow-up information was available for 32 patients. Two patients with teratoma had recurrence, but were alive with no evidence of disease after long-term follow-up. One patient with combined teratoma and yolk sac tumour had recurrence 7 months after resection. The other patients were alive with no evidence of disease at last follow-up. Conclusions: Our data suggest that paediatric sacrococcygeal teratomas should be considered as two distinct groups with divergent histogenetic pathways. The prognosis of these tumours is excellent, despite rare recurrence.",
keywords = "Germ cell tumour, Histogenesis, Isochromosome 12p, Molecular genetics, Sacrococcygeal teratoma, Testis",
author = "Robert Emerson and Kao, {Chia Sui} and John Eble and David Grignon and Mingsheng Wang and Shaobo Zhang and Xiaoyan Wang and Rong Fan and Timothy Masterson and Roth, {Lawrence M.} and Liang Cheng",
year = "2016",
doi = "10.1111/his.13062",
language = "English (US)",
journal = "Histopathology",
issn = "0309-0167",
publisher = "Wiley-Blackwell",

}

TY - JOUR

T1 - Evidence of a dual histogenetic pathway of sacrococcygeal teratomas

AU - Emerson, Robert

AU - Kao, Chia Sui

AU - Eble, John

AU - Grignon, David

AU - Wang, Mingsheng

AU - Zhang, Shaobo

AU - Wang, Xiaoyan

AU - Fan, Rong

AU - Masterson, Timothy

AU - Roth, Lawrence M.

AU - Cheng, Liang

PY - 2016

Y1 - 2016

N2 - Aims: Sacrococcygeal teratomas are rare tumours that occur most frequently in neonates, although adult cases also occur. The molecular pathogenesis of these tumours and their long-term prognosis is uncertain. We investigated the i(12p) status of a large number of primary sacrococcygeal teratomas in both children and adults, including cases with malignant germ cell tumour elements. Methods and results: Fifty-four sacrococcygeal teratoma specimens from 52 patients were identified, and available follow-up information was obtained. Fluorescence in-situ hybridization analysis was performed to identify isochromosome 12p [i(12p)] abnormalities on paraffin blocks of the tumours. Among the 48 paediatric patients, there were 44 teratomas and four tumours with combined teratoma and yolk sac tumour (one of whom also had primitive neuroectodermal tumour). The teratomas included 37 mature teratomas and 11 immature teratomas (four grade 1, two grade 2, and five grade 3). The 44 teratomas lacking a yolk sac tumour component were all negative for i(12p). The four tumours with a yolk sac tumour component were all positive for i(12p). The four adult cases all lacked non-teratomatous germ cell tumour components, immature elements, and i(12p). Follow-up information was available for 32 patients. Two patients with teratoma had recurrence, but were alive with no evidence of disease after long-term follow-up. One patient with combined teratoma and yolk sac tumour had recurrence 7 months after resection. The other patients were alive with no evidence of disease at last follow-up. Conclusions: Our data suggest that paediatric sacrococcygeal teratomas should be considered as two distinct groups with divergent histogenetic pathways. The prognosis of these tumours is excellent, despite rare recurrence.

AB - Aims: Sacrococcygeal teratomas are rare tumours that occur most frequently in neonates, although adult cases also occur. The molecular pathogenesis of these tumours and their long-term prognosis is uncertain. We investigated the i(12p) status of a large number of primary sacrococcygeal teratomas in both children and adults, including cases with malignant germ cell tumour elements. Methods and results: Fifty-four sacrococcygeal teratoma specimens from 52 patients were identified, and available follow-up information was obtained. Fluorescence in-situ hybridization analysis was performed to identify isochromosome 12p [i(12p)] abnormalities on paraffin blocks of the tumours. Among the 48 paediatric patients, there were 44 teratomas and four tumours with combined teratoma and yolk sac tumour (one of whom also had primitive neuroectodermal tumour). The teratomas included 37 mature teratomas and 11 immature teratomas (four grade 1, two grade 2, and five grade 3). The 44 teratomas lacking a yolk sac tumour component were all negative for i(12p). The four tumours with a yolk sac tumour component were all positive for i(12p). The four adult cases all lacked non-teratomatous germ cell tumour components, immature elements, and i(12p). Follow-up information was available for 32 patients. Two patients with teratoma had recurrence, but were alive with no evidence of disease after long-term follow-up. One patient with combined teratoma and yolk sac tumour had recurrence 7 months after resection. The other patients were alive with no evidence of disease at last follow-up. Conclusions: Our data suggest that paediatric sacrococcygeal teratomas should be considered as two distinct groups with divergent histogenetic pathways. The prognosis of these tumours is excellent, despite rare recurrence.

KW - Germ cell tumour

KW - Histogenesis

KW - Isochromosome 12p

KW - Molecular genetics

KW - Sacrococcygeal teratoma

KW - Testis

UR - http://www.scopus.com/inward/record.url?scp=84990937880&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84990937880&partnerID=8YFLogxK

U2 - 10.1111/his.13062

DO - 10.1111/his.13062

M3 - Article

C2 - 27521765

AN - SCOPUS:84990937880

JO - Histopathology

JF - Histopathology

SN - 0309-0167

ER -