Presacral masses and sacrococcygeal teratomas in patients with and without anorectal malformations: A single institution comparative study.


Journal

Journal of pediatric surgery
ISSN: 1531-5037
Titre abrégé: J Pediatr Surg
Pays: United States
ID NLM: 0052631

Informations de publication

Date de publication:
Jul 2019
Historique:
received: 19 04 2018
revised: 31 10 2018
accepted: 27 11 2018
pubmed: 12 1 2019
medline: 14 11 2019
entrez: 12 1 2019
Statut: ppublish

Résumé

Despite variability at presentation, presacral masses in patients with and without anorectal malformations (ARM) appear histologically similar. The purpose of this study was to identify differences in oncologic outcomes between these two groups. A retrospective review was performed utilizing our institutional cancer and colorectal and pelvic reconstruction databases for patients with presacral masses and sacrococcygeal teratomas between 1990 and 2017. Data captured included age at surgical resection, type of ARM, tumor location within the pelvis, tumor histopathology, tumor size, adjuvant chemotherapy, recurrence, and follow-up. Forty-six patients comprised our cohort, of whom 12 had an ARM. The median age was older at resection for those with an ARM (1.4 years; range 1 day to 29.4 years) compared to those without an ARM (9 days; range 0 days to 6.9 years) (p = 0.01). The mean tumor size was 2.5 cm in patients with an ARM compared to 6.0 cm in patients without an ARM (p = 0.036). All patients with ARM had exclusively intrapelvic tumors, and histopathology included mature teratoma (8), yolk sac tumor (1), lipoma (1), and unknown (2). Tumor location for patients with sacral and presacral masses without ARM included exclusively extrapelvic (10), primarily extrapelvic with large intrapelvic component (7), primarily intrapelvic with extrapelvic component (1), exclusively intrapelvic (8), and unknown (8). Histopathology for patients with presacral masses without ARM included mature teratoma (20), immature teratoma (7), yolk sac tumor (3), ganglioneuroma (1), neuroblastoma (1), benign epithelial cyst (1), and unknown (1). Tumor recurrence rate was similar between patients with ARM (n = 3, 25%) and those without an ARM (n = 5, 15%) (p = 0.41). The 5-year event free survival was 65% (95% CI: 25%-87%) in the group with ARM and 81% (95% CI: 60%-92%) in the group without ARM (p = 0.44). Sacral and presacral masses in patients with ARM are resected at a later age and are more likely to be intrapelvic. They appear histologically similar and have similar rates of recurrence and malignancy when compared to patients without ARM. III TYPE OF STUDY: Retrospective comparative study.

Sections du résumé

BACKGROUND BACKGROUND
Despite variability at presentation, presacral masses in patients with and without anorectal malformations (ARM) appear histologically similar. The purpose of this study was to identify differences in oncologic outcomes between these two groups.
METHODS METHODS
A retrospective review was performed utilizing our institutional cancer and colorectal and pelvic reconstruction databases for patients with presacral masses and sacrococcygeal teratomas between 1990 and 2017. Data captured included age at surgical resection, type of ARM, tumor location within the pelvis, tumor histopathology, tumor size, adjuvant chemotherapy, recurrence, and follow-up.
RESULTS RESULTS
Forty-six patients comprised our cohort, of whom 12 had an ARM. The median age was older at resection for those with an ARM (1.4 years; range 1 day to 29.4 years) compared to those without an ARM (9 days; range 0 days to 6.9 years) (p = 0.01). The mean tumor size was 2.5 cm in patients with an ARM compared to 6.0 cm in patients without an ARM (p = 0.036). All patients with ARM had exclusively intrapelvic tumors, and histopathology included mature teratoma (8), yolk sac tumor (1), lipoma (1), and unknown (2). Tumor location for patients with sacral and presacral masses without ARM included exclusively extrapelvic (10), primarily extrapelvic with large intrapelvic component (7), primarily intrapelvic with extrapelvic component (1), exclusively intrapelvic (8), and unknown (8). Histopathology for patients with presacral masses without ARM included mature teratoma (20), immature teratoma (7), yolk sac tumor (3), ganglioneuroma (1), neuroblastoma (1), benign epithelial cyst (1), and unknown (1). Tumor recurrence rate was similar between patients with ARM (n = 3, 25%) and those without an ARM (n = 5, 15%) (p = 0.41). The 5-year event free survival was 65% (95% CI: 25%-87%) in the group with ARM and 81% (95% CI: 60%-92%) in the group without ARM (p = 0.44).
CONCLUSION CONCLUSIONS
Sacral and presacral masses in patients with ARM are resected at a later age and are more likely to be intrapelvic. They appear histologically similar and have similar rates of recurrence and malignancy when compared to patients without ARM.
LEVEL OF EVIDENCE METHODS
III TYPE OF STUDY: Retrospective comparative study.

Identifiants

pubmed: 30630596
pii: S0022-3468(18)30782-6
doi: 10.1016/j.jpedsurg.2018.11.009
pii:
doi:

Types de publication

Comparative Study Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

1372-1378

Informations de copyright

Copyright © 2018 Elsevier Inc. All rights reserved.

Auteurs

Devin R Halleran (DR)

Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, OH.

Alejandra Vilanova-Sanchez (A)

Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, OH.

Carlos A Reck (CA)

Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, OH.

Tassiana Maloof (T)

Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, OH.

Laura Weaver (L)

Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, OH.

Joseph Stanek (J)

Department of Biostatistics, Division of Hematology/Oncology/Bone Marrow Transplantation, Nationwide Children's Hospital, Columbus, OH.

Marc A Levitt (MA)

Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, OH.

Richard J Wood (RJ)

Center for Colorectal and Pelvic Reconstruction, Nationwide Children's Hospital, Columbus, OH.

Jennifer H Aldrink (JH)

Department of Surgery, Division of Pediatric Surgery, Nationwide Children's Hospital, The Ohio State University College of Medicine, Columbus, OH. Electronic address: jennifer.aldrink@nationwidechildrens.org.

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