Pediatric Oral/Maxillofacial Soft Tissue Sarcomas: A Clinicopathologic Report of Four Cases

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Abstract

Pediatric soft tissue sarcomas of the oral/maxillofacial region are rare neoplasms that present significant difficulty with respect to treatment and local control measures. We report four cases of pediatric oral/maxillofacial soft tissue sarcomas from our tertiary care pediatric hospital and emphasize the rarity of these malignancies and the challenges encountered in treating these lesions, and suggest areas for further research. We conclude that multimodal therapy and interdisciplinary cooperation are paramount to successful management of these lesions.

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Most cited references 15

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Potential benefits of scanned intensity-modulated proton therapy versus advanced photon therapy with regard to sparing of the salivary glands in oropharyngeal cancer.

Fernando Vega, R. Van Der Leeuw, Johannes A Langendijk … (2011)

To test the hypothesis that scanned intensity-modulated proton therapy (IMPT) results in a significant dose reduction to the parotid and submandibular glands as compared with intensity-modulated radiotherapy with photons (IMRT) and three-dimensional conformal radiotherapy (3D-CRT) for oropharyngeal cancer. In addition, we investigated whether the achieved dose reductions would theoretically translate into a reduction of salivary dysfunction and xerostomia. Ten patients with N0 oropharyngeal carcinoma were used. The intensity-modulated plans delivered simultaneously 70 Gy to the boost planning target volume (PTV2) and 54 Gy to the elective nodal areas (PTV1). The 3D-CRT technique delivered sequentially 70 Gy and 46 Gy to PTV2 and PTV1, respectively. Normal tissue complication probabilities were calculated for salivary dysfunction and xerostomia. Planning target volume coverage results were similar for IMPT and IMRT. Intensity-modulated proton therapy clearly improved the conformity. The 3D-CRT results were inferior to these results. The mean dose to the parotid glands by 3D-CRT (50.8 Gy), IMRT (25.5 Gy), and IMPT (16.8 Gy) differed significantly. For the submandibular glands no significant differences between IMRT and IMPT were found. The dose reductions obtained with IMPT theoretically translated into a significant reduction in normal tissue complication probability. Compared with IMRT and 3D-CRT, IMPT improved sparing of the organs at risk, while keeping similar target coverage results. The dose reductions obtained with IMPT vs. IMRT and 3D-CRT varied widely per individual patient. Intensity-modulated proton therapy theoretically translated into a clinical benefit for most cases, but this requires clinical validation. Copyright © 2011 Elsevier Inc. All rights reserved.

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Proton radiation therapy for head and neck cancer: a review of the clinical experience to date.

Emma Holliday, Steven Frank (2014)

Proton beam radiation has been used for cancer treatment since the 1950s, but recent increasing interest in this form of therapy and the construction of hospital-based and clinic-based facilities for its delivery have greatly increased both the number of patients and the variety of tumors being treated with proton therapy. The mass of proton particles and their unique physical properties (ie, the Bragg peak) allow proton therapy to spare normal tissues distal to the tumor target from incidental irradiation. Initial observations show that proton therapy is particularly useful for treating tumors in challenging locations close to nontarget critical structures. Specifically, improvements in local control outcomes for patients with chordoma, chonodrosarcoma, and tumors in the sinonasal regions have been reported in series using proton. Improved local control and survival outcomes for patients with cancer of the head and neck region have also been seen with the advent of improvements in better imaging and multimodality therapy comprising surgery, radiation therapy, and chemotherapy. However, aggressive local therapy in the proximity of critical normal structures to tumors in the head and neck region may produce debilitating early and late toxic effects. Great interest has been expressed in evaluating whether proton therapy can improve outcomes, especially early and late toxicity, when used in the treatment of head and neck malignancies. This review summarizes the progress made to date in addressing this question.

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Rhabdomyosarcoma: current challenges and their implications for developing therapies.

Simone Hettmer, Zhizhong Li, Andrew Billin … (2014)

Rhabdomyosarcoma (RMS) represents a rare, heterogeneous group of mesodermal malignancies with skeletal muscle differentiation. One major subgroup of RMS tumors (so-called "fusion-positive" tumors) carries exclusive chromosomal translocations that join the DNA-binding domain of the PAX3 or PAX7 gene to the transactivation domain of the FOXO1 (previously known as FKHR) gene. Fusion-negative RMS represents a heterogeneous spectrum of tumors with frequent RAS pathway activation. Overtly metastatic disease at diagnosis is more frequently found in individuals with fusion-positive than in those with fusion-negative tumors. RMS is the most common pediatric soft-tissue sarcoma, and approximately 60% of all children and adolescents diagnosed with RMS are cured by currently available multimodal therapies. However, a curative outcome is achieved in <30% of high-risk individuals with RMS, including all those diagnosed as adults, those diagnosed with fusion-positive tumors during childhood (including metastatic and nonmetastatic tumors), and those diagnosed with metastatic disease during childhood (including fusion-positive and fusion-negative tumors). This white paper outlines current challenges in RMS research and their implications for developing more effective therapies. Urgent clinical problems include local control, systemic disease, need for improved risk stratification, and characterization of differences in disease course in children and adults. Biological challenges include definition of the cellular functions of PAX-FOXO1 fusion proteins, clarification of disease heterogeneity, elucidation of the cellular origins of RMS, delineation of the tumor microenvironment, and identification of means for rational selection and testing of new combination therapies. To streamline future therapeutic developments, it will be critical to improve access to fresh tumor tissue for research purposes, consider alternative trial designs to optimize early clinical testing of candidate drugs, coalesce advocacy efforts to garner public and industry support, and facilitate collaborative efforts between academia and industry.

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Author and article information

Journal

Journal ID (publisher-id): CRO

Journal ID (pmc): CRO

Journal ID (doi): 10.1159/issn.1662-6575

Title: Case Reports in Oncology

Publisher: S. Karger AG

ISSN (Electronic): 1662-6575

Publication date Subscription-year: 2016

Publication date Collection: May – August 2016

Publication date (Electronic): 17 August 2016

Volume: 9

Issue: 2

Pages: 447-453

Affiliations

[_a] _aDivision of Pediatric Hematology/Oncology/BMT, Nationwide Children’s Hospital, Columbus, Ohio, USA

[_b] _bDivision of Pediatric Hematology/Oncology, Children’s Hospital of the King’s Daughters, Norfolk, Virginia, USA

[_c] _cDepartment of Pathology and Laboratory Medicine, Nationwide Children’s Hospital, Columbus, Ohio, USA

[_d] _dCenter for Childhood Cancer and Blood Disorders, Nationwide Children’s Hospital, Columbus, Ohio, USA

[_e] _eDepartment of Pediatrics, The Ohio State University, Columbus, Ohio, USA

[_f] _fDepartment of Pathology, The Ohio State University, Columbus, Ohio, USA

[_g] _gDepartment of Pediatric Otolaryngology, Nationwide Children’s Hospital, Columbus, Ohio, USA

[_h] _hDepartment of Otolaryngology, The Ohio State University, Columbus, Ohio, USA

Author notes

*Bhuvana A. Setty, MD, Division of Pediatric Hematology/Oncology/BMT, Nationwide Children’s Hospital, 700 Children’s Drive, Columbus, OH 43205 (USA), E-Mail bhuvana.setty@nationwidechildrens.org

Article

Publisher ID: 447689 Pmcid ID: PMC5043172 Other ID: Case Rep Oncol 2016;9:447–453

DOI: 10.1159/000447689

PMC ID: PMC5043172

PubMed ID: 27721766

SO-VID: 3454543a-c810-46ec-b26b-fb3a1d97679d

License:

This article is licensed under the Creative Commons Attribution-NonCommercial 4.0 International License (CC BY-NC). Usage and distribution for commercial purposes requires written permission. Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug. Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

History

Date received : 15 June 2016

Date accepted : 17 June 2016

Page count

Figures: 1, Tables: 1, Pages: 7

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Pediatric Oral/Maxillofacial Soft Tissue Sarcomas: A Clinicopathologic Report of Four Cases

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Abstract

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Karger: Oncology

Most cited references 15

Potential benefits of scanned intensity-modulated proton therapy versus advanced photon therapy with regard to sparing of the salivary glands in oropharyngeal cancer.

Proton radiation therapy for head and neck cancer: a review of the clinical experience to date.

Rhabdomyosarcoma: current challenges and their implications for developing therapies.

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