Hindawi Publishing Corporation
Sarcoma
Volume 2013, Article ID 520858, 8 pages
http://dx.doi.org/10.1155/2013/520858
Research Article
MDM2 Amplification and PI3KCA Mutation in a Case of
Sclerosing Rhabdomyosarcoma
Ken Kikuchi,
1
George R. Wettach,
2
Christopher W. Ryan,
3
Arthur Hung,
4
Jody E. Hooper,
2
Carol Beadling,
2
Andrea Warrick,
2
Christopher L. Corless,
2
Susan B. Olson,
5
Charles Keller,
1
and Atiya Mansoor
2
1
Pediatric Cancer Biology Program, Department of Pediatrics, Pap´ e Family Pediatric Research Institute, Oregon Health & Science
University, 3181 S.W. Sam Jackson Park Road, Mail Code L321, Portland, OR 97239-3098, USA
2
Department of Pathology, Oregon Health & Science University, 3181 S.W. Sam Jackson Park Road, Mail Code L41,
Portland, OR 97239-3098, USA
3
Division of Hematology-Oncology, Department of Medicine, Oregon Health & Science University, Portland, OR 97239, USA
4
Department of Radiation Oncology, Oregon Health & Science University, Portland, OR 97239, USA
5
Molecular and Medical Genetics, Oregon Health & Science University, Portland, OR 97239, USA
Correspondence should be addressed to Charles Keller; keller@ohsu.edu and Atiya Mansoor; mansoora@ohsu.edu
Received 9 March 2013; Accepted 24 April 2013
Academic Editor: Cyril Fisher
Copyright © 2013 Ken Kikuchi et al. Tis is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
A rare sclerosing variant of rhabdomyosarcoma characterized by prominent hyalinization and pseudovascular pattern has recently
been described as a subtype biologically distinct from embryonal, alveolar, and pleomorphic forms. We present cytogenetic and
molecular fndings as well as experimental studies of an unusual case of sclerosing rhabdomyosarcoma. Te primary lesion arose
within the plantar subcutaneous tissue of the lef foot of an otherwise healthy 23-year-old male who eventually developed pulmonary
nodules despite systemic chemotherapy. Two genetic abnormalities identifed in surgical and/or autopsy samples of the tumor were
introduced into 10T1/2 murine fbroblasts to determine whether these genetic changes cooperatively facilitated transformation and
growth. Cytogenetic analysis revealed a complex abnormal hyperdiploid clone, and MDM2 gene amplifcation was confrmed by
fuorescence in situ hybridization. Cancer gene mutation screening using a combination of multiplexed PCR and mass spectroscopy
revealed a PIK3CA exon 20 H1047R mutation in the primary tumor, lung metastasis, and liver metastasis. However, this mutation
was not cooperative with MDM2 overexpression in experimental assays for transformation or growth. Nevertheless, MDM2 and
PIK3CA are genes worthy of further investigation in patients with sclerosing rhabdomyosarcoma and might be considered in the
enrollment of these patients into clinical trials of targeted therapeutics.
1. Introduction
Rhabdomyosarcoma (RMS) is subdivided into three major
variants: embryonal, alveolar, and pleomorphic. Embryonal
and alveolar subtypes are commonest sarcomas of childhood
and adolescence. Better clinical outcome is associated with
botryoid and spindle cell variants of embryonal RMS. In
particular, the spindle cell variant in childhood is considered
to be of low malignant potential with excellent overall patient
survival. Pleomorphic RMS is rare and highly aggressive
adult sarcomas typically arising in the deep sof tissue of
the extremities. Even rarer are recently described spindle
cell and sclerosing variants of RMS in adults. Due to their
rarity, the experience with the newer subsets is limited
but appears to show poor outcome in adults. Sclerosing
variant of RMS as a distinct entity was initially reported
in three cases by Mentzel and Katenkamp in 2000 [1].
Histologically the tumor is characterized by polygonal to
spindle-shaped neoplastic cells forming anastomosing cords
in pseudovascular clefs and a highly sclerotic, hyalinized
matrix. Rare rhabdomyoblasts can be seen and the skeletal
muscle diferentiation is evidenced by immunoreactivity for
desmin, MyoD1, and myogenin. In a subsequent series of four
additional cases, Folpe considered these tumors to be either