Primary desmoplastic small round cell tumor of the femur

被引:10
作者
Yoshida, Akihiko [2 ]
Edgar, Mark A. [2 ,3 ]
Garcia, Joaquin [2 ]
Meyers, Paul A. [3 ,4 ]
Morris, Carol D. [3 ,5 ]
Panicek, David M. [1 ,3 ]
机构
[1] Mem Sloan Kettering Canc Ctr, Dept Radiol, New York, NY 10021 USA
[2] Mem Sloan Kettering Canc Ctr, Dept Pathol, New York, NY 10021 USA
[3] Cornell Univ, Weill Med Coll, New York, NY 10021 USA
[4] Mem Sloan Kettering Canc Ctr, Dept Pediat, New York, NY 10021 USA
[5] Mem Sloan Kettering Canc Ctr, Orthopaed Serv, Dept Surg, New York, NY 10021 USA
关键词
desmoplastic small round cell tumor; bone neoplasms; EWS-WT1 fusion protein;
D O I
10.1007/s00256-008-0501-0
中图分类号
R826.8 [整形外科学]; R782.2 [口腔颌面部整形外科学]; R726.2 [小儿整形外科学]; R62 [整形外科学(修复外科学)];
学科分类号
摘要
Desmoplastic small round cell tumor (DSRCT) is a rare malignant neoplasm typically involving the abdominal cavity of a young male. Extra-abdominal occurrence of this tumor is very rare. We report a 10-year-old girl with primary DSRCT arising within the left femur. The patient presented with knee pain, and radiological findings were strongly suggestive of osteogenic sarcoma. In addition to the typical microscopic appearance and immunophenotype, RT-PCR demonstrated the chimeric transcript of EWS-WT1, which is diagnostic of DSRCT. Pulmonary metastases were present at initial staging studies, but no abdominal or pelvic lesion was present. Despite chemotherapy and complete tumor excision, the patient developed progressive lung and bone metastases and died 3 years after initial presentation. This is the second reported case of primary DSRCT of bone with genetic confirmation.
引用
收藏
页码:857 / 862
页数:6
相关论文
共 11 条
[1]   Primary desmoplastic small cell tumor of soft tissues and bone of the hand [J].
Adsay, V ;
Cheng, J ;
Athanasian, E ;
Gerald, W ;
Rosai, J .
AMERICAN JOURNAL OF SURGICAL PATHOLOGY, 1999, 23 (11) :1408-1413
[2]   Spindle cell tumor with EWS-WT1 transcript and a favorable clinical course: A variant of DSCT, a variant of leiomyosarcoma, or a new entity? Report of 2 pediatric cases [J].
Alaggio, Rita ;
Rosolen, Angelo ;
Sartori, Francesca ;
Leszl, Anna ;
d'Amore, Emanuele S. G. ;
Bisogno, Gianni ;
Carli, Modesto ;
Cecchetto, Giovanni ;
Coffin, Cheryl M. ;
Ninfo, Vito .
AMERICAN JOURNAL OF SURGICAL PATHOLOGY, 2007, 31 (03) :454-459
[3]   Adamantinoma-like Ewing's sarcoma: Genomic confirmation, phenotypic drift [J].
Bridge, JA ;
Fidler, ME ;
Neff, JR ;
Degenhardt, J ;
Wang, M ;
Walker, C ;
Dorfman, HD ;
Baker, KS ;
Seemayer, TA .
AMERICAN JOURNAL OF SURGICAL PATHOLOGY, 1999, 23 (02) :159-165
[4]  
GERALD W L, 1989, Pediatric Pathology, V9, P177
[5]   Clinical, pathologic, and molecular spectrum of tumors associated with t(11;22)(p13;q12): Desmoplastic small round-cell tumor and its variants [J].
Gerald, WL ;
Ladanyi, M ;
de Alava, E ;
Cuatrecasas, M ;
Kushner, BH ;
LaQuaglia, MP ;
Rosai, J .
JOURNAL OF CLINICAL ONCOLOGY, 1998, 16 (09) :3028-3036
[6]   INTRAABDOMINAL DESMOPLASTIC SMALL ROUND-CELL TUMOR - REPORT OF 19 CASES OF A DISTINCTIVE TYPE OF HIGH-GRADE POLYPHENOTYPIC MALIGNANCY AFFECTING YOUNG INDIVIDUALS [J].
GERALD, WL ;
MILLER, HK ;
BATTIFORA, H ;
MIETTINEN, M ;
SILVA, EG ;
ROSAI, J .
AMERICAN JOURNAL OF SURGICAL PATHOLOGY, 1991, 15 (06) :499-513
[7]   Long-term event-free survival after intensive chemotherapy for Ewing's family of tumors in children and young adults [J].
Kolb, EA ;
Kushner, BH ;
Gorlick, R ;
Laverdiere, C ;
Healey, JH ;
LaQuaglia, MP ;
Huvos, AG ;
Qin, J ;
Vu, HT ;
Wexler, L ;
Walden, S ;
Meyers, PA .
JOURNAL OF CLINICAL ONCOLOGY, 2003, 21 (18) :3423-3430
[8]   Contribution of molecular genetic data to the classification of sarcomas [J].
Ladanyi, M ;
Bridge, JA .
HUMAN PATHOLOGY, 2000, 31 (05) :532-538
[9]  
LADANYI M, 1994, CANCER RES, V54, P2837
[10]   Desmoplastic small round cell tumor - A clinicopathologic, immunohistochemical, and molecular study of 32 tumors [J].
Lae, ME ;
Roche, PC ;
Jin, L ;
Lloyd, RV ;
Nascimento, AG .
AMERICAN JOURNAL OF SURGICAL PATHOLOGY, 2002, 26 (07) :823-835