Primary intraosseous myoepithelioma arising in the iliac bone and displaying trisomies of 11, 15, 17 with del (16q) and del (22q11)-A rare case report with review of literature

Bharat Rekhi, Pratibha Amare, Ashish Gulia, Chanda Baisane, Asawari Patil, Shweta Agarwal, Ajay Puri, Nirmala A. Jambhekar

Research output: Contribution to journalArticle

13 Citations (Scopus)

Abstract

Mixed tumors are uncommonly observed in the musculoskeletal system, where they form a common spectrum with a myoepithelioma and a parachordoma. Herein, we present a rare case of a mixed tumor/myoepithelioma arising in the iliac bone of a young adult male who presented with swelling in his right hip. Radiological imaging disclosed a large, intraosseous, lytic, heterogenous mass with a soft tissue component. Biopsy and subsequent tumor resection showed an 18. cm sized tumor involving the iliac bone and soft tissues and comprising polygonal and spindly cells, arranged in cords and aggregates, embedded in a myxohyaline stroma with osteochondroid differentiation. Tumor cells exhibited mild nuclear variation, rare mitotic figures, focal cytoplasmic clearing, and prominent squamous differentiation. On immunohistochemistry (IHC), tumor cells were diffusely positive for S100-P, EMA, CK5/6, p63, GFAP, calponin, and focally positive for CK/MNF116, but negative for Brachyury/T. Diagnosis of a myoepithelioma/mixed tumor was offered. Further, cytogenetic analysis revealed lack of EWSR1 gene rearrangement and showed clonal trisomies of 11, 15, 17 with del (16q) and del (22q11). The present case is a rare documentation of a myoepithelioma in the appendicular bones and the second such case identified in the iliac bone. IHC and cytogenetic findings supported a myoepithelial cell origin, and reinforced its relationship with a parachordoma and its distinction from mixed salivary gland tumors, a chordoma, and an extraskeletal myxoid chondrosarcoma that form its differential diagnoses.

Original languageEnglish (US)
Pages (from-to)780-785
Number of pages6
JournalPathology Research and Practice
Volume207
Issue number12
DOIs
StatePublished - Dec 15 2011

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Myoepithelioma
Trisomy
Bone and Bones
Neoplasms
Immunohistochemistry
Chordoma
Pleomorphic Adenoma
Musculoskeletal System
Gene Rearrangement
Cytogenetic Analysis
Cytogenetics
Documentation
Hip
Young Adult
Differential Diagnosis
Biopsy

All Science Journal Classification (ASJC) codes

  • Pathology and Forensic Medicine
  • Cell Biology

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Primary intraosseous myoepithelioma arising in the iliac bone and displaying trisomies of 11, 15, 17 with del (16q) and del (22q11)-A rare case report with review of literature. / Rekhi, Bharat; Amare, Pratibha; Gulia, Ashish; Baisane, Chanda; Patil, Asawari; Agarwal, Shweta; Puri, Ajay; Jambhekar, Nirmala A.

In: Pathology Research and Practice, Vol. 207, No. 12, 15.12.2011, p. 780-785.

Research output: Contribution to journalArticle

Rekhi, Bharat ; Amare, Pratibha ; Gulia, Ashish ; Baisane, Chanda ; Patil, Asawari ; Agarwal, Shweta ; Puri, Ajay ; Jambhekar, Nirmala A. / Primary intraosseous myoepithelioma arising in the iliac bone and displaying trisomies of 11, 15, 17 with del (16q) and del (22q11)-A rare case report with review of literature. In: Pathology Research and Practice. 2011 ; Vol. 207, No. 12. pp. 780-785.
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AU - Amare, Pratibha

AU - Gulia, Ashish

AU - Baisane, Chanda

AU - Patil, Asawari

AU - Agarwal, Shweta

AU - Puri, Ajay

AU - Jambhekar, Nirmala A.

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AB - Mixed tumors are uncommonly observed in the musculoskeletal system, where they form a common spectrum with a myoepithelioma and a parachordoma. Herein, we present a rare case of a mixed tumor/myoepithelioma arising in the iliac bone of a young adult male who presented with swelling in his right hip. Radiological imaging disclosed a large, intraosseous, lytic, heterogenous mass with a soft tissue component. Biopsy and subsequent tumor resection showed an 18. cm sized tumor involving the iliac bone and soft tissues and comprising polygonal and spindly cells, arranged in cords and aggregates, embedded in a myxohyaline stroma with osteochondroid differentiation. Tumor cells exhibited mild nuclear variation, rare mitotic figures, focal cytoplasmic clearing, and prominent squamous differentiation. On immunohistochemistry (IHC), tumor cells were diffusely positive for S100-P, EMA, CK5/6, p63, GFAP, calponin, and focally positive for CK/MNF116, but negative for Brachyury/T. Diagnosis of a myoepithelioma/mixed tumor was offered. Further, cytogenetic analysis revealed lack of EWSR1 gene rearrangement and showed clonal trisomies of 11, 15, 17 with del (16q) and del (22q11). The present case is a rare documentation of a myoepithelioma in the appendicular bones and the second such case identified in the iliac bone. IHC and cytogenetic findings supported a myoepithelial cell origin, and reinforced its relationship with a parachordoma and its distinction from mixed salivary gland tumors, a chordoma, and an extraskeletal myxoid chondrosarcoma that form its differential diagnoses.

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