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An unusual case of multiple primary tumours involving the long bone and oral cavity

Sekar K, Ramanathan A, Khalid R, Mun KS, Valliappan V, Ismail SM

Oral Maxillofac Surg, 2025 Mar 06;29(1):62.

PMID: 40048046 DOI: 10.1007/s10006-025-01356-0

PURPOSE OF REVIEW: Multiple primary tumours (MPTs) are a rare clinical entity, and the co-occurrence of synchronous and metachronous MPTs in a single patient is even more exceptional. In the context of Erdheim-Chester Disease (ECD), an already uncommon disorder, the presentation of this disease in the oral cavity is notably rare. Furthermore, the simultaneous presence of ECD in the oral cavity and a Giant Cell Tumour (GCT) in a long bone has not been previously reported in medical literature.

RECENT FINDINGS: This case report presents the first documented instance of such a unique clinical scenario. The marked histological disparities between GCT and ECD further underscore the enigmatic nature of this case. The intricate interplay of genetic, environmental, and pathophysiological factors that led to the simultaneous development of two distinct neoplasms in the same patient is yet to be fully elucidated. This case not only challenges our understanding of the etiology of these conditions but also emphasizes the importance of a multidisciplinary approach to the evaluation and management of such complex cases. The confluence of rare entities, the diagnostic complexities they introduce, and the imperative need for tailored treatment strategies exemplify the intricate landscape of oncological care. This case serves as a compelling reminder of the many unknown facets of disease etiology and the significance of collaborative medical efforts in offering the best possible care for patients confronting exceptionally rare clinical presentations.

Matched MeSH terms: Giant Cell Tumor of Bone/pathology

Reconstruction of large sacral defects following tumour resection: a report of two cases

Cheong YW, Sulaiman WA, Halim AS

J Orthop Surg (Hong Kong), 2008 Dec;16(3):351-4.

PMID: 19126905

Sacral tumours often present surgical resection and reconstruction challenges. Wide resections result in large sacral defects and neoadjuvant radiotherapy impairs wound healing. The wounds need to be covered with bulky, well-vascularised, healthy tissues. We present 2 cases where large sacral defects were reconstructed following tumour resection. Both defects were reconstructed with inferiorly based, transpelvic, pedicled vertical rectus abdominis myocutaneous flaps. This is a robust flap and carries a well-vascularised muscle bulk and skin paddle. The donor site is distant from the lesion site and is thus unaffected by both the resection and radiotherapy. This is a useful flap for reconstructing large sacral defects.

Matched MeSH terms: Giant Cell Tumor of Bone/pathology

Fulltext Natural history of giant cell tumour of the bone

Faisham WI, Zulmi W, Mutum SS, Shuaib IL

Singapore Med J, 2003 Jul;44(7):362-5.

PMID: 14620730

The clinical presentation and behaviour of giant cell tumours of bone vary. The progression of the disease and metastases are unpredictable, but the overall prognosis is good. We describe the natural history and different clinical presentations of two cases of giant cell tumour of bone where the patients had refused the initial treatment and presented several years later with the disease.

Matched MeSH terms: Giant Cell Tumor of Bone/pathology*

Giant cell tumour of bone with late presentation: review of treatment and outcome

Ng ES, Saw A, Sengupta S, Nazarina AR, Path M

J Orthop Surg (Hong Kong), 2002 Dec;10(2):120-8.

PMID: 12493923

To review cases of giant cell tumour of bone or osteoclastoma managed at the University Malaya Medical Center, University of Malaya, Kuala Lumpur, from January 1990 to December 1999.

Matched MeSH terms: Giant Cell Tumor of Bone/pathology

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