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Role of Denosumab in Endoscopic Endonasal Treatment for Juvenile Clival Giant Cell Tumor: A Case Report and Review of the Literature.
World Neurosurg. 2016 Jul; 91:674.e1-6.WN

Abstract

BACKGROUND

Giant cell tumors (GCTs) are histologically benign bone neoplasms with a locally aggressive nature that primarily occur in the epiphyses of the long bones. A small proportion of these tumors, however, occur in the pelvis, spine, or skull bones. Among these, GCTs of the skull base cannot be completely resected and require adjuvant therapy. We report a juvenile case of clival GCT that was successfully treated by endoscopic endonasal transsphenoidal surgery and subsequent adjuvant therapy with denosumab, a monoclonal antibody to receptor activator of nuclear factor-κB ligand.

CASE DESCRIPTION

A 16-year-old boy was admitted to our hospital with progressively intolerable headache and right oculomotor nerve palsy. Computed tomography and magnetic resonance imaging showed a large tumor mass in the sphenoid sinus with extensive erosion of the clivus and compression of the right cavernous sinus. The tumor was resected by endonasal transsphenoidal surgery and histologically diagnosed as GCT. The giant cells showed positive immunostaining for CD68 and Mib-1 labeling index was less than 1.0%. Postoperative course was uneventful and the oculomotor disturbance was markedly improved. However, magnetic resonance imaging 2 weeks after surgery revealed marked enlargement of the tumor. Adjuvant therapy with denosumab was therefore initiated, resulting in marked reductions in tumor size.

CONCLUSIONS

This is the first report to describe beneficial effects of denosumab in the treatment of GCT of the skull base.

Authors+Show Affiliations

Department of Neurosurgery, Ehime University School of Medicine, Ehime, Japan. Electronic address: iakihiro3@gmail.com.Department of Neurosurgery, Washoukai Sadamoto Hospital, Ehime, Japan.Department of Neurosurgery, Ehime University School of Medicine, Ehime, Japan.Department of Neurosurgery, Ehime University School of Medicine, Ehime, Japan.Department of Otolaryngology, Ehime University School of Medicine, Ehime, Japan.Department of Neurosurgery, Ehime University School of Medicine, Ehime, Japan.

Pub Type(s)

Case Reports
Journal Article
Review

Language

eng

PubMed ID

27113403

Citation

Inoue, Akihiro, et al. "Role of Denosumab in Endoscopic Endonasal Treatment for Juvenile Clival Giant Cell Tumor: a Case Report and Review of the Literature." World Neurosurgery, vol. 91, 2016, pp. 674.e1-6.
Inoue A, Ohnishi T, Kohno S, et al. Role of Denosumab in Endoscopic Endonasal Treatment for Juvenile Clival Giant Cell Tumor: A Case Report and Review of the Literature. World Neurosurg. 2016;91:674.e1-6.
Inoue, A., Ohnishi, T., Kohno, S., Nishikawa, M., Nishida, N., & Ohue, S. (2016). Role of Denosumab in Endoscopic Endonasal Treatment for Juvenile Clival Giant Cell Tumor: A Case Report and Review of the Literature. World Neurosurgery, 91, e1-6. https://doi.org/10.1016/j.wneu.2016.04.054
Inoue A, et al. Role of Denosumab in Endoscopic Endonasal Treatment for Juvenile Clival Giant Cell Tumor: a Case Report and Review of the Literature. World Neurosurg. 2016;91:674.e1-6. PubMed PMID: 27113403.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Role of Denosumab in Endoscopic Endonasal Treatment for Juvenile Clival Giant Cell Tumor: A Case Report and Review of the Literature. AU - Inoue,Akihiro, AU - Ohnishi,Takanori, AU - Kohno,Shohei, AU - Nishikawa,Masahiro, AU - Nishida,Naoya, AU - Ohue,Shiro, Y1 - 2016/04/23/ PY - 2016/03/16/received PY - 2016/04/14/revised PY - 2016/04/15/accepted PY - 2016/4/27/entrez PY - 2016/4/27/pubmed PY - 2017/9/29/medline KW - Clivus KW - Denosumab KW - Endoscopic endonasal transsphenoidal surgery KW - Giant cell tumor SP - 674.e1 EP - 6 JF - World neurosurgery JO - World Neurosurg VL - 91 N2 - BACKGROUND: Giant cell tumors (GCTs) are histologically benign bone neoplasms with a locally aggressive nature that primarily occur in the epiphyses of the long bones. A small proportion of these tumors, however, occur in the pelvis, spine, or skull bones. Among these, GCTs of the skull base cannot be completely resected and require adjuvant therapy. We report a juvenile case of clival GCT that was successfully treated by endoscopic endonasal transsphenoidal surgery and subsequent adjuvant therapy with denosumab, a monoclonal antibody to receptor activator of nuclear factor-κB ligand. CASE DESCRIPTION: A 16-year-old boy was admitted to our hospital with progressively intolerable headache and right oculomotor nerve palsy. Computed tomography and magnetic resonance imaging showed a large tumor mass in the sphenoid sinus with extensive erosion of the clivus and compression of the right cavernous sinus. The tumor was resected by endonasal transsphenoidal surgery and histologically diagnosed as GCT. The giant cells showed positive immunostaining for CD68 and Mib-1 labeling index was less than 1.0%. Postoperative course was uneventful and the oculomotor disturbance was markedly improved. However, magnetic resonance imaging 2 weeks after surgery revealed marked enlargement of the tumor. Adjuvant therapy with denosumab was therefore initiated, resulting in marked reductions in tumor size. CONCLUSIONS: This is the first report to describe beneficial effects of denosumab in the treatment of GCT of the skull base. SN - 1878-8769 UR - https://www.unboundmedicine.com/medline/citation/27113403/Role_of_Denosumab_in_Endoscopic_Endonasal_Treatment_for_Juvenile_Clival_Giant_Cell_Tumor:_A_Case_Report_and_Review_of_the_Literature_ DB - PRIME DP - Unbound Medicine ER -