Rare presentation of juvenile xanthogranuloma in the thoracic spine of an adult patient: case report and literature review

Isolated spinal involvement of juvenile xanthogranuloma (JXG) is extremely rare. There are only seven prior published cases of spinal JXG, of which only one has been reported in an adult. We report here the eighth case of spinal JXG and the second in an adult. The patient, a 22-year-old female, pres...

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Bibliographic Details
Published in:Acta neurochirurgica 2011-09, Vol.153 (9), p.1813-1818
Main Authors: Jain, Ayushi, Mathur, Kusum, Khatri, Suman, Kasana, Shweta, Jain, S. K.
Format: Article
Language:English
Subjects:
Antigens, CD - metabolism
Antigens, Differentiation, Myelomonocytic - metabolism
Back Pain - etiology
Biomarkers, Tumor - metabolism
Case Report
Female
Humans
Interventional Radiology
Medicine
Medicine & Public Health
Minimally Invasive Surgery
Neurology
Neuroradiology
Neurosurgery
Neurosurgical Procedures - methods
Radiography
Spinal Neoplasms - diagnosis
Spinal Neoplasms - surgery
Surgical Orthopedics
Thoracic Vertebrae - diagnostic imaging
Thoracic Vertebrae - pathology
Thoracic Vertebrae - surgery
Treatment Outcome
Xanthogranuloma, Juvenile - diagnosis
Xanthogranuloma, Juvenile - surgery
Young Adult
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Summary:Isolated spinal involvement of juvenile xanthogranuloma (JXG) is extremely rare. There are only seven prior published cases of spinal JXG, of which only one has been reported in an adult. We report here the eighth case of spinal JXG and the second in an adult. The patient, a 22-year-old female, presented with progressive upper backache. Radiological examination revealed a well-defined osteolytic hypointense mass in the T7 vertebral body, with a large soft tissue paraspinal extension causing cord compression. Complete resection of the mass was performed, with resolution of symptoms. Histology showed a histiocytic tumour with numerous Touton, foreign body and osteoclastic giant cells, immunopositive for CD68 and vimentin and negative for S100 and CD1a, corresponding to a diagnosis of JXG. Literature regarding spinal JXG is reviewed and discussed.
ISSN:0001-6268
0942-0940
DOI:10.1007/s00701-011-1057-7