Solitary juvenile xanthogranuloma in the upper cervical spine: case report and review of the literatures

Cao, Dong; Ma, Jinghong; Yang, Xinghai; Xiao, Jianru

doi:10.1007/s00586-008-0606-0

Cited by 28 publications

(44 citation statements)

References 17 publications

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“…2 Extracutaneous HN JXG has been reported in locations such as the tympanic membrane, nasal cavity, tongue, orbit and paranasal sinus, subglottis, cervical spine, skull base including the temporal bone, intracranial compartment, and HN muscles. [2][3][4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19][20][21] Cutaneous JXG typically presents as a solitary macule or papule with a flesh-colored, erythematous, or yellowish hue. 2 In such cases, radiographic imaging is not indicated.…”

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confidence: 99%

Imaging Features of Juvenile Xanthogranuloma of the Pediatric Head and Neck

Ginat

Vargas

Silvera

et al. 2016

American Journal of Neuroradiology

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Imaging Features of Juvenile Xanthogranuloma of the Pediatric Head and Neck

Ginat

Vargas

Silvera

et al. 2016

American Journal of Neuroradiology

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“…In case of presentation as IDEM tumors, resection of the tumor appears to be curative (14,19,20). However, fusion may be required following radical excision for lesions having extensive bony involvement (3,12). Immunohistochemistry shows the lesions to be positive for factor XIIIa, CD68, CD163, fascin, and CD14 but are negative for S100 and CD1a (12,16,17).…”

Section: Resultsmentioning

confidence: 99%

“…Orbital and skull base involvement is common, though xanthogranuloma in a colloid cyst has been described before (13). Isolated spinal involvement is extremely rare, and to the best of our knowledge, there are only 11 reported cases in the English literature, and third in the subaxial cervical spine (1,3,5,6,8,14,15,(18)(19)(20).…”

Section: Konar S Et Al: Solitary Juvenile Xanthogranuloma In Cervicamentioning

confidence: 99%

“…Pathologically, it is a proliferative disorder of dendritic cell, non-Langerhans histiocytes, and is on a spectrum of histiocytic disorders that includes benign cephalic histiocytosis, generalized eruptive histiocytosis, adult xanthogranuloma, and progressive nodular histiocytosis (7). JXG is rarely associated with systemic manifestations, and isolated involvement of the sub-axial cervical spine is extremely rare (3,8,18,19). Only 11 previous cases have been described of isolated involvement of spine with JXG.…”

Section: Introductionmentioning

confidence: 99%

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Solitary juvenile xanthogranuloma in cervical spine: case report and review of literature

Konar

Pandey²,

Yasha³

2013

Turkish Neurosurgery

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Solitary juvenile xanthogranuloma (JXG) in the spinal column is extremely rare. Here, we report and characterize the case of xanthogranuloma of the upper cervical spine. A 18-year-old male presented with neck pain for 3 months, along with progressive quadriparesis and sensory loss of 2 months duration with urinary retention. Motor examination revealed spastic quadriparesis with power of 2/5 in all the 4 limbs. Magnetic Resonance Imaging (MRI) spine with contrast showed a dorsally placed intradural extramedullary lesion at the level of C2-C4 vertebral body. The lesion, measuring 2.9x1.7x1.4 cm, was isointense on T1WI, hypointense on T2WI, and enhanced homogenously on contrast. He underwent an emergency C2-C4 laminectomy and complete excision of the lesion. At 3-month follow-up, he was asymptomatic except for mild neck pain. MRI scan of the cervical spine done at follow-up, revealed complete excision of tumor without any residual lesion. Histopathological examination of the mass revealed a polymorphous population of sheets of bloated pale foamy histiocytes (xanthoma cells), numerous admixed mature lymphocytes and several Touton giant cells. The cells were positive for CD68, a histiocytic marker, and negative for CD1a (excludes LCH) and S-100 (excludes RDD). KeywOrds: Juvenile xanthogranuloma, Cervical spine, MRI, Complete resection ÖZOmurgada soliter jüvenil ksantogranülom çok nadirdir. Burada üst servikal omurgada bir ksantogranülom olgusunu bildiriyoruz. 18 yaşında bir erkek 3 aydır boyun ağrısı, 2 aydır progresif kuadriparezi ve duyu kaybı ile üriner retansiyonla geldi. Motor inceleme 4 uzuvda 2/5 güçle spastik kuadriparezi gösterdi. Omurganın kontrastlı Manyetik Rezonans Görüntülemesi (MRG) C2-C4 vertebral cisim seviyesinde dorsal yerleşimli bir intradural ekstramedüller lezyon ortaya koydu. 2,9x1,7x1,4 cm boyutlarında olan lezyon T1WI ile izointens, T2WI ile hipointens ve kontrast ile homojen tutulum göstermekteydi. Acil C2-C4 laminektomi ve lezyonun tam eksizyonu yapıldı. 3 aylık takipte hafif boyun ağrısı dışında semptom yoktu. Takipte yapılan servikal omurga MRG taraması herhangi bir rezidüel lezyon olmadan tümörün tam eksizyonunu gösterdi. Kitlenin histopatolojik incelemesi şiş ve soluk köpüklü histiosit tabakalarının (ksantoma hücreleri) polimorfik bir popülasyonunu, araya karışmış çok sayıda matür lenfositi ve birkaç Touton dev hücresini gösterdi. Hücreler bir histiositik belirteç olan CD68 için pozitifti ve CD1a (LCH ekarte eder) ve S-100 (RDD ekarte eder) için negatifti.

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“…Exceptionally, cardiac, retroperitoneal, gonadal, adrenal gland, and skeletal examples have been reported 3 . While vastly outnumbered by cutaneous examples, occasional JXGs have been reported involving all levels of the neuroaxis, including the central and peripheral nervous system (PNS) and their enveloping meninges 2,4–37 . Clinically, JXGs are infrequently included in the differential diagnosis of lesions of the nervous system, particularly in the absence of pathologically confirmed cutaneous lesions.…”

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Juvenile xanthogranulomas of the nervous system: A report of two cases and review of the literature

Deisch

Patel²,

Koral³

et al. 2012

Neuropathology

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Juvenile xanthogranulomas (JXG) are uncommon non-Langerhans cell histiocytic proliferations which arise most often in children. While most cases present as solitary cutaneous lesions, occasional cases involve extracutaneous sites. Rare examples of JXGs have been reported involving all levels of the neuroaxis. We present two cases of JXGs involving the nervous system, and review the literature. The first patient was a 14-year-old female with headaches and a mass involving the left trigeminal nerve; pathologic examination showed a JXG. At 11 months follow-up, after administration of systemic chemotherapy, the patient remained stable with residual tumor. The second patient was a 15-year-old female with leg weakness and numbness, who underwent complete surgical resection of a dural JXG. At eight months follow-up, she showed no evidence of tumor, and was able to walk without difficulty. Review of the literature revealed 38 previously published reports of JXGs involving the nervous system. The CNS was involved in the majority (75%) of cases. The clinical characteristics of JXGs arising in the CNS varied significantly from cases in the peripheral nervous system (PNS); CNS tumors occurred in younger patients, more often males, and were more likely to be associated with concurrent cutaneous and extra-nervous systemic lesions. The clinical outcomes were similar for CNS and PNS lesions, with the caveat that all three lethal JXGs occurred in the CNS. The clinical and radiologic presentation of JXGs is nonspecific, thus necessitating biopsy and pathologic examination to arrive at the diagnosis. The pathologic differential diagnosis includes a heterogeneous group of histiocytic proliferations; immunostaining for histiocytic markers CD68, factor XIIIa, and Fascin, and the absence of Birbeck granules and CD1a immunoexpression suggests the diagnosis of JXG. In many cases, total surgical resection is curative. However, some cases will require additional chemotherapy and/or radiotherapy.

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Solitary juvenile xanthogranuloma in the upper cervical spine: case report and review of the literatures

Cited by 28 publications

References 17 publications

Imaging Features of Juvenile Xanthogranuloma of the Pediatric Head and Neck

Imaging Features of Juvenile Xanthogranuloma of the Pediatric Head and Neck

Solitary juvenile xanthogranuloma in cervical spine: case report and review of literature

Juvenile xanthogranulomas of the nervous system: A report of two cases and review of the literature

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