Isolated Cervical Juvenile Xanthogranuloma in Childhood

Rampini, Paolo; Alimehmeti, Ridvan; Egidi, Marcello G.; Zavanone, M.; Bauer, Dario; Fossali, Emilio F.; Villani, R.

doi:10.1097/00007632-200106150-00026

Cited by 29 publications

(38 citation statements)

References 7 publications

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“…24 Likewise, the MR signal intensity of intracranial and spinal JXG has been reported as similar to that of neural tissue with faint homogeneous enhancement after gadolinium administration. 25 The higher signal intensity on T1-weighted images compared with the cortex in many of the JXGs in our series is presumably attributable to the presence of lipid within these lesions. Lipidrich zones correspond to xanthomatized areas that are seen histologically, characterized by abundant intracellular fat within histiocytes, and they occur as a variably prominent component of JXG.…”

Section: Discussionmentioning

confidence: 56%

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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Section: Discussionmentioning

confidence: 56%

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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“…16,26,37 Among the 42 cases of isolated intracraniospinal nonLCHs (reported as JXG, fibrous xanthoma, xanthoma, fibroxanthoma, and XG) including our case (Table 1), 18 cases (43%) were dural-based lesions. 1,2,5,9,11,16,21,22,[25][26][27][28]30,32,35,37,38 Eleven of these 18 cases were intracranial 1,2,5,9,16,21,27,28,30,38 and the other 7 were spinal. 11,22,25,26,32,35,37 No cases showed recurrence during the follow-up period and essentially followed benign courses.…”

Section: Discussionmentioning

confidence: 99%

Spontaneous regression of infantile dural-based non-Langerhans cell histiocytosis after surgery: case report

Miyake¹,

Ito²,

Tanaka

et al. 2015

PED

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The authors report the case of a large left occipital mass lesion in an 8-month-old boy who presented with seizure. Neuroimaging demonstrated an approximately 5-cm extraaxial tumor, and the patient underwent partial resection. The tumor was strongly attached to the tentorium and falx. In the postoperative course the residual lesion regressed spontaneously, and after 5 years only a slight residual tumor remained along the tentorium. Histopathological examination of the tumor revealed non-Langerhans cell histiocytosis (non-LCH). However, the tumor was not diagnosed as juvenile xanthogranuloma (JXG) because it lacked Touton giant cells. Hence, the authors described this lesion as a fibroxanthogranuloma. Most intracraniospinal non-LCHs have been reported as JXG; however, several cases of xanthomatous tumors with histopathological features resembling those of JXG have been described as fibrous xanthoma, xanthoma, fibroxanthoma, and xanthogranuloma. Among JXG and the xanthomatous tumors, a review of the literature revealed several cases of dural-based tumors; these dural-based tumors have had favorable courses, including the case described in this report. In addition, the patient in the present case experienced spontaneous regression of the residual tumor. The authors report this unique case and review the literature on isolated intracraniospinal non-LCHs, especially in cases of dural-based lesion.

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“…And other reported involving organs include the oropharynx, heart, lung, liver, spleen, adrenals, muscles, subcutaneous tissues, and the central nervous system, but the involvement of spine is extremely rare [2,11,15]. We could find only four such cases reported in English [8,9,15,16] so far, and no report of a xanthogranuloma involving C1 and C2 (Table 1). The present case, to the best of our knowledge, is the first solitary JXG case involving the upper cervical spine, which originated from the C2 nerve root.…”

Section: Discussionmentioning

confidence: 86%

“…Spontaneous regression of the skin lesions is the natural course, but in cases involving the spine, there's no regression documented so far. For such an invasive benign tumor in the upper cervical spine, total removal of the tumor seems to be curative [15]. The recurrence of the tumor is unlikely to happen after the total resection, and neurological functions can be well preserved if no damage to the neural structures had been done during the tumor resection.…”

Section: Discussionmentioning

confidence: 99%

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

Cao

Yang

et al. 2008

Eur Spine J

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Solitary juvenile xanthogranuloma (JXG) in the spinal column is extremely rare and there has been no report of such a lesion involving C1 and C2 in English literature so far. Here, we report and characterize the first case of xanthogranuloma of the upper cervical spine. This case report draws attention to the fact that solitary xanthogranuloma should be considered among possible diagnoses of spinal tumor in children and young adults. An 18-year-old female patient presented to the hospital with intermittent pain in the right side of the neck. MRI studies revealed a huge soft tissue mass to the right side of the C1 and C2 vertebras, and osseous destruction can be found in the cervical spine CT scan. Complete surgical removal of the tumor and occipital-cervical instrumentation with autogenous bone graft were performed with no complications. The patient was free of pain immediately after the surgery with intact neurological functions. Follow-up MRI 6 and 12 months after the surgery showed no residue or recurrence of the tumor. Our report and the literature review indicate that isolated JXG does not show any predilections of localization inside the central nervous system. So a solitary xanthogranuloma should be considered among possible diagnoses of spinal tumor in susceptible patients. Localized JXG shows isointense signals in MRI and enhances homogeneously with gadolinium. Immunohistochemical studies can ensure the diagnosis. Whenever possible, total surgical removal alone seems to be curative.

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Isolated Cervical Juvenile Xanthogranuloma in Childhood

Cited by 29 publications

References 7 publications

Imaging Features of Juvenile Xanthogranuloma of the Pediatric Head and Neck

Imaging Features of Juvenile Xanthogranuloma of the Pediatric Head and Neck

Spontaneous regression of infantile dural-based non-Langerhans cell histiocytosis after surgery: case report

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

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