Juvenile Xanthogranuloma with Ocular Involvement

Liang, Sai; Liu, Yuehua; Fang, Kai

doi:10.1111/j.1525-1470.2009.00893.x

Cited by 26 publications

(20 citation statements)

References 5 publications

(5 reference statements)

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“…Our case presented with a large mass-like lesion in the left neck, ptosis and leaking amber-coloured tears, which might be a rare manifestation. In reported cases, eyes were the most common site of extracutaneous involvement and almost 50% of the patients with ocular involvement also suffered from skin lesions, in comparison with our patient who did not have any cutaneous lesions 3 6. Involvement of the iris may lead to heterochromia, uveitis, spontaneous hyphema and secondary glaucoma, whereas our presented case did not have these symptoms.…”

Section: Discussioncontrasting

confidence: 44%

“…Only a few literature reviews have been published about the pathogenesis, prognosis and treatment guidelines of this condition 1. The eye, especially the uveal tract, is the most frequent site of involvement in an extracutaneous manifestation 3 5 6. Cutaneous JXG is seen 1.5 times more frequently in male children than in female children, but the extracutaneous predilection rate has not been reported 9.…”

Section: Introductionmentioning

confidence: 99%

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Juvenile xanthogranuloma presenting as a large neck mass and ocular complications: a diagnostic and therapeutic dilemma

Zahir

Sharahjin²,

Vahedian

et al. 2014

BMJ Case Reports

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Juvenile xanthogranuloma is a non-Langerhans cell histiocytosis that originates from dendritic cells. Dendritic cells can accumulate in the skin of the head, neck, trunk, arms and legs. They may also involve other tissues such as the bones, lung, liver, heart, bone marrow, central nervous system, spleen and large intestine in rare cases. We report a rare case of juvenile xanthogranuloma in a 16-year-old girl who presented with a neck mass and left-sided ptosis 2.5 months previously. Excisional biopsy of the neck lesion revealed proliferated histiocytes admixed with numerous eosinophils and multinucleated giant cells that simulate eosinophilic granuloma; however, the histiocytes were negative for CD1a, CD123 and S-100 protein and positive for CD68 and CD14. The course of the disease led to treatment of the patient with chemotherapy, followed by low-dose radiotherapy.

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Section: Discussioncontrasting

confidence: 44%

Section: Introductionmentioning

confidence: 99%

Juvenile xanthogranuloma presenting as a large neck mass and ocular complications: a diagnostic and therapeutic dilemma

Zahir

Sharahjin²,

Vahedian

et al. 2014

BMJ Case Reports

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“…Juvenile xanthogranuloma (JXG) is a self-limiting dermatological condition characterised by spontaneous hyphaema due to uveal involvement causing neovascularisation of iris (NVI) rarely 1 2. Neovascular glaucoma can cause intractable glaucoma as seen in our case necessitating surgery 3…”

Section: Discussionmentioning

confidence: 78%

The child with spontaneous recurrent bleeding in the eye

Rao¹,

Padhy²

2014

BMJ Case Reports

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Juvenile xanthogranuloma is a self-limiting dermatological condition characterised by spontaneous hyphaema due to uveal involvement causing neovascularisation. Recurrent hyphaema in a child should alert the clinician towards this rare diagnosis. Appropriate investigations and treatment should be directed towards treating this possible diagnosis associated with neovascular glaucoma where routine filtering surgeries may be associated with several complications.

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“…19 Extracutaneous and/or organ involvement is rare and, when seen, most commonly involves the orbit. 19,20 Brain parenchymal lesions have been reported with juvenile xanthogranuloma. 19 Skin lesions generally follow a benign course, with spontaneous resolution.…”

Section: Types Of Histiocytosesmentioning

confidence: 99%

“…21,22 Fair-to-good response with steroids, surgery, radiation, and chemotherapy has been reported. 20,21 …”

Section: Types Of Histiocytosesmentioning

confidence: 99%

Central Nervous System and Head and Neck Histiocytoses: A Comprehensive Review on the Spectrum of Imaging Findings

Hashmi¹,

Guha-Thakurta²,

Ketonen³

et al. 2016

neurograph

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The histiocytoses are a rare group of varied but related disorders characterized by abnormal tissue proliferation of macrophages and dendritic cells within tissues. The purpose of this article was to review the imaging findings in patients presenting with CNS and with head and neck manifestations of these disorders. Histiocytoses include but are not limited to Rosai-Dorfman disease, Erdheim Chester disease, Langerhans cell histiocytosis, histiocytic sarcoma, and juvenile xanthogranuloma. A review of the literature was performed to determine the sites of disease involvement. This article includes the demographics, histopathologic criteria for diagnosis, and imaging features of these histiocytoses, and describes the manifestations in locations known to harbor disease: intraaxial and extra-axial intracranial regions, the calvaria, skull base, hypothalamopituitary axis, orbits, paranasal sinuses, spine, and the head and neck region. Histiocytoses have variable imaging appearances in the CNS and in the head and neck region, and radiologists should be aware of the spectrum of findings to avoid mistaking them for other disease processes. Learning Objective To understand the general pathophysiology, clinical presentation, and typical imaging characteristics of the most common histiocytoses; comprehend the morphologic and immunohistochemical characteristics of these histiocytoses and the hallmark findings on pathology; and be able to differentiate between these disorders based on their most common presentations.

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Juvenile Xanthogranuloma with Ocular Involvement

Cited by 26 publications

References 5 publications

Juvenile xanthogranuloma presenting as a large neck mass and ocular complications: a diagnostic and therapeutic dilemma

Juvenile xanthogranuloma presenting as a large neck mass and ocular complications: a diagnostic and therapeutic dilemma

The child with spontaneous recurrent bleeding in the eye

Central Nervous System and Head and Neck Histiocytoses: A Comprehensive Review on the Spectrum of Imaging Findings

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