Orbital manifestations of Langerhans Cell Histiocytosis: A report of three cases

Das, Jyotirmoy; Soibam, Ronel; Tiwary, B K; Magdalene, Damaris; Paul, S B; Bhuyan, Cida

doi:10.4103/0974-620x.57315

Cited by 23 publications

(28 citation statements)

References 7 publications

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“…In cases with severe cutaneous involvement, topical nitrogen mustard may be used. 20 Treatment resistant sites may require systemic chemotherapy. In multiple organ involvement, systemic chemotherapy is indicated.…”

Section: Discussionmentioning

confidence: 99%

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Oral mucosal involvement in Langerhans’ cell histiocytosis: long‐term follow‐up of a rare case

Kılıç

Mavili

et al. 2011

Australian Dental Journal

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Langerhans' cell histiocytosis (LCH) is a rare disease where different organs and systems may be affected. Oral involvement generally consists of mucosal ulceration associated with lesions of the underlying bone. Many reports exist about the misdiagnosis of this disease. Various symptoms may lead the clinician to an incorrect diagnosis, especially with multiple organ involvement. Oral manifestations are common, and dentists should be aware of this disease and evaluate intraoral findings accordingly. This study presents an LCH case characterized by oral mucosal ulcerations with no involvement of the underlying bone. A definitive diagnosis was made by open biopsy from the oral mucosa.

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Section: Discussionmentioning

confidence: 99%

“…Low to moderate doses of methotrexate, prednisone and vinblastine have been used successfully. 20,21 This treatment scheme was used for our patient. The patient's clinical symptoms rapidly disappeared during the 35-week chemotherapy regimen, and oral lesions healed completely.…”

Section: Discussionmentioning

confidence: 99%

Oral mucosal involvement in Langerhans’ cell histiocytosis: long‐term follow‐up of a rare case

Kılıç

Mavili

et al. 2011

Australian Dental Journal

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“…18 The choice of therapeutic regime is based on disease severity and degree of systemic involvement. 66 Different treatment possibilities are shown in Table 4. In general, the diagnosis of LCH should be proven by a biopsy.…”

Section: Treatmentmentioning

confidence: 99%

Langerhans Cell Histiocytosis of the Orbit: Five Clinicopathologic Cases and Review of the Literature

Herwig

Wojno

Zhang

et al. 2013

Survey of Ophthalmology

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Langerhans cell histiocytosis (LCH) is a proliferation of Langerhans cells intermixed with inflammatory cells, in particular eosinophils, that may manifest as unisystem (unifocal or multifocal) or multisystem disease. Orbital involvement typically manifests as a solitary lesion that carries a favorable prognosis. Herein, we describe the clinical and histologic spectrum of LCH of the orbit on the basis of five cases. One patient exhibited multifocal unisystem disease, the other four patients presented with a localized process. The typical histologic features included numerous histiocytes with varying degrees of giant cell formation and scattered eosinophilic granulocytes. The presence of Langerhans cells was confirmed by CD1a and S100 immunostaining. Transmission electron microscopy exhibited characteristic intracytoplasmic Birbeck granules. The different ophthalmic manifestations of LCH and treatment strategies are reviewed in the context of previously reported cases. As LCH may solely involve the orbit, treatment is based on the degree of organ involvement. LCH has to be included in the differential diagnosis in tumors of the ocular adnexae, in particular in young children.

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“…2 LCH more commonly seen in children and rarely seen in adults. The younger patients of LCH usually have multifocal involvement with aggressive presentation as compared with patient with unifocal involvement.…”

Section: Introductionmentioning

confidence: 99%

“…The three types of LCH are known: -Eosinophilic granuloma, Hand-Schuller-Christian disease and Letterer-Siwe disease. 2 The aim of this case report is that LCH should be considered as differential diagnosis of swelling in the orbital region in children where diagnosis is uncertain.…”

Section: Introductionmentioning

confidence: 99%

Langerhans Cell Histiocytosis of Ocular Region: A Report of Two Cases

Khatri¹

2018

RAPL

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Langerhans cell histiocytosis (LCH) is a rare monoclonal disorder of dendritic cells originating from bone marrow. We are reporting two cases of isolated involvement of ocular region both of which clinically appeared as soft tissue tumour. A young girl of two and half years presented with swelling of left eyelid. Microscopic examination of biopsy showed collections of round cells which were large having abundant cytoplasm and centrally placed nuclei. Plenty of eosinophils were seen in the background. The final diagnosis of LCH involving eyelid was given after histopathological examination. Second case report is of three-year-old girl presented to ophthalmology outpatient department with the complaints of painless progressive swelling of lateral aspect of left orbital wall for four months which were rapidly increasing in size for the past two months. Fine-needle aspiration (FNA) of both swellings was done and finding was suggestive of LCH. The final diagnosis of LCH was given on histopathological examination of biopsy tissue from both the swellings. Ocular region involvement by Langerhans cell histiocytosis is a very rare presentation.

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Orbital manifestations of Langerhans Cell Histiocytosis: A report of three cases

Cited by 23 publications

References 7 publications

Oral mucosal involvement in Langerhans’ cell histiocytosis: long‐term follow‐up of a rare case

Oral mucosal involvement in Langerhans’ cell histiocytosis: long‐term follow‐up of a rare case

Langerhans Cell Histiocytosis of the Orbit: Five Clinicopathologic Cases and Review of the Literature

Langerhans Cell Histiocytosis of Ocular Region: A Report of Two Cases

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