Journal of Pathology and Translational Medicine

Original Article

A study of pathological characteristics and BRAF V600E status in Langerhans cell histiocytosis of Vietnamese children: Thu Dang Anh Phan, Bao Gia Phung, Tu Thanh Duong, Vu Anh Hoang, Dat Quoc Ngo, Nguyen Dinh The Trinh, Tung Thanh Tran; J Pathol Transl Med. 2021;55(2):112-117. Published online January 27, 2021; DOI: https://doi.org/10.4132/jptm.2020.11.30

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Background
Langerhans cell histiocytosis (LCH) is more common in children than adults and involves many organs. In children, the BRAF V600E mutation is associated with recurrent and high-risk LCH.
Methods
We collected paraffin blocks of 94 pediatric LCH patients to detect BRAF V600E mutation by sequencing. The relationship between BRAF V600E status and clinicopathological parameters were also critically analyzed.
Results
BRAF V600E mutation exon 15 was detected in 45 cases (47.9%). Multiple systems LCH showed a significantly higher BRAF V600E mutation rate than a single system (p=.001). No statistical significance was evident for other clinical characteristics such as age, sex, location, risk organs involvement, and CD1a expression.
Conclusions
In Vietnamese LCH children, the proportion of BRAF V600E mutational status was relatively high and related to multiple systems.

Citations

Citations to this article as recorded by

Pathologic characteristics of histiocytic and dendritic cell neoplasms
Sun Och Yoon
Blood Research.2024;[Epub] CrossRef
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Zixuan Ding, Peilin Wang, Zhenrun Li, Yupeng Guo, Qiang Ma
Talanta.2023; 265: 124925. CrossRef

Case Reports

Langerhans Cell Sarcoma Arising in a Lymph Node: A Case Report and Review of the Literature.: Dong Wook Kang, Hyun Jin Son, Tae Hwa Baek, Hye Kyung Lee, Joo Ryung Huh, Joo Heon Kim, Mee Ja Park; Korean J Pathol. 2011;45(1):101-105.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.1.101

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Abstract PDF: We report a case of Langerhans cell sarcoma presented as a solitary mass in the left supraclavicular area in a 31-year-old woman. Computed tomography revealed a relatively well-defined and lightly enhancing mass in the left supraclavicular area, measuring 5.5x4.5x3.2 cm. Excision was subsequently performed. Microscopically, the specimen consisted of an enlarged and partially effaced lymph node. Nests of different size composed of atypical tumor cells were located in the paracortex and the medulla of the lymph node. The tumor cells exhibited abundant eosinophilic or clear cytoplasm and displayed marked nuclear atypia and increased mitotic figures. Infiltration of many eosinophils was identified in the periphery and between the tumor cells. The tumor cells were reactive for CD1a and S100 protein. Ultrastructually, they were found to have Birbeck granules in the cytoplasm.

Fine Needle Aspiration Cytology of Langerhans Cell Histiocytosis of Mandible: A Case Report.: Sang Ryung Lee, Jae Hee Suh, Hee Jung Cha, Young Min Kim, Hye Jeong Choi; Korean J Pathol. 2010;44(1):106-109.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.1.106

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Abstract PDF

We present a case of mandibular involvement with Langerhans cell histiocytosis (LCH), diagnosed by ultrasound-guided aspiration and subsequently confirmed by incisional biopsy and immunohistochemistry in an eight-year-old boy. The cytologic findings included the presence of characteristic Langerhans cells of both mononucleate and multinucleate form. Diagnostic confirmation was obtained by immunopositivity for S-100 protein and CD1a of Langerhans histiocytes on paraffin-embedded sections obtained during incisional biopsy of the right mandibular area. By reporting a case of childhood LCH, we correlate the cytologic findings with histologic features and discuss the role of aspiration cytologic diagnosis in such a rare and cytomorphologically characteristic case.

Citations

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Bronchial Washing Cytology of Pulmonary Langerhans Cell Histiocytosis: A Case Report
Taeyeong Kim, Hyeong Ju Kwon, Minseob Eom, Sang Wook Kim, Min Hi Sin, Soon-Hee Jung
Journal of Pathology and Translational Medicine.2017; 51(4): 444. CrossRef

Fine Needle Aspiration Cytology of Langerhans Cell Histiocytosis in a Lymph Node: A Case Report.: Seung Yeon Ha, Min Jung Kim, Gou Young Kim, Hyun Yee Cho, Dong Hae Chung, Na Rae Kim; Korean J Cytopathol. 2007;18(1):87-91.

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Abstract PDF: Langerhans cell histiocytosis (LCH) is a pleomorphic disease entity characterized by local or disseminated atypical Langerhans cells (LCs) found most commonly in bone, lungs, mucocutaneous structures and endocrine organs. Among different sites, unifocal disease confined to a lymph node (LN) is rare. We report a case of LCH confined to a LN in a 38-year-old male who presented with a 2 cm-sized cervical mass. The fine needle aspiration (FNA) smears of cervical LN showed high cellularity having isolated LCs with contorted nuclei and nuclear grooves mixed in multinucleated giant cells, small lymphocytes and eosinophils. Charcot-Leyden crystals were also seen, as were a few dendritic-like cells and intranuclear inclusions. Confirmation of LCH was made by histopathologic studies, positive reactions for S-100 protein and CD1a immunohistochemical staining and by the demonstration of Birbeck granules on electron microscopy. The differentials to be considered include dermatopathic lymphadenitis, sinus histiocytosis with massive lymphadenopathy, Hodgkin's lymphoma and malignant histiocytosis. The characteristic cytomorphologic pattern of LCH in a LN FNA smear plays an important role in suggesting the diagnosis of LCH.

Original Articles

The Epidermal Proliferation and the Number of Langerhans Cells in 7, 12-dimethylbenzanthracene Induced Epidermal Changes.: Chang Soon Han, Young Nyun Park, Kwang Gil Lee, In Joon Choi; Korean J Pathol. 1993;27(6):590-604.

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Abstract PDF: Chemically induced epiderml carcinogenesis is usually divided into two stages, the initiation and promotion. The initiation involves conversion of some epidermal cells into latent neoplastic cells and the promotion is proliferation of the transformed cells. As immunosurveillence is thought to be a host defense against tumors, Langerhans cells, being essential in initiation of local cutaneous immunologic reaction, is suggested to be important in the carcingenesis of the epidermis. This study is attempted to investigate the epidermal proliferative changes in mice induced by application of 12-0-tetradecanoy1-phorbol-13-acetate(TPA) on the skin initiated with 7, 12-dimethylbenzanthracene(DMBA) and its relationship with Langerhans cell. Ninty five male inbred BALB/c mice weighing 20~25 g were divided into five groups; the 33 week-group, the 21 week-group, the 12 week-group and the 4 week-group according to the duration of carcinogen application, and the control group. The carcingen was applied with a brush on the dorsal skin of mice after depilation. Ten days after application of 800 nmole DMBA in 0.4 cc acetone, 20 nmole TPA in 0.4 cc acetone was applied twice per week and the control group was applied with the same amount of acetone for 4 weeks. Animals were sacrificed 3 days after the last application of TPA. One hour before sacrifice, bromodeoxyuridine(BrdU) (1 mg/g) was injected via the tail artert for BrdU stain of S phase cells. A strip of dorsal skin was used for hematoxylineosin stain, immunohistochemical stain for BrdU and la antigen of Langerhans cell, and flow cytometry. The results are as follows: 1. Cellular proliferation, hyperkeratosis and dysplasia of the epidermis were increased in relation to duration of carcingen application. Papillomas were developed 12 weeks after application of the carcingen. 2. BrdU labelling and proliferative indices of the 20 weeks' application group were significantly higher than those of the 12 weeks' application group. The number of Langerhans cell was decreased markedly ater 4 week' application of the carcinogen. 3. All epiedrmal lesions including a case of squamous cell carcinoma were diploidy in flow cytometry. It is thought that disturbance of immunosurveillence, caused by depletion of Langerhans cell, may permit proliferation of epidermal cells. Although abnormal quantitative change of nuclear DNA has not occurred even when the epidermal proliferative activity and dysplastic change were increased markedly, it is thought that the occurrence of structural change of chromosome is remained to be clarified.

Macrophage/dendritic Cell Marker Staining Characteristics of Langerhans cell Granulomatosis(Histiocytosis X).: Sang Ae Yoon, In Sun Kim; Korean J Pathol. 1992;26(3):310-313.

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Abstract PDF: Histiocytosis X is characterized by aggregates of Langerhans cells with other inflammatory cells. These Langerhans cells are antigen-presenting cells to T lymphocytes and identified by characteristic morphology, ultrastructural demonstration of Birbeck granules and immunologic reactivity with OKT-6 and HLA-DR antibodies. In this report, the tumor arising in a 2-years-old baby was examined byimmunostaining with several macrophage/dendritic cell markers. The main tumor cells showed cytoplasmic and nuclear staining with S-100 protein and ring-like surface and paranuclear staining with PNA. However, they were negative for follicular dendritic cell marker CD21, macrophage markers lysozyme, Mac 387, alpha-1 antitrypsin and CD68, and interdigitating reticulum cell marker ID4 and ID5. These observations demonstrate the usefulness of S-100 protein and PNA for the identification of Langerhans cells in paraffin-embedded tissue.

A Study on the Histopathologic Features of Bowenoid Papulosis and the Numerical Change in Langerhans Cells.: Kwang Gil Lee, Soo Im Choi; Korean J Pathol. 1990;24(3):275-286.

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Abstract PDF: Fourteen cases of bowenoid papulosis were studied both clinically and histologically and were compared with 22 cases of Bowen's disease to find histologic differences between the two diseases. Bowenoid papulosis was prevalent in young, sexually active adults, consisted of multiple erythematous or hyperpigmented papules, and located mostly on the penis and vulva. The main histologic characteristics of bowenoid papulosis were an orderly background of keratinocyte maturation of the epidermis with monotonous pattern, the frequent presence of 'skip area', and the lesser degree of cytologic atypia than seen in lesions of Bowen's disease. In addition, mitotic figures more than 5 per x400 filed, formation of keratin pearl, and spindling of keratinocytes, which have not been described in other articles yet, are considered very important findings for the diagnosis of bowenoid papulosis. It is our opinion that bowenoid papulosis can be reliably distinguished from Bowen's disease by light microscopic features. Paraffin-embedded samples of bowenoid papulosis, Bowen's disease, and normal skin were investigated with antibodies against S 100-protein to relate the number of intraepidermal Langerhans cells and the disease. Langerhans cells were found to be reduced when compared with normal skin. No significant differences between Bowen's disease and bowenoid papulosis were found in density of Langerhans cells.

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