Soft Tissue Special Issue: Perivascular and Vascular Tumors of the Head and Neck

Flucke, Uta; Karanian, Marie; Broek, Roel W. Ten; Thway, Khin

doi:10.1007/s12105-020-01129-z

Cited by 24 publications

(31 citation statements)

References 75 publications

(139 reference statements)

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“…KHE consists of irregularly in ltrating, compressed vascular spaces, intervening between spindle-shaped endothelial cells. Occasionally the vessels contain erythrocyte and platelet thrombi (10).…”

Section: Discussionmentioning

confidence: 99%

Kaposiform Hemangioendothelioma of the Sublingual Gland in an Adult: Resection is the Key.

Chowdhary

Sekar

Saxena

et al. 2021

Preprint

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Introduction: Kaposiform haemangioendotheliomas (KHE) are extremely rare, borderline malignant, locally aggressive vascular tumours. Although more commonly seen in childhood and adolescents, some very rare reports in adults do exist. KHE is usually found in the retroperitoneum, over the extremities and the soft tissues of the trunk, mediastinum and the head and neck regions. Prior published reports have shown a strong association with the Kasabach-Merritt phenomenon (KMP). We present a very rare case of KHE of the sublingual gland in an adult, which was not associated with KMP and was resected completely without any sequelae. Case report: A 42-year-old female presented with a painful swelling over the floor of the mouth on the right for a year. Clinical examination revealed a bi-digitally palpable, non-transilluminant lesion on the right side of the lingual frenulum. Radiological investigations suggested the possibility of a benign soft tissue tumour arising from the sublingual gland. The patient underwent an intraoral excision of the lesion. The biopsy revealed spindle-shaped tumour cells in fascicles, surrounded by vascular spaces, suggestive of a KHE. The patient was offered radiation therapy, which she declined and continued to be on follow-up for two years. She remains symptom-free to date.Conclusion: Excision of a KHE of the floor of the mouth proved to be sufficient, without the evidence of any residual lesion or progression of symptoms. To the best of our knowledge, our case represents the first reported case in the literature of a KHE arising from the salivary glands in an adult.

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Section: Discussionmentioning

confidence: 99%

Kaposiform Hemangioendothelioma of the Sublingual Gland in an Adult: Resection is the Key.

Chowdhary

Sekar

Saxena

et al. 2021

Preprint

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“…Vascular tumors arise by clonal cellular proliferation of vessels showing a disproportionate growth. In contrast, vascular malformations originate in utero as a result of mosaic mutations leading to erroneous development of vessels with proportionate growth [187].…”

Section: Hemangioma Of Bone With An Ewsr1-nfatc1 Fusionmentioning

confidence: 99%

EWSR1—The Most Common Rearranged Gene in Soft Tissue Lesions, Which Also Occurs in Different Bone Lesions: An Updated Review

et al. 2021

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EWSR1 belongs to the FET family of RNA-binding proteins including also Fused in Sarcoma (FUS), and TATA-box binding protein Associated Factor 15 (TAF15). As consequence of the multifunctional role of EWSR1 leading to a high frequency of transcription of the chromosomal region where the gene is located, EWSR1 is exposed to aberrations such as rearrangements. Consecutive binding to other genes leads to chimeric proteins inducing oncogenesis. The other TET family members are homologous. With the advent of widely used modern molecular techniques during the last decades, it has become obvious that EWSR1 is involved in the development of diverse benign and malignant tumors with mesenchymal, neuroectodermal, and epithelial/myoepithelial features. As oncogenic transformation mediated by EWSR1-fusion proteins leads to such diverse tumor types, there must be a selection on the multipotent stem cell level. In this review, we will focus on the wide variety of soft tissue and bone entities, including benign and malignant lesions, harboring EWSR1 rearrangement. Fusion gene analysis is the diagnostic gold standard in most of these tumors. We present clinicopathologic, immunohistochemical, and molecular features and discuss differential diagnoses.

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“…The prevalence of LMs is approximately 1:5000 among live births 10 . The diagnosis can be made before the baby is born (in the womb), but most lesions are clinically recognized by the age of 2 3,5,9,11–16 . Generally, lesion development follows patient growth 5,17 .…”

Section: Introductionmentioning

confidence: 99%

“…10 The diagnosis can be made before the baby is born (in the womb), but most lesions are clinically recognized by the age of 2. 3,5,9,[11][12][13][14][15][16] Generally, lesion development follows patient growth. 5,17 However, stimuli, such as trauma, infections, and hormonal changes, are also associated with rapid development.…”

Section: Introductionmentioning

confidence: 99%

Oral lymphatic malformations: A multicenter study of 208 cases and literature review

et al. 2021

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Background To evaluate the frequency and analyze demographic and clinical characteristics of individuals with a histopathological diagnosis of oral lymphatic malformations (OLMs). Methods A multicenter study was performed, collecting biopsy record data from a consortium of Brazilian Oral and Maxillofacial Pathology Centers. A review was also conducted to compare this data with cases already available in the literature. Results This study retrieved 208 cases of OLM in the multicenter study and 1035 cases in the literature review. In both, OLMs affected male and female individuals equally, with the most affected site being the tongue. Individuals ≥60 years of age were uncommonly affected. Symptomatic and larger lesions were more commonly reported in the literature review. Conclusions This study comprises the largest sample of OLMs to date. OLMs are rare conditions, without sex predilection. The elderly proved to be less frequently affected, and the tongue is the most commonly affected site.

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Soft Tissue Special Issue: Perivascular and Vascular Tumors of the Head and Neck

Cited by 24 publications

References 75 publications

Kaposiform Hemangioendothelioma of the Sublingual Gland in an Adult: Resection is the Key.

Kaposiform Hemangioendothelioma of the Sublingual Gland in an Adult: Resection is the Key.

EWSR1—The Most Common Rearranged Gene in Soft Tissue Lesions, Which Also Occurs in Different Bone Lesions: An Updated Review

Oral lymphatic malformations: A multicenter study of 208 cases and literature review

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