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1.
Pediatr Dermatol ; 37(2): 320-325, 2020 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-31930561

RESUMO

BACKGROUND/OBJECTIVES: Neurothekeoma is a rare, benign, cutaneous neoplasm consisting of Schwann cells and perineural cells in myxoid stroma. Cellular neurothekeoma (CNT) was previously thought to represent a morphologic variant of neurothekeoma, but recent studies have shown that CNTs are unrelated to neurothekeomas and are more likely of histiocytic lineage. METHODS: Herein, we describe seven cases of CNT in pediatric patients. A comprehensive search of PubMed was performed, and 71 cases of cellular neurothekeoma in pediatric patients were reviewed. RESULTS: The clinical differential diagnosis for these lesions included Spitz nevi, keloid, juvenile xanthogranuloma, cutaneous lymphoid hyperplasia, and lymphomatoid papulosis. All cases were treated by excision or excisional biopsy. Histopathologically, all demonstrated multilobular, primarily intradermal neoplasms composed of plump spindled or epithelioid mononuclear cells with abundant eosinophilic pale-staining cytoplasm. Immunophenotypic findings included CD68 and NKI/C3 positivity, and negative staining with cytokeratin, S-100, Melan-A, and SOX-10. CONCLUSION: Cellular neurothekeoma is distinguished from conventional neurothekeoma by increased cellularity, a lack of myxoid stroma, and a lack of neural expression with immunohistochemical stains. These uncommon neoplasms should be included in the differential diagnosis of dermal nodules in children. Accurate diagnosis of these lesions is essential, as they can be mistaken for malignancy leading to unnecessary treatment.


Assuntos
Neurotecoma/patologia , Neoplasias Cutâneas/patologia , Adolescente , Criança , Pré-Escolar , Estudos de Coortes , Feminino , Humanos , Lactente , Masculino , Neurotecoma/metabolismo , Neurotecoma/cirurgia , Neoplasias Cutâneas/metabolismo , Neoplasias Cutâneas/cirurgia
2.
Hand (N Y) ; 15(1): NP7-NP10, 2020 01.
Artigo em Inglês | MEDLINE | ID: mdl-30762430

RESUMO

Background: Neurothekeomas are slow-growing, well-circumscribed benign neoplasms. They usually involve cutaneous or subcutaneous tissues. Although originally described as myxomas deriving from nerve sheath cells, their exact histological classification is still uncertain. Peripheral nerve localization is rarely reported. Here, we describe a unique case of sleeve-shaped neurothekeoma of the ulnar nerve, which was incidentally discovered during a cubital tunnel release surgery. Methods: A 57-year-old man was admitted at our institution with clinical, ultrasonographic, and electromyographic findings highly suggestive of cubital tunnel syndrome. During ulnar nerve decompression surgery, however, no bony or ligament compression was noticed, but a segment of the nerve wrapped by a thick sleeve-shaped tissue, which had no clear-cut cleavage plane from the nerve. Given this unexpected finding, the en bloc excision of the lesion was avoided. A nerve decompression with biopsy of the swelling portion of the lesion was performed instead. Results: Histological examination described abundant myxoid stroma, with epithelioid and ring-shaped cells arranged in cords, negative to S100 protein at immunohistochemical analysis. This pattern was suggestive of neurothekeoma. The patient showed improvement in hypermyotrophy and intrinsic weakness of the hand. Conclusions: To the best of our knowledge, this is the first report of a sleeve-shaped neurothekeoma of the ulnar nerve. The exact pathological characterization of such rare entities remains uncertain. In case of peripheral nerve localization, and when a clear cleavage plane is absent, the correct management of these lesions should be that of simple nerve decompression followed by biopsy.


Assuntos
Neurotecoma/patologia , Nervo Ulnar/patologia , Síndrome do Túnel Ulnar/cirurgia , Descompressão Cirúrgica , Humanos , Achados Incidentais , Masculino , Pessoa de Meia-Idade , Neurotecoma/diagnóstico , Neurotecoma/cirurgia
3.
Pediatr Dermatol ; 37(1): 187-189, 2020 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-31774578

RESUMO

Neurothekeomas, or simply nerve sheath myxomas, are rare benign skin neoplasms. They are believed to derive from peripheral nerve sheath transformation and are typically more prevalent in females. They tend to be found mainly on the upper limbs as well as head and neck locations. In this report, we detail two children with neurothekeomas, describe the fundamental clinical manifestations, and review the current literature.


Assuntos
Neurotecoma/patologia , Neoplasias Cutâneas/patologia , Biópsia , Criança , Pré-Escolar , Feminino , Humanos , Masculino , Neurotecoma/cirurgia , Neoplasias Cutâneas/cirurgia
5.
Pathologica ; 112(4): 210-213, 2020 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-33393524

RESUMO

Atypical cellular neurothekeoma (ACN) is an aggressive and rare variant of cellular neurothekeoma. Only few cases have been reported in the literature and the biological behavior seems to be uncertain. We describe the case of an ACN presenting on the scalp of an elderly man, emphasizing the cytologic features of malignancy. In addition, we provide a brief overview of the literature and discuss the differential diagnosis with other entities, and the possible diagnostic pitfalls.


Assuntos
Neurotecoma , Couro Cabeludo/patologia , Idoso , Biomarcadores Tumorais/análise , Diagnóstico Diferencial , Humanos , Imuno-Histoquímica , Masculino , Neurotecoma/diagnóstico , Neurotecoma/patologia , Neoplasias Cutâneas/diagnóstico , Neoplasias Cutâneas/patologia , Neoplasias de Tecidos Moles/diagnóstico , Neoplasias de Tecidos Moles/patologia
6.
Surv Ophthalmol ; 64(6): 852-857, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-30978337

RESUMO

Neurothekeomas (NTKs) are benign soft tissue tumors most commonly occurring in the head, neck, and upper extremities of young adults, with a female preponderance. Herein, we report 2 cases of ocular NTK presenting as an orbital mass or chalazion. Both cases underwent excision of the mass with histopathologic diagnosis of cellular NTK. Tumor cells for both cases were immunopositive for smooth muscle actin, microphthalmia-associated transcription factor, and cluster of differentiation 10. S100, human melanoma black 45, and melanoma antigen recognized by t-cell (Melan A) were negative. Both were associated with scattered histiocytes, which were positive for cluster of differentiation 68. To date, including our 2 cases, there have been 22 cases of ocular and periorbital adnexal NTK reported in the English-language literature. Although rare, clinicians should include NTK in their differential diagnosis in patients presenting with a periocular soft tissue tumor.


Assuntos
Neoplasias da Túnica Conjuntiva/patologia , Neurotecoma/patologia , Neoplasias Orbitárias/patologia , Pré-Escolar , Feminino , Humanos , Masculino , Músculos Oculomotores/patologia
7.
J Cutan Pathol ; 46(1): 67-73, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-30270462

RESUMO

Cellular neurothekeoma is a benign cutaneous neoplasm that typically occurs on the head, neck, and upper body of young adults with a slight female predominance. It is a rare lesion to diagnose and multiple neurothekeomas in one patient are even more uncommon finding. We present a case of multiple neurothekeomas in a middle-aged woman with lower extremity involvement and summarize the current literature on multiple neurothekeoma patients. A 46-year-old female presented with nearly one dozen skin-colored papules on the head, upper limb, and lower limb. The lesions were clinically diagnosed as dermatofibromas and a nevus. Eight lesions were biopsied and confirmed to be cellular neurothekeomas, with one initially misinterpreted on histology as a dermatofibroma. Awareness of cellular neurothekeoma as a diagnostic entity and the possibility of atypical presentations as seen in our case (eg, in multiple numbers, in older adults, and on the lower extremity) are important in allowing for accurate clinical and histological diagnosis of these lesions. The possibility of a syndromic association with multiple cellular neurothekeomas should be explored further.


Assuntos
Extremidade Inferior , Neurotecoma , Neoplasias Cutâneas , Humanos , Pessoa de Meia-Idade , Neurotecoma/diagnóstico , Neurotecoma/metabolismo , Neurotecoma/patologia , Neoplasias Cutâneas/diagnóstico , Neoplasias Cutâneas/metabolismo , Neoplasias Cutâneas/patologia
8.
J Cutan Pathol ; 46(1): 80-83, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-30311258

RESUMO

Cellular neurothekeoma (CNT) is a dermal lesion with still unknown histogenesis, characterized by immunohistochemical staining for NKI/C3, NSE, MiTF, CD10 and CD68, whereas S100 protein, desmin and cytokeratins are negative. Particularly, in several studies NKI/C3 has been reported as a strong marker of CNT. We describe herein the clinical, histopathological and immunohistochemical features of two cases morphologically consistent with myxoid CNT, one of which showing some atypical features, both characterized by negative immunohistochemical staining for NKI/C3. Our findings stress the importance of morphology in diagnosing CNT and underline the fact that NKI/C3 can fail to stain cases belonging to the "neurothekeoma family." In selected cases of CNT, an expanded immunohistochemical panel is mandatory to differentiate this tumor from other dermal lesions.


Assuntos
Proteínas de Neoplasias/metabolismo , Neurotecoma , Neoplasias Cutâneas , Adulto , Feminino , Humanos , Imuno-Histoquímica , Masculino , Pessoa de Meia-Idade , Neurotecoma/diagnóstico , Neurotecoma/metabolismo , Neurotecoma/patologia , Neoplasias Cutâneas/diagnóstico , Neoplasias Cutâneas/metabolismo , Neoplasias Cutâneas/patologia
9.
Br J Neurosurg ; 33(2): 213-214, 2019 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-28282991

RESUMO

Nerve sheath myxomas (NSM) are uncommon benign tumours of nerve sheath origin. These tumours are most often located in the dermis and subcutaneous tissue of the head and neck region, but less frequently can also involve extracutaneous sites but they are rarely seen in the oral cavity. Many authors consider nerve sheath myxoma and neurothekeoma to be variants of the same tumour, though currently it has been demonstrated that both the lesions are distinct entities with different origins and unique morphological features. Herein we report a case of Nerve Sheath Myxoma on the palatal gingiva.


Assuntos
Neoplasias Gengivais/patologia , Neurotecoma/patologia , Neoplasias Palatinas/patologia , Adolescente , Feminino , Neoplasias Gengivais/cirurgia , Humanos , Neurotecoma/cirurgia , Neoplasias Palatinas/cirurgia
13.
J Cutan Pathol ; 45(8): 619-622, 2018 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-29744902

RESUMO

Cellular neurothekeomas (CNTs) are rare, benign cutaneous tumors that arise primarily on the head and neck, with a slight female predominance. CNTs with atypical features have been described, including those with an infiltrative growth pattern. Although CNTs with atypical features are benign, recognition of this entity can pose diagnostic challenges. Here, we report a case of CNT with an unusual clinical presentation on the left second digit, and with atypical histological features including an infiltrative growth pattern, which could have been mistaken for features of malignancy.


Assuntos
Histiocitoma Fibroso Benigno/diagnóstico , Neurotecoma/diagnóstico , Neoplasias Cutâneas/diagnóstico , Adulto , Diagnóstico Diferencial , Feminino , Histiocitoma Fibroso Benigno/patologia , Humanos , Neurotecoma/patologia , Neoplasias Cutâneas/patologia
15.
Head Neck Pathol ; 12(1): 75-81, 2018 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-28597210

RESUMO

Cellular neurothekeomas (CNs) are distinctive benign tumors of uncertain histogenesis, with predilection for the skin of the head and neck region. We describe the first case of multiple desmoplastic CNs (DCNs) affecting the oral cavity in a 9-year-old girl. Histopathologic evaluation showed a proliferation of spindle and epithelioid cells, forming nests and bundles, supported by exuberant fibrous stroma, as well as scattered multinucleated floret-like giant cells. The tumor cells were immunopositive for vimentin, CD63, CD56, whereas AE1/AE3, S100, CD34, α-SMA, GFAP, EMA, CD57 and NSE were negative. Ki-67 was <2%. Multiple DCNs should be considered in the differential diagnosis of oral nodular lesions.


Assuntos
Neoplasias Bucais/patologia , Neoplasias Primárias Múltiplas/patologia , Neurotecoma/patologia , Biomarcadores Tumorais/análise , Criança , Feminino , Humanos
19.
Pathology ; 49(1): 44-49, 2017 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-27914685

RESUMO

Cellular neurothekeoma (CNTK) frequently enters the differential diagnosis of a benign dermal cellular proliferation. Diagnosis often relies on immunohistochemistry including the use of protein gene product 9.5 (PGP9.5). A previous study demonstrated PGP9.5 expression across a wide variety of soft tissue neoplasms. We explored the utility of this antibody in distinguishing CNTK from other benign dermal-based lesions. A cohort of CNTK (n=7) and benign cutaneous lesions of neural (n=28), fibrohistiocytic (n=23), fibroblastic (n=25), histiocytic (n=18), myofibroblastic (n=7), smooth muscle (n=14), and melanocytic (n=12) differentiations were immunostained with PGP9.5. Staining was graded by H-score and compared with CNTK. A significantly higher H-score was found in CNTK compared with the fibrohistiocytic (p=0.0001), histiocytic (p=0.0016), myofibroblastic (p=0.0003), smooth muscle (p<0.0001), and melanocytic (p=0.0004) groups, with the exceptions of plexiform fibrohistiocytic tumour, xanthoma, and xanthogranuloma. No significant difference was found when comparing CNTK with fibroblastic and neural lesions, with the exceptions of neurofibroma and perineurioma. In conclusion, PGP9.5 is helpful in distinguishing CNTK from most benign cutaneous fibrohistiocytic, histiocytic, myofibroblastic, smooth muscle, and melanocytic lesions. In addition to CNTK and neural lesions, PGP9.5 is also expressed in benign fibroblastic lesions, and therefore distinction of these lesions should not be based on PGP9.5 positivity.


Assuntos
Biomarcadores Tumorais/análise , Imuno-Histoquímica , Neoplasias da Bainha Neural/patologia , Neurotecoma/patologia , Neoplasias Cutâneas/patologia , Neoplasias de Tecidos Moles/patologia , Ubiquitina Tiolesterase/análise , Biomarcadores Tumorais/genética , Diagnóstico Diferencial , Regulação Neoplásica da Expressão Gênica , Humanos , Imuno-Histoquímica/métodos , Neoplasias da Bainha Neural/diagnóstico , Neurotecoma/diagnóstico , Neoplasias Cutâneas/diagnóstico , Neoplasias de Tecidos Moles/diagnóstico , Ubiquitina Tiolesterase/genética
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