‘Neuroendocrine’ middle ear adenomas: consistent expression of the transcription factor <scp>ISL</scp>1 further supports their neuroendocrine derivation

Agaimy, Abbas; Lell, Michael; Schaller, Tina; Märkl, Bruno; Hornung, Joachim

doi:10.1111/his.12447

Cited by 21 publications

(11 citation statements)

References 31 publications

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“…The patient was followed up with 6-monthly cross-sectional imaging; in 2020, her contrast-enhanced CT thorax, abdomen and pelvis showed progression of all metastases in the liver. These lesions showed avid uptake on both 68 Ga-DOTATATE-PET and 18 FDG-PET with complete concordance. Considering her age and the sideeffects of the available therapies for metastatic progressive NETs, the multi-disciplinary team decided on a further single treatment with 177 Lu-DOTATATE, which she has recently completed.…”

Section: A S Ementioning

confidence: 66%

“…A further 4 years later in 2019, recurrence in the left petrous bone was treated by left petrosectomy, showing a well differentiated neuroendocrine neoplasm with no mitotic activity and a Ki-67 of 8%. However, at this point, he was noted on 18 FDG-PET scanning to show uptake in his para-aortic and left axillary lymph nodes, which were non-avid on 68 Ga-DOTATATE-PET/CT, suggesting a recurrence of his Hodgkin lymphoma. He was therefore given a further course of chemotherapy.…”

mentioning

confidence: 93%

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Middle ear neuroendocrine tumours: Insight into their pathogenesis, diagnosis and management

Ratnayake

Luong

Toumpanakis

et al. 2021

J Neuroendocrinology

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Neuroendocrine tumours (NETs) represent a spectrum of tumours with a diverse range of molecular abnormalities, functionality and anatomical locations. 1 The common sites of neuroendocrine tumours include the gastrointestinal tract, pancreas and lungs, although other primary sites have been reported, such as the ovary, 2 thymus and C cells of the thyroid. 3 Very rarely, these tumours appear to arise from the middle ear and, although extremely uncommon, accounting for < 2% of all middle ear tumours, 4 they are important to differentiate from other middle ear tumours because the management of these tumours significantly differs from these other types of tumour. In addition, they raise significant questions as to the provenance of such tumours arising from an area not generally known to harbour neuroendocrine tissue.We describe two patients with middle ear NETs, one with multiple local recurrences and the other with distant metastases, discuss the management of these tumours, and review the extant literature on these tumours and their putative derivation.

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Section: A S Ementioning

confidence: 66%

mentioning

confidence: 93%

Middle ear neuroendocrine tumours: Insight into their pathogenesis, diagnosis and management

Ratnayake

Luong

Toumpanakis

et al. 2021

J Neuroendocrinology

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“…The value of radio‐/chemotherapy, somatostatin analogues, and PRRT in MEANT is yet unascertained 9,11 . Current studies advocate long‐term follow‐up, irrespective of tumor characteristics at presentation, in order to facilitate early detection of recurrent disease 6‐9,11,12 . This study, nor the literature, provides sufficient information to define specific follow‐up protocols.…”

Section: Discussionmentioning

confidence: 99%

“…In 1980, Murphy et al reported on the histochemical and ultrastructural neuroendocrine features, further distinguishing MEANT from other neoplasms of the temporal bone and surrounding structures 4 . Its terminology is still equivocal and comprises (neuroendocrine/amphicrine) adenoma and carcinoid; however, all of these tumors share a common phenotype and immunohistochemical profile 5‐7 . In the fourth edition of the WHO Classification of Tumors of the Head and Neck, it is featured as Middle ear adenoma (ICD‐O code: 8140/0).…”

Section: Introductionmentioning

confidence: 99%

Neuroendocrine neoplasms of the middle ear: Unpredictable tumor behavior and tendency for recurrence

et al. 2021

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Background Middle ear adenomatous neuroendocrine tumors (MEANTs) are rare temporal bone tumors. This study evaluates its clinical behavior and therapy outcome. Method Retrospective case review in a tertiary referral center evaluating histopathology, immunohistochemistry, treatment, and outcome. Results Nine patients were diagnosed with MEANT. One patient presented with locally invasive tumor and underwent extensive en‐bloc tumor resection with adjuvant radiotherapy. Seven of eight patients with locally non‐aggressive tumor confined to the tympanomastoid space underwent tumor resection. Two patients were disease‐free, five presented recurrence, even after apparent successful surgery. All tumors showed neuroendocrine features. Histopathology and immunohistochemistry did not yield prognostic tumor characteristics. Conclusion MEANTs are rare tumors with uncertain biological behavior and subsequent unpredictable clinical course. The preferred treatment is complete surgical tumor resection. They have a high tendency for recurrence, irrespective of negative intermediary surgery. As of yet, there are no prognostic biomarkers, including histopathology and immunohistochemistry.

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“…Agaimy et al 9 reported that a plasmacytoid cell appearance seemed to be infrequent but was observed in one of their six cases. Consequently, plasmacytoma should be considered.…”

Section: Differential Diagnosismentioning

confidence: 91%

Middle ear adenoma with uncommon presentation and literature review

Wang¹,

Ouyang

Guo

et al. 2017

BMJ Case Reports

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Middle ear adenoma (MEA) is a rare benign tumour. Here we report a case of MEA in a 39-year-old man with hearing loss. Some significant histopathological features of MEA and neuroendocrine differentiation identified by immunohistochemistry are described. The tumour showed remarkable and rare degenerative histological characteristics. There were a few tumour cells which were covered by degenerated tissue. Consequently, following frozen biopsy, the tumour was misdiagnosed as chronic ear inflammation. No recurrence was seen in this patient after 45 months of follow-up; regular clinical and radiological follow-up is necessary since recurrence is possible. A literature review for MEA is presented and this type of tumour is discussed clinically, microscopically and immunohistochemically. The differential diagnosis and associated treatment are described.

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‘Neuroendocrine’ middle ear adenomas: consistent expression of the transcription factor ISL1 further supports their neuroendocrine derivation

Cited by 21 publications

References 31 publications

Middle ear neuroendocrine tumours: Insight into their pathogenesis, diagnosis and management

Middle ear neuroendocrine tumours: Insight into their pathogenesis, diagnosis and management

Neuroendocrine neoplasms of the middle ear: Unpredictable tumor behavior and tendency for recurrence

Middle ear adenoma with uncommon presentation and literature review

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