Human Papillomavirus–Associated Oropharyngeal High-Grade Neuroendocrine Carcinoma in an Adolescent: Case Report and Review of Literature

Sinno, Sara; Assaad, Adel; Shabb, Nina S.

doi:10.1177/1179556519870520

Cited by 5 publications

(4 citation statements)

References 26 publications

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“…A PRISMA diagram outlining the literature search is shown in Figure 1. Fifty full‐text articles published from 1987 to 2020 were ultimately included in this systematic review 4,5,8–12,18–60 . Further details regarding these publications including the Oxford level of evidence can be found in Table 1.…”

Section: Resultsmentioning

confidence: 99%

Systematic review of neuroendocrine carcinomas of the oropharynx

et al. 2022

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Data describing features and management of oropharyngeal neuroendocrine carcinomas (NEC) remain sparse. A systematic review was performed. Patients were stratified by treatment modality and examined for disease progression and survival outcomes. Ninety‐four patients from 50 publications were included. Average age at diagnosis was 59.7 years (range 14–83). 73.4% were male. Most studies did not document HPV status. Forty patients (85.1%) were p16 positive, and 34 (85.0%) were HPV‐ISH positive. Overall survival was 75.4% at 1 year, and 40.0% at 2 years. Of patients with locoregional disease, 33.8% developed distant metastasis. 12.5% of patients developed locoregional recurrence. Patients who developed distant metastases had worse overall survival (p = 0.0004). No significant difference was found between treatment modalities. Human papilloma virus may be associated with oropharyngeal NEC. Current treatments provide locoregional control, but distant metastases are common and confer low overall survival.

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Section: Resultsmentioning

confidence: 99%

Systematic review of neuroendocrine carcinomas of the oropharynx

et al. 2022

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“…The importance of p16/HPV status for the prediction of treatment response and prognosis in patients with PD NEC is uncertain. The results of p16 IHC and HPV status determination showed that, indeed, most (but not all) p16-positive and HPV-positive cases originated from the oropharynx; however, in contrast to the case for squamous cell carcinoma, the p16 status in PD NEC cannot be used as a surrogate marker of HPV status due to its low specificity [ 38 , 39 , 40 ]. Namely, the frequently seen overexpression of p16 in LC/SC-NEC of the head and neck may also be due to a virus-independent Rb pathway dysregulation [ 41 ].…”

Section: Discussionmentioning

confidence: 99%

“…Since the extent of the disease was not included in the multivariate model which, in several analyses (including ours), has proved to be an important prognostic factor, the results of this study should be interpreted with caution. Furthermore, upon reviewing the literature specifically focused on oropharyngeal PD NEC, Sinno et al found no survival advantage of HPV positivity, although without regard to the stage of disease [ 40 ].…”

Section: Discussionmentioning

confidence: 99%

Neuroendocrine Carcinoma of the Larynx and Pharynx: A Clinical and Histopathological Study

Strojan

Šifrer

Ferlito³

et al. 2021

Cancers

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Neuroendocrine carcinomas (NECs) of the head and neck are rare and the experience scanty. The Cancer Registry of Slovenia database was used to identify cases of laryngeal and pharyngeal NECs diagnosed between 1995–2020. Biopsies were analyzed for the expression of standard neuroendocrine markers (synaptophysin, chromogranin, CD56), INSM1, Ki-67, p16, and PD-L1 (using the combined positive score, CPS). In situ hybridization for human papillomavirus (HPV) and Epstein–Barr virus (EBV) was performed. Twenty patients (larynx, 12; pharynx, 8) were identified. One tumor was well differentiated (WD), five were moderately differentiated (MD), and 14 were poorly differentiated (PD). Disease control was achieved solely by surgery in 4/4 MD/PD T1-2N0-1 tumors. Eight patients died of the disease, seven of which were due to distant metastases. All three traditional markers were positive in 11/17 NECs and the INSM1 marker in all 20 tumors. Two of fourteen p16-positive tumors were HPV-positive, but all three nasopharyngeal NECs were EBV-negative. Three tumors had CPSs ≥ 1. In conclusion, INSM1 was confirmed to be a reliable marker of neuroendocrine differentiation. Except in WD and early-stage MD/PD tumors, aggressive multimodal therapy is needed; the optimal systemic therapy remains to be determined. p16, HPV, and EBV seem to bear no prognostic information.

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“…Herein, NEC located in the base of the tongue and tonsils were not included since these sites are considered to be part of the oropharynx. Moreover, Sinno et al [39] have recently reviewed the literature concerning oropharyngeal NEC and our aim was to focus on oral cavity and jawbone cases [39].…”

Section: Discussionmentioning

confidence: 99%

Oral and Maxillofacial Neuroendocrine Carcinoma: A Systematic Review

Schuch

Schmidt

Zigmundo

et al. 2021

Head and Neck Pathol

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The aim of the present study was to integrate the available data published in the literature on oral and maxillofacial neuroendocrine carcinomas concerning the demographic, clinical and histopathological features of this condition. An electronic search with no publication date restriction was undertaken in April 2021 in four databases. Eligibility criteria included reports published in English having enough data to confirm a definite diagnosis, always showing a neuroendocrine marker. Cases originating in the oropharynx, including base of the tongue and tonsils, were excluded. Outcomes were evaluated by the Kaplan-Meier method along with Cox regression. Twenty-five articles (29 cases) from nine different countries were detected. Mean patient age was 56.3 (± 17.5) years, with a slight male predilection. Symptomatology was present in 72.2% of informed cases. Regarding clinical presentation, a non-ulcerated nodule located in the gingiva with a mean size of 3.4 (± 2.0) cm was most frequently reported. Concomitant metastasis was identified in seven individuals. Histopathologically, most neoplasms were of the small cell type, and immunohistochemistry for both epithelial and neuroendocrine differentiation was used in 65.5% cases. Radical surgery was the treatment of choice in almost all cases, with or without adjuvant therapy. Mean follow-up was 20.5 (± 21.2) months, and only four patients developed recurrences. Eleven (44.0%) individuals died due to the disease. Ulcerated lesions were a prognostic factor. This study provides knowledge that can assist surgeons, oncologists, and oral and maxillofacial pathologists with the diagnosis and management of neuroendocrine carcinomas. Our findings demonstrated that the long-term prognosis of this lesion continues to be poor.

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Human Papillomavirus–Associated Oropharyngeal High-Grade Neuroendocrine Carcinoma in an Adolescent: Case Report and Review of Literature

Cited by 5 publications

References 26 publications

Systematic review of neuroendocrine carcinomas of the oropharynx

Systematic review of neuroendocrine carcinomas of the oropharynx

Neuroendocrine Carcinoma of the Larynx and Pharynx: A Clinical and Histopathological Study

Oral and Maxillofacial Neuroendocrine Carcinoma: A Systematic Review

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