Pediatric neuroendocrine carcinoid tumors: Management, pathology, and imaging findings in a pediatric referral center

Degnan, Andrew J.; Tocchio, Shannon; Kurtom, Waleed; Tadros, Sameh

doi:10.1002/pbc.26477

Cited by 22 publications

(24 citation statements)

References 53 publications

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“…Another issue is the high proportion of patients lost to FU or who opted out, as it happened in six patients of our series (23% of the total population). This was often reported in the past, 14,16,27 and in our experience it seems a consequence of parents’ decision to look for a second or third opinion in other centers outside the pediatric network.…”

Section: Discussionsupporting

confidence: 50%

Extra‐appendicular neuroendocrine tumors: A report from the TREP project (2000‐2020)

Virgone

Ferrari

Chiaravalli

et al. 2021

Pediatric Blood & Cancer

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Background Extra‐appendicular neuroendocrine tumors (NETs) are very rare tumors. While diagnostic and therapeutic guidelines are well established for adults, data on children and adolescents are lacking. Patients and Methods Patients with a diagnosis of extra‐appendicular NET registered on the Tumori Rari in Età Pediatrica ‐ Rare Tumors in Pediatric Age (TREP) from 2000 to 2020 were analyzed. Clinical characteristics including patients’ presentation, tumor features, treatment, and outcome were reviewed. Results Twenty‐seven patients with extra‐appendicular NET registered on TREP with a median age of 173 months. The primary site was the pancreas (12) or bronchi (10) in the majority of cases. Other primary sites included the thymus, Meckel's diverticulum, and liver. Thirteen (48%) of tumors extended beyond the organ of origin: four invaded neighboring organs and/or regional nodes and nine involved distant metastases. The 3‐year event‐free survival (EFS) for those with localized disease was superior to those with metastatic disease (66.6% 95% CI 5‐95% vs 33% 95% CI 5‐68%, respectively; P = .005). A complete resection was feasible in 17 patients. The 3‐year EFS in these patients was superior to those with no or incomplete resection (R0 vs R1/R2, respectively; P = .007). Overall, 16 children had no evidence of disease at follow‐up, and one is alive with disease; five died, and five were lost to follow‐up. Conclusions Data from our experience demonstrated a wide heterogeneity of presentation and outcome of these tumors. Localized disease and complete surgical resection were the main prognostic factors of good outcome. Other therapies may have a role in prolonging survival in metastatic disease.

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Section: Discussionsupporting

confidence: 50%

Extra‐appendicular neuroendocrine tumors: A report from the TREP project (2000‐2020)

Virgone

Ferrari

Chiaravalli

et al. 2021

Pediatric Blood & Cancer

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“…Neuroendocrine tumors commonly metastasize [19] and research has demonstrated that Tyro3 may contribute to cancer cell motility [20] . We noted a decrease in Tyro3 expression with increasing doses of crizotinib ( Fig.…”

Section: Resultsmentioning

confidence: 99%

Downregulation of PDGFRß Signaling Overcomes Crizotinib Resistance in a TYRO3 and ALK Mutated Neuroendocrine-Like Tumor

Quinn

Beierle

Williams

et al. 2021

Translational Oncology

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Highlights Rare, high grade neuroendocrine-like pediatric tumor has TYRO3 and ALK mutations. Crizotinib downregulates STAT3 and ERK1/2 signaling in tumor. In vivo analysis demonstrated crizotinib resistance. Crizotinib resistant cells have upregulation of PDGFRß signaling. PDGFRß inhibition overrides resistance.

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“…The effect of lymph node resection on overall survival for typical bronchial carcinoid in pediatrics has not been clearly defined; however, multiple series have shown that lymphatic spread occurs in up to 20% of pediatric patients of both histologies. 1,5,9,17 Given this information, we believe that lymph node resection should be performed for all enlarged nodes and lymph node sampling should be considered in other patients.…”

Section: Discussionmentioning

confidence: 99%

Pediatric Bronchial Carcinoid Tumors: A Case Series and Review of the Literature

Potter

HaDuong

Okcu

et al. 2019

Journal of Pediatric Hematology/Oncology

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Bronchial carcinoid tumor, while rare, remains the most common primary malignant lung tumor in children. We present a retrospective analysis of 7 patients with typical bronchial carcinoid tumors diagnosed at 2 pediatric tertiary care referral centers between 1990 and 2014. The most common presenting symptom was pneumonia, followed by respiratory distress. Somatostatin scans were performed in selected patients. All patients had negative resection margin following surgery and were alive without disease at last follow-up. Typical carcinoid tumors have a good prognosis following definitive surgical resection. A review of published literature on pediatric bronchial carcinoid tumors is provided.

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Pediatric neuroendocrine carcinoid tumors: Management, pathology, and imaging findings in a pediatric referral center

Cited by 22 publications

References 53 publications

Extra‐appendicular neuroendocrine tumors: A report from the TREP project (2000‐2020)

Extra‐appendicular neuroendocrine tumors: A report from the TREP project (2000‐2020)

Downregulation of PDGFRß Signaling Overcomes Crizotinib Resistance in a TYRO3 and ALK Mutated Neuroendocrine-Like Tumor

Pediatric Bronchial Carcinoid Tumors: A Case Series and Review of the Literature

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