Lymph node metastases are more frequent in paediatric appendiceal NET ≥1.5 cm but without impact on outcome - Data from the German MET studies

Eur J Surg Oncol. 2024 Apr;50(4):108051. doi: 10.1016/j.ejso.2024.108051. Epub 2024 Feb 27.

Abstract

Background: Paediatric appendiceal neuroendocrine tumours (appNET) are very rare tumours, mostly detected incidentally by histopathological evaluation after appendectomy. Treatment recommendations are based on adult data considering high-risk NET as defined by European Neuroendocrine Tumour Society (ENETS) guidelines for completion right-sided hemicolectomy (RHC). Recent data suggest that less aggressive therapy may be justified.

Procedure: Analysis of children and adolescents with appNET prospectively registered with the German Malignant Endocrine Tumour (MET) studies between 1997 and 2022.

Results: By December 2022, 662 patients (64.7% females, 35.3% male) had been reported. Median age was 13.3 years [4.5-17.9], median duration of follow-up 2.2 years [0-10.9]. No distant metastases were reported. Tumour size was <1 cm in 63.5%, 1-2 cm in 33.2%, and >2 cm in 3.2% of patients. WHO grade 1 and 2 tumours were diagnosed in 76.9% and 23.1% of patients, respectively. Lymphovascular invasion and lymph node metastases were associated with tumour size ≥1.5 cm. 27.0% of patients presented with high-risk NET according to ENETS criteria. Of those, only 55.9% underwent secondary oncological right hemicolectomy. Neither distant metastases, nor recurrences or disease-related deaths occurred in patients with appendectomy only as well as in patients with completion RHC. Overall and event-free survival were both 100%.

Conclusions: Internationally harmonized consensus recommendations on treatment of children and adolescents with appendiceal NET are urgently needed to avoid completion RHC in high-risk patients.

Keywords: Appendix; Children and adolescents; Hemicolectomy; Neuroendocrine tumour.

MeSH terms

  • Adolescent
  • Adult
  • Appendectomy
  • Appendiceal Neoplasms* / pathology
  • Child
  • Colectomy
  • Endocrine Gland Neoplasms* / surgery
  • Female
  • Humans
  • Intestinal Neoplasms*
  • Lymphatic Metastasis
  • Male
  • Neuroendocrine Tumors* / pathology
  • Pancreatic Neoplasms*
  • Retrospective Studies
  • Stomach Neoplasms*

Supplementary concepts

  • Gastro-enteropancreatic neuroendocrine tumor