Faculty Collaboration Database

Multicenter Survival Analysis and Application of an Olfactory Neuroblastoma Staging Modification Incorporating Hyams Grade. JAMA Otolaryngol Head Neck Surg 2023 Sep 01;149(9):837-844

Date

08/03/2023

Pubmed ID

37535372

Pubmed Central ID

PMC10401389

DOI

10.1001/jamaoto.2023.1939

Scopus ID

2-s2.0-85171309350 (requires institutional sign-in at Scopus site)   6 Citations

Abstract

IMPORTANCE: Current olfactory neuroblastoma (ONB) staging systems inadequately delineate locally advanced tumors, do not incorporate tumor grade, and poorly estimate survival and recurrence.

OBJECTIVE: The primary aims of this study were to (1) examine the clinical covariates associated with survival and recurrence of ONB in a modern-era multicenter cohort and (2) incorporate Hyams tumor grade into existing staging systems to assess its ability to estimate survival and recurrence.

DESIGN, SETTING, AND PARTICIPANTS: This retrospective, multicenter, case-control study included patients with ONB who underwent treatment between January 1, 2005, and December 31, 2021, at 9 North American academic medical centers.

INTERVENTION: Standard-of-care ONB treatment.

MAIN OUTCOME AND MEASURES: The main outcomes were overall survival (OS), disease-free survival (DFS), and disease-specific survival (DSS) as C statistics for model prediction.

RESULTS: A total of 256 patients with ONB (mean [SD] age, 52.0 [15.6] years; 115 female [44.9%]; 141 male [55.1%]) were included. The 5-year rate for OS was 83.5% (95% CI, 78.3%-89.1%); for DFS, 70.8% (95% CI, 64.3%-78.0%); and for DSS, 94.1% (95% CI, 90.5%-97.8%). On multivariable analysis, age, American Joint Committee on Cancer (AJCC) stage, involvement of bilateral maxillary sinuses, and positive margins were associated with OS. Only AJCC stage was associated with DFS. Only N stage was associated with DSS. When assessing the ability of staging systems to estimate OS, the best-performing model was the novel modification of the Dulguerov system (C statistic, 0.66; 95% CI, 0.59-0.76), and the Kadish system performed most poorly (C statistic, 0.57; 95% CI, 0.50-0.63). Regarding estimation of DFS, the modified Kadish system performed most poorly (C statistic, 0.55; 95% CI, 0.51-0.66), while the novel modification of the AJCC system performed the best (C statistic, 0.70; 95% CI, 0.66-0.80). Regarding estimation of DSS, the modified Kadish system was the best-performing model (C statistic, 0.79; 95% CI, 0.70-0.94), and the unmodified Kadish performed the worst (C statistic, 0.56; 95% CI, 0.51-0.68). The ability for novel ONB staging systems to estimate disease progression across stages was also assessed. In the novel Kadish staging system, patients with stage VI disease were approximately 7 times as likely to experience disease progression as patients with stage I disease (hazard ratio [HR], 6.84; 95% CI, 1.60-29.20). Results were similar for the novel modified Kadish system (HR, 8.99; 95% CI, 1.62-49.85) and the novel Dulguerov system (HR, 6.86; 95% CI, 2.74-17.18).

CONCLUSIONS AND RELEVANCE: The study findings indicate that 5-year OS for ONB is favorable and that incorporation of Hyams grade into traditional ONB staging systems is associated with improved estimation of disease progression.

Author List

Choby G, Geltzeiler M, Almeida JP, Champagne PO, Chan E, Ciporen J, Chaskes MB, Fernandez-Miranda J, Gardner P, Hwang P, Ji KSY, Kalyvas A, Kong KA, McMillan R, Nayak J, O'Byrne J, Patel C, Patel Z, Peris Celda M, Pinheiro-Neto C, Sanusi O, Snyderman C, Thorp BD, Van Gompel JJ, Young SC, Zenonos G, Zwagerman NT, Wang EW

Author

Nathan Zwagerman MD Associate Professor in the Neurosurgery department at Medical College of Wisconsin

MESH terms used to index this publication - Major topics in bold

Case-Control Studies
Disease Progression
Esthesioneuroblastoma, Olfactory
Female
Humans
Male
Middle Aged
Nasal Cavity
Neoplasm Staging
Nose Neoplasms
Retrospective Studies
Survival Analysis