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- Title
Multicenter Survival Analysis and Application of an Olfactory Neuroblastoma Staging Modification Incorporating Hyams Grade.
- Authors
Choby, Garret; Geltzeiler, Mathew; Almeida, Joao Paulo; Champagne, Pierre-Olivier; Chan, Erik; Ciporen, Jeremy; Chaskes, Mark B.; Fernandez-Miranda, Juan; Gardner, Paul; Hwang, Peter; Ji, Keven Seung Yong; Kalyvas, Aristotelis; Kong, Keonho A.; McMillan, Ryan; Nayak, Jayakar; O'Byrne, Jamie; Patel, Chirag; Patel, Zara; Peris Celda, Maria; Pinheiro-Neto, Carlos
- Abstract
Key Points: Question: Is incorporation of Hyams grade into olfactory neuroblastoma (ONB) staging systems associated with better prediction of disease recurrence? Findings: In this multicenter case-control study of 256 patients with ONB, patients experienced excellent overall and disease-specific survival, although disease progression and recurrence were common. By incorporating Hyams grade, novel versions of tumor staging systems showed an excellent ability to estimate recurrence. Meaning: These findings suggest that incorporation of Hyams grade into traditional ONB staging systems may increase these systems' ability to estimate disease progression. This case-control study describes survival variables for olfactory neuroblastoma and examines the association of incorporating Hyams tumor grade into existing staging systems. 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.
- Publication
JAMA Otolaryngology-Head & Neck Surgery, 2023, Vol 149, Issue 9, p837
- ISSN
2168-6181
- Publication type
Article
- DOI
10.1001/jamaoto.2023.1939