CLINICAL PRESENTATION, DIAGNOSTIC UTILIZATION, AND CLINICAL BURDEN IN BING-NEEL SYNDROME: A SYSTEMATIC REVIEW OF PUBLISHED CASE REPORTS
Author(s)
Srushti M. Jaiswal1, Tanvi R. Mhetre, Pharm. D2, Pranav R. Mhetre, Pharm. D3.
1Student, Dr. D.Y. Patil Dnyan Prasad University's School of Pharmacy and Research, Pune, Pimpri , Pune, India, 2Dr. D.Y. Patil Dnyan Prasad University's School of Pharmacy and Research, Pune, Pimpri, India, 3Dr. D.Y. Patil Dnyan Prasad University's School of Pharmacy and Research, Pune, Pune, India.
1Student, Dr. D.Y. Patil Dnyan Prasad University's School of Pharmacy and Research, Pune, Pimpri , Pune, India, 2Dr. D.Y. Patil Dnyan Prasad University's School of Pharmacy and Research, Pune, Pimpri, India, 3Dr. D.Y. Patil Dnyan Prasad University's School of Pharmacy and Research, Pune, Pune, India.
OBJECTIVES: Waldenström Macroglobulinemia (WM) can develop neurological complications called Bing -Neel Syndrome (BNS) with heterogeneous clinical and diagnostic history. This study measures clinical presentation, diagnostic utilization and association between disease burden and diagnostic approaches of published BNS cases.
METHODS: A systematic review of published BNS case reports was done. Data regarding clinical manifestation, Investigation were retrieved. Descriptive analyses were done to estimate prevalence. Association of clinical domains with diagnostic modalities were evaluated with chi-square tests, multivariable logistic regression, correlation analysis and clinical burden assessment.
RESULTS: Motor symptoms (46.77%) were predominantly reported followed by sensory symptoms (39.52%), cognitive symptoms (33.87%) and psychiatric symptoms (2.42%). MRI usage was highest (49.19%), followed by biopsy (42.74%), flow cytometry (31.45%) and CT findings (25.00%). None of the clinical domains, except for cranial nerves which showed a trend for statistical association with biopsy findings (p=0.084), were associated with various diagnostic modalities (all p>0.05). Logistic regression models also failed to demonstrate any independent predictors for MRI, CT, biopsy or flow cytometry positivity. Only weak correlations between clinical and lab results (|r|<0.30) were found and all present significant disease heterogeneity. The clinical burden analysis also suggested a significantly lower burden score in patients with CT imaging than in patients without clinicians imaging (1.10 vs. 1.53; p=0.036). We did not see any significant burden differences for MRI (p=0.924), biopsy (p=0.091) or flow cytometry (p=0.950).
CONCLUSIONS: BNS is very heterogeneous and individual symptom domains have limited prognosis for diagnosis. Clinical evaluation seems to be the main determinant of diagnostic utilization rather than from particular neurological phenomena. These results show the lack of uniformity in the diagnosis, and may point to future guidance for clinical pathways in rare neurological events occurring in WM.
METHODS: A systematic review of published BNS case reports was done. Data regarding clinical manifestation, Investigation were retrieved. Descriptive analyses were done to estimate prevalence. Association of clinical domains with diagnostic modalities were evaluated with chi-square tests, multivariable logistic regression, correlation analysis and clinical burden assessment.
RESULTS: Motor symptoms (46.77%) were predominantly reported followed by sensory symptoms (39.52%), cognitive symptoms (33.87%) and psychiatric symptoms (2.42%). MRI usage was highest (49.19%), followed by biopsy (42.74%), flow cytometry (31.45%) and CT findings (25.00%). None of the clinical domains, except for cranial nerves which showed a trend for statistical association with biopsy findings (p=0.084), were associated with various diagnostic modalities (all p>0.05). Logistic regression models also failed to demonstrate any independent predictors for MRI, CT, biopsy or flow cytometry positivity. Only weak correlations between clinical and lab results (|r|<0.30) were found and all present significant disease heterogeneity. The clinical burden analysis also suggested a significantly lower burden score in patients with CT imaging than in patients without clinicians imaging (1.10 vs. 1.53; p=0.036). We did not see any significant burden differences for MRI (p=0.924), biopsy (p=0.091) or flow cytometry (p=0.950).
CONCLUSIONS: BNS is very heterogeneous and individual symptom domains have limited prognosis for diagnosis. Clinical evaluation seems to be the main determinant of diagnostic utilization rather than from particular neurological phenomena. These results show the lack of uniformity in the diagnosis, and may point to future guidance for clinical pathways in rare neurological events occurring in WM.
Conference/Value in Health Info
2026-11, ISPOR Europe 2026, Vienna, Austria
Value in Health, Volume 29, Issue 12S
Code
HSD67
Topic
Health Service Delivery & Process of Care, Study Approaches
Disease
Oncology, Personalized & Precision Medicine