Diagnosis and management of urinary bladder paragangliomas: A Sino-American-European retrospective observational study

Yingxian Pang; Jing Zhang; Jingjing Jiang; Christina Pamporaki; Minghao Li; Nicole Bechmann; Leah Meuter; Yongbao Wei; Haijian Huang; Shenghui Huang; Xunbin Yu; Mercedes Robledo; Miguel J Soria; Dewen Zhong; Shangyuan Xu; Henri J L M Timmers; Johan F Langenhuijsen; Xiaofeng Chen; Wanglong Deng; Timo Deutschbein; Hanna Remde; Long Wang; Hanyu Yao; Bin Yan; Annika M A Berends; Michiel N Kerstens; Yazhuo Jiang; Joakim Crona; Ning Xu; Hai Cai; Yanlin Wen; Anguo Wang; Ji Wu; Zongpin Zhang; Jinzhuo Ning; Fan Cheng; Xiang Chen; Jing Wang; Bin Xie; Danlei Chen; Yujun Liu; Longfei Liu; Karel Pacak; Graeme Eisenhofer; Jacques W M Lenders

doi:10.1111/cen.15058

Diagnosis and management of urinary bladder paragangliomas: A Sino-American-European retrospective observational study

Yingxian Pang, Jing Zhang, Jingjing Jiang, Christina Pamporaki, Minghao Li, Nicole Bechmann, Leah Meuter, Yongbao Wei, Haijian Huang, Shenghui Huang, Xunbin Yu, Mercedes Robledo, Miguel J Soria, Dewen Zhong, Shangyuan Xu, Henri J L M Timmers, Johan F Langenhuijsen, Xiaofeng Chen, Wanglong Deng, Timo DeutschbeinHanna Remde, Long Wang, Hanyu Yao, Bin Yan, Annika M A Berends, Michiel N Kerstens, Yazhuo Jiang, Joakim Crona, Ning Xu, Hai Cai, Yanlin Wen, Anguo Wang, Ji Wu, Zongpin Zhang, Jinzhuo Ning, Fan Cheng, Xiang Chen, Jing Wang, Bin Xie, Danlei Chen, Yujun Liu^*, Longfei Liu^*, Karel Pacak, Graeme Eisenhofer, Jacques W M Lenders

^*Corresponding author voor dit werk

Guided Treatment in Optimal Selected Cancer Patients (GUTS)

Onderzoeksoutput › Academic › peer review

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OBJECTIVE: Paragangliomas of the urinary bladder (UBPGLs) are rare neuroendocrine tumours and pose a diagnostic and surgical challenge. It remains unclear what factors contribute to a timely presurgical diagnosis. The purpose of this study is to identify factors contributing to missing the diagnosis of UBPGLs before surgery.

DESIGN, PATIENTS AND MEASUREMENTS: A total of 73 patients from 11 centres in China, and 51 patients from 6 centres in Europe and 1 center in the United States were included. Clinical, surgical and genetic data were collected and compared in patients diagnosed before versus after surgery. Logistic regression analysis was used to identify clinical factors associated with initiation of presurgical biochemical testing.

RESULTS: Among all patients, only 47.6% were diagnosed before surgery. These patients were younger (34.0 vs. 54.0 years, p < .001), had larger tumours (2.9 vs. 1.8 cm, p < .001), and more had a SDHB pathogenic variant (54.7% vs. 11.9%, p < .001) than those diagnosed after surgery. Patients with presurgical diagnosis presented with more micturition spells (39.7% vs. 15.9%, p = .003), hypertension (50.0% vs. 31.7%, p = .041) and catecholamine-related symptoms (37.9% vs. 17.5%, p = .012). Multivariable logistic analysis revealed that presence of younger age (<35 years, odds ratio [OR] = 6.47, p = .013), micturition spells (OR = 6.79, p = .007), hypertension (OR = 3.98, p = .011), and sweating (OR = 41.72, p = .013) increased the probability of initiating presurgical biochemical testing.

CONCLUSIONS: Most patients with UBPGL are diagnosed after surgery. Young age, hypertension, micturition spells and sweating are clues in assisting to initiate early biochemical testing and thus may establish a timely presurgical diagnosis.

Originele taal-2	English
Tijdschrift	Clinical Endocrinology
DOI's	https://doi.org/10.1111/cen.15058
Status	E-pub ahead of print - 12-apr.-2024

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Pang, Y., Zhang, J., Jiang, J., Pamporaki, C., Li, M., Bechmann, N., Meuter, L., Wei, Y., Huang, H., Huang, S., Yu, X., Robledo, M., Soria, M. J., Zhong, D., Xu, S., Timmers, H. J. L. M., Langenhuijsen, J. F., Chen, X., Deng, W., ... Lenders, J. W. M. (2024). Diagnosis and management of urinary bladder paragangliomas: A Sino-American-European retrospective observational study. Clinical Endocrinology. Online publicatie vooraf. https://doi.org/10.1111/cen.15058

@article{241f745fa805401990a13ab675bb1cdd,

title = "Diagnosis and management of urinary bladder paragangliomas: A Sino-American-European retrospective observational study",

abstract = "OBJECTIVE: Paragangliomas of the urinary bladder (UBPGLs) are rare neuroendocrine tumours and pose a diagnostic and surgical challenge. It remains unclear what factors contribute to a timely presurgical diagnosis. The purpose of this study is to identify factors contributing to missing the diagnosis of UBPGLs before surgery.DESIGN, PATIENTS AND MEASUREMENTS: A total of 73 patients from 11 centres in China, and 51 patients from 6 centres in Europe and 1 center in the United States were included. Clinical, surgical and genetic data were collected and compared in patients diagnosed before versus after surgery. Logistic regression analysis was used to identify clinical factors associated with initiation of presurgical biochemical testing.RESULTS: Among all patients, only 47.6% were diagnosed before surgery. These patients were younger (34.0 vs. 54.0 years, p < .001), had larger tumours (2.9 vs. 1.8 cm, p < .001), and more had a SDHB pathogenic variant (54.7% vs. 11.9%, p < .001) than those diagnosed after surgery. Patients with presurgical diagnosis presented with more micturition spells (39.7% vs. 15.9%, p = .003), hypertension (50.0% vs. 31.7%, p = .041) and catecholamine-related symptoms (37.9% vs. 17.5%, p = .012). Multivariable logistic analysis revealed that presence of younger age (<35 years, odds ratio [OR] = 6.47, p = .013), micturition spells (OR = 6.79, p = .007), hypertension (OR = 3.98, p = .011), and sweating (OR = 41.72, p = .013) increased the probability of initiating presurgical biochemical testing.CONCLUSIONS: Most patients with UBPGL are diagnosed after surgery. Young age, hypertension, micturition spells and sweating are clues in assisting to initiate early biochemical testing and thus may establish a timely presurgical diagnosis.",

author = "Yingxian Pang and Jing Zhang and Jingjing Jiang and Christina Pamporaki and Minghao Li and Nicole Bechmann and Leah Meuter and Yongbao Wei and Haijian Huang and Shenghui Huang and Xunbin Yu and Mercedes Robledo and Soria, {Miguel J} and Dewen Zhong and Shangyuan Xu and Timmers, {Henri J L M} and Langenhuijsen, {Johan F} and Xiaofeng Chen and Wanglong Deng and Timo Deutschbein and Hanna Remde and Long Wang and Hanyu Yao and Bin Yan and Berends, {Annika M A} and Kerstens, {Michiel N} and Yazhuo Jiang and Joakim Crona and Ning Xu and Hai Cai and Yanlin Wen and Anguo Wang and Ji Wu and Zongpin Zhang and Jinzhuo Ning and Fan Cheng and Xiang Chen and Jing Wang and Bin Xie and Danlei Chen and Yujun Liu and Longfei Liu and Karel Pacak and Graeme Eisenhofer and Lenders, {Jacques W M}",

note = "{\textcopyright} 2024 John Wiley & Sons Ltd.",

year = "2024",

month = apr,

day = "12",

doi = "10.1111/cen.15058",

language = "English",

journal = "Clinical Endocrinology",

issn = "0300-0664",

publisher = "Wiley",

}

Pang, Y, Zhang, J, Jiang, J, Pamporaki, C, Li, M, Bechmann, N, Meuter, L, Wei, Y, Huang, H, Huang, S, Yu, X, Robledo, M, Soria, MJ, Zhong, D, Xu, S, Timmers, HJLM, Langenhuijsen, JF, Chen, X, Deng, W, Deutschbein, T, Remde, H, Wang, L, Yao, H, Yan, B, Berends, AMA , Kerstens, MN, Jiang, Y, Crona, J, Xu, N, Cai, H, Wen, Y, Wang, A, Wu, J, Zhang, Z, Ning, J, Cheng, F, Chen, X, Wang, J, Xie, B, Chen, D, Liu, Y, Liu, L, Pacak, K, Eisenhofer, G & Lenders, JWM 2024, 'Diagnosis and management of urinary bladder paragangliomas: A Sino-American-European retrospective observational study', Clinical Endocrinology. https://doi.org/10.1111/cen.15058

TY - JOUR

T1 - Diagnosis and management of urinary bladder paragangliomas

T2 - A Sino-American-European retrospective observational study

AU - Pang, Yingxian

AU - Zhang, Jing

AU - Jiang, Jingjing

AU - Pamporaki, Christina

AU - Li, Minghao

AU - Bechmann, Nicole

AU - Meuter, Leah

AU - Wei, Yongbao

AU - Huang, Haijian

AU - Huang, Shenghui

AU - Yu, Xunbin

AU - Robledo, Mercedes

AU - Soria, Miguel J

AU - Zhong, Dewen

AU - Xu, Shangyuan

AU - Timmers, Henri J L M

AU - Langenhuijsen, Johan F

AU - Chen, Xiaofeng

AU - Deng, Wanglong

AU - Deutschbein, Timo

AU - Remde, Hanna

AU - Wang, Long

AU - Yao, Hanyu

AU - Yan, Bin

AU - Berends, Annika M A

AU - Kerstens, Michiel N

AU - Jiang, Yazhuo

AU - Crona, Joakim

AU - Xu, Ning

AU - Cai, Hai

AU - Wen, Yanlin

AU - Wang, Anguo

AU - Wu, Ji

AU - Zhang, Zongpin

AU - Ning, Jinzhuo

AU - Cheng, Fan

AU - Chen, Xiang

AU - Wang, Jing

AU - Xie, Bin

AU - Chen, Danlei

AU - Liu, Yujun

AU - Liu, Longfei

AU - Pacak, Karel

AU - Eisenhofer, Graeme

AU - Lenders, Jacques W M

PY - 2024/4/12

Y1 - 2024/4/12

N2 - OBJECTIVE: Paragangliomas of the urinary bladder (UBPGLs) are rare neuroendocrine tumours and pose a diagnostic and surgical challenge. It remains unclear what factors contribute to a timely presurgical diagnosis. The purpose of this study is to identify factors contributing to missing the diagnosis of UBPGLs before surgery.DESIGN, PATIENTS AND MEASUREMENTS: A total of 73 patients from 11 centres in China, and 51 patients from 6 centres in Europe and 1 center in the United States were included. Clinical, surgical and genetic data were collected and compared in patients diagnosed before versus after surgery. Logistic regression analysis was used to identify clinical factors associated with initiation of presurgical biochemical testing.RESULTS: Among all patients, only 47.6% were diagnosed before surgery. These patients were younger (34.0 vs. 54.0 years, p < .001), had larger tumours (2.9 vs. 1.8 cm, p < .001), and more had a SDHB pathogenic variant (54.7% vs. 11.9%, p < .001) than those diagnosed after surgery. Patients with presurgical diagnosis presented with more micturition spells (39.7% vs. 15.9%, p = .003), hypertension (50.0% vs. 31.7%, p = .041) and catecholamine-related symptoms (37.9% vs. 17.5%, p = .012). Multivariable logistic analysis revealed that presence of younger age (<35 years, odds ratio [OR] = 6.47, p = .013), micturition spells (OR = 6.79, p = .007), hypertension (OR = 3.98, p = .011), and sweating (OR = 41.72, p = .013) increased the probability of initiating presurgical biochemical testing.CONCLUSIONS: Most patients with UBPGL are diagnosed after surgery. Young age, hypertension, micturition spells and sweating are clues in assisting to initiate early biochemical testing and thus may establish a timely presurgical diagnosis.

AB - OBJECTIVE: Paragangliomas of the urinary bladder (UBPGLs) are rare neuroendocrine tumours and pose a diagnostic and surgical challenge. It remains unclear what factors contribute to a timely presurgical diagnosis. The purpose of this study is to identify factors contributing to missing the diagnosis of UBPGLs before surgery.DESIGN, PATIENTS AND MEASUREMENTS: A total of 73 patients from 11 centres in China, and 51 patients from 6 centres in Europe and 1 center in the United States were included. Clinical, surgical and genetic data were collected and compared in patients diagnosed before versus after surgery. Logistic regression analysis was used to identify clinical factors associated with initiation of presurgical biochemical testing.RESULTS: Among all patients, only 47.6% were diagnosed before surgery. These patients were younger (34.0 vs. 54.0 years, p < .001), had larger tumours (2.9 vs. 1.8 cm, p < .001), and more had a SDHB pathogenic variant (54.7% vs. 11.9%, p < .001) than those diagnosed after surgery. Patients with presurgical diagnosis presented with more micturition spells (39.7% vs. 15.9%, p = .003), hypertension (50.0% vs. 31.7%, p = .041) and catecholamine-related symptoms (37.9% vs. 17.5%, p = .012). Multivariable logistic analysis revealed that presence of younger age (<35 years, odds ratio [OR] = 6.47, p = .013), micturition spells (OR = 6.79, p = .007), hypertension (OR = 3.98, p = .011), and sweating (OR = 41.72, p = .013) increased the probability of initiating presurgical biochemical testing.CONCLUSIONS: Most patients with UBPGL are diagnosed after surgery. Young age, hypertension, micturition spells and sweating are clues in assisting to initiate early biochemical testing and thus may establish a timely presurgical diagnosis.

U2 - 10.1111/cen.15058

DO - 10.1111/cen.15058

M3 - Article

C2 - 38606576

SN - 0300-0664

JO - Clinical Endocrinology

JF - Clinical Endocrinology

ER -

Diagnosis and management of urinary bladder paragangliomas: A Sino-American-European retrospective observational study

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