Comparing Characteristics and Treatment of Brain Vascular Malformations in Children and Adults with HHT
Abstract
:1. Introduction
2. Materials and Methods
2.1. Cohort
2.2. Analysis
3. Results
3.1. Demographics
3.2. Clinical Presentation of HHT-Related Brain VMs
3.3. Treatment of HHT Related Brain VMs
3.4. Treatment for Brain VMs Complicated by ICH
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Acknowledgments
Conflicts of Interest
References
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Characteristic (n, %) | Pediatric Patients n = 114 | Adult Patients n = 253 | p-Value |
---|---|---|---|
Female | 62 (54.4%) | 161 (63.6%) | 0.076 |
Age at diagnosis (years) | - | ||
Mean | 9.2 | 41.1 | |
Standard deviation | ±5.5 | ±14.6 | |
Median | 10 | 40 | |
Range | Birth-17 | 18–77 | |
Interquartile Range | 5–14 | 28–54 | |
Number of brain VMs (range) | 1–4 | 1–10 | - |
>1 brain VM | 15 (13.1%) | 38 (15.0%) | 0.638 |
Diagnosed by screening | 63 (56.1%) | 168 (66.4%) | 0.034 |
Asymptomatic at presentation | 54 (47.4%) | 160 (63.2%) | 0.004 |
ICH at presentation | 27 (23.7%) | 25 (9.9%) | <0.001 |
Ischemic stroke at presentation | 14 (12.3%) | 24 (9.5%) | 0.417 |
Focal neurological deficit | 10 (8.8%) | 30 (11.9%) | 0.379 |
Seizure | 21 (18.4%) | 19 (7.5%) | 0.002 |
Headache | 34 (29.8%) | 53 (20.9%) | 0.064 |
Treatment Modality (n, %) | Pediatric Patients n = 114 | Adult Patients n = 253 | p-Value |
---|---|---|---|
Treatment for brain VM | 69 (60.5%) | 128 (50.6%) | 0.076 |
n = 69 | n = 128 | ||
Surgery only | 34 (49.3%) | 43 (33.5%) | 0.032 |
Embolization only | 19 (27.5%) | 16 (12.5%) | 0.008 |
Radiosurgery only | 6 (8.7%) | 38 (29.7%) | <0.001 |
Multiple modalities | 10 (14.58%) | 31 (24.2%) | 0.110 |
Surgery + Embolization | 7 (10.1%) | 18 (14.1%) | 0.429 |
Surgery + Radiosurgery | 1 (1.5%) | 5 (3.9%) | 0.246 |
Embolization + Radiosurgery | 2 (3.0%) | 7 (5.4%) | 0.337 |
Embolization + Radiosurgery + Surgery | 0 (0.0%) | 1 (0.01%) | 0.459 |
Post-treatment hemorrhage | 3 (4.3%) | 9 (6.9%) | 0.453 |
Incidence of Post-Treatment Hemorrhage | |||
---|---|---|---|
Intervention | Pediatric Patients | Adult Patients | p-Value |
Surgery | 0/42 (0.0%) | 1/67 (1.4%) | 0.423 |
Embolization | 2/28 (7.1%) | 4/42 (9.5%) | 0.726 |
Radiosurgery | 1/9 (11.1%) | 4/51 (7.8%) | 0.741 |
Timing of Post-Treatment Hemorrhage | ||||||
---|---|---|---|---|---|---|
Intervention | Acute (<30 Days) | Remote (>30 Days) | Range | Missing Data | ||
Pediatric | Adults | Pediatric | Adults | |||
Surgery | 0/42 (0.0%) | 0/67 (0.0%) | 0/42 (0.0%) | 1/67 (1.4%) | 15 years | 0 |
Embolization | 0/28 (0.0%) | 2/42 (4.8%) | 1/28 (3.5%) | 1/42 (2.3%) | 1–118 days | 2 |
Radiosurgery | 0/9 (0.0%) | 0/51 (0.0%) | 1/9 (11.1%) | 1/51 (1.9%) | 1–2 years | 3 |
Initial ICH | p-Value | ||
---|---|---|---|
Pediatric (27/114, 23.7%) | Adult Patients (25/253, 9.9%) | ||
n = 27 | n = 25 | ||
Underwent treatment for brain VM | 23 (85.1%) | 22 (88.0%) | 0.764 |
Surgery | 17 (63.0%) | 15 (60.0%) | 0.825 |
Embolization | 6 (22.2%) | 5 20.0%) | 0.888 |
Radiosurgery | 0 (0%) | 5 (20.0%) | 0.423 |
Multiple modalities | 0 (0%) | 3 (12.0%) | 0.118 |
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Kilian, A.; Latino, G.A.; White, A.J.; Ratjen, F.; McDonald, J.; Whitehead, K.J.; Gossage, J.R.; Krings, T.; Lawton, M.T.; Kim, H.; et al. Comparing Characteristics and Treatment of Brain Vascular Malformations in Children and Adults with HHT. J. Clin. Med. 2023, 12, 2704. https://doi.org/10.3390/jcm12072704
Kilian A, Latino GA, White AJ, Ratjen F, McDonald J, Whitehead KJ, Gossage JR, Krings T, Lawton MT, Kim H, et al. Comparing Characteristics and Treatment of Brain Vascular Malformations in Children and Adults with HHT. Journal of Clinical Medicine. 2023; 12(7):2704. https://doi.org/10.3390/jcm12072704
Chicago/Turabian StyleKilian, Alexandra, Giuseppe A. Latino, Andrew J. White, Felix Ratjen, Jamie McDonald, Kevin J. Whitehead, James R. Gossage, Timo Krings, Michael T. Lawton, Helen Kim, and et al. 2023. "Comparing Characteristics and Treatment of Brain Vascular Malformations in Children and Adults with HHT" Journal of Clinical Medicine 12, no. 7: 2704. https://doi.org/10.3390/jcm12072704
APA StyleKilian, A., Latino, G. A., White, A. J., Ratjen, F., McDonald, J., Whitehead, K. J., Gossage, J. R., Krings, T., Lawton, M. T., Kim, H., Faughnan, M. E., & The Brain Vascular Malformation Consortium HHT Investigator Group. (2023). Comparing Characteristics and Treatment of Brain Vascular Malformations in Children and Adults with HHT. Journal of Clinical Medicine, 12(7), 2704. https://doi.org/10.3390/jcm12072704