Embolization of Recurrent Pulmonary Arteriovenous Malformations by Ethylene Vinyl Alcohol Copolymer (Onyx®) in Hereditary Hemorrhagic Telangiectasia: Safety and Efficacy
Abstract
:1. Introduction
2. Materials and Methods
2.1. Study Design
2.2. Study Population
2.3. Clinical and Biological Data
2.4. Follow-Up Imaging Protocol
2.5. Pulmonary Arteriovenous Malformation Imaging
2.6. Embolization
2.7. Statistical Analysis
3. Results
3.1. Study Population
3.2. PAVM Characteristics before Embolization
3.3. Safety
3.4. Short-Term Efficacy
3.5. Long-Term Efficacy
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Acknowledgments
Conflicts of Interest
Appendix A
References
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Population | 45 | |
---|---|---|
Women | 23 (51) | |
Mean age (SD) (years) | 53 (18) | |
Mean BMI (SD) | 25.8 (6.2) | |
Diabetes mellitus | 4 (9) | |
Tobacco | 18 (40) | |
Mean oxygen saturation (range) | 96.3 (92–100) | |
Curacao criteria | ||
Family history of HHT symptoms | 45 | |
Epistaxis | 43 (96) | |
Telangiectasia | 41 (91) | |
Liver AVM | 7 (16) | |
Gastro-intestinal AVM | 5 (11) | |
Brain AVM | 3 (7) | |
HHT severe complications | ||
Hemoptysis | 3 (7) | |
Brain abscess | 5 (11) | |
Stroke | 7 (16) | |
Mutation | ||
HHT1/ENG | 36 (80) | |
HHT2/ALK1 | 5 (11) | |
SMAD4 | 0 (0) | |
Unknown/unconfirmed | 4 (9) | |
Unique PAVM | 18 (40) | |
Multiple PAVM | 27 (60) |
Criteria | Total | Long-Term Occlusion | Short-Term Occlusion | p | |
---|---|---|---|---|---|
Previous procedures | 70 (100) | 42 (60) | 28 (40) | NA | |
PAVMs naive from Onyx® | 64 (91) | 40 (95) | 24 (86) | 0.166 | |
PAVMs previously treated with Onyx® | 6 (9) | 2 (5) | 4 (14) | 0.166 | |
Simple PAVMs | 61 (87) | 36 (86) | 25 (89) | 0.664 | |
Complex PAVMs | 9 (14) | 6 (15) | 3 (13) | 0.664 | |
Mean number of embolizations before onyx per PAVM (SD) | 2.0 (1.1) | 1.8 (1.1) | 2.5 (1.3) | 0.01 | |
Mean number of recurrence before Onyx® per PAVM | 0.9 (1.1) | 0.7 (1.0) | 1.4 (1.2) | <0.01 | |
PAVMs first treated with coils | 70 (100) | 42 (100) | 28 (100) | 1.00 | |
PAVMs first treated with plugs and coils | 2 (3) | 2 (5) | 0 | 0.245 | |
Length between aneurysm and plug/coil < 10 mm | 43 (61) | 27 (64) | 16 (57) | 0.550 | |
Vein diameter (mm) | 4.2 (3.7) | 3.4 (1.0) | 5.5 (5.6) | <0.01 | |
Aneurysm diameter (mm) | 5.1 (5.4) | 3.4 (4.1) | 8 (6.1) | <0.01 | |
Lobar location | |||||
Upper right lobe | 13 (19) | 9 (21) | 4 (14) | 0.455 | |
Middle lobe | 7 (10) | 4 (10) | 3 (11) | 0.872 | |
Lower right lobe | 22 (31) | 11 (26) | 11 (39) | 0.251 | |
Upper left lobe | 6 (9) | 5 (12) | 1 (4) | 0.226 | |
Lower left lobe | 22 (31) | 13 (31) | 9 (32) | 0.917 | |
Absence of safety distance | 33 (47) | 17 (60) | 16 (43) | 0.174 | |
Mechanism of recurrence | |||||
Recanalization | 69 (98) | 41 (98) | 28 (100) | 0.414 | |
Reperfusion | 10 (14) | 7 (17) | 3 (11) | 0.489 | |
Both | 9 (13) | 6 (67) | 3 (23) | 0.664 | |
Incomplete primary treatment | 0 | 0 | 0 | 1.000 | |
Territory potentially at risk | |||||
Lobar | 2 (3) | 1 (2) | 1 (4) | 0.771 | |
Segmental | 18 (26) | 9 (21) | 9 (32) | 0.318 | |
Sub-segmental | 50 (71) | 32 (76) | 18 (64) | 0.284 |
Immediate Embolization Characteristics | Total | Long-Term Occlusion | Short-Term Occlusion | p | |
---|---|---|---|---|---|
Per-embolization occlusion | 70 (100) | 42 (60) | 28 (40) | 1.00 | |
Treatment type | |||||
Onyx® only | 53 (76) | 31 (74) | 22 (79) | 0.65 | |
Onyx® + coils | 17(24) | 11 (26) | 6 (21) | 0.65 | |
Onyx® volume, mL (SD) | 0.6 (0.5) | 0.5 (0.3) | 0.7 (0.6) | 0.23 | |
Onyx® distribution ** | |||||
Upstream | 48 (69) | 30 (71) | 18 (64) | 0.53 | |
Inside coiling | 51 (73) | 28 (67) | 23 (82) | 0.16 | |
Downstream | 13 (19) | 7 (17) | 6 (21) | 0.62 | |
Upstream + inside | 29 (41) | 16 (38) | 13 (46) | 0.49 | |
Downstream + inside | 13 (19) | 7 (17) | 6 (21) | 0.62 | |
Upstream + inside + downstream | 5 (7) | 2 (5) | 3 (11) | 0.35 | |
Upstream leak outside the target | |||||
At a lobar level | 0 | 0 | 0 | 1.00 | |
At a segmental level | 4 (6) | 2 (5) | 2 (7) | 1.00 | |
At a sub-segmental level | 39 (56) | 25 (60) | 14 (50) | 0.68 | |
Perfusion defect in healthy territory | |||||
Lobar territory | 0 | 0 | 0 | 1.00 | |
Segmental territory | 0 | 0 | 0 | 1.00 | |
Sub-segmental territory | 20 (29) | 13 (31) | 7 (25) | 0.59 | |
Downstream leak in draining vein or aneurysmal sac | 0 | 0 | 0 | 1.00 | |
Downstream leak in systemic circulation | 0 | 0 | 0 | 1.00 | |
Procedure time, min (SD) | 105 (34) | 110 (33) | 99 (35) | 0.15 | |
Volume of contrast agent, mL (SD) | 110 (49) | 116 (50) | 102 (47) | 0.19 | |
Adverse events during hospitalization time | 5 (7.1) | 2 (2.8) | 3 (4.3) | 0.35 | |
Allergy | 1 (20) | 0 | 1 (33.3) | 0.22 | |
Chest pain | 1 (20) | 0 | 1 (33.3) | 0.22 | |
Pleural effusion | 0 | 0 | 0 | 1.00 | |
Lung distal infarction | 3 (60) | 2 (100) | 1 (33.3) | 0.81 | |
Lung infection | 0 | 0 | 0 | 1.00 | |
Brain abscess | 0 | 0 | 0 | 1.00 | |
Stroke | 0 | 0 | 0 | 1.00 | |
Access site complication | 0 | 0 | 0 | 1.00 |
Long Time Follow-Up | Total | Long-Term Occlusion | Short-Term Occlusion | p | |
---|---|---|---|---|---|
Number of PAVMs | 70 (100) | 42 (60) | 28 (40) | ||
Follow-up time, months (SD) | 34.8 (15.3) | 25.2 (13.0) | 34.1 (16.7) | 0.5 | |
Tobacco consumption | 28 (40) | 14 (33.3) | 14 (50) | 0.111 | |
Pack-year of tobacco (SD) | 9.3 (17.3) | 8.8 (2.8) | 11.8 (3.1) | 0.133 | |
PAVM characteristics | |||||
Aneurysm diameter, mm (SD) | 3.9 (4.7) | 2.1 (3.2) | 7.0 (5.1) | <0.001 | |
Vein diameter, mm (SD) | 2.9 (1.2) | 2.4 (0.9) | 3.8 (1.0) | <0.001 | |
Difference in aneurysm diameter, mm (SD) * | 22.9 (37.2) | 40.5 (39.1) | 4.4 (24.4) | <0.01 | |
Difference in vein diameter, mm (SD) * | 18.7 (21.7) | 29.9 (18.2) | 0.04 (12.1) | <0.001 | |
Complications | |||||
Brain abscess | 0 | 0 | 0 | ||
Stroke | 0 | 0 | 0 | ||
Hemoptysis | 2 | 0 | 2 | ||
Hemothorax | 0 | 0 | 0 |
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Si-Mohamed, S.A.; Cierco, A.; Gamondes, D.; Restier, L.M.; Delagrange, L.; Cottin, V.; Dupuis-Girod, S.; Revel, D. Embolization of Recurrent Pulmonary Arteriovenous Malformations by Ethylene Vinyl Alcohol Copolymer (Onyx®) in Hereditary Hemorrhagic Telangiectasia: Safety and Efficacy. J. Pers. Med. 2022, 12, 1091. https://doi.org/10.3390/jpm12071091
Si-Mohamed SA, Cierco A, Gamondes D, Restier LM, Delagrange L, Cottin V, Dupuis-Girod S, Revel D. Embolization of Recurrent Pulmonary Arteriovenous Malformations by Ethylene Vinyl Alcohol Copolymer (Onyx®) in Hereditary Hemorrhagic Telangiectasia: Safety and Efficacy. Journal of Personalized Medicine. 2022; 12(7):1091. https://doi.org/10.3390/jpm12071091
Chicago/Turabian StyleSi-Mohamed, Salim A., Alexandra Cierco, Delphine Gamondes, Lauria Marie Restier, Laura Delagrange, Vincent Cottin, Sophie Dupuis-Girod, and Didier Revel. 2022. "Embolization of Recurrent Pulmonary Arteriovenous Malformations by Ethylene Vinyl Alcohol Copolymer (Onyx®) in Hereditary Hemorrhagic Telangiectasia: Safety and Efficacy" Journal of Personalized Medicine 12, no. 7: 1091. https://doi.org/10.3390/jpm12071091
APA StyleSi-Mohamed, S. A., Cierco, A., Gamondes, D., Restier, L. M., Delagrange, L., Cottin, V., Dupuis-Girod, S., & Revel, D. (2022). Embolization of Recurrent Pulmonary Arteriovenous Malformations by Ethylene Vinyl Alcohol Copolymer (Onyx®) in Hereditary Hemorrhagic Telangiectasia: Safety and Efficacy. Journal of Personalized Medicine, 12(7), 1091. https://doi.org/10.3390/jpm12071091