Highlights
- •There is no proven treatment for adults with CCM and recurrent hemorrhages so far.
- •This case and two other recent cases suggest a beneficial effect of propranolol.
- •Future clinical studies should evaluate propranolol as treatment for aggressive CCM.
Abstract
Objective
To report a case of a patient with multiple cerebral cavernous malformations (CCM).
Observation
A 22 year old man with mild von Willebrand disease presented with two occipital CCM, one
of them with acute hemorrhage. Genetic testing for CCM1–3 did not reveal a mutation.
Over a period of 54 months, the patient suffered 26 new CCM hemorrhages despite multiple treatment attempts
including thalidomide, simvastatin and hemostatic factors. Only after initiation of
propranolol, which has already been successfully used in three children with giant
cerebral cavernoma, the occurrence of new CCM with hemorrhages was completely stopped
already at dose of 30 mg daily – now for a period of 26 months and without any other medications.
Conclusion
This case suggests a protective role for propranolol in preventing the occurrence
of new cerebral cavernoma with hemorrhages in adults with multiple CCM.
Keywords
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References
- Cavernous malformations: natural history, diagnosis and treatment.Nat. Rev. Neurol. 2009; 5: 659-670
- Genetics of cavernous angiomas.Lancet Neurol. 2007; 6: 237-244
- Antithrombotic therapy and bleeding risk in a prospective cohort study of patients with cerebral cavernous malformations.Stroke. 2012; 43: 3196-3199
- Propranolol for severe hemangiomas of infancy.N. Engl. J. Med. 2008; 358: 2649-2651
- Propranolol treatment for a giant infantile brain cavernoma.J. Child Neurol. 2010; 25: 653-655
- Propranolol for cerebral cavernous angiomatosis: a magic bullet.Clin. Pediatr. (Phila.). 2014; 53: 189-190
- A case of giant fetal intracranial capillary hemangioma cured with propranolol.J. Neurosurg. Pediatr. 2016; 29: 1-6
- Propranolol treatment of cavernous malformations with symptomatic hemorrhage.World Neurosurg. 2015; (epub, Nov 11)
- A call for rigorous study of statins in resolution of cerebral cavernous malformation pathology.Stroke. 2014; 45: 1859-1861
Article info
Publication history
Published online: May 06, 2016
Accepted:
April 27,
2016
Received in revised form:
April 5,
2016
Received:
February 11,
2016
Identification
Copyright
© 2016 Elsevier B.V. All rights reserved.
