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Moyamoya disease associated with persistent primitive trigeminal artery. Report of two cases
Abstract:
Two cases of moyamoya disease associated with persistent primitive trigeminal artery (PTA) are reported. The first patient was a 44-year-old man who experienced a sudden severe headache brought about by an intracerebral hematoma in the left temporoparietal lobe. Four-vessel study showed a right-sided PTA and moyamoya disease. The second patient was a 56-year-old woman with similar symptoms and a hematoma in the right temporoparietal lobe. Four-vessel study showed a left-sided PTA and moyamoya disease. Among the 212 PTA cases reported in the literature, none has been associated with moyamoya disease. Moreover, there are no cases of moyamoya disease among the 119 cases of persistent primitive hypoglossal artery (PHA), which is thought to be a vascular anomaly fundamentally similar to PTA. None of the 907 cases of moyamoya disease reported in Japan has been associated with either PTA or PHA. Nonetheless, the embryonic stage when PTA or PHA normally disappears partially overlaps that period when moyamoya-like vascular anomalies have been thought to arise. The possible developmental relationship between these two varieties of vascular abnormality is discussed.
Insights
This study reports two rare cases of moyamoya disease linked with persistent primitive trigeminal artery (PTA). These findings suggest a potential developmental link between these distinct cerebrovascular conditions.
Area of Science:
- Neurology
- Vascular Biology
- Developmental Biology
Background:
- Moyamoya disease is a rare cerebrovascular disorder characterized by progressive stenosis of the internal carotid arteries.
- Persistent primitive trigeminal artery (PTA) is a congenital vascular anomaly representing a remnant of an embryonic artery.
Observation:
- Two patients presented with intracerebral hematomas and were diagnosed with moyamoya disease.
- Both patients were found to have a persistent primitive trigeminal artery (PTA) on four-vessel studies.
Findings:
- This is the first reported association of moyamoya disease with PTA in the literature.
- No cases of moyamoya disease were found associated with PTA or persistent primitive hypoglossal artery (PHA) in extensive literature reviews.
Implications:
- The findings suggest a potential shared embryonic developmental pathway for moyamoya disease and PTA.
- Further research into the embryogenesis of cerebrovascular anomalies may elucidate the relationship between moyamoya disease and PTA.