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Two CASES OF INTRACRANIAL FIBROMUSCULAR DYSPLASIA WHOSE REPEATED ANGIOGRAPHY DISCLOSED PROGRESSION OF THE LESION Shunichi Fujimoto 1,3 , Takamasa Kayama 1 , Akira Ogawa 1 , Yoshiharu Sakurai 1 , Takashi Yoshimoto 2 , Jiro Suzuki 2 1Neurosurgical Department, Stroke Center of Sendai National Hospital 2Division of Neurosurgery, Institute of Brain Diseases, Tohoku University School of Medicine 3Present Address : Department of Neurosurgery, Obihiro Daiichi Hospital pp.937-945
Published Date 1987/10/1
DOI https://doi.org/10.11477/mf.1406205986
  • Abstract
  • Look Inside

Fibromuscular dysplasia (FMD) is well known owing to the characteristic angiographical finding of a "string of beads" appearance, but intracranial involvement with this disease is extremely rare. Moreover, to our knowledge, only seven cases that had repeated angiograms disclosed progres-sion of FMD lesion in the literature.

Such cases of intracranial FMD which showed progression in the follow-up angiography are re-ported.

Case 1: A 8-year-old boy was referred to our hospital because of aphasia and right hemiplegia following right hemiconvulsion. Left carotid angio-graphy on the 7th day from the onset revealed a "string of beads" appearance involving the left middle cerebral artery from M1 to M2 portion. He was treated with low molecular dextran, uro-kinase and steroid. After these drugs were admi-nistered, his speech was normalized. A repeat left angiogram performed two months later disclosed difinite increase in the degree of stenosis associa-ted with FMD. Perivascular sympathectomy around common and internal carotid artery and superior cervical ganglionectomy on the left side carried out on the 70th day from the onset. Postoperative left carotid angiogram showed improvement of the stenosis markedly, and the motor disturbance was improved gradually.

Case 2: A 34-year-old woman presented with head dullness and disorientation suddenly. Left carotid angiogram on the third day from the onset showed a "string of beads" appearance from C1 to M1 portion. Follow-up angiography three days later revealed some progression of the ste-nosis. Furthermore a repeat left angiogram dis-closed occlusion of left internal carotid artery at the C2 portion. Left STA-MCA bypass surgery was performed on the 61 st day from the onset. Postoperative course was uneventful and she left hospital without neurological deficit.

According to the previous report, prognosis for FMD was good and progression of FMD was rare. But the analysis of the intracranial FMD in the reported cases indicated that the patients were below 40 years of age, especially younger persons, and the lesion were ipsilateral in many cases, moreover that there were not always good prognosis (Table 1).

It was certain that clinical course were not always consist with the angiographical progression of the FMD lesion (Table 2).

Judging from our cases and analysis of the previous literature, it is suggested that some FMDcases which have been believed to be static lesion lead to angiographically dynamic chage and there may be cause of asymptomatic, completed occlu-sion of internal carotid artery or middle cerebral artery.


Copyright © 1987, Igaku-Shoin Ltd. All rights reserved.

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電子版ISSN 2185-405X 印刷版ISSN 0006-8969 医学書院

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