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STROKE

VOL 12, No

5, SEPTEMBER-OCTOBER

1981

230-240, 1977

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A Case for Cerebral Thromboangiitis Obliterans

JOSE BILLER, M.D., JORGE ASCONAPE, M.D., VENKATA R. CHALLA, JAMES F. TOOLE, M.D., AND WILLIAM T. MCLEAN, M.D.

M.D.,

SUMMARY The clinical, laboratory

existence of cerebral thromboangiitis obliterans (CTAO) has been controrersial. The and angiographic features of a young woman with recurrent thrombophlebitis, digital

gangrene and a bilateral anterior opercular syndrome (Foix-Charany-Marie) are reported. The angiognun demonstrated significant narrowing of fronto-opercular branches of both middle cerebral

cerebral arteries.

Histology

of small digital muscular arteries rerealed segmental adventitial flbrosis, narrowing or occlusion of

lumen and mild lyraphocitic infiltrates; occasional reins showed phlebitis. An etiologic relationship cerebral occlusire disease and peripheral thromboangiitis obliterans (TAO) is suggested.

between

Stroke, Vol 12, No 5, 1981

IN 1879, Von Winiwarter1 published an account of angiopathy in a 57-year-old man with foot gangrene; he named the angiopathy endarteritis obliterans. Buerger,3 in 1908, designated the condition thrombo- angiitis obliterans (TAO). Spatz,8 and Spatz and Lindenberg,4 in 1939, published comprehensive ac- counts of the cerebral form of TAO (CTAO) dis- tinguishing 2 types according to distribution of the lesions. There is a controversy in recent literature""" on whether TAO and CTAO are distinct clinico- pathologic entities.

A young woman affected by recurrent thrombo- phlebitis, digital gangrene and a Foix-Chavany-Marie (biopercular) syndrome* is reported as representative of the association between cerebral and peripheral TAO. These observations buttress the present trend in angiology to consider TAO and CTAO as distinctive but rare entities.

Patient History

MOC, a 33-year-old black woman, was admitted to the hospital on August 31, 1980, because of sudden in- ability to talk and swallow. She had been in good health until 1966 when she had a thrombophlebitis in her left leg. One year later she developed a similar episode. In 1968 she had a right deep vein thrombosis. In 1973, at the age of 26, she suddenly had left

From the Department of Neurology and the Department of Pathology, (Section of Neuropathology) (Dr. Challa), Bowman Gray School of Medicine of Wake Forest University, 300 S. Hawthorne Rd., Winston-Salem, NC 27103.

This work was supported in part by NIH Grant No. NS-06655. Reprints: Dr. McLean.

hemiparesis. Radionuclide brain scan at the time showed increased uptake over the right parietal region consistent with an area of infarction. Bilateral carotid angiography revealed mild segmental narrowing of several of the left operculofrontal branches and an area of "luxury perfusion" over the right parietal fron- tal region. Routine blood tests and CSF examination were normal. Electrocardiogram and echocardio- gram were normal. Electroencephalogram showed mild slowing over the left hemisphere. During the en- suing weeks she gradually recovered from her hemi- paresis. During the next year she had numerous episodes of pain in her fingers and toes aggravated by cold weather and/or water. A selective left brachial angiogram revealed multiple occlusions and no opacification of the interdigital arteries (fig. 1A). A femoral angiogram was unremarkable down to the level of the ankle but failed to visualize any vessel beyond that area. Due to recurrent episodes of painful digital ischemia, she required a right transmetatarsal amputation of the left ring, right index finger, left fourth toe, and left big toe. Histologic examination of the amputated digits revealed segmental involvement of small muscular arteries several of which showed narrowing or occlusion of lumens (fig. IB) mainly due to intimal hyperplasia and sparse mononuclear in- filtration. No evidence of atheroma or calcification was seen in these vessels. None of these arteries showed fresh thromboses or vasculitis with fibrinoid necrosis. Occasional veins showed phlebitis. Focally involved vessels were surrounded by concentric layers of increased connective tissue.

In November, 1979 she had a bilateral lumbar sym-

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