Ординатура / Офтальмология / Английские материалы / Neuro-Ophthalmology_Kidd, Newman, Biousse_2008
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CHURG-STRAUSS SYNDROME
Churg-Strauss syndrome is a small vessel vasculitis associated with p-ANCA, lung involvement associated with symptoms of airways obstruction, a peripheral blood hypereosinophilia, and vasculitis affecting various tissues, including the nervous system and muscle (Fig. 7–13).
Ophthalmic involvement is not common but cases of conjunctivitis, keratitis,
scleritis, and uveitis have been described, whereas retinal vasculitis appears to be rare.170,171 An optic neuropathy may arise but is most uncommon with only 10 cases published in the literature.170–178 It may be bilateral170,179 or symptom-
atic on one side only despite evidence for choroidal hypoperfusion on fluorescein angiography173 and may be seen as disc edema before the onset of visual loss.178 In a Mayo Clinic review of a series of 47 consecutive patients with Churg-Strauss syndrome, optic neuropathy was seen in only 1 case of 29 patients with neurologic complications.175
Treatment and Prognosis
When associated with signs of central retinal artery occlusion, visual recovery
seems not to occur despite reasonable treatment with corticosteroids and immuno- suppression.170–173,175 In 2 of the 10 cases,174,176 it has been shown that visual
recovery may occur provided treatment is prompt. Treatment with high-dose corticosteroids and cyclophosphamide appears to be optimal.
WEGENER’S GRANULOMATOSIS
Wegener’s granulomatosis is small vessel vasculitis associated with antibodies to cytoplasmic-pattern ANCA (c-ANCA) in which a necrotizing vasculitis with granuloma formation develops in lung, upper airways, kidney, and other organs, including the nervous system.180 The ophthalmic manifestations are common and include conjunctivitis, dacryocystitis, keratitis, episcleritis, scleritis, uveitis, and retinal vasculitis. They may arise because of independent involvement by the vasculitis itself or by means of a spread of abnormal tissue from adjacent structures, usually the paranasal air sinuses. Hence, the development of an inflammatory tissue mass within the orbit is a common complication, leading
Figure 7–13 Temporal artery biopsy of a patient with Churg-Strauss syndrome presenting with a subacute painful optic neuropathy. There is an infiltration predominately of eosinophils around two small vessels. (Courtesy of Dr. Julie Crow.)
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to orbital pain, evolving proptosis, and ophthalmoparesis. Adjacent structures become involved, leading to keratitis, uveitis, and scleritis.181 Limited forms of the disease have on occasion been seen to affect orbital structures alone.
Similarly, involvement of the optic nerve may occur as an independent process or in consequence of compression by an inflammatory mass within the orbit. Patients may present with symptoms and signs suggestive of an anterior or posterior ischemic optic neuropathy in association with the presence of abnormal tissue within the orbit and adjacent paranasal air sinuses on MRI or, less commonly, with signs of an optic neuropathy with normal imaging. The optic neuropathy may be bilateral.182 Although a vasculitic disorder implies that the pathophysiology of the optic neuropathy is one of ischemia, most recent case reports suggest a favorable outcome provided prompt and aggressive treatment is made available. High-dose corticosteroids improve acuity, but the treatment of choice in Wegener’s granulomatosis is cyclophosphamide. A trial of etanercept failed to show benefit,183 but recent reports point to the effectiveness of the CD20 antagonist rituximab184 in cases refractory to cyclophosphamide.
GRANULOMATOUS ANGIITIS OF THE CENTRAL NERVOUS SYSTEM
Granulomatous angiitis is a rare vasculitic disorder of the CNS in which small and medium-sized vessel vasculitis develops with a perivascular inflammatory infiltration involving lymphocytes, macrophages, and giant cells. Optic neuropathy has been reported only rarely and always in association with other manifestations of the disorder. It may complicate raised CSF pressure, and one case of optic perineuritis has been seen.185
POLYARTERITIS NODOSA
Polyarteritis nodosa (PAN) is a systemic vasculitis affecting both small and medium-sized arteries, which can affect all tissues including the kidney, lungs, and nervous system. It is twice as common in men than women and presents at all ages but more commonly between 40 and 60 years. The eye is said to be involved in 10% to 20% of cases with an ischemic retinopathy associated with branch and central retinal artery occlusions, uveitis, exudative retinal detachment, scleritis, episcleritis, and keratitis.186,187
Optic neuropathies may arise with signs of anterior or posterior ischemic optic neuropathy with only 10 case reports in the literature; an early case showed inflammatory occlusion of the posterior ciliary arteries on both sides in a man
with PAN who became blind shortly before death.188 Others presented as anterior ischemic optic neuropathies.189–193 Another case, which also came to autopsy,
involved a man who reported unilateral transient visual loss194 in which a vasculitis of the small vessels of the orbit was seen without significant changes in the ciliary arteries. It was postulated that the visual loss arose as a result of choroidal ischemia rather than an optic neuropathy.
Treatment involves systemic corticosteroids and cyclophosphamide; the prognosis for recovery of vision appears to be poor; presumably then the pathology of the optic neuropathy is one of thrombotic occlusion of vessels involved in vasculitis, rather than inflammation adjacent to it.
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Figure 7–14 Temporal artery biopsy showing giant cell arteritis; high-power view showing a giant cell (arrow) and disruption of the internal elastic lamina. On the left, there is a mononuclear inflammatory cell infiltrate, on the right the thickened intima. (Courtesy of Prof. Tamas Revesz.)
GIANT CELL ARTERITIS
Giant cell arteritis is much more common in older adults than in middle age. It is characterized by involvement of the large and medium-sized vessels of the branches of the arteries that arise from the aortic arch (Fig. 7–14). The prevalence increases from 2.3 105 in the sixth decade of life to 44.7 105 in the ninth decade195 and only very rarely in those younger than age 50. It is more frequent in Caucasian peoples than in Africans, and each sex is equally represented. The pathology is one of a severe granulomatous vasculitis with infiltration of lymphocytes, histiocytes, and giant cells; edema of the adventitia; and necrosis of the internal elastic lamina.196
The visual symptoms that arise are a result of thrombotic occlusion of the arteries and not to the direct affects of inflammation (see Chapter 5). An anterior ischemic optic neuropathy resulting from occlusion of the short ciliary arteries is the most common, when the disc is seen to be pale and swollen with flameshaped hemorrhages and cotton wool spots in the adjacent retina. A posterior ischemic optic neuropathy may also arise, as may a central or branch retinal artery occlusion. Ophthalmoparesis and any neuro-ophthalmic manifestation of stroke may also arise, and mononeuropathies and peripheral polyneuropathies occur rarely.
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