Rheumatology Advance Access originally published online on March 10, 2006
Rheumatology 2006 45(5):641-643; doi:10.1093/rheumatology/kel064
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LETTER TO THE EDITOR |
Systemic nocardiosis mimicking an ocular relapse of giant-cell arteritis
Department of Internal Medicine, Hôpital des Quinze-Vingts, 28 rue de Charenton 1 Departments of Internal Medicine 2 Pathology, Hôpital Saint-Antoine, 184 rue du faubourg Saint-Antoine, Université Paris VI, 75012 Paris 3 Department of Ophthalmology 4, Hôpital des Quinze-Vingts, 28 rue de Charenton and 4 Department of Infectious Diseases, Hôpital Necker, 149 rue de Sèvres, Université Paris V, 75015 Paris, France
Correspondence to: E. Héron, Service de Médecine Interne, Hôpital des Quinze-Vingts, 28 rue de Charenton, 75571 Paris Cedex 12, France. E-mail: heron{at}quinze-vingts.fr
We describe a patient treated for temporal arteritis (TA) who experienced recurrent ocular events from different aetiologies, clearly showing that a high degree of suspicion against infection is permanently required in such patients. In addition it seems to be the first reported case of ocular and systemic nocardiosis in that context.
A 83-yr-old man was diagnosed with TA in September 2000, on account of new headaches, tender superficial temporal arteries, polymyalgia rheumatica, a C-reactive protein (CRP) level of 39 mg/l and granulomatous giant-cell arteritis on temporal artery biopsy. Prednisone 40 mg/day (0.5 mg per kg of body weight) led to prompt clinical improvement and CRP normalization. After 3 weeks, however, the left visual acuity suddenly decreased to 6/20 due to choroidal ischaemia attributed to TA (Fig. 1A,B), and progressively returned to 20/20 after four pulses of methylprednisolone 250 mg/day followed by oral prednisone 80 mg/day. At the beginning of May 2001, on prednisone 25 mg/day with the CRP level being 34 mg/l, a painless visual loss of the right eye occurred, leading to a vision of ‘counting fingers at 50 centimeters’. Retinal angiography showed a delayed retinal artery filling time of 35 s and marked localized subretinal oedema without choroidal defect or papillary oedema (Fig. 1C,D). The patient had no signs or symptoms of infection and a negative work-up including chest radiography, echocardiography and microbiological analysis of urine and blood, and we concluded there was TA progression involving the right central retinal artery. A 3-day course of 120 mg/day pulsed methylprednisolone was started on 14 May. During the following days, the CRP level increased rapidly to 240 mg/l and no visual improvement occurred. At the end of May, a pustular lesion was noticed on the patient's abdomen, and the patient became dyspnoeic with multiple nodular and reticular shadows appearing on chest X-ray. He died on 4 June from septic shock and multi-organ failure. Autopsy showed multiple abscesses varying in size from 1–2 mm to 4 cm in the heart, lungs, pancreas, peritoneum, kidneys, thyroid and in the whole brain (cerebral hemispheres and brain stem, oculomotor nerves and choroid plexus, associated with a purulent meningitis). An accumulation of 1 µm-wide filamentous gram-positive bacilli, Grocott-positive and Ziehl-positive, randomly oriented and branching at right angles was found in the abscesses and meninges, suggesting Nocardia asteroides infection. The walls of several cerebellar and coronary arteries were totally destroyed by invasive nocardial infection. Arterial segments taken from the internal carotids and first centimetres of the ophthalmic arteries were normal. A fibrotic lesion on the right vertebral artery, with disappearance of the internal elastic lamina, suggested a sequela of cured TA. Unfortunately, the eyes and retro-orbital content could not be examined due to lack of replacement ocular prostheses at the time of autopsy. We concluded that death occurred from fulminant systemic nocardiosis in an immunocompromised host, that was first manifested by an ocular event mimicking a TA relapse, but a posteriori related to a nocardial choroidal abscess.
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This observation dramatically illustrates the physician's dilemma between the fear of ocular involvement and the risk of corticosteroid complications in the treatment of temporal arteritis. Ocular manifestations occurred twice, 3 weeks and 6 months after start of corticosteroids, which theoretically reduces the ocular risk of TA to around 1% after 1 week of treatment [1]. The first event, isolated choroidal ischaemia, is an unusual finding in TA because choroidal ischaemia is generally associated in this disease with anterior ischaemic optic neuropathy or central retinal artery occlusion [2]. On the other hand, it may constitute a reversible cause of visual loss in TA [3], as in our patient. The rate of primary corticoresistance in TA is quite high: 13.5% in a prospective trial of 164 patients, and only apparent clinically (normal CRP) in half the cases [4]. Thus, in the absence of an alternative explanation, primary TA corticoresistance appeared to us to be the most plausible cause of the choroidal ischaemic event. That it was the first ocular manifestation of nocardiosis is indeed very unlikely, due to the 6-month interval before the next event without any antibiotic treatment. The second ocular manifestation was certainly related to a nocardial choroidal abscess, based on a compatible angiographic appearance, and widespread intracranial abscesses together with a lack of any active TA lesion at autopsy.
Nocardiosis occurs in immunocompromised hosts and can involve the central nervous system and the eyes by haematogenous spreading from a lung infection, leading to endogenous endophthalmitis or retinal abscesses [5–7]. To our knowledge, only two cases of nocardial infection without ocular localization have been described in patients treated for TA [8, 9]. In this patient, the sequence of a first TA-related then a second infection-related ocular complication highlights both the difficulty of diagnosing opportunistic infections in rheumatological immunocompromised patients [10], and the fact that suspicion of infection should grow in parallel with the cumulative dose of corticosteroids administered.
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The authors have declared no conflicts of interest.
References
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