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• W2163769162 abstract "Polyangiitis overlap syndrome is defined as a type of systemic vasculitis that does not fit into a single diagnostic category but converges several categories.1Leavitt R.Y. Fauci A.S. Polyangiitis overlap syndrome. Classification and prospective clinical experience.Am J Med. 1986; 81: 79-85Abstract Full Text PDF PubMed Scopus (134) Google Scholar We present a rare case of polyangiitis overlap syndrome consisting of granulomatosis with polyangiitis (GPA; formerly known as Wegener’s granulomatosis) and giant cell arteritis (GCA). A 61-year-old female of Irish descent presented to the emergency department with a 1-week history of progressive headache, mainly frontal and occipital, and a 3-day history of worsening binocular vertical diplopia. She gave a history of possible jaw claudication and neck, shoulder, and arm pain. Her recent medical history was notable for otitis media and asthmatic rhinosinusitis with intermittent epistaxis diagnosed 1 month earlier. On examination, her visual acuity was 20/20 in both eyes, and confrontation fields were intact. Her extraocular movements appeared full, but she complained of vertical diplopia on left and upward gaze. She had decreased hearing in her left ear, and tuning fork tests suggested possible sensorineuronal hearing loss. She had full range of motion of her neck and shoulders but complained of severe shoulder pain. The rest of her ocular and physical examinations was unremarkable. Blood tests were normal except for thrombocytosis (435×103/mm3) and an increased erythrocyte sedimentation rate (65 mm/hr). We were unable to locate the C-reactive protein result and saw the patient in consultation after steroids had been initiated. A computed tomography scan without contrast and a magnetic resonance imaging scan with gadolinium enhancement of the head were unrevealing except for some mild-to-moderate paranasal sinus mucosal thickening of the ethmoid, maxillary, and sphenoid sinuses. A chest radiograph showed no acute abnormalities. Abdominal ultrasound showed 2 small hepatic cysts in the left lobe and several small cysts in the left kidney. Her primary physician was a nephrologist, who initiated 25 mg prednisone daily. A 2.2-cm temporal artery biopsy was taken. Serial cross sections of the entire vessel were examined. Histologic sections showed focal relatively heavy adventitial with transmural inflammation of the medium-sized vessel that comprised lymphocytes and macrophages (Fig. 1). Neutrophils, eosinophils, granulomas, and multinucleated giant cells were not apparent. The intima was irregularly thickened that represented fibromyxoid change. The lumen of the vessel was patent. These features represent intermediate stage of resolution of the inflammation, findings consistent with diagnosis of GCA. Her blood work later confirmed high titers of classic anti-neutrophil cytoplasmic antibodies directed to proteinase 3 (PR3-ANCA/c-ANCA). The nephrologist elected to do a biopsy of the left kidney that showed approximately 50% fibrocellular crescents within the glomeruli in the background of mild mesangial hypercellularity, interstitial fibrosis, and tubular atrophy, consistent with pauci–immune crescentic glomerulonephritis (Fig. 2). She was treated with intravenous methylprednisolone and was started on pulsed intravenous cyclophosphamide every 2 weeks.Fig. 2Kidney biopsy that shows glomeruli with fibrocellular crescents (arrow) in a background of mild tubular atrophy and moderate arteriolar hyalinosis (periodic acid–Schiff stain, original magnification×10).View Large Image Figure ViewerDownload (PPT) To the best of our knowledge, GPA associated with vasculitis of the temporal artery has been previously reported in only 8 patients, of which only 2 had biopsy-proved GCA.2Vermeulen J.P. Mahowald M.L. A case of Wegener’s granulomatosis presenting with jaw claudication.J Rheumatol. 1984; 11: 707-709PubMed Google Scholar, 3Zenone T. Souquet P.J. Bohas C. Vital Durand D. Bernard J.P. Unusual manifestations of giant cell arteritis: pulmonary nodules, cough, conjunctivitis and otitis with deafness.Eur Respir J. 1994; 7: 2252-2254Crossref PubMed Scopus (35) Google Scholar, 4Nishino H. DeRemee R.A. Rubino F.A. Parisi J.E. Wegener’s granulomatosis associated with vasculitis of the temporal artery: report of five cases.Mayo Clin Proc. 1993; 68: 115-121Abstract Full Text Full Text PDF PubMed Scopus (95) Google Scholar, 5Small P. Brisson M.L. Wegener’s granulomatosis presenting as temporal arteritis.Arthritis Rheum. 1991; 34: 220-223Crossref PubMed Scopus (51) Google Scholar It is important to identify patients with polyangiitis overlap syndrome because it is a systemic vasculitic syndrome that can result in irreversible organ damage if not treated quickly.1Leavitt R.Y. Fauci A.S. Polyangiitis overlap syndrome. Classification and prospective clinical experience.Am J Med. 1986; 81: 79-85Abstract Full Text PDF PubMed Scopus (134) Google Scholar Systemic steroids are usually sufficient to treat GCA, but GPA usually requires cyclophosphamide therapy. This patient had clinical features of GCA but also had recent clinical symptoms suggestive of GPA. Our case provides further evidence that distinct systemic vasculitides with overlapping features can occur in a single patient as a polyangiitis overlap syndrome. Support: This work was supported through the Eye Foundation of Canada (to S.O.T.)." @default.
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• W2163769162 title "Polyangiitis overlap syndrome with granulomatosis with polyangiitis (Wegener’s) and giant cell arteritis" @default.
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