Postgrad Med J: first published as 10.1136/pgmj.60.708.670 on 1 October 1984. Downloaded from

Postgraduate Medical Journal (October 1984) 60, 670-671

Giant cell presenting with FIONA MULCAHY LINDA D. JUBY M.B., M.R.C.P.I. M.B., M.R.C.P. GEOFFREY N. CHANDLER D.M., F.R.C.P. Chapel Allerton Hospital, Leeds LS7 4RB

Summary loss of sensation in a glove-like distribution. The A 76-year-old woman with who chest X-ray showed a right mid-zone opacity al- with is described. though bronchoscopy was normal and direct needle presented peripheral neuropathy lung biopsy showed only fibrotic lung tissue. Sinus X- KEY WORDS: central retinal artery occulsion, temporal artery biopsy. rays were normal. The initial haemoglobin was 12-2 g/dl and fell to 7-7 g/dl (normochromic, normocytic); Introduction white cell count was 14-7 x 109/1 (neutrophil leucocy-

tosis); ESR 110 mm/hr; plasma urea and electrolytes,copyright. Giant cell arteritis is an inflammatory condition of liver function tests, serum vitamin B2 and folate were the medium sized vessels which is associated with a all variety of neurological complications including optic normal. Autoantibody and antigen atrophy, isolated cranial nerve palsies, and tests were negative. Cerebrospinal fluid (CSF) was dementia (Paulley and Hughes, 1980). Peripheral normal. Computed tomographic (CT) scan showed neuropathy may occur with polyarteritis nodosa but evidence of the previous right cerebral . is a little recognised complication of giant cell Initial nerve conduction studies showed a mixed arteritis. We report an unusual case of giant cell sensori-motor neuropathy with absence of the sen- arteritis presenting with peripheral neuropathy. sory nerve action potentials in the arms and legs. Motor conduction velocities were within normal http://pmj.bmj.com/ limits in the arms but no motor response was Case report obtained following maximum stimulation of the A 76-year-old woman presented with a 3-day nerves in the legs. Temporal artery biopsy showed history of difficulty in walking, paraesthesiae in both evidence of giant cell arteritis with lymphocyte hands and feet and diplopia on looking to the left. infiltration, loss of the internal elastic lamina and There were no associated headaches or . Her multinucleated giant cells (Fig. 1.). previous medical history included a right cerebral Treatment with oral prednisolone (60 mg/day) was infarction 25 years previously from which she made a commenced without objective improvement. Repeat on September 28, 2021 by guest. Protected full recovery, and a 3-month history of sinusitis. nerve conduction studies at 1 month showed no Initial examination showed a left sixth cranial nerve significant change in the mixed axonal and myelin palsy and a mixed motor and sensory neuropathy in damage. Indeed the motor conduction velocities in both lower limbs with complete loss of sensation the arms were now slow. Azathioprine 25 mg tds was below the knees and absent ankle reflexes. The next added for a period of 10 days. Unfortunately the day, she developed complete right-sided visual loss patient developed herpes zoster in tlEe right L2 with normal fundal appearances, a right radial nerve dermatome. With no clinical benefit, azathioprine was palsy and resolution of the left sixth nerve palsy. discontinued and prednisolone was reduced over 3 Her temporal arteries were both pulsatile and not weeks to 30 mg daily. The patient, however, then thickened on palpation. She then developed a low developed a marked left radial nerve palsy and grade and the right fundus showed signs of a prednisolone was further increased. Although hae- central retinal artery occlusion and optic atrophy. moglobin rose to 13-4 g/dl and ESR fell to 39 mm/hr The right radial nerve palsy became submerged in an there was no neurological change. The patient finally upper limb peripheral neuropathy with and required long-term nursing care. Postgrad Med J: first published as 10.1136/pgmj.60.708.670 on 1 October 1984. Downloaded from

Clinical reports 671 v i ~~~~~~~ .....

Wq4t

~~E _ / , .,

FIG. 1. Temporal artery biopsy showing giant cell arteritis with loss of internal elastic lamina, lymphocyte infiltration and multinucleated giant cells. Magnification x 400. Discussion described. Indeed the clinical picture would favour The rapidly progressive neurological signs, low the diagnosis of polyarteritis nodosa or one of the grade fever, normochromic, normocytic anaemia, other forms of granulomatous arteritis. However leucocytosis and elevated ESR suggested a diagnosis there was no such evidence on temporal artery copyright. of . Peripheral neuropathy is well described biopsy. Therefore it would be unreasonable to associated with the inflammatory arteritides, in parti- consider an alternative diagnosis to explain a coinci- cular polyarteritis nodosa, but is unusual as a dental and acute onset peripheral neuropathy. We presentation of giant cell arteritis (Fauci, 1978). Ross must therefore conclude that an aggressive form of Russell (1959) first described peripheral neuropathy neuropathy may occur in giant cell arteritis and must in a series of patients with cranial arteritis of whom re-emphasis the need to include giant cell arteritis in one developed a lateral popliteal nerve palsy, and the differential diagnosis of peripheral neuropathy. another a lesion of painful causalgia-like the median http://pmj.bmj.com/ nerve. Later Warrell, Godfrey and Oslen (1968) and Acknowledgment Wilske and Healey (1971) described symmetrical We wish to thank Nigel J Buxton M.B., M.C.R. Path., Consultant peripheral neuropathy, both motor and sensory in Pathologist, for his valuable assistance. type as a clinical manifestation of biopsy proven giant cell arteritis. We describe a case with gross References histological changes on temporal artery biopsy. FAUCI, A.S. (1978) The spectrum of vasculitis. Annals of Internal Unlike the previous patients reported however, our Medicine, 89, 660. case did not present the clinical features ofheadache, PAULLEY, J.W. & HUGHES, J.P. Giant cell arteritis or arteritis of the on September 28, 2021 by guest. Protected myalgia or thickened non-pulsatile temporal arteries. aged. British Medical Journal, 2, 1562. The response to and azathioprine was Ross RUSSELL, R.W. (1959) Giant cell arteritis. Quarterly Journal of Medicine, 28, 471. disappointing. Contrary to the experience of Warrell WARRELL, D.A., GODFREY, S. & OSLEN, E.G.T. (1968) Giant cell et al. (1968) and Wilske and Healey (1971) there was arteritis with peripheral neuropathy. Lancet, i, 1010. no neurological improvement, although the haemo- WILSKE, K.R. & HEALEY, L.A. (1971) Clinical manifestations of globin and ESR reverted to normal as expected. The biopsy-proven giant cell arteritis. Arthritis and , 14, occurrence of a left radial nerve palsy following 424. reduction in prednisolone dosage must suggest a more active and aggressive arteritis than previously (Accepted 26 October 1983)