Annals of the Rheumatic Diseases 1993; 52: 308-309

308

Rapid development of a tophus following
ipsilateral hemiparesis
Ciaran F Donegan, Peter Berman, Michael Doherty

Abstract
An 85 year old man with a longstanding
history of non-tophaceous gout developed
a single large tophus, unassociated with
inflammation, on his paretic leg over a six
week period following an acute hemiplegia. The rapidity of tophus formation,
its localisation to the paretic limb, and the
apparent blunting of the acute inflammatory response represent a previously
unreported interaction between gout and
neurological deficit.
(Ann Rheum Dis 1993; 52: 308-309)

Considerable interest exists in the interaction
between neurological deficits and the clinical
expression of rheumatic diseases. The sparing
effect of upper or lower neurological deficit on
the subsequent development of arthropathy,
including gout, has been well described.`6
Conversely, a few cases have been described in
which exacerbation of established joint disease
has followed hemiparesis.
We report an
apparently unique patient with a history of
recurrent acute gout, in whom the development of a hemiparesis was followed by the
rapid appearance of a non-inflammatory tophus
on the heel of his paretic foot.

and intact with no signs of local inflammation.
No other swellings were apparent elsewhere. A
lateral radiograph (fig 1) showed soft tissue
swelling superficial to the Achilles tendon
insertion with an underlying small focus of
calcification. Aspiration of the swelling confirmed urate crystals in a typical tophus 'sheet'
configuration (compensated polarised light
microscopy). After aspiration macroscopically
white crystal deposits exuded through the
needle track (fig 2), without provoking an
inflammatory response.
Further investigations showed: urea 11-7
mmol/l (normal range 1-6 5); creatinine 167
Rmol/I (60-120); uric acid 406 ,umol/l
(100-400); calcium and fasting lipoproteins
within normal limits. Radiographs of feet,
knees, pelvis, and hands showed only minor
changes of osteoarthritis (knees, first metatarsophalangeal joints) and no chondrocalcinosis. Treatment was established with
allopurinol 100 mg daily. Because the

Case report
A previously fit 85 year old right handed man
was admitted with an acute left hemiplegia.
Since the age of 50 he had had recurrent
acute attacks of gout limited to both feet

Department of Health
Care of the Elderly,
City Hospital,
Nottingham NG5 1PB,
United Kingdom
C F Donegan
P Berman
Rheumatology Unit,
City Hospital,
Nottingham NG5 1PB,
United Kingdom
M Doherty
Correspondence to:

Dr Donegan.
Accepted for publication
17 December 1992

(including classic podagra), treated with short
intermittent courses of non-steroidal antiinflammatory drugs and allopurinol. He had
not taken such drugs for many months. There
were no associated risk factors for gout
(obesity, excess alcohol, chronic diuretic
treatment, family history) and he had never
developed tophi. Examination confirmed
marked left sided weakness affecting the arm
more than the leg, mild sensory signs but no
visual field deficit. A computed tomography
brain scan showed right frontoparietal

infarction.
Over the subsequent six weeks his motor
function improved, though he still required the
assistance of one person for most activities. At
this time he complained of a swelling over his
left heel which prevented him from wearing his
normal footwear. The swelling, at the level of
the pre-Achilles bursa, was 2 X 3 cm, firm, and Figure 1 Lateral left heel radiograph showing soft tissue
non-tender. The overlying skin was healthy swelling and central area of calcification.

Rapid development of a tophus following ipsilateral hemiparesis

309

present at this site for some time. Whether a
crystal mass originated before or after the
stroke, however, the hemiparesis seems to have
encouraged accelerated crystal deposition and
tophus development.
The only comparable case is that reported by
Cosgrave and Lewkonia' of a patient with posttraumatic 'frozen shoulder' who developed
tophi in the ipsilateral hand six months later.
Although there was no neurological deficit,
disuse or altered regional blood flow were again
both proposed as mechanisms of causation.
Tophus formation is normally regarded as a
Figure 2 The left heel of the patient following aspiration,
showing white tophaceous material that had travelled up the slow process. Although in vitro formation of
monosodium urate monohydrate crystals can
needle track.
be rapid, in vivo formation is estimated to take
many months,'3 and tophi usually occur in
mechanical effect of the large tophus was association with symptomatic chronic gout. In
impeding mobilisation it was excised nine our patient the speed of formation of his first
weeks after his stroke. His subsequent recovery ever tophus was rapid. The absence of
was uneventful.
associated inflammation is also remarkable,
particularly as newly forming (compared with
older) urate crystals might be expected to
Discussion
expose more 'naked', non-protein coated
To our knowledge this is the first report of a surfaces for interaction with cell surfaces and
tophus developing in a paretic limb following inflammatory mediators.'4 The morphology of
acute neurological deficit. The close temporal the urate crystals seemed typical of those seen
link between hemiparesis and development of in tophi, though it is possible that such rapidly
the tophus, its ipsilateral location, and the forming crystals might present less active
absence of a previous history or evidence surfaces with respect to surface charge and
of other tophi strongly support a causal 'roughness'. Diminution of the inflammatory
association. Two further features of interest are reaction consequent upon altered neurothe rapidity of development and the absence of vascular responses would seem a more
associated inflammation.
plausible explanation, possibly mediated
Mechanisms to explain exacerbation of through reduction in neurotransmitters. 5
arthropathy (rheumatoid arthritis, osteoarthritis) following hemiparesis favour neurovascular alteration of blood flow or altered
1 Glynn J J, Clayton M L. Sparing effect of hemiplegia on
neurotrophic effects on inflammation.7 8 The
tophaceous gout. Ann Rheum Dis 1976; 35: 534-5.
converse sparing effect of upper and lower
2 Thompson M, Bywaters E G L. Unilateral rheumatoid
arthritis following hemiplegia. Ann Rheum Dis 1962; 21:
motor neurone lesions on development of
370-7.
locomotor disease (including gout) has more
3 Coste F, Forestier J. Hemiplegie et nodosites d'Heberden
contralaterales. Bull Mem Soc Med Hop Paris 1935; 51:
commonly been attributed to disuse with
772-6.
reduction of damaging mechanical factors and 4 Glick E N. Asymmetrical rheumatoid arthritis after
poliomyelitis. BMJ 1967; iii: 26-8.
intra-articular pressure.2 F6 Acute self limiting
5 Glynn J H, Sutherland I, Walker G F, Young A C. Low
arthropathy has been seen in hemiplegic limbs
incidence of osteoarthrosis in hip and knee after anterior
poliomyelitis: a late review. BMJ 1966; ii: 739-42.
of patients with no prior history of arthritis
6 Needs C J, Webb J, Tyndall A. Paralysis and unilateral
shortly after a cerebrovascular accidentl';
arthritis: is the association established? Clin Rheumatol
1985; 4:176-80.
although reaction to underlying infection has 7 Cosgrave
P M, Lewkonia R M. Asymmetric tophaceous
been incriminated," the cause remains
gout.Jf Rheumatol 1979; 6: 299-301.
8 Pattrick M, Doherty M, Dieppe P A. Unilateral
speculative.
exacerbation of rheumatoid arthritis by hemiparesis. BrJ
In the present case it is possible that minor
Rheumatol 1984; 23: 107-9.
9 Pattrick M, Doherty M. Rapidly destructive hip disease
tissue damage or unrecognised trauma,
following unilateral hemiparesis: report of two cases. Ann
consequent upon weakness and sensory deficit,
Rheum Dis 1987; 46: 477-81.
10
Hermann
E. Acute arthritis in hemiplegics. Scand J
promoted tophus formation in a predisposed
Rheumatol 1972; 1: 87-9.
patient. Alternatively, altered neurovascular 11 Mitchell W S, Capell H A. Unexplained acute arthritis in
hemiplegia. BMJ 1982; Vol: 284-6.
response in a paretic limb following stroke may
12 Simkin P. The pathogenesis of podagra. Ann Intern Med

lead to increased tissue urate concentrations
and possibly predispose to the development of
tophi. 12 Although there was no overt preAchilles trauma, ulceration or bursitis, the
calcific focus on the radiograph might support
local tissue injury and indicate that a small,
unidentified (mixed) crystal deposit had been

1977; 86: 230-3.
13 Fiddis R W, Vlachos N, Calvert P D. Studies of urate
crystallisation in relation to gout. Ann Rheum Dis 1983;
42 (suppl 1); 12-15.
14 Doherty M, Dieppe P A. Crystal deposition disease in the
elderly. Clin Rheum Dis 1986; 12: 97-116.
15 Levine J D, Goetzl E J, Basbaum A I. Contribution of the
nervous system to the pathophysiology of rheumatoid
arthritis and other polyarthritides. Rheum Dis Clin North
Am 1987; 13: 369-83.