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Giant cell arteritis presenting with necrosis of the tongue — A case report
British Journal of Oral Surgery (1980), 18, 162-165 @ The British Association
0007-117)3/80/00220162502.00
of Oral Surgeons
GIANT CELL ARTERITIS PRESENTING WITH NECROSIS OF THE TONGUE-A CASE REPORT PAUL ALLEN, B.D.S.(LOND.),
L.D.s.R.c.s.(ENG.),
Department of Oral Surgery, North Manchester General Hospital, Crumpsall, Manchester M8 6RB Summary. The condition the tongue reported.
of giant cell arteritis
is described and a case presenting
with necrosis of
Introduction Giant cell arteritis is an inflammatory condition of the arteries in elderly persons where the cause is unknown and any large or medium sized artery may be involved (Harrison, 1974). The sexes are affected equally, the condition usually manifesting itself over the age of 60 years. Systemic symptoms include fever, sweats, malaise, fatigue, anorexia and weight loss, which usually occur before any symptomatic arterial involvement. Headache is a frequent symptom and in those with clinical arteritis of the temporal arteries, a temporal headache will occur. The temporal arteries will be thickened and tender to palpation and the patient may also complain of intermittent claudication of the tongue and muscles of mastication. It must be stressed that the most serious complication is loss of vision due to involvement of the ophthalmic arteries. Characteristically, the blood shows a raised ESR and elevation of the ~(2globulin and fibrinogen levels. The condition is, however, usually confirmed by an arterial biopsy which shows lymphocytes, macrophages, plasma cells and giant cells in the walls of affected blood vessels. Giant cell arteritis may affect any of the major cranial arteries but most commonly, branches of the superficial temporal artery are involved. A case is now reported of a patient who presented with giant cell arteritis of the lingual artery. Case report A 66-year-old lady presented complaining of a sore tongue and also of persistent headache. She stated that the headaches had started three weeks previously and that the pain was made worse by touching both her temples. In the-last week her tongue had become sore and started to swell. Her past medical history revealed that she suffered from rheumatoid arthritis and was anaemic, taking an oral iron preparation and analgesics as prescribed by her doctor. Examination revealed an extensive lesion of the left side of the tongue which appeared as an area of furring about 1 cm x 2 cm surrounded by a 1 cm margin of smooth mucosa. The fur could not be scraped off the tongue but the area felt normal and was sensitive to probing with a needle. She was reviewed again two days later when it had become obvious that the central furred area of the lesion on the left side of her tongue was sloughing (Fig. 1). She was admitted to hospital and placed on antibiotics and steroids (100 mg prednisolone, t.d.s.). Her blood picture on admission showed Hb 10.6, WBC 10.0 and ESR 77. She was referred to the physicians for management of her general condition. Three (Received 2 August 1979; accepted 19 September 1979) 162
GIANT
CELL
163
ARTERITIS
1. 1. Necrotic portion left side of tongue beginning to separate. Note the slough is bounded an area of smooth mucosa.
FIG. 2. The slough has now almost completely
separated.
by
164
BRITISH
JOURNAL
OF
ORAL
SURGERY
FIG. 3. Shows the size of the defect after the necrotic portion of the tongue had completely separated.
Note the defect is bounded by normal looking lingual mucosa.
days after admission the necrotic area on the left side of her tongue had completely separated (Fig. 2) to reveal healthy granulation tissue which subsequently contracted to form a notch in the left side of her tongue (Fig. 3). Her level of steroid intake was reduced over the following few days and she was discharged on a maintenance dose of 5 mg prednisolone, t.d.s. She was subsequently readmitted for a temporal artery biopsy which was reported as ‘Temporal arteritis of moderate activity’. She was finally discharged from hospital some three weeks after her initial presentation, and at this time she was free of symptoms.
Necrosis of the tongue resulting from giant cell arteritis was first described by Brearley and MacDonald (1961). Although a similar lesion had been reported two years earlier, it was not described as giant cell arteritis (Howard & Cremin, 1959). Missen (1961) describes post-mortem studies on five patients who had had giant cell arteritis of the temporal artery. The tongues of these patients were examined. Ten lingual arteries were studied histologically and nine out of ten showed inflammatory changes; of these, five vessels were severely stenosed, although macroscopically the tongues appeared normal. The reason why lingual necrosis is not common is due to the excellent collateral blood supply. This study would suggest that lingual arteritis is common amongst sufferers of more generalised giant cell arteritis. However, from the literature this mode of presentation is extremely rare and those cases that have been published lack good photographic records. Acknowledgements My thanks to Mr P. S. Holland, Consultant Oral Surgeon, for allowing me to present his patient, and for his advice in preparing this paper, to Mrs P. Shiel, for secretarial assistance and to the Photographic Department, North Manchester General Hospital.
GIANT
CELL
ARTERITIS
165
References Brearley, B. F., & MacDonald, J. G. (1961). Temporal Arteritis resulting in infected gangrene of tongue. British Medical Journal, 1, 1151. Harrison, T. B. (1974). Principles of Internal Medicine, M. M. Wintrobe et al. (eds.) 7th ed., p. 394. London & New York: McGraw Hill. Howard, S. &, Cremin, M. D. (1959). Acute parenchymatous glossitis with gangrene of the tongue. Lancet, ii, 410. Missen, G. A. K. (1961). Gangrene of the tongue. British Medical Journal, 1, 1393.
0007-117)3/80/00220162502.00
of Oral Surgeons
GIANT CELL ARTERITIS PRESENTING WITH NECROSIS OF THE TONGUE-A CASE REPORT PAUL ALLEN, B.D.S.(LOND.),
L.D.s.R.c.s.(ENG.),
Department of Oral Surgery, North Manchester General Hospital, Crumpsall, Manchester M8 6RB Summary. The condition the tongue reported.
of giant cell arteritis
is described and a case presenting
with necrosis of
Introduction Giant cell arteritis is an inflammatory condition of the arteries in elderly persons where the cause is unknown and any large or medium sized artery may be involved (Harrison, 1974). The sexes are affected equally, the condition usually manifesting itself over the age of 60 years. Systemic symptoms include fever, sweats, malaise, fatigue, anorexia and weight loss, which usually occur before any symptomatic arterial involvement. Headache is a frequent symptom and in those with clinical arteritis of the temporal arteries, a temporal headache will occur. The temporal arteries will be thickened and tender to palpation and the patient may also complain of intermittent claudication of the tongue and muscles of mastication. It must be stressed that the most serious complication is loss of vision due to involvement of the ophthalmic arteries. Characteristically, the blood shows a raised ESR and elevation of the ~(2globulin and fibrinogen levels. The condition is, however, usually confirmed by an arterial biopsy which shows lymphocytes, macrophages, plasma cells and giant cells in the walls of affected blood vessels. Giant cell arteritis may affect any of the major cranial arteries but most commonly, branches of the superficial temporal artery are involved. A case is now reported of a patient who presented with giant cell arteritis of the lingual artery. Case report A 66-year-old lady presented complaining of a sore tongue and also of persistent headache. She stated that the headaches had started three weeks previously and that the pain was made worse by touching both her temples. In the-last week her tongue had become sore and started to swell. Her past medical history revealed that she suffered from rheumatoid arthritis and was anaemic, taking an oral iron preparation and analgesics as prescribed by her doctor. Examination revealed an extensive lesion of the left side of the tongue which appeared as an area of furring about 1 cm x 2 cm surrounded by a 1 cm margin of smooth mucosa. The fur could not be scraped off the tongue but the area felt normal and was sensitive to probing with a needle. She was reviewed again two days later when it had become obvious that the central furred area of the lesion on the left side of her tongue was sloughing (Fig. 1). She was admitted to hospital and placed on antibiotics and steroids (100 mg prednisolone, t.d.s.). Her blood picture on admission showed Hb 10.6, WBC 10.0 and ESR 77. She was referred to the physicians for management of her general condition. Three (Received 2 August 1979; accepted 19 September 1979) 162
GIANT
CELL
163
ARTERITIS
1. 1. Necrotic portion left side of tongue beginning to separate. Note the slough is bounded an area of smooth mucosa.
FIG. 2. The slough has now almost completely
separated.
by
164
BRITISH
JOURNAL
OF
ORAL
SURGERY
FIG. 3. Shows the size of the defect after the necrotic portion of the tongue had completely separated.
Note the defect is bounded by normal looking lingual mucosa.
days after admission the necrotic area on the left side of her tongue had completely separated (Fig. 2) to reveal healthy granulation tissue which subsequently contracted to form a notch in the left side of her tongue (Fig. 3). Her level of steroid intake was reduced over the following few days and she was discharged on a maintenance dose of 5 mg prednisolone, t.d.s. She was subsequently readmitted for a temporal artery biopsy which was reported as ‘Temporal arteritis of moderate activity’. She was finally discharged from hospital some three weeks after her initial presentation, and at this time she was free of symptoms.
Necrosis of the tongue resulting from giant cell arteritis was first described by Brearley and MacDonald (1961). Although a similar lesion had been reported two years earlier, it was not described as giant cell arteritis (Howard & Cremin, 1959). Missen (1961) describes post-mortem studies on five patients who had had giant cell arteritis of the temporal artery. The tongues of these patients were examined. Ten lingual arteries were studied histologically and nine out of ten showed inflammatory changes; of these, five vessels were severely stenosed, although macroscopically the tongues appeared normal. The reason why lingual necrosis is not common is due to the excellent collateral blood supply. This study would suggest that lingual arteritis is common amongst sufferers of more generalised giant cell arteritis. However, from the literature this mode of presentation is extremely rare and those cases that have been published lack good photographic records. Acknowledgements My thanks to Mr P. S. Holland, Consultant Oral Surgeon, for allowing me to present his patient, and for his advice in preparing this paper, to Mrs P. Shiel, for secretarial assistance and to the Photographic Department, North Manchester General Hospital.
GIANT
CELL
ARTERITIS
165
References Brearley, B. F., & MacDonald, J. G. (1961). Temporal Arteritis resulting in infected gangrene of tongue. British Medical Journal, 1, 1151. Harrison, T. B. (1974). Principles of Internal Medicine, M. M. Wintrobe et al. (eds.) 7th ed., p. 394. London & New York: McGraw Hill. Howard, S. &, Cremin, M. D. (1959). Acute parenchymatous glossitis with gangrene of the tongue. Lancet, ii, 410. Missen, G. A. K. (1961). Gangrene of the tongue. British Medical Journal, 1, 1393.