Tongue necrosis provoked by ergotamine tartrate and disclosing a giant cell arteritis

ARTICLE

Giant cell arteritis (GCA), is a systemic granulomatous vasculitis that usually occurs in elderly patients and that preferentially involves the medium and large sized arteries from the aortic arch to the scalp. GCA can cause a number of local symptoms in the head and neck region, of which headache is the most common and blindness the most serious. Involvement of lingual arteries is not uncommon, but ischaemia or necrosis of the tongue is a rare manifestation of GCA [1]. We describe a case of tongue necrosis provoked by ergotamine tartrate treatment and revealing a GCA.

Case report

A 70 year-old woman was referred to our clinic with a 3-month history of headache, mandibular claudication and tongue pain. She had a several-year history of hypertension, for which she has being treated with atenolol (50 mg daily) and nifedipine (20 mg daily). The day before the hospitalization she began complaining of a violent headache that she treated with ergotamine tartrate (2 mg). A few hours later, she experienced a strong pain in the tongue and a greyish coating appeared covering the anterior part of her tongue. On admission to hospital both sides of the anterior third of the tongue were necrotic (Fig. 1). Temporal arteries were palpable and pulsatile. Physical examination (including ophtalmological examination) was otherwise within normal limits.

Laboratory tests showed an erytrocyte sedimentation rate of 44 mm/h and serum-fibrinogen of 6.0 g/l. Haemoglobin was 14.2 g/dl and total white blood cell count was 17,600/mm³.

The temporal artery biopsy confirmed the diagnosis of GCA. The histological sections showed arteritis with giant cell infiltration (Fig. 2). Treatment with prednisolone (60 mg daily) brought about an improvement in her symptoms in 72 hours. The tongue necrosis regressed, healing within a month without marked loss of tongue substance and without any reduced mobility (Fig. 3). She remained asymptomatic on low-dose prednisolone.

Discussion

The most frequent cutaneous finding in GCA is necrosis of the scalp, resulting in ulceration that may be localized on one side, may be bilateral, or may extend to a large area with much of the scalp being involved [2-5]. Sixty-seven percent of patients with scalp necrosis will also develop visual loss and the mortality rate is higher than in those without necrosis [6]. Although the lingual arteries are commonly involved in GCA, clinical manifestations are rare [7]. This is likely to be a consequence of the rich vascular supply to the tongue, and the numerous collaterals invariably present. Clinical manifestations of lingual arteritis are estimated to be present to some degree in 25% of patients suffering from GCA [8], but only a few of them showed necrosis or gangrene. The usual tongue symptoms include pain, lingual claudication, burning, recurrent blanching, redness or erythema and bluish swellings [2].

Inversely GCA is the most common cause of tongue necrosis. Cases of tongue necrosis resulting from tongue carcinoma, abscess of the floor of mouth, arterial embolism or Hodgkin's disease are sporadic [1]. More than 60 cases of tongue necrosis secondary to GCA have been reported. Lingual necrosis is usually unilateral and involves the tip or the anterior half. Although bilateral involvement has been reported [9], the base of the tongue is never engaged. Some of these cases have been related to triggering factors such as local anaesthesia [10, 11], surgical procedure [12], or use of an ergotamine derivative. To our knowledge, few well-documented cases of tongue necrosis secondary to GCA have been attributed to ergotamine [1, 13-18]. These cases, as well as our own, indicate that the vaso-constrictive action of ergotamine tartrate or dihydroergotamine may trigger a tongue necrosis in patients with unknown lingual GCA. Indeed, it is likely that the tongue critically depends upon an unstable blood supply via stenosed arteries and may thus undergo necrosis triggered by vasoactive drugs such as ergotamine derivatives [16]. Despite its cardioselectivity, atenolol prescribed in our patient for an essential HTA, has probably enhanced the ergotamine tartrate vasoconstrictive activity.

Corticosteroids, with or without nitroglycerin, are usually effective for limited necrosis when they are started early although medium to large necrotic areas must be resected under general anaesthesia and closed, primarily to avoid recurrent infections [17].

CONCLUSION

REFERENCES

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