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Intra-alveolar haemorrhage in temporal arteritis
  1. D Le Thi Huong,
  2. M R Andreu,
  3. P Duhaut,
  4. P Godeau,
  5. J C Piette
  1. Department of Internal Medicine, Groupe Hospitalier Pitié-Salpêtrière, 83 bd de l’Hôpital, 75013 Paris, France
  1. Correspondence to:
    Dr D Le Thi Huong;
    du.boutin{at}wanadoo.fr

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Temporal arteritis (TA) is the most common systemic vasculitis. We report herein a case of TA complicated with intra-alveolar haemorrhage. To our knowledge, this manifestation has not previously been reported.

CASE REPORT

A woman born in 1926 presented in 1999 with persistent dry cough and raised erythrocyte sedimentation rate at 60 mm at the first hour. C reactive protein was 14 mg/l. She had a history of pulmonary tuberculosis treated in 1951 with streptomycin and p-aminosalicylic acid. She complained of headache without fever, jaw claudication, scalp tenderness, and visual or musculoskeletal manifestations. She denied any other upper airways symptoms. Physical examination was normal. Arterial pressure was 140/70 mm Hg. Leucocyte count was 6.8×109/l with 4.2×109/l polynuclear neutrophils and 0.2×109/l eosinophils, haemoglobin 130 g/l, and platelets 310×109/l. A dipstick urinary test showed no proteinuria and no haematuria. Chest radiography disclosed calcified nodular density in the upper right lobe, which was confirmed by computed tomography. An electrocardiogram and echocardiogram were normal. Fibre optic bronchoscopy was normal. Bronchoalveolar lavage fluid examination showed 120×109 cells/l, comprising macrophages 56% with siderophages 30%, lymphocytes 39%, polynuclear neutrophils 1%, and polynuclear eosinophils 4%. Bronchoalveolar lavage was sterile on cultures for bacterial infection, Mycobacterium tuberculosis, cytomegalovirus, parasites, and fungi. A search for antinuclear antibodies and cryoglobulin was negative. Histological study of the temporal artery disclosed a granulomatous inflammation of the vessel wall containing mononuclear cells and histiocytes without giant cells, leading to fragmentation of the elastic lamina.

Prednisone 30 mg daily was started. The headache and cough disappeared rapidly. At the two year follow up, the patient is asymptomatic with 7 mg daily prednisone. C reactive protein is negative.

DISCUSSION

This patient had four of five American College of Rheumatology criteria for the classification of TA, which have a sensitivity of 93.5% and a specificity of 91.2%.1 Chronic dry cough without any chest radiograph anomaly is a classical symptom of TA. Machado in Minnesota reported respiratory symptoms in up to 25% of cases of TA.2 Cough may be the sole manifestation of the disease for a long time—up to one year.3 A three year course of cough associated with upper limb arteritis was also reported as an initial symptom of TA.4 The origin of the cough is not clear. In rare cases, cough might have been attributed to pleural effusion, interstitial pneumonitis, pulmonary vasculitis, or hyperreactive airways.5

Blockmans et al found T4 lymphocytic alveolitis in three patients with histologically proved TA.6 Only one patient complained of dry cough and dyspnoea. Chest radiographs and lung function tests were always normal. Cell count was normal on bronchoalveolar lavage fluid examination, but the number of lymphocytes was increased, as in our case, with a percentage varying between 16 and 61%. Discovery of nodular parenchymal densities raises the question of a borderline systemic vasculitis with Wegener granulomatosis.4

In our case, there was no obvious cause for intra-alveolar haemorrhage besides TA. Congestive cardiac failure and exogenous agents were excluded. Bacteriological, viral, fungal, and parasitic studies of bronchoalveolar lavage fluid were negative. There was no manifestation suggesting another systemic disease such as microscopic polyangiitis, Wegener’s granulomatosis or vasculitis secondary to systemic lupus erythematosus. The favourable outcome with medium dose prednisone in the absence of any other immunosuppressive agent also suggests TA. Cytomegalovirus infection was ruled out, but not other viral infections. Several viruses such as cytomegalovirus,7 coxsackievirus,8 adenovirus,9 enterovirus,10 mumps,11 and hepatitis C virus12 can cause pulmonary haemorrhage. Some authors have suggested that TA has a viral cause, and various agents were incriminated. Varicella zoster13 and parvovirus B19 DNA14 were found in temporal biopsy specimens of patients with histologically proved TA. IgM directed against human para-influenza type 1 virus were associated with TA onset.15

In conclusion, intra-alveolar haemorrhage may be one of the various causes of cough in TA. Further studies are necessary to ascertain whether it is the expression of a primary vasculitis or the consequence of an as yet unknown viral infection.

REFERENCES

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