Letters to the Editor Right
recurrent
laryngeal
nerve
palsy in
a
patient with systemic lupus erythematosus
Sir Left recurrent laryngeal nerve palsy is infrequently communicated in systemic lupus erythematosus (SLE), and it has been attributed to pulmonary hypertensionl-3. Nevertheless, two cases with isolated right recurrent laryngeal nerve palsy have been reported recently’,’. We present a patient with SLE and multiple cranial nerve palsy, including right recurrent laryngeal nerve involvement. One year before admission, a 55-year-old female developed fever ~3~~C~, arthritis of ankles, wrists, interphalangeal joints and elbows, depression, and loss of weight (10 kg). She was started on a non-steroidal antiinflammatory drug with some relief. Two days before hospitalization, the patient developed arthritis with high fever. At presentation she was pale, with a blood pressure of 100/60, a temperature of 38°C and alopecia. Her metacarpophalangeal joints and wrists were red and swollen. Neurological examination was unremarkable, although the patient was depressed. The rest of the physical examination was normal. Investigations on admission were: hemoglobin 9.1 gldl,
leukocytes 4.7 x 10’/ (0.27 x i.~’lllymphocytes), platelets 293 x 10~/1. Erythrocyte sedimentation rate was 5 mm/h. Blood urea, creatine phosphokinase and coagulation screen were normal. Rheumatoid factor was negative. Antinuclear antibody level was 1:236~, with a homogeneous pattern. The DNA-binding activity test (Farr test) was 50Vo . Extractable nuclear antigen, ANCA and anticardiolipin antibody tests were negative, as was the Coombs’ test. C3 level was 0.4 g/l (1-2); C4 level was 0. 14 g/i (0.2-0.7). Chest X-ray was initially normal. After admission, indomethacin (200 mg) was started with good articular response. Four days later an erythematous rash appeared on the patient’s hands and arms, with vesicle formation; mouth ulcers also appeared. Seven days later, fever rose to 40° C, and she developed disseminated rales in both lungs. Chest X-ray revealed diffuse bilateral infiltrates. Cultures were negative and prednisone (80 mg) was given, with resolution of both the fever and the pulmonary infiltrates. Doses of prednisone were tapered. Twenty days later the patient complained of dysphagia and hoarseness. Neurological examination revealed left peripheral facial palsy and upward gaze paresis. The gag reflex was absent in the left side and there was palsy of the left levator veli palatini. Indirect laryngoscopy showed right vocal cord palsy.
Electroencephalogram and magnetic resonance imaging of the brain were normal. The patient’s corticosteroid dose was increased, with slow but complete resolution of all symptoms. At follow-up, the patient was asymptomatic and out of medication for 1 year. After that time a flare-up of arthritis and cutaneous rash occurred. She was treated with a non-steroidal anti-inflammatory drug and hydroxychloroquine with control of the disease. At present she is on hydroxychloroquine without any signs of lupus activity. Central or peripheral neurological damage is common in SLE6. 7, although laryngeal nerve involvement is unusual’-’. In most of the reported cases the left recurrent laryngeal nerve was affected, and it was thought to be caused by the pressure on the nerve by the enlarged left pulmonary artery due to pulmonary hypertension 1-3. Nevertheless, two cases have recently been reported with right recurrent laryngeal nerve palsy as the only neurological manifestation,’. Our patient showed multiple cranial nerve involvement and severe depression, all appearing during a flare-up of the disease and resolving completely with corticosteroid therapy. Although right side catheterization was not performed, the lack of symptoms and the fact that the right recurrent laryngeal nerve was affected suggest that pulmonary hypertension is not the cause of hoarseness. We consider that recurrent laryngeal nerves can be involved in SLE as a consequence of nerve vasculitis.
References 1. Yu PN. 2.
3.
4.
5.
Primary pulmonary hypertension: report of six cases and review of literature. Ann Intern Med 1958; 49: 1138-61. Asherson RA, Mackworth-Young CG, Boey ML et al. Pulmonary hypertension in systemic lupus erythematosus. Br Med J 1983; 287: 1024-5. Asherson RA, Higenbottam TW, Dinh Xuan AT et al. Pulmonary hypertension in a lupus clinic: experience with twenty-four patients. J Rheumatol 1990; 17: 1292-8. Gordon T, Dunn EC. Systemic lupus erythematosus and right recurrent laryngeal nerve palsy. Br J Rheumatol 1990;29: 308-9. España A, Gutierrez JM, Soria C et al. Recurrent laryngeal palsy
in systemic lupus erythematosus. Neurology 1990; 40 :1143-4. 6. Feinglass EJ, Arnett FC, Dorsch CA et al. Neuropsychiatric manifestations of systemic lupus erythematosus: diagnosis, clinical spectrum, and relationship to other features of the disease. Medicine 1976; 55: 323-39. 7. Bennet R, Hughes GRV, Bywaters EGL, Holt PJL. Neuropsychiatric problems in systemic lupus erythematosus. Br Med J 1972; 4: 342-5.
407
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recurrent
laryngeal
nerve
palsy in
a
patient with systemic lupus erythematosus
Sir Left recurrent laryngeal nerve palsy is infrequently communicated in systemic lupus erythematosus (SLE), and it has been attributed to pulmonary hypertensionl-3. Nevertheless, two cases with isolated right recurrent laryngeal nerve palsy have been reported recently’,’. We present a patient with SLE and multiple cranial nerve palsy, including right recurrent laryngeal nerve involvement. One year before admission, a 55-year-old female developed fever ~3~~C~, arthritis of ankles, wrists, interphalangeal joints and elbows, depression, and loss of weight (10 kg). She was started on a non-steroidal antiinflammatory drug with some relief. Two days before hospitalization, the patient developed arthritis with high fever. At presentation she was pale, with a blood pressure of 100/60, a temperature of 38°C and alopecia. Her metacarpophalangeal joints and wrists were red and swollen. Neurological examination was unremarkable, although the patient was depressed. The rest of the physical examination was normal. Investigations on admission were: hemoglobin 9.1 gldl,
leukocytes 4.7 x 10’/ (0.27 x i.~’lllymphocytes), platelets 293 x 10~/1. Erythrocyte sedimentation rate was 5 mm/h. Blood urea, creatine phosphokinase and coagulation screen were normal. Rheumatoid factor was negative. Antinuclear antibody level was 1:236~, with a homogeneous pattern. The DNA-binding activity test (Farr test) was 50Vo . Extractable nuclear antigen, ANCA and anticardiolipin antibody tests were negative, as was the Coombs’ test. C3 level was 0.4 g/l (1-2); C4 level was 0. 14 g/i (0.2-0.7). Chest X-ray was initially normal. After admission, indomethacin (200 mg) was started with good articular response. Four days later an erythematous rash appeared on the patient’s hands and arms, with vesicle formation; mouth ulcers also appeared. Seven days later, fever rose to 40° C, and she developed disseminated rales in both lungs. Chest X-ray revealed diffuse bilateral infiltrates. Cultures were negative and prednisone (80 mg) was given, with resolution of both the fever and the pulmonary infiltrates. Doses of prednisone were tapered. Twenty days later the patient complained of dysphagia and hoarseness. Neurological examination revealed left peripheral facial palsy and upward gaze paresis. The gag reflex was absent in the left side and there was palsy of the left levator veli palatini. Indirect laryngoscopy showed right vocal cord palsy.
Electroencephalogram and magnetic resonance imaging of the brain were normal. The patient’s corticosteroid dose was increased, with slow but complete resolution of all symptoms. At follow-up, the patient was asymptomatic and out of medication for 1 year. After that time a flare-up of arthritis and cutaneous rash occurred. She was treated with a non-steroidal anti-inflammatory drug and hydroxychloroquine with control of the disease. At present she is on hydroxychloroquine without any signs of lupus activity. Central or peripheral neurological damage is common in SLE6. 7, although laryngeal nerve involvement is unusual’-’. In most of the reported cases the left recurrent laryngeal nerve was affected, and it was thought to be caused by the pressure on the nerve by the enlarged left pulmonary artery due to pulmonary hypertension 1-3. Nevertheless, two cases have recently been reported with right recurrent laryngeal nerve palsy as the only neurological manifestation,’. Our patient showed multiple cranial nerve involvement and severe depression, all appearing during a flare-up of the disease and resolving completely with corticosteroid therapy. Although right side catheterization was not performed, the lack of symptoms and the fact that the right recurrent laryngeal nerve was affected suggest that pulmonary hypertension is not the cause of hoarseness. We consider that recurrent laryngeal nerves can be involved in SLE as a consequence of nerve vasculitis.
References 1. Yu PN. 2.
3.
4.
5.
Primary pulmonary hypertension: report of six cases and review of literature. Ann Intern Med 1958; 49: 1138-61. Asherson RA, Mackworth-Young CG, Boey ML et al. Pulmonary hypertension in systemic lupus erythematosus. Br Med J 1983; 287: 1024-5. Asherson RA, Higenbottam TW, Dinh Xuan AT et al. Pulmonary hypertension in a lupus clinic: experience with twenty-four patients. J Rheumatol 1990; 17: 1292-8. Gordon T, Dunn EC. Systemic lupus erythematosus and right recurrent laryngeal nerve palsy. Br J Rheumatol 1990;29: 308-9. España A, Gutierrez JM, Soria C et al. Recurrent laryngeal palsy
in systemic lupus erythematosus. Neurology 1990; 40 :1143-4. 6. Feinglass EJ, Arnett FC, Dorsch CA et al. Neuropsychiatric manifestations of systemic lupus erythematosus: diagnosis, clinical spectrum, and relationship to other features of the disease. Medicine 1976; 55: 323-39. 7. Bennet R, Hughes GRV, Bywaters EGL, Holt PJL. Neuropsychiatric problems in systemic lupus erythematosus. Br Med J 1972; 4: 342-5.
407
Downloaded from lup.sagepub.com at Purdue University on May 29, 2015
