- Email: [email protected]
PLASMAPHERESIS FOR LATE-STAGE TRYPANOSOMIASIS
1200
comparable with black patients in Ingulli and colleagues’ study, the results suggest a propensity among black patients for the development of FSGS. It would be interesting to know whether these observations in MCD
are
due to environment or
to race.
Paediatrics & Child Health,
Faculty of Medicine, University of Natal, Congella 4013, South Africa
H. M. COOVADI M. ADHIKARI
1. Coovadia HM. South Africa: nephrology around the World. In: Holliday MA, Banath TM, Vernier RL, eds. Paediatric nephrology, 2nd ed. Baltimore: Williams and Wilkins, 1986: 361. 2. Kala UK, Milner LS, Jacobs D, Thompson PD. Focal glomerulosclerosis (FSGS) in Southern Africa: Johannesburg experience. 17th Congress of the South African Renal Society (July, 1988). Abstr.
PLASMAPHERESIS FOR LATE-STAGE TRYPANOSOMIASIS
SIR,-Sleeping sickness is endemic
in 36 countries of sub-
Saharan Africa in areas where the tsetse fly vector occurs. According to the World Health Organisation at least 20 000 new patients are reported every year, which is probably an underestimate of the true frequency. We describe an 18-year-old Angolan woman who had lived for 18 months in Rome and was admitted to our hospital in April, 1988. On admission she had low grade fever, hepatomegaly, and signs of cerebral involvement such as loss of sphincter control, ataxia, and tremor of the tongue but no nuchal rigidity. An indirect haemagglutination test for African trypanosomiasis was positive (1/1024 serum, 1/8 cerebrospinal fluid [CSF]). Trypanosomes were subsequently found in CSF and blood. Nuclear magnetic resonance (NMR) imaging of the brain revealed high intensity signal in the frontal and medial temporal white matter, which suggested leucoencephalitis (figure, left).1 Treatment with fx-difluoromethylomithine (DFMO)2,3 was started at a daily intravenous dose of 300 mg/kg for 11days followed by 400 mg/kg per day orally for 21 days. DFMO was well tolerated and blood and CSF were cleared of trypanosomes in 4 days. The neurological condition of the patient, however, failed to improve and large amounts of immunoglobulins (IgG 3380 mg/dl, IgM 3540 mg/dl) and circulating immune complexes and cold agglutinins were found in blood and CSF. With the hypothesis that the clinical features were being caused by the deposition of immune complexes in the brain and vessels’5 plasmapheresis with cascade filtration was started on the 15th day of antiparasitic therapy. Plasmapheresis was done five times over 10 days, while DFMO administration was continued. One volume of plasma was exchanged in 3 h. Plasmapheresis was well tolerated and the neurological picture improved rapidly. On day 1 the patient awoke briefly and by day 5 she was conscious. On day 7 her tremor became less intense and on day 10 she regained sphincter control. The immunoglobulin levels decreased to normal and immune complexes disappeared from the blood and CSF. By January, 1989,
Brain NMR.
Left, before treatment with DFMO (April 19, 1988); right, after therapy T2 weighted images.
(Oct 20, 1988).
the patient was symptom-free, although IgM levels were high (869 mg/dl). Parasitological tests remained negative. Repeat NMR showed reduction of the high intensity signal (figure, right). Our findings support the hypothesis that the neurological symptoms of this patient were due to the abnormal amount of IgM arising from polyclonal activation of B cells caused by the variant surface glycoprotein of the trypanosomes or by a mitogenic factor produced by the parasites.6 To our knowledge such an abnormal increase of IgG and IgM has not been described before in patients with African sleeping sickness. We think that plasmapheresis could be used on a wider scale with this disease. We thank Dr D. A. Evans of the London School of Hygiene and Tropical Medicine for commenting on the manuscript, and Dr L Savioli for suggestions and help.
Neurology and Neuroradiology Departments, Hospital "L. Spallanzani" for Infectious Diseases and Hospital "S. Camillo", 00149 Rome, Italy
F. SPINAZZOLA . C. D’AMATO A. DE FELICI R. GIANNUZZI M. G. PAGLIA C. STRUGLIA G. TOCCI G. VISCO S. GALGANI E. COTRONEO
1. Medina EA, Ventura FA. Champalimaud JL. Computer assisted tomographic findings in a patient with African trypanosomiasis. J Trop Med Hyg 1986; 89: 75-77. 2. Doua F, Boa FY, Scheckter PJ, Miezan TW, Diai D. Treatment of human late stage gambiense trypanosomiasis with &agr;-difluoromethylornithine (eflornithine): efficacy and tolerance in 14 cases in Cote D’Ivoire. Am J Trop Med Hyg 1987; 37: 525-33. 3. Taelman H, Scheckter PJ, Marcelis L, Sonnet J, Kazyumba G. Difluoromethylomithine, an effective new treatment of Gambian trypanosomiasis. Am J Med 1987; 82: 607-14. 4. Greenwood BM, Whittle HC. Complement activation in patients with Gambian sleeping sickness. Clin Exp Immurol 1976; 24: 133-38. 5. Lambert PH, Bemey M, Kazyumba G. Immune complexes in serum and in cerebrospinal fluid in African trypanosomiasis. J Clin Invest 1981; 67: 77-85. 6. Greenwood BM. Possible role of a B-cell mitogen in hypergammaglobulinaemia in malaria and trypanosomiasis. Lancet 1974, i: 435-36.
THROMBOLYTIC THERAPY FOR SUPERIOR VENA CAVA SYNDROME
SIR,-A 72-year-old woman presented with progressive facial and upper extremity oedema, exertional dyspnoea, and fatigue. 3 years earlier she had had surgery for grade 3 endometrial adenocarcinoma, followed by radiotherapy. 1 year before admission she had been put on home parenteral nutrition, via a left subclavian Hickman catheter, because of abdominal pain and inadequate oral intake. After 3 months the vein occluded so the right subclavian route was used. 3 months after that 90 cm of right colon had been removed because of a primary adenocarcinoma. 2 months before admission the right subclavian catheter was found to be broken and was
replaced.
On admission she was not in acute distress but her face was suffused and the jugular veins were severely distended, with a prominent superficial venous pattern in the upper chest. A murmur was heard at the right upper sternal border. Laboratory tests were not significant and the electrocardiogram was unremarkable except for sinus tachycardia (130/min). Venography demonstrated occlusion of the superior vena cava to the level of the innominate vein, with prominent collateral vessels. Echocardiography demonstrated no extension of thrombus into the right atrium. A computed tomographic scan ruled out extrinsic compression and confirmed extensive thrombosis from the right axilla to the junction of the superior vena cava and right atrium. Continuous intravenous heparin for 3 days was unsuccessful so a catheter, via a right antecubital vein, was advanced into the thrombus and alteplase (rtPA) was infused at 10 mg/h. After 100 mg the patient’s facial suffusion and oedema was much reduced. Continuous intravenous heparin was maintained until a repeat venogram showed that the superior vena cava was patent. Significant residual thrombus remained but after a further 50 mg of alteplase the thrombus resolved further. Heparin was continued to keep the activated thromboplastin time at 1to 2times normal, and long-term warfarin was started. The second course of thrombolytic therapy was complicated by haematomas at skin puncture sites, epistaxis, and haem-positive
comparable with black patients in Ingulli and colleagues’ study, the results suggest a propensity among black patients for the development of FSGS. It would be interesting to know whether these observations in MCD
are
due to environment or
to race.
Paediatrics & Child Health,
Faculty of Medicine, University of Natal, Congella 4013, South Africa
H. M. COOVADI M. ADHIKARI
1. Coovadia HM. South Africa: nephrology around the World. In: Holliday MA, Banath TM, Vernier RL, eds. Paediatric nephrology, 2nd ed. Baltimore: Williams and Wilkins, 1986: 361. 2. Kala UK, Milner LS, Jacobs D, Thompson PD. Focal glomerulosclerosis (FSGS) in Southern Africa: Johannesburg experience. 17th Congress of the South African Renal Society (July, 1988). Abstr.
PLASMAPHERESIS FOR LATE-STAGE TRYPANOSOMIASIS
SIR,-Sleeping sickness is endemic
in 36 countries of sub-
Saharan Africa in areas where the tsetse fly vector occurs. According to the World Health Organisation at least 20 000 new patients are reported every year, which is probably an underestimate of the true frequency. We describe an 18-year-old Angolan woman who had lived for 18 months in Rome and was admitted to our hospital in April, 1988. On admission she had low grade fever, hepatomegaly, and signs of cerebral involvement such as loss of sphincter control, ataxia, and tremor of the tongue but no nuchal rigidity. An indirect haemagglutination test for African trypanosomiasis was positive (1/1024 serum, 1/8 cerebrospinal fluid [CSF]). Trypanosomes were subsequently found in CSF and blood. Nuclear magnetic resonance (NMR) imaging of the brain revealed high intensity signal in the frontal and medial temporal white matter, which suggested leucoencephalitis (figure, left).1 Treatment with fx-difluoromethylomithine (DFMO)2,3 was started at a daily intravenous dose of 300 mg/kg for 11days followed by 400 mg/kg per day orally for 21 days. DFMO was well tolerated and blood and CSF were cleared of trypanosomes in 4 days. The neurological condition of the patient, however, failed to improve and large amounts of immunoglobulins (IgG 3380 mg/dl, IgM 3540 mg/dl) and circulating immune complexes and cold agglutinins were found in blood and CSF. With the hypothesis that the clinical features were being caused by the deposition of immune complexes in the brain and vessels’5 plasmapheresis with cascade filtration was started on the 15th day of antiparasitic therapy. Plasmapheresis was done five times over 10 days, while DFMO administration was continued. One volume of plasma was exchanged in 3 h. Plasmapheresis was well tolerated and the neurological picture improved rapidly. On day 1 the patient awoke briefly and by day 5 she was conscious. On day 7 her tremor became less intense and on day 10 she regained sphincter control. The immunoglobulin levels decreased to normal and immune complexes disappeared from the blood and CSF. By January, 1989,
Brain NMR.
Left, before treatment with DFMO (April 19, 1988); right, after therapy T2 weighted images.
(Oct 20, 1988).
the patient was symptom-free, although IgM levels were high (869 mg/dl). Parasitological tests remained negative. Repeat NMR showed reduction of the high intensity signal (figure, right). Our findings support the hypothesis that the neurological symptoms of this patient were due to the abnormal amount of IgM arising from polyclonal activation of B cells caused by the variant surface glycoprotein of the trypanosomes or by a mitogenic factor produced by the parasites.6 To our knowledge such an abnormal increase of IgG and IgM has not been described before in patients with African sleeping sickness. We think that plasmapheresis could be used on a wider scale with this disease. We thank Dr D. A. Evans of the London School of Hygiene and Tropical Medicine for commenting on the manuscript, and Dr L Savioli for suggestions and help.
Neurology and Neuroradiology Departments, Hospital "L. Spallanzani" for Infectious Diseases and Hospital "S. Camillo", 00149 Rome, Italy
F. SPINAZZOLA . C. D’AMATO A. DE FELICI R. GIANNUZZI M. G. PAGLIA C. STRUGLIA G. TOCCI G. VISCO S. GALGANI E. COTRONEO
1. Medina EA, Ventura FA. Champalimaud JL. Computer assisted tomographic findings in a patient with African trypanosomiasis. J Trop Med Hyg 1986; 89: 75-77. 2. Doua F, Boa FY, Scheckter PJ, Miezan TW, Diai D. Treatment of human late stage gambiense trypanosomiasis with &agr;-difluoromethylornithine (eflornithine): efficacy and tolerance in 14 cases in Cote D’Ivoire. Am J Trop Med Hyg 1987; 37: 525-33. 3. Taelman H, Scheckter PJ, Marcelis L, Sonnet J, Kazyumba G. Difluoromethylomithine, an effective new treatment of Gambian trypanosomiasis. Am J Med 1987; 82: 607-14. 4. Greenwood BM, Whittle HC. Complement activation in patients with Gambian sleeping sickness. Clin Exp Immurol 1976; 24: 133-38. 5. Lambert PH, Bemey M, Kazyumba G. Immune complexes in serum and in cerebrospinal fluid in African trypanosomiasis. J Clin Invest 1981; 67: 77-85. 6. Greenwood BM. Possible role of a B-cell mitogen in hypergammaglobulinaemia in malaria and trypanosomiasis. Lancet 1974, i: 435-36.
THROMBOLYTIC THERAPY FOR SUPERIOR VENA CAVA SYNDROME
SIR,-A 72-year-old woman presented with progressive facial and upper extremity oedema, exertional dyspnoea, and fatigue. 3 years earlier she had had surgery for grade 3 endometrial adenocarcinoma, followed by radiotherapy. 1 year before admission she had been put on home parenteral nutrition, via a left subclavian Hickman catheter, because of abdominal pain and inadequate oral intake. After 3 months the vein occluded so the right subclavian route was used. 3 months after that 90 cm of right colon had been removed because of a primary adenocarcinoma. 2 months before admission the right subclavian catheter was found to be broken and was
replaced.
On admission she was not in acute distress but her face was suffused and the jugular veins were severely distended, with a prominent superficial venous pattern in the upper chest. A murmur was heard at the right upper sternal border. Laboratory tests were not significant and the electrocardiogram was unremarkable except for sinus tachycardia (130/min). Venography demonstrated occlusion of the superior vena cava to the level of the innominate vein, with prominent collateral vessels. Echocardiography demonstrated no extension of thrombus into the right atrium. A computed tomographic scan ruled out extrinsic compression and confirmed extensive thrombosis from the right axilla to the junction of the superior vena cava and right atrium. Continuous intravenous heparin for 3 days was unsuccessful so a catheter, via a right antecubital vein, was advanced into the thrombus and alteplase (rtPA) was infused at 10 mg/h. After 100 mg the patient’s facial suffusion and oedema was much reduced. Continuous intravenous heparin was maintained until a repeat venogram showed that the superior vena cava was patent. Significant residual thrombus remained but after a further 50 mg of alteplase the thrombus resolved further. Heparin was continued to keep the activated thromboplastin time at 1to 2times normal, and long-term warfarin was started. The second course of thrombolytic therapy was complicated by haematomas at skin puncture sites, epistaxis, and haem-positive