Acute cysticercal meningitis - missed diagnosis
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《美国医学杂志》
1 Department of Pediatric Medicine, Chacha Nehru Bal Chikitsalaya, (Maulana Azad Medical College), Geeta Colony, Delhi-110031, India
2 Department of Laboratory Medicine, Chacha Nehru Bal Chikitsalaya, (Maulana Azad Medical College), Geeta Colony, Delhi-110031, India
Abstract
Neurocysticercosis is the commonest CNS parasitic disease worldwide but cysticercal meningoencephalitis is relatively rare, especially in Indian patients. We herein report a girl with cysticercal meningitis that was initially not suspected and later diagnosed on the basis of cerebrospinal fluid (CSF) eosinophilia. The need for CSF examination with wright-giemsa staining to avoid missing CSF eosinophilia is discussed.
Keywords: CSF analysis; CSF eosinophilia; Neurocysticercosis; Lumbar puncture
Neurocysticercosis (NC) is the most frequent parasitic infestation of the central nervous system in the world. It has varied manifestations like seizures, raised intracranial tension, stroke, hydrocephalus, meningoencephalitis etc., depending on the number and localization of the cysts. Cysticercal meningitis is uncommon in India.[1] In the world literature also; acute cysticercal meningitis has been described infrequently, especially in children. We herein describe a 9-year-old girl who presented with acute cysticercal meningitis.
Case Report
The patient was a 9-year-old girl who presented with one-day history of high grade, continuous fever, frontal headache, and projectile vomiting. There was no history of alteration in sensorium or convulsions. She had a history of fever with rhinorrhea 1-week back for which she had received oral amoxycillin for 5 days . There was no history of rash, cough, bleeding, ear-discharge, and any urinary or bowel complaints. She was developmentally normal. Family history and past history were not significant, they had a vegetarian dietary pattern, and there was no history of contact with tubercular patient. On examination, she was conscious but irritable and febrile. Vitals were stable and fundus examination was normal. General physical examination and systemic examination were normal except for positive meningeal signs without focal neurological deficits.
The child was admitted with a provisional diagnosis of meningitis. Investigations revealed total leukocyte count of 10,470/cumm with; polymorphonuclear cells, 68%; lymphocytes, 24%; monocyte 6%; and eosinophils; 2%. Lumbar puncture revealed turbid cerebrospinal fluid (CSF) with 950 cells (polymorphonuclear cells, 90%; lymphocytes, 10%), protein-610 mg/dL, and sugar-45mg/dL (concurrent blood sugar, 72 mg/dL). CSF gram stain and culture revealed no abnormality. Chest X-ray, blood and urine cultures, serum biochemistry, and platelet count were non-contributory. On the basis of compatible clinical and CSF profile, a diagnosis of acute bacterial meningitis was made and treatment with Ceftriaxone, steroids (for 48 hours), and mannitol (single dose, due to raised intracranial pressure) was started. Fever, headache and vomiting subsided within 24 hours. The patient remained well for the next 6 days.
On the 7th day of hospital stay, she developed headache, projectile vomiting and irritability. She had no recrudescence of fever or any convulsions. Examination revealed neck rigidity and positive Kernig sign. There were no focal neurological findings or papilledema. A contrast enhanced computed tomograph (CECT) of the cranium revealed an 8X7 mm hypodense lesion with an eccentric mural nodule, minimal enhancement, but no perilesional edema, in the left periventricular region. On the basis of the radiological picture the lesion was considered to be due to neurocysticercosis. A lumbar puncture was repeated with a specific request to the pathologist to look for eosinophils. CSF was grossly clear with leukocytes-500/cumm; lymphocytes-65%, polymorphonuclear cells-25%, eosinophils-10%; protein-35mg/dL, and sugar-51mg/dL (concurrent blood sugar-82mg/dL). CSF gram stain and culture did not reveal any abnormality. Tuberculin test with 5TU was negative. Serum IgG-ELISA enzyme-linked immunosorbent assay tests for Cysticercosis and for Tuberculosis were Negative. Serum and CSF immunoblot studies and MRI brain to look for subarachnoid cysts could not be done.
The patient was started on dexamethasone, and decongestive measures (mannitol, acetazolamide). Her symptoms improved over the next 12 hours. Indirect ophthalmoscopy ruled out intra-ocular cysts. Subsequently she received albendazole at dose of 15mg/kg/day for 28 days and anticonvulsants. She made an uneventful recovery. At last follow-up (ten month later), she was asymptomatic with no recurrence of symptoms, no evidence of meningeal irritation or raised intracranial pressure (ICT), and no neurodeficiet. Repeat CECT revealed resolution of the lesion.
Discussion
The initial clinical presentation was consistent with the diagnosis of pyogenic meningitis. The previous course of oral antibiotics could have been responsible for the negative CSF gram stain and culture; the rapid improvement on antibiotics, however, supported the diagnosis. When the patient deteriorated after 6 days; subdural effusion, ventriculitis or a concurrent brain abscess were the likely possibilities. The re-appearance of signs of meningeal irritation prompted us to repeat the lumbar puncture. In view of the presence of granuloma on CT, the possibility of cysticercal meningitis was considered, and a Wright Giemsa staining of the CSF was asked for. In the absence of this staining, eosinophils can be mistaken for polymorphonuclear cells.[1]
The finding of the viable NC granuloma on CECT in the absence of any past history of teniasis, seizures, headaches, or any neurological problem was surprising. This finding could also have been coincidental, as viable cysts are known to remain asymptomatic for long periods and reported to be present in up to 1.3% of normal persons (autopsy study).[2] The presence of CSF eosinophilia and the coexistent inflammatory granuloma suggested the possibility of cysticercal meningitis. Cysticercosis is by far the commonest cause of CSF eosinophilia in endemic areas.[3] The CSF findings in cysticercal meningitis consist of pleocytosis (usually lymphocytic but frequently polymorphonuclear), reduced glucose and elevated protein, with up to 70% patients with cysticercal meningitis having CSF eosinophilia between 2-40%.[4],[5],[6] The presence of eosinophils more than 4% of leukocytes in the CSF is of diagnostic significance[4] and is seen in the initial phases of the illness only. For visualizing eosinophils in the CSF, Wright-Giemsa staining of the CSF is required which is not routinely done in wardside-laboratories in most hospitals. In the absence of this staining, eosinophils can be mistaken for polymorphonuclear cells.[1] Other causes of eosinophilic meningitis include coccidioidal meningitis; parasitic infestation with Angiostrongylus cantonensis , Paragonimus westermani , Gnathostoma spinigerum ; intrathecal injection of foreign proteins, and the insertion of rubber tubing into the central nervous system in the course of neurosurgery.[7]
Cysticercal meningoencephalitis is caused by infiltration of the meninges and the parenchyma of the brain by a large number of parasites and inflammation in the surrounding tissue is responsible for meningitis and encephalitis.[8] Although described in 42-48% of patients in Latin American series, cysticercal meningoencephalitis has been reported in less than 10% adults patients with NC in India.[1],[9] Of the two large series of Indian children with NC, only one reported meningoencephalitis in 0.3% of the subjects.[10],[11] Slightly higher proportions have been reported in a small case-series.[8] Around 60% cysticercal meningoencephalitis cases have been reported to have associated parenchymal lesions.[11] Most cases described are chronic in evolution[12] and isolated acute meningitis has rarely been described. A case report from Thailand describes two children presenting as pyogenic meningitis, with normal cranial CT but not responding to antibiotic therapy.[6] Cysticercal meningitis was diagnosed based on the presence of CSF eosinophilia and positive serum serology, and both responded to praziquantel. Both cysticercus ELISA and Electro-Immuno Transfer Blot Assay (EITB) assays in the CSF have high sensitivity for cysticercal meningitis, but these studies in CSF and EITB in serum were not feasible in our setup.
There are no guidelines for the treatment of acute cysticercal meningitis. Anti-parasitic therapy is recommended for chronic meningitis.[13] The decision to treat in this patient was also justified by the radiological presence of a viable cyst. To conclude, we describe a 9-year-old Indian child with acute cysticercal meningitis that initially presented as pyogenic meningitis. Given the high prevalence of cysticercosis in Indian patients, this entity should be suspected in all patients; and CSF eosinophilia specifically looked for, more so when a concurrent parenchymal lesion suggestive of cysticercosis is present.
References
1. Singh G. Neurocysticercosis in the south-central America and the Indian subcontinent. Arq Neuropsiquiatr 1997; 55: 349-356.
2. Mahajan RC. Geographical distribution of human cysticercosis. In, Cysticercosis: Present state of Knowledge and Perspectives , (Ed.) Ana Flisser, et al. Academic Press 1982, 39-46.
3. Tasker WG, Plotkin SA. Cerebral Cysticercosis. Pediatrics 1979; 63: 761-763.
4. Wang CH, Gao SF, Guo YP. Diagnostic significance of eosinophilia of the cerebro-spinal fluid in cerebral cysticercosis. Chinese Medical Journal 1993; 106: 282-284.
5. Wilber RR, King EB, Howes EL. Cerebrospinal fluid cytology in five patients with cerebral cysticercosis. Acta Cytologica 1980; 24 : 421-426.
6. Visudhipan P, Chiemchanya S. Acute cysticercal meningitis in children: response to praziquantel. Ann Trop Ped 1997; 17: 9-13.
7. Char DFB, Rosen L. Eosinophilic meningitis among children in Hawaii. J Pediatr 1967; 70 : 28-35.
8. Puri V, Sharma DK. Neurocysticercosis in children. Indian Pediatr 1991; 28 : 1309-1317.
9. Venkatraman S, Roy AK, Dhamija RM, Sanchetee PC. Cysticercal meningoencephalitis - Clinical presentation and autopsy findings. J Assoc Phys India 1990; 38: 763-765.
10. Singhi P, Ray M, Singhi S, Khandelwal P. Clinical spectrum of 500 children with neurocysticercosis and response to albendazole therapy. J Child Neurol 2000; 15: 207-213.
11. Kalra V, Mittal R, Rana KS, Gupta A. Neurocysticercosis: Indian experience. Perat MV (Ed), New Developments in Neurology , Monduzzi Editore S.P.A. Bologna, Italy; 1998 pp 353-359.
12. Joubert J. Cysticercal meningitis- a pernicious form of neurocysticercosis which responds poorly to praziquantel. S Afr Med J 1990; 77: 528-530.
13. Garcia HH, Evans CAW, Nash TE et al. Current consensus guidelines for the treatment of Neurocysticercosis. Clin Microbiol Rev 2002; 15: 747-756.(Mishra Devendra, Sharma S)
2 Department of Laboratory Medicine, Chacha Nehru Bal Chikitsalaya, (Maulana Azad Medical College), Geeta Colony, Delhi-110031, India
Abstract
Neurocysticercosis is the commonest CNS parasitic disease worldwide but cysticercal meningoencephalitis is relatively rare, especially in Indian patients. We herein report a girl with cysticercal meningitis that was initially not suspected and later diagnosed on the basis of cerebrospinal fluid (CSF) eosinophilia. The need for CSF examination with wright-giemsa staining to avoid missing CSF eosinophilia is discussed.
Keywords: CSF analysis; CSF eosinophilia; Neurocysticercosis; Lumbar puncture
Neurocysticercosis (NC) is the most frequent parasitic infestation of the central nervous system in the world. It has varied manifestations like seizures, raised intracranial tension, stroke, hydrocephalus, meningoencephalitis etc., depending on the number and localization of the cysts. Cysticercal meningitis is uncommon in India.[1] In the world literature also; acute cysticercal meningitis has been described infrequently, especially in children. We herein describe a 9-year-old girl who presented with acute cysticercal meningitis.
Case Report
The patient was a 9-year-old girl who presented with one-day history of high grade, continuous fever, frontal headache, and projectile vomiting. There was no history of alteration in sensorium or convulsions. She had a history of fever with rhinorrhea 1-week back for which she had received oral amoxycillin for 5 days . There was no history of rash, cough, bleeding, ear-discharge, and any urinary or bowel complaints. She was developmentally normal. Family history and past history were not significant, they had a vegetarian dietary pattern, and there was no history of contact with tubercular patient. On examination, she was conscious but irritable and febrile. Vitals were stable and fundus examination was normal. General physical examination and systemic examination were normal except for positive meningeal signs without focal neurological deficits.
The child was admitted with a provisional diagnosis of meningitis. Investigations revealed total leukocyte count of 10,470/cumm with; polymorphonuclear cells, 68%; lymphocytes, 24%; monocyte 6%; and eosinophils; 2%. Lumbar puncture revealed turbid cerebrospinal fluid (CSF) with 950 cells (polymorphonuclear cells, 90%; lymphocytes, 10%), protein-610 mg/dL, and sugar-45mg/dL (concurrent blood sugar, 72 mg/dL). CSF gram stain and culture revealed no abnormality. Chest X-ray, blood and urine cultures, serum biochemistry, and platelet count were non-contributory. On the basis of compatible clinical and CSF profile, a diagnosis of acute bacterial meningitis was made and treatment with Ceftriaxone, steroids (for 48 hours), and mannitol (single dose, due to raised intracranial pressure) was started. Fever, headache and vomiting subsided within 24 hours. The patient remained well for the next 6 days.
On the 7th day of hospital stay, she developed headache, projectile vomiting and irritability. She had no recrudescence of fever or any convulsions. Examination revealed neck rigidity and positive Kernig sign. There were no focal neurological findings or papilledema. A contrast enhanced computed tomograph (CECT) of the cranium revealed an 8X7 mm hypodense lesion with an eccentric mural nodule, minimal enhancement, but no perilesional edema, in the left periventricular region. On the basis of the radiological picture the lesion was considered to be due to neurocysticercosis. A lumbar puncture was repeated with a specific request to the pathologist to look for eosinophils. CSF was grossly clear with leukocytes-500/cumm; lymphocytes-65%, polymorphonuclear cells-25%, eosinophils-10%; protein-35mg/dL, and sugar-51mg/dL (concurrent blood sugar-82mg/dL). CSF gram stain and culture did not reveal any abnormality. Tuberculin test with 5TU was negative. Serum IgG-ELISA enzyme-linked immunosorbent assay tests for Cysticercosis and for Tuberculosis were Negative. Serum and CSF immunoblot studies and MRI brain to look for subarachnoid cysts could not be done.
The patient was started on dexamethasone, and decongestive measures (mannitol, acetazolamide). Her symptoms improved over the next 12 hours. Indirect ophthalmoscopy ruled out intra-ocular cysts. Subsequently she received albendazole at dose of 15mg/kg/day for 28 days and anticonvulsants. She made an uneventful recovery. At last follow-up (ten month later), she was asymptomatic with no recurrence of symptoms, no evidence of meningeal irritation or raised intracranial pressure (ICT), and no neurodeficiet. Repeat CECT revealed resolution of the lesion.
Discussion
The initial clinical presentation was consistent with the diagnosis of pyogenic meningitis. The previous course of oral antibiotics could have been responsible for the negative CSF gram stain and culture; the rapid improvement on antibiotics, however, supported the diagnosis. When the patient deteriorated after 6 days; subdural effusion, ventriculitis or a concurrent brain abscess were the likely possibilities. The re-appearance of signs of meningeal irritation prompted us to repeat the lumbar puncture. In view of the presence of granuloma on CT, the possibility of cysticercal meningitis was considered, and a Wright Giemsa staining of the CSF was asked for. In the absence of this staining, eosinophils can be mistaken for polymorphonuclear cells.[1]
The finding of the viable NC granuloma on CECT in the absence of any past history of teniasis, seizures, headaches, or any neurological problem was surprising. This finding could also have been coincidental, as viable cysts are known to remain asymptomatic for long periods and reported to be present in up to 1.3% of normal persons (autopsy study).[2] The presence of CSF eosinophilia and the coexistent inflammatory granuloma suggested the possibility of cysticercal meningitis. Cysticercosis is by far the commonest cause of CSF eosinophilia in endemic areas.[3] The CSF findings in cysticercal meningitis consist of pleocytosis (usually lymphocytic but frequently polymorphonuclear), reduced glucose and elevated protein, with up to 70% patients with cysticercal meningitis having CSF eosinophilia between 2-40%.[4],[5],[6] The presence of eosinophils more than 4% of leukocytes in the CSF is of diagnostic significance[4] and is seen in the initial phases of the illness only. For visualizing eosinophils in the CSF, Wright-Giemsa staining of the CSF is required which is not routinely done in wardside-laboratories in most hospitals. In the absence of this staining, eosinophils can be mistaken for polymorphonuclear cells.[1] Other causes of eosinophilic meningitis include coccidioidal meningitis; parasitic infestation with Angiostrongylus cantonensis , Paragonimus westermani , Gnathostoma spinigerum ; intrathecal injection of foreign proteins, and the insertion of rubber tubing into the central nervous system in the course of neurosurgery.[7]
Cysticercal meningoencephalitis is caused by infiltration of the meninges and the parenchyma of the brain by a large number of parasites and inflammation in the surrounding tissue is responsible for meningitis and encephalitis.[8] Although described in 42-48% of patients in Latin American series, cysticercal meningoencephalitis has been reported in less than 10% adults patients with NC in India.[1],[9] Of the two large series of Indian children with NC, only one reported meningoencephalitis in 0.3% of the subjects.[10],[11] Slightly higher proportions have been reported in a small case-series.[8] Around 60% cysticercal meningoencephalitis cases have been reported to have associated parenchymal lesions.[11] Most cases described are chronic in evolution[12] and isolated acute meningitis has rarely been described. A case report from Thailand describes two children presenting as pyogenic meningitis, with normal cranial CT but not responding to antibiotic therapy.[6] Cysticercal meningitis was diagnosed based on the presence of CSF eosinophilia and positive serum serology, and both responded to praziquantel. Both cysticercus ELISA and Electro-Immuno Transfer Blot Assay (EITB) assays in the CSF have high sensitivity for cysticercal meningitis, but these studies in CSF and EITB in serum were not feasible in our setup.
There are no guidelines for the treatment of acute cysticercal meningitis. Anti-parasitic therapy is recommended for chronic meningitis.[13] The decision to treat in this patient was also justified by the radiological presence of a viable cyst. To conclude, we describe a 9-year-old Indian child with acute cysticercal meningitis that initially presented as pyogenic meningitis. Given the high prevalence of cysticercosis in Indian patients, this entity should be suspected in all patients; and CSF eosinophilia specifically looked for, more so when a concurrent parenchymal lesion suggestive of cysticercosis is present.
References
1. Singh G. Neurocysticercosis in the south-central America and the Indian subcontinent. Arq Neuropsiquiatr 1997; 55: 349-356.
2. Mahajan RC. Geographical distribution of human cysticercosis. In, Cysticercosis: Present state of Knowledge and Perspectives , (Ed.) Ana Flisser, et al. Academic Press 1982, 39-46.
3. Tasker WG, Plotkin SA. Cerebral Cysticercosis. Pediatrics 1979; 63: 761-763.
4. Wang CH, Gao SF, Guo YP. Diagnostic significance of eosinophilia of the cerebro-spinal fluid in cerebral cysticercosis. Chinese Medical Journal 1993; 106: 282-284.
5. Wilber RR, King EB, Howes EL. Cerebrospinal fluid cytology in five patients with cerebral cysticercosis. Acta Cytologica 1980; 24 : 421-426.
6. Visudhipan P, Chiemchanya S. Acute cysticercal meningitis in children: response to praziquantel. Ann Trop Ped 1997; 17: 9-13.
7. Char DFB, Rosen L. Eosinophilic meningitis among children in Hawaii. J Pediatr 1967; 70 : 28-35.
8. Puri V, Sharma DK. Neurocysticercosis in children. Indian Pediatr 1991; 28 : 1309-1317.
9. Venkatraman S, Roy AK, Dhamija RM, Sanchetee PC. Cysticercal meningoencephalitis - Clinical presentation and autopsy findings. J Assoc Phys India 1990; 38: 763-765.
10. Singhi P, Ray M, Singhi S, Khandelwal P. Clinical spectrum of 500 children with neurocysticercosis and response to albendazole therapy. J Child Neurol 2000; 15: 207-213.
11. Kalra V, Mittal R, Rana KS, Gupta A. Neurocysticercosis: Indian experience. Perat MV (Ed), New Developments in Neurology , Monduzzi Editore S.P.A. Bologna, Italy; 1998 pp 353-359.
12. Joubert J. Cysticercal meningitis- a pernicious form of neurocysticercosis which responds poorly to praziquantel. S Afr Med J 1990; 77: 528-530.
13. Garcia HH, Evans CAW, Nash TE et al. Current consensus guidelines for the treatment of Neurocysticercosis. Clin Microbiol Rev 2002; 15: 747-756.(Mishra Devendra, Sharma S)