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Sequential imaging studies of cerebral gnathostomiasis with subdural hemorrhage as its complication
Transactions of the Royal Society of Tropical Medicine and Hygiene (2009) 103, 102—104
available at www.sciencedirect.com
journal homepage: www.elsevierhealth.com/journals/trst
CASE REPORT
Sequential imaging studies of cerebral gnathostomiasis with subdural hemorrhage as its complication Kittisak Sawanyawisuth a,∗, Maciej Piotr Chlebicki b, Edward Pratt b, Jaturat Kanpittaya c, Pewpan M. Intapan d a
Department of Medicine, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand Department of Medicine, Changi General Hospital, Singapore 529889, Singapore c Department of Radiology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand d Department of Parasitology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand b
Received 1 June 2008; received in revised form 15 September 2008; accepted 15 September 2008 Available online 31 October 2008
KEYWORDS Gnathostomiasis; Cerebral; Subdural hemorrhage; Pathogenesis; Computed tomography; Magnetic resonance imaging
Summary Cerebral gnathostomiasis is a severe form of human infection caused by Gnathostoma spinigerum. We report sequential brain imaging in serologically proven cerebral gnathostomiasis. The clinical and radiographic correlation showed the probable route taken by the parasite during its migration through the central nervous system. We offer additional insight into pathogenesis of cerebral gnathostomiasis and its rare complication, subdural hemorrhage, a finding not previously reported in the literature. © 2008 Royal Society of Tropical Medicine and Hygiene. Published by Elsevier Ltd. All rights reserved.
1. Introduction Gnathostomiasis, caused by Gnathostoma spinigerum, is endemic in Thailand and Japan. It is emerging in nonendemic areas due to increasing international travel. However, clinical awareness of this disease remains low. Cerebral gnathostomiasis usually results in severe neurological deficit, coma or death.1,2
We report a case of cerebral gnathostomiasis with extensive central nervous system (CNS) involvement. Sequential radiologic imaging studies correlated with clinical findings allowed us to reconstruct the probable route taken by the parasite during its migration through the CNS. These findings offer additional insight into the pathogenesis of cerebral gnathostomiasis and its complication, subdural hemorrhage, a finding not previously reported in the literature.
2. Case report ∗
Corresponding author. Tel.: +66 43 363 664; fax: +66 43 347 542. E-mail address: [email protected] (K. Sawanyawisuth).
A 46-year-old woman from northeastern Thailand was admitted to Changi General Hospital, Singapore, on 22 May 2007,
0035-9203/$ — see front matter © 2008 Royal Society of Tropical Medicine and Hygiene. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.trstmh.2008.09.011
Pathogenesis of cerebral gnathostomiasis
Figure 1 Three days after admission, non-contrast computed tomography showed acute subdural hemorrhage along the tentorial fissures.
because of severe headache and diplopia for 2 days. She also had intermittent severe left neck pain that radiated to the occipital area 3 days before the headache. On the day of admission, she developed confusion. She reported a history of regularly eating raw fish and symptoms of intermittent, migratory, subcutaneous swelling on her left arm for 5 years. Informed consent was given by the patient for her assessment and treatment. Physical examination revealed a drowsy middle-aged woman without fever. Neurological examination revealed bilateral sixth and right seventh cranial nerve palsies. Computed tomography (CT) of the brain showed no abnormality. A complete blood count (CBC) showed a total white blood cell (WBC) count of 10,700 cells/mm3 with 30% eosinophils. Plasma blood chemistry was within normal limits. Magnetic resonance imaging (MRI) performed on the same day revealed two small round lesions with heterogeneous high signal intensity on axial T2-weighted images in the left medial and right lateral thalamus, and another round high signal intensity lesion found in the left deep insular cortex region. There were also multiple small periventricular white matter lesions. The axial gradient image showed multiple small areas of abnormal dark signal lesions in the left thalamus and the left side of the basal ganglia (Supplementary Figure 1). Lumbar puncture revealed slightly turbid cerebrospinal fluid (CSF) with an opening pressure of 22 cmH2 O. Total CSF WBC was 1050 cells/mm3 (60% neutrophils, 40% lymphocytes on routine Gram stain). However, hematoxylin-eosin stain demonstrated prominent eosinophils (30%). CSF total protein and glucose were 0.82 g/l (0.10—0.40) and 0.9 mmol/l (2.4—5.5), respectively. The results of CSF Gram stain, acidfast stain and Indian ink, cryptococcal antigen, and cultures for bacteria, fungi and mycobacteria were all negative. Three days after admission, the patient developed rightsided weakness. The CT scan of the brain showed an acute subdural hemorrhage along the tentorial (Figure 1) and interhemispheric fissures (Supplementary Figure 2). Meanwhile, the serum was tested strongly positive on the specific band to a 24 kDa G. spinigerum antigen3 but negative for both Angiostrongylus cantonensis and Cysticercus antigens.
103
Figure 2 Ten days later, non-contrast computed tomography showed a new hemorrhagic lesion at the left frontal gray-white junction, with high attenuation changes next to the interhemispheric fissure subdural hemorrhage.
The patient was treated with oral albendazole 800 mg/d for 14 d with additional corticosteroid. Ten days later, a repeated CT scan of the brain showed partial resolution of the subdural hemorrhagic lesion with a new hemorrhagic lesion at the left frontal gray-white junction with high attenuation changes (Figure 2). Twenty days later, the patient was discharged with minimal cranial nerve palsy and right arm weakness. At 6 months follow-up, the patient had minimal right facial palsy and right-sided weakness with ipsilateral hypereflexia and positive palmomental reflex. The MRI scan showed an unchanged lesion in the left insular cortex, decreased size lesion in the left medial thalamus, and complete resolution of subdural hemorrhage and right lateral thalamic lesion. However, there was increased size of periventricular white matter lesions (more on the left, Supplementary Figure 3). In addition, a well-delineated hemorrhagic tract was detected in the left frontal lobe in the gray-white junction and deep white matter region (Figure 3). The subsequent CBC showed the resolution of eosinophilia.
3. Discussion Gnathostomiasis can be diagnosed clinically. However, in uncertain cases, the detection of Gnathostoma antibodies in either serum or CSF is helpful and reliable in confirming the diagnosis.4 Gnathostoma larvae can stay in the human body for 10—12 years. Our patient probably acquired the infection several years earlier, as she had had a long history of migratory swelling in her left arm. The larva entered the CNS via the left cervical nerve root and caused typical left cervical radicular pain. Subsequently, the invasion of meninges through the brain stem contributed to eosinophilic meningoencephalitis. The subdural and frontal hemorrhage indicated the upward migration of the larva to the left frontal lobe. It died there after the albendazole treatment and resulted in positive palmomental reflex. The demyelination of neuronal tissues, caused by tissue injuries and reactions, may represent the non-specific findings in the thalamus and periventricular area on MRIs.
104
K. Sawanyawisuth et al. Hospital, Khon Kaen University, Thailand; KS drafted the manuscript, which was reviewed and augmented by all the authors. All authors read and approved the final manuscript. KS and MPC are guarantors of the paper.
Appendix A. Supplementary data Supplementary data associated with this article can be found, in the online version, at doi:10.1016/j.trstmh. 2008.09.011. Funding: None. Conflicts of interests: None declared. Ethical approval: Not required; the assessment and treatment of the patient was part of the standard clinical procedures at the Changi General Hospital, Singapore. Figure 3 Six months later, a coronal T1-weighted magnetic resonance image showed a hemorrhagic tract at the gray-white junction and deep white matter region of the left frontal lobe (in the same area as in Figure 2).
All these abnormal clinical and radiological findings may be explained by the migration of a single larva of G. spinigerum as previously reported.5 We believe that this is the first report that has shown the larva migration by sequential imaging studies and subdural hemorrhage as its complication. Another lesson we learned was that the initial CT might not be an appropriate tool to initially evaluate cerebral gnathostomiasis. Authors’ note: Informed consent for publication of this case report and inclusion of the clinical photographs was given by the patient. Authors’ contributions: KS, MPC, EP, JK and PMI all contributed to the patient’s clinical care and assessment either at Changi General Hospital, Singapore or Srinagarind
References 1. Moore DA, McCroddan J, Dekumyoy P, Chiodini PL. Gnathostomiasis: an emerging imported disease. Emerg Infect Dis 2003;9:647—50. 2. Sawanyawisuth K, Tiamkao S, Kanpittaya J, Dekumyoy P, Jitpimolmard S. MR imaging findings in cerebrospinal gnathostomiasis. AJNR Am J Neuroradiol 2004;25:446—9. 3. Laummaunwai P, Sawanyawisuth K, Intapan PM, Chotmongkol V, Wongkham C, Maleewong W. Evaluation of human IgG class and subclass antibodies to a 24 kDa antigenic component of Gnathostoma spinigerum for the serodiagnosis of gnathostomiasis. Parasitol Res 2007;101:703—8. 4. Tuntipopipat S, Chawengkiattikul R, Witoonpanich R, Chiemchanya S, Sirisinha S. Antigens, antibodies and immune complexes in cerebrospinal fluid of patients with cerebral gnathostomiasis. Southeast Asian J Trop Med Public Health 1989;20:439—46. 5. Punyagupta S, Bunnag T, Juttijudata P. Eosinophilic meningitis in Thailand. Clinical and epidemiological characteristics of 162 patients with myeloencephalitis probably caused by Gnathostoma spinigerum. J Neurol Sci 1990;96:241—56.
available at www.sciencedirect.com
journal homepage: www.elsevierhealth.com/journals/trst
CASE REPORT
Sequential imaging studies of cerebral gnathostomiasis with subdural hemorrhage as its complication Kittisak Sawanyawisuth a,∗, Maciej Piotr Chlebicki b, Edward Pratt b, Jaturat Kanpittaya c, Pewpan M. Intapan d a
Department of Medicine, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand Department of Medicine, Changi General Hospital, Singapore 529889, Singapore c Department of Radiology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand d Department of Parasitology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand b
Received 1 June 2008; received in revised form 15 September 2008; accepted 15 September 2008 Available online 31 October 2008
KEYWORDS Gnathostomiasis; Cerebral; Subdural hemorrhage; Pathogenesis; Computed tomography; Magnetic resonance imaging
Summary Cerebral gnathostomiasis is a severe form of human infection caused by Gnathostoma spinigerum. We report sequential brain imaging in serologically proven cerebral gnathostomiasis. The clinical and radiographic correlation showed the probable route taken by the parasite during its migration through the central nervous system. We offer additional insight into pathogenesis of cerebral gnathostomiasis and its rare complication, subdural hemorrhage, a finding not previously reported in the literature. © 2008 Royal Society of Tropical Medicine and Hygiene. Published by Elsevier Ltd. All rights reserved.
1. Introduction Gnathostomiasis, caused by Gnathostoma spinigerum, is endemic in Thailand and Japan. It is emerging in nonendemic areas due to increasing international travel. However, clinical awareness of this disease remains low. Cerebral gnathostomiasis usually results in severe neurological deficit, coma or death.1,2
We report a case of cerebral gnathostomiasis with extensive central nervous system (CNS) involvement. Sequential radiologic imaging studies correlated with clinical findings allowed us to reconstruct the probable route taken by the parasite during its migration through the CNS. These findings offer additional insight into the pathogenesis of cerebral gnathostomiasis and its complication, subdural hemorrhage, a finding not previously reported in the literature.
2. Case report ∗
Corresponding author. Tel.: +66 43 363 664; fax: +66 43 347 542. E-mail address: [email protected] (K. Sawanyawisuth).
A 46-year-old woman from northeastern Thailand was admitted to Changi General Hospital, Singapore, on 22 May 2007,
0035-9203/$ — see front matter © 2008 Royal Society of Tropical Medicine and Hygiene. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.trstmh.2008.09.011
Pathogenesis of cerebral gnathostomiasis
Figure 1 Three days after admission, non-contrast computed tomography showed acute subdural hemorrhage along the tentorial fissures.
because of severe headache and diplopia for 2 days. She also had intermittent severe left neck pain that radiated to the occipital area 3 days before the headache. On the day of admission, she developed confusion. She reported a history of regularly eating raw fish and symptoms of intermittent, migratory, subcutaneous swelling on her left arm for 5 years. Informed consent was given by the patient for her assessment and treatment. Physical examination revealed a drowsy middle-aged woman without fever. Neurological examination revealed bilateral sixth and right seventh cranial nerve palsies. Computed tomography (CT) of the brain showed no abnormality. A complete blood count (CBC) showed a total white blood cell (WBC) count of 10,700 cells/mm3 with 30% eosinophils. Plasma blood chemistry was within normal limits. Magnetic resonance imaging (MRI) performed on the same day revealed two small round lesions with heterogeneous high signal intensity on axial T2-weighted images in the left medial and right lateral thalamus, and another round high signal intensity lesion found in the left deep insular cortex region. There were also multiple small periventricular white matter lesions. The axial gradient image showed multiple small areas of abnormal dark signal lesions in the left thalamus and the left side of the basal ganglia (Supplementary Figure 1). Lumbar puncture revealed slightly turbid cerebrospinal fluid (CSF) with an opening pressure of 22 cmH2 O. Total CSF WBC was 1050 cells/mm3 (60% neutrophils, 40% lymphocytes on routine Gram stain). However, hematoxylin-eosin stain demonstrated prominent eosinophils (30%). CSF total protein and glucose were 0.82 g/l (0.10—0.40) and 0.9 mmol/l (2.4—5.5), respectively. The results of CSF Gram stain, acidfast stain and Indian ink, cryptococcal antigen, and cultures for bacteria, fungi and mycobacteria were all negative. Three days after admission, the patient developed rightsided weakness. The CT scan of the brain showed an acute subdural hemorrhage along the tentorial (Figure 1) and interhemispheric fissures (Supplementary Figure 2). Meanwhile, the serum was tested strongly positive on the specific band to a 24 kDa G. spinigerum antigen3 but negative for both Angiostrongylus cantonensis and Cysticercus antigens.
103
Figure 2 Ten days later, non-contrast computed tomography showed a new hemorrhagic lesion at the left frontal gray-white junction, with high attenuation changes next to the interhemispheric fissure subdural hemorrhage.
The patient was treated with oral albendazole 800 mg/d for 14 d with additional corticosteroid. Ten days later, a repeated CT scan of the brain showed partial resolution of the subdural hemorrhagic lesion with a new hemorrhagic lesion at the left frontal gray-white junction with high attenuation changes (Figure 2). Twenty days later, the patient was discharged with minimal cranial nerve palsy and right arm weakness. At 6 months follow-up, the patient had minimal right facial palsy and right-sided weakness with ipsilateral hypereflexia and positive palmomental reflex. The MRI scan showed an unchanged lesion in the left insular cortex, decreased size lesion in the left medial thalamus, and complete resolution of subdural hemorrhage and right lateral thalamic lesion. However, there was increased size of periventricular white matter lesions (more on the left, Supplementary Figure 3). In addition, a well-delineated hemorrhagic tract was detected in the left frontal lobe in the gray-white junction and deep white matter region (Figure 3). The subsequent CBC showed the resolution of eosinophilia.
3. Discussion Gnathostomiasis can be diagnosed clinically. However, in uncertain cases, the detection of Gnathostoma antibodies in either serum or CSF is helpful and reliable in confirming the diagnosis.4 Gnathostoma larvae can stay in the human body for 10—12 years. Our patient probably acquired the infection several years earlier, as she had had a long history of migratory swelling in her left arm. The larva entered the CNS via the left cervical nerve root and caused typical left cervical radicular pain. Subsequently, the invasion of meninges through the brain stem contributed to eosinophilic meningoencephalitis. The subdural and frontal hemorrhage indicated the upward migration of the larva to the left frontal lobe. It died there after the albendazole treatment and resulted in positive palmomental reflex. The demyelination of neuronal tissues, caused by tissue injuries and reactions, may represent the non-specific findings in the thalamus and periventricular area on MRIs.
104
K. Sawanyawisuth et al. Hospital, Khon Kaen University, Thailand; KS drafted the manuscript, which was reviewed and augmented by all the authors. All authors read and approved the final manuscript. KS and MPC are guarantors of the paper.
Appendix A. Supplementary data Supplementary data associated with this article can be found, in the online version, at doi:10.1016/j.trstmh. 2008.09.011. Funding: None. Conflicts of interests: None declared. Ethical approval: Not required; the assessment and treatment of the patient was part of the standard clinical procedures at the Changi General Hospital, Singapore. Figure 3 Six months later, a coronal T1-weighted magnetic resonance image showed a hemorrhagic tract at the gray-white junction and deep white matter region of the left frontal lobe (in the same area as in Figure 2).
All these abnormal clinical and radiological findings may be explained by the migration of a single larva of G. spinigerum as previously reported.5 We believe that this is the first report that has shown the larva migration by sequential imaging studies and subdural hemorrhage as its complication. Another lesson we learned was that the initial CT might not be an appropriate tool to initially evaluate cerebral gnathostomiasis. Authors’ note: Informed consent for publication of this case report and inclusion of the clinical photographs was given by the patient. Authors’ contributions: KS, MPC, EP, JK and PMI all contributed to the patient’s clinical care and assessment either at Changi General Hospital, Singapore or Srinagarind
References 1. Moore DA, McCroddan J, Dekumyoy P, Chiodini PL. Gnathostomiasis: an emerging imported disease. Emerg Infect Dis 2003;9:647—50. 2. Sawanyawisuth K, Tiamkao S, Kanpittaya J, Dekumyoy P, Jitpimolmard S. MR imaging findings in cerebrospinal gnathostomiasis. AJNR Am J Neuroradiol 2004;25:446—9. 3. Laummaunwai P, Sawanyawisuth K, Intapan PM, Chotmongkol V, Wongkham C, Maleewong W. Evaluation of human IgG class and subclass antibodies to a 24 kDa antigenic component of Gnathostoma spinigerum for the serodiagnosis of gnathostomiasis. Parasitol Res 2007;101:703—8. 4. Tuntipopipat S, Chawengkiattikul R, Witoonpanich R, Chiemchanya S, Sirisinha S. Antigens, antibodies and immune complexes in cerebrospinal fluid of patients with cerebral gnathostomiasis. Southeast Asian J Trop Med Public Health 1989;20:439—46. 5. Punyagupta S, Bunnag T, Juttijudata P. Eosinophilic meningitis in Thailand. Clinical and epidemiological characteristics of 162 patients with myeloencephalitis probably caused by Gnathostoma spinigerum. J Neurol Sci 1990;96:241—56.