- Email: [email protected]
Curvularia brain abscess
Case Reports / Journal of Clinical Neuroscience 20 (2013) 173–175
translocation.1,3 In a recent histopathologic series of 32 MZBCL, the authors found one patient with an IgG4 positive thoracic dural lesion without further elaboration, and concluded that there may be a strong association between MZBCL of the dura and IgG4-related diseases.3 A recent description of an IgG4-related sclerosing pachymeningitis in the thoracic dura also indicates that IgG4 may have an important role in dural MZL.5 In our patient, the lymphoma consisted of monoclonal B cells that carried IGH@ translocations, a finding which has been reported commonly in orbital MZBCL3 but not in the setting of dural MZBCL.
4. Conclusion To our knowledge this is the first report of Ex-MZBCL arising purely within the tissue of spinal dura. The clinical, pathologic and cytogenetic profile suggests that it is similar to MZBCL of other locations. Primary MZBCL of the spinal dura should be considered in patients with slowly progressive spinal lesions.
173
Appendix A. Supplementary data Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.jocn.2012.01.042. References 1. Bhagavathi S, Greiner TC, Kazmi SA, et al. Extranodal marginal zone lymphoma of the dura mater with IgH/MALT1 translocation and review of literature. J Hematop 2008;1:131–7. 2. Tu PH, Giannini C, Judkins AR, et al. Clinicopathologic and genetic profile of intracranial marginal zone lymphoma: a primary low-grade CNS lymphoma that mimics meningioma. J Clin Oncol 2005;23:5718–27. 3. Venkataraman G, Rizzo KA, Chavez JJ, et al. Marginal zone lymphomas involving meningeal dura: possible link to IgG4-related diseases. Mod Pathol. 2011; 24:355–366. 4. Park I, Huh J, Kim JH, et al. Primary central nervous system marginal zone B-cell lymphoma of the Basal Ganglia mimicking low-grade glioma: a case report and review of the literature. Clin Lymphoma Myeloma 2008;8:305–8. 5. Chan SK, Cheuk W, Chan KT, et al. IgG4-related sclerosing pachymeningitis: a previously unrecognized form of central nervous system involvement in IgG4related sclerosing disease. Am J Surg Pathol 2009;33:1249–52.
doi:http://dx.doi.org/10.1016/j.jocn.2012.01.042
Curvularia brain abscess Nisha Gadgil a, Michael Kupferman b, Sheila Smitherman a, Gregory N. Fuller c, Ganesh Rao a,⇑ a b c
Department of Neurosurgery, The University of Texas M.D. Anderson Cancer Center, 1515 Holcombe Boulevard, Houston, TX 77030, USA Department of Head and Neck Surgery, The University of Texas M.D. Anderson Cancer Center, 1515 Holcombe Boulevard, Houston, TX 77030, USA Department of Pathology, The University of Texas M.D. Anderson Cancer Center, 1515 Holcombe Boulevard, Houston, TX 77030, USA
a r t i c l e
i n f o
Article history: Received 23 May 2012 Accepted 27 May 2012
Keywords: Curvularia abscess Brain abscess Phaeohyphomycosis
a b s t r a c t Curvularia is a ubiquitous dematiaceous fungus that is a very rare but often fatal cause of infection in the central nervous system (CNS). In this report, we describe a patient with chronic sinusitis who presented with a Curvularia abscess of the skull base extending into the left frontal lobe. She was successfully treated with aggressive surgical resection and antibiotic therapy. In the published literature, this patient to our knowledge represents the longest period of disease-free follow-up in those afflicted with CNS Curvularia infection, indicating the importance of proper diagnosis and aggressive surgical debridement for a successful outcome. Ó 2012 Elsevier Ltd. All rights reserved.
1. Introduction Curvularia is a pigmented filamentous fungus ubiquitous in the soil yet rarely a cause of human infection. However, it has been described in patients with sinusitis, keratitis, cutaneous infection, respiratory infection, endocarditis, and brain abscess. Central nervous system (CNS) phaeohyphomycosis has an extremely poor prognosis.1 Here, we report a previously healthy individual with a Curvularia brain abscess. After extensive surgical and antibiotic therapy, she has been disease-free for a follow-up period of more than 3 years. This report demonstrates the importance of an aggressive therapeutic approach in CNS infection caused by Curvularia. 2. Case report A 50-year-old immunocompetent female with a history of chronic sinusitis and previous sinus surgery presented with a sei⇑ Corresponding author. Tel.: +1 713 792 2400; fax: +1 713 794 4950. E-mail address: [email protected] (G. Rao).
zure followed by weakness, slurred speech, and memory loss. The patient reported a 4-year history of headaches and blurred vision in the right eye progressing, in the prior few months, to unbearable headaches, syncopal episodes, and bloody nasal drainage. The patient was noted to have dysarthria, ataxia, numbness of the right ophthalmic division of cranial nerve V, and right eye proptosis. CT scan showed destruction of the anterior cranial fossa floor (Fig. 1A). MRI of the head revealed a 4.6 cm 4.2 cm 3.2 cm destructive mass in the right inferior frontal lobe extending into the ethmoid, right orbit, and cranial base (Fig. 1B), accompanied by subfalcine herniation and posterior mass effect. A biopsy (Fig. 1C) of the mass revealed necrotizing granulomatous inflammation and pigmented fungal organisms identified as Curvularia species. The patient was started on voriconazole, minocycline, and levofloxacin. A stereotactic-guided combined endoscopic-assisted craniofacial resection was performed. After bifrontal craniotomy, the mass was resected along with the inferior frontal lobe. Frontal sinus exenteration was performed. Extensive erosions of the medial orbit, cribiform plate, and roof of the sphenoid were noted.
174
Case Reports / Journal of Clinical Neuroscience 20 (2013) 173–175
Fig. 1. Imaging studies of an immunocompetent 50-year-old female with a Curvularia brain abscess: (A) sagittal reconstruction of a non-contrast CT scan (bone windows) showing destruction of the anterior cranial base (arrow); (B) sagittal post gadolinium T1-weighted MRI showing a large enhancing mass extending from the skull base into the left frontal lobe; (C) transnasal endoscopic view of the right nasal cavity, demonstrating an extensive necrotic soft tissue mass (# = nasal septum; = right middle turbinate; arrowhead = sphenoid sinus); and (D) sagittal MRI (post gadolinium T1-weighted MRI) 3 years after surgery and antibiotic treatment showing no evidence of disease recurrence. (E–H) Photomicrograph showing: (E) the specimen removed at surgery demonstrating meningeal thickening (Men) with lymphocytic infiltration, giant cells, and invasion into the brain (NB = normal brain) (hematoxylin and eosin [H&E], 100); (F) septated hyphae and terminal dilatation (arrow) consistent with Curvularia (H&E, 400); (G) hyphal forms against a background of meninges and brain tissue (Grocott methenamine silver stain [GMS], 100); (H) terminal dilatation (arrow) consistent with Curvularia (GMS, 400).
Endonasal disease along the right medial orbital wall and right sphenoid sinus was resected transnasally. A pericranial graft was utilized to reconstruct the skull base. The patient’s hospital course was uncomplicated and she was discharged on antibiotics. She was followed with serial MRI. After more than 3 years of follow-up, the patient remained disease-free (Fig. 1D). Pathological examination of the specimen revealed extensive meningeal thickening with infiltration including multi-nucleated giant cells and lymphocytes (Fig. 1E). Higher magnification revealed fungal hyphae (Fig. 1F). Grocott methenamine silver (GMS) stain showed extensive fungal involvement (Fig. 1G). Higher magnification image of GMS stain revealed terminal dilatation, an artifact of tissue fixation that is characteristic of Curvularia (Fig. 1H). 3. Discussion Phaeohyphomycosis, an infection caused by pigmented fungi with mycelial morphology,2 is a rare but fatal cause of cerebral infection. In a review of 101 cases of CNS phaeohyphomycosis,1 73% were fatal, although most patients had no underlying risk factor. These organisms respond poorly to standard antifungal therapy, but patients treated with combination antifungals and complete surgical excision have better outcomes.1
To our knowledge only seven patients with CNS phaeohyphomycosis caused by Curvularia species have been reported. Friedman et al.3 described a patient with a pulmonary infiltrate and frontoparietal abscess caused by C. pallescens, which recurred fatally 3years after surgical excision. Rohwedder et al.4 described a patient with a decade-long infection with C. lunata, which caused soft tissue, pulmonary, paravertebral, and cerebral abscesses in which the patient ultimately refused further therapy. Pierce et al.5 described a patient with a lung and multiple cerebral lesions caused by C. lunata treated with prolonged amphotericin B with no relapse up to one year follow-up. Ebright et al.6 reported a patient with invasive sinusitis and bifrontal cerebritis caused by C. clavata treated with aggressive resection with no relapse up to 2 years of follow up. Carter and Boudreaux7 reported a patient with a large basal ganglia lesion caused by C. lunata that was fatal within 1month. Smith et al.8 reported a patient with a pituitary fossa lesion resulting in optic nerve atrophy caused by C. lunata that was treated with stereotactic transphenoidal resection and antifungals without relapse up to 8 months follow-up. Singh et al.9 reported a patient with a cranial base lesion caused by C. geniculata treated with limited transnasal endoscopic debulking. The patient died 4 months later. Because of this patient’s long history of chronic sinusitis and evidence of sinus erosion, the mechanism of infection was probably inhalation of spores, colonization of the nasal sinuses, and local
Case Reports / Journal of Clinical Neuroscience 20 (2013) 175–177
extension of infection into the frontal lobe. Curvularia is a common cause of allergic fungal sinusitis,10 although there is only one report of a patient with associated cerebral Curvularia infection.8 It is unclear why this ubiquitous organism causes invasive cerebral disease in rare instances. Although definitive treatment of cerebral Curvularia infection has not been established, the high mortality and incidence of relapse suggest that an aggressive therapeutic approach is paramount. In our patient, follow-up for more than 3 years confirms no residual disease or relapse. This is the longest period of disease-free follow-up reported for intracranial Curvularia infection, the success of which is attributed to aggressive surgical treatment and long-term intravenous antifungals. References 1. Revankar SG, Sutton DA, Rinaldi MG. Primary central nervous system phaeohyphomycosis: a review of 101 cases. Clin Infect Dis 2004;38:206–16.
175
2. University of Adelaide. Phaeohyphomycosis. Mycology Online. accessed 23.03.11. 3. Friedman AD, Campos JM, Roke LB, et al. Fatal recurrent Curvularia brain abscess. J Pediatr 1981;99:413–5. 4. Rohwedder JJ, Simmons JL, Colfer H, et al. Disseminated Curvularia lunata infection in a football player. Arch Intern Med 1979;139:940–1. 5. Pierce NF, Millan JC, Bender BS, et al. Disseminated Curvularia infection. Additional therapeutic and clinical considerations with evidence of medical cure. Arch Pathol Lab Med 1986;110:959–61. 6. Ebright JR, Chandrasekar PH, Marks S, et al. Invasive sinusitis and cerebritis due to Curvularia clavata in an immunocompetent adult. Clin Infect Dis 1999;28:687–9. 7. Carter E, Boudreaux C. Fatal cerebral phaeohyphomycosis due to Curvularia lunata in an immunocompetent patient. J Clin Microbiol 2004;42:5419–23. 8. Smith T, Goldschlager T, Mott N, et al. Optic atrophy due to Curvularia lunata mucocoele. Pituitary 2007;10:295–7. 9. Singh H, Irwin S, Falowski S, et al. Curvularia fungi presenting as a large cranial base meningioma: case report. Neurosurgery 2008;63:E177. 10. El-Morsy SM, Khafagy YW, El-Naggar MM, et al. Allergic fungal rhinosinusitis: detection of fungal DNA in sinus aspirate using polymerase chain reaction. J Laryngol Otol 2010;124:152–60.
doi:http://dx.doi.org/10.1016/j.jocn.2012.05.012
Clinical and radiologic spectrum of corpus callosum infarctions: Clues to the etiology Santosh B. Murthy, Mohamad Chmayssani, Shreyansh Shah, Corey E. Goldsmith, Joseph S. Kass ⇑ Department of Neurology, Baylor College of Medicine, 6501 Fannin, NB302, Houston, TX 77030, USA
a r t i c l e
i n f o
Article history: Received 22 May 2012 Accepted 27 May 2012
Keywords: Alien hand syndrome Anterior cerebral artery Corpus callosum Infarction
a b s t r a c t Infarctions of the corpus callosum are rare. The clinical picture varies from an acute onset to slow evolving symptoms, frequently with poor localizing signs; however, the location of the infarct in the callosum often correlates with a specific etiology. We describe three patients with varying degrees of callosal infarction, each corresponding to a particular etiology. Published by Elsevier Ltd.
1. Introduction We report three patients with corpus callosum (CC) infarctions and aim to highlight the subtle radiological features that would aid in identifying the specific etiology.
2. Case report 2.1. Patient 1 A 56-year-old right-handed Hispanic man with hypertension, hyperlipidemia and coronary artery disease presented with new onset chest pain. He underwent three-vessel coronary artery bypass grafting. On post-operative day 2, he was noted to have left-sided hemiparesis. A CT scan of the head showed a hypodensity in the CC. MRI showed diffusion restriction confined exclusively to the body of the CC (Fig. 1A). A magnetic resonance angiogram (MRA), revealed severe (>70%) atherosclerotic narrowing of the bilateral distal petrous and cavernous internal ⇑ Corresponding author. Tel.: +1 713 798 6151; fax: +1 713 798 8350. E-mail address: [email protected] (J.S. Kass).
carotid arteries (ICA). A double lumen was noted at the junction of the petrous and cavernous segments of the left ICA, raising concerns for a dissection as opposed to a calcified plaque (Fig. 1B). Despite the possibility of a dissection, therapeutic anticoagulation was not considered given the recent cardiac surgery. Following extubation, he was noted to have ideomotor apraxia of the non-dominant hand consistent with alien hand syndrome. At the 6-month follow-up, the patient was ambulatory with assistance, but continued to have apraxia affecting the left hand. 2.2. Patient 2 A 46-year-old right-handed Hispanic man with diabetes and hypertension presented with acute onset of decreased volitional speech output and bilateral lower extremity weakness. On neurologic exam, he appeared withdrawn and had complete speech arrest with intact comprehension and normal writing and ability to follow commands. Initial National Institutes of Health Stroke Scale (NIHSS) score was 9 and he received intravenous alteplase (tissue plasminogen activator, tPA). Brain MRI revealed diffusion restriction in the striato-capsular region on the right, genu of the CC, and frontal subcortical white matter bilaterally (Fig. 2A, B). A CT
translocation.1,3 In a recent histopathologic series of 32 MZBCL, the authors found one patient with an IgG4 positive thoracic dural lesion without further elaboration, and concluded that there may be a strong association between MZBCL of the dura and IgG4-related diseases.3 A recent description of an IgG4-related sclerosing pachymeningitis in the thoracic dura also indicates that IgG4 may have an important role in dural MZL.5 In our patient, the lymphoma consisted of monoclonal B cells that carried IGH@ translocations, a finding which has been reported commonly in orbital MZBCL3 but not in the setting of dural MZBCL.
4. Conclusion To our knowledge this is the first report of Ex-MZBCL arising purely within the tissue of spinal dura. The clinical, pathologic and cytogenetic profile suggests that it is similar to MZBCL of other locations. Primary MZBCL of the spinal dura should be considered in patients with slowly progressive spinal lesions.
173
Appendix A. Supplementary data Supplementary data associated with this article can be found, in the online version, at http://dx.doi.org/10.1016/j.jocn.2012.01.042. References 1. Bhagavathi S, Greiner TC, Kazmi SA, et al. Extranodal marginal zone lymphoma of the dura mater with IgH/MALT1 translocation and review of literature. J Hematop 2008;1:131–7. 2. Tu PH, Giannini C, Judkins AR, et al. Clinicopathologic and genetic profile of intracranial marginal zone lymphoma: a primary low-grade CNS lymphoma that mimics meningioma. J Clin Oncol 2005;23:5718–27. 3. Venkataraman G, Rizzo KA, Chavez JJ, et al. Marginal zone lymphomas involving meningeal dura: possible link to IgG4-related diseases. Mod Pathol. 2011; 24:355–366. 4. Park I, Huh J, Kim JH, et al. Primary central nervous system marginal zone B-cell lymphoma of the Basal Ganglia mimicking low-grade glioma: a case report and review of the literature. Clin Lymphoma Myeloma 2008;8:305–8. 5. Chan SK, Cheuk W, Chan KT, et al. IgG4-related sclerosing pachymeningitis: a previously unrecognized form of central nervous system involvement in IgG4related sclerosing disease. Am J Surg Pathol 2009;33:1249–52.
doi:http://dx.doi.org/10.1016/j.jocn.2012.01.042
Curvularia brain abscess Nisha Gadgil a, Michael Kupferman b, Sheila Smitherman a, Gregory N. Fuller c, Ganesh Rao a,⇑ a b c
Department of Neurosurgery, The University of Texas M.D. Anderson Cancer Center, 1515 Holcombe Boulevard, Houston, TX 77030, USA Department of Head and Neck Surgery, The University of Texas M.D. Anderson Cancer Center, 1515 Holcombe Boulevard, Houston, TX 77030, USA Department of Pathology, The University of Texas M.D. Anderson Cancer Center, 1515 Holcombe Boulevard, Houston, TX 77030, USA
a r t i c l e
i n f o
Article history: Received 23 May 2012 Accepted 27 May 2012
Keywords: Curvularia abscess Brain abscess Phaeohyphomycosis
a b s t r a c t Curvularia is a ubiquitous dematiaceous fungus that is a very rare but often fatal cause of infection in the central nervous system (CNS). In this report, we describe a patient with chronic sinusitis who presented with a Curvularia abscess of the skull base extending into the left frontal lobe. She was successfully treated with aggressive surgical resection and antibiotic therapy. In the published literature, this patient to our knowledge represents the longest period of disease-free follow-up in those afflicted with CNS Curvularia infection, indicating the importance of proper diagnosis and aggressive surgical debridement for a successful outcome. Ó 2012 Elsevier Ltd. All rights reserved.
1. Introduction Curvularia is a pigmented filamentous fungus ubiquitous in the soil yet rarely a cause of human infection. However, it has been described in patients with sinusitis, keratitis, cutaneous infection, respiratory infection, endocarditis, and brain abscess. Central nervous system (CNS) phaeohyphomycosis has an extremely poor prognosis.1 Here, we report a previously healthy individual with a Curvularia brain abscess. After extensive surgical and antibiotic therapy, she has been disease-free for a follow-up period of more than 3 years. This report demonstrates the importance of an aggressive therapeutic approach in CNS infection caused by Curvularia. 2. Case report A 50-year-old immunocompetent female with a history of chronic sinusitis and previous sinus surgery presented with a sei⇑ Corresponding author. Tel.: +1 713 792 2400; fax: +1 713 794 4950. E-mail address: [email protected] (G. Rao).
zure followed by weakness, slurred speech, and memory loss. The patient reported a 4-year history of headaches and blurred vision in the right eye progressing, in the prior few months, to unbearable headaches, syncopal episodes, and bloody nasal drainage. The patient was noted to have dysarthria, ataxia, numbness of the right ophthalmic division of cranial nerve V, and right eye proptosis. CT scan showed destruction of the anterior cranial fossa floor (Fig. 1A). MRI of the head revealed a 4.6 cm 4.2 cm 3.2 cm destructive mass in the right inferior frontal lobe extending into the ethmoid, right orbit, and cranial base (Fig. 1B), accompanied by subfalcine herniation and posterior mass effect. A biopsy (Fig. 1C) of the mass revealed necrotizing granulomatous inflammation and pigmented fungal organisms identified as Curvularia species. The patient was started on voriconazole, minocycline, and levofloxacin. A stereotactic-guided combined endoscopic-assisted craniofacial resection was performed. After bifrontal craniotomy, the mass was resected along with the inferior frontal lobe. Frontal sinus exenteration was performed. Extensive erosions of the medial orbit, cribiform plate, and roof of the sphenoid were noted.
174
Case Reports / Journal of Clinical Neuroscience 20 (2013) 173–175
Fig. 1. Imaging studies of an immunocompetent 50-year-old female with a Curvularia brain abscess: (A) sagittal reconstruction of a non-contrast CT scan (bone windows) showing destruction of the anterior cranial base (arrow); (B) sagittal post gadolinium T1-weighted MRI showing a large enhancing mass extending from the skull base into the left frontal lobe; (C) transnasal endoscopic view of the right nasal cavity, demonstrating an extensive necrotic soft tissue mass (# = nasal septum; = right middle turbinate; arrowhead = sphenoid sinus); and (D) sagittal MRI (post gadolinium T1-weighted MRI) 3 years after surgery and antibiotic treatment showing no evidence of disease recurrence. (E–H) Photomicrograph showing: (E) the specimen removed at surgery demonstrating meningeal thickening (Men) with lymphocytic infiltration, giant cells, and invasion into the brain (NB = normal brain) (hematoxylin and eosin [H&E], 100); (F) septated hyphae and terminal dilatation (arrow) consistent with Curvularia (H&E, 400); (G) hyphal forms against a background of meninges and brain tissue (Grocott methenamine silver stain [GMS], 100); (H) terminal dilatation (arrow) consistent with Curvularia (GMS, 400).
Endonasal disease along the right medial orbital wall and right sphenoid sinus was resected transnasally. A pericranial graft was utilized to reconstruct the skull base. The patient’s hospital course was uncomplicated and she was discharged on antibiotics. She was followed with serial MRI. After more than 3 years of follow-up, the patient remained disease-free (Fig. 1D). Pathological examination of the specimen revealed extensive meningeal thickening with infiltration including multi-nucleated giant cells and lymphocytes (Fig. 1E). Higher magnification revealed fungal hyphae (Fig. 1F). Grocott methenamine silver (GMS) stain showed extensive fungal involvement (Fig. 1G). Higher magnification image of GMS stain revealed terminal dilatation, an artifact of tissue fixation that is characteristic of Curvularia (Fig. 1H). 3. Discussion Phaeohyphomycosis, an infection caused by pigmented fungi with mycelial morphology,2 is a rare but fatal cause of cerebral infection. In a review of 101 cases of CNS phaeohyphomycosis,1 73% were fatal, although most patients had no underlying risk factor. These organisms respond poorly to standard antifungal therapy, but patients treated with combination antifungals and complete surgical excision have better outcomes.1
To our knowledge only seven patients with CNS phaeohyphomycosis caused by Curvularia species have been reported. Friedman et al.3 described a patient with a pulmonary infiltrate and frontoparietal abscess caused by C. pallescens, which recurred fatally 3years after surgical excision. Rohwedder et al.4 described a patient with a decade-long infection with C. lunata, which caused soft tissue, pulmonary, paravertebral, and cerebral abscesses in which the patient ultimately refused further therapy. Pierce et al.5 described a patient with a lung and multiple cerebral lesions caused by C. lunata treated with prolonged amphotericin B with no relapse up to one year follow-up. Ebright et al.6 reported a patient with invasive sinusitis and bifrontal cerebritis caused by C. clavata treated with aggressive resection with no relapse up to 2 years of follow up. Carter and Boudreaux7 reported a patient with a large basal ganglia lesion caused by C. lunata that was fatal within 1month. Smith et al.8 reported a patient with a pituitary fossa lesion resulting in optic nerve atrophy caused by C. lunata that was treated with stereotactic transphenoidal resection and antifungals without relapse up to 8 months follow-up. Singh et al.9 reported a patient with a cranial base lesion caused by C. geniculata treated with limited transnasal endoscopic debulking. The patient died 4 months later. Because of this patient’s long history of chronic sinusitis and evidence of sinus erosion, the mechanism of infection was probably inhalation of spores, colonization of the nasal sinuses, and local
Case Reports / Journal of Clinical Neuroscience 20 (2013) 175–177
extension of infection into the frontal lobe. Curvularia is a common cause of allergic fungal sinusitis,10 although there is only one report of a patient with associated cerebral Curvularia infection.8 It is unclear why this ubiquitous organism causes invasive cerebral disease in rare instances. Although definitive treatment of cerebral Curvularia infection has not been established, the high mortality and incidence of relapse suggest that an aggressive therapeutic approach is paramount. In our patient, follow-up for more than 3 years confirms no residual disease or relapse. This is the longest period of disease-free follow-up reported for intracranial Curvularia infection, the success of which is attributed to aggressive surgical treatment and long-term intravenous antifungals. References 1. Revankar SG, Sutton DA, Rinaldi MG. Primary central nervous system phaeohyphomycosis: a review of 101 cases. Clin Infect Dis 2004;38:206–16.
175
2. University of Adelaide. Phaeohyphomycosis. Mycology Online.
doi:http://dx.doi.org/10.1016/j.jocn.2012.05.012
Clinical and radiologic spectrum of corpus callosum infarctions: Clues to the etiology Santosh B. Murthy, Mohamad Chmayssani, Shreyansh Shah, Corey E. Goldsmith, Joseph S. Kass ⇑ Department of Neurology, Baylor College of Medicine, 6501 Fannin, NB302, Houston, TX 77030, USA
a r t i c l e
i n f o
Article history: Received 22 May 2012 Accepted 27 May 2012
Keywords: Alien hand syndrome Anterior cerebral artery Corpus callosum Infarction
a b s t r a c t Infarctions of the corpus callosum are rare. The clinical picture varies from an acute onset to slow evolving symptoms, frequently with poor localizing signs; however, the location of the infarct in the callosum often correlates with a specific etiology. We describe three patients with varying degrees of callosal infarction, each corresponding to a particular etiology. Published by Elsevier Ltd.
1. Introduction We report three patients with corpus callosum (CC) infarctions and aim to highlight the subtle radiological features that would aid in identifying the specific etiology.
2. Case report 2.1. Patient 1 A 56-year-old right-handed Hispanic man with hypertension, hyperlipidemia and coronary artery disease presented with new onset chest pain. He underwent three-vessel coronary artery bypass grafting. On post-operative day 2, he was noted to have left-sided hemiparesis. A CT scan of the head showed a hypodensity in the CC. MRI showed diffusion restriction confined exclusively to the body of the CC (Fig. 1A). A magnetic resonance angiogram (MRA), revealed severe (>70%) atherosclerotic narrowing of the bilateral distal petrous and cavernous internal ⇑ Corresponding author. Tel.: +1 713 798 6151; fax: +1 713 798 8350. E-mail address: [email protected] (J.S. Kass).
carotid arteries (ICA). A double lumen was noted at the junction of the petrous and cavernous segments of the left ICA, raising concerns for a dissection as opposed to a calcified plaque (Fig. 1B). Despite the possibility of a dissection, therapeutic anticoagulation was not considered given the recent cardiac surgery. Following extubation, he was noted to have ideomotor apraxia of the non-dominant hand consistent with alien hand syndrome. At the 6-month follow-up, the patient was ambulatory with assistance, but continued to have apraxia affecting the left hand. 2.2. Patient 2 A 46-year-old right-handed Hispanic man with diabetes and hypertension presented with acute onset of decreased volitional speech output and bilateral lower extremity weakness. On neurologic exam, he appeared withdrawn and had complete speech arrest with intact comprehension and normal writing and ability to follow commands. Initial National Institutes of Health Stroke Scale (NIHSS) score was 9 and he received intravenous alteplase (tissue plasminogen activator, tPA). Brain MRI revealed diffusion restriction in the striato-capsular region on the right, genu of the CC, and frontal subcortical white matter bilaterally (Fig. 2A, B). A CT