- Email: [email protected]
Traumatic acute subdural hygroma mimicking acute subdural hematoma
Traumatic acute subdural hygroma mimicking acute subdural hematoma
sedimentation rate, albumin and calcium levels makes the diagnosis of Crohn’s disease unlikely. Likewise, there are no clinicoradiological features of sarcoidosis, and angiotensin converting enzyme levels were normal. Many clinical findings have been reported in association with the MRS. Some are coincidental, but others may be related to MRS in a fundamental manner. Rare associations of MRS that have been described are; hyperhidrosis, hypogeusia, glossodynia, acroparesthesia, hyperacusis, epiphoria and migraine-like headaches.12 Our review of the medical literature could not identify previous description of heart block with MRS. It is rare for complete heart block to occur in a previously healthy 37-year old. In adults, drug toxicity, ischaemic heart disease and degenerative heart diseases cause the majority of heart blocks.14 None of these were present in our patient. Sclerodegenerative disease of the conduction system is unlikely as this usually occurs after the age of 50. We suggest granulomatous infiltration of the conductive tissues as the basis of the heart block in a similar way to cardiac sarcoidosis. Cardiac sarcoidosis commonly presents with complete heart block and typically occurs in a younger age group than with other etiologies.15 Involvement of cardiac muscles, conducting system or atrioventricular node by granulomas has been demonstrated at necropsy in patients dying of complete heart block.16;17 Definitive diagnosis would require positive findings on myocardial biopsy. The procedure has a low sensitivity in cases of cardiac sarcoidosis, approaching 20% in one series of 26 patients18 and the patient declined the procedure. Previously reported ophthalmologic findings in patients with MRS include lag-ophthalmos, exposure keratitis, retrobulbar neuritis, and paralysis of the medial rectus muscle.12 One patient in Greene and Rogers’10 study had diplopia without further characterisation. To our knowledge, limitation of eye abduction was never previously described. MRI scan of the brain and the orbital structures did not provide an explanation. We postulate either abducens nerve palsy or paralysis of the lateral rectus muscle due to periorbital tissue inflammation. The inflammatory cellular infiltration of the nerve bundles with marked interstitial oedema and the lymphocytic cellular infiltration of skeletal muscle fibres with resulting segmental necrosis and myophagia seen in our biopsy specimens could support both possibilities. Wiesenfeld et al.13 introduced the term orofacial granulomatosis to describe, as a group, patients with granulomatous lesions of the mouth and face. The term allows for the possibility of more than one underlying aetiology and gives recognition to the possibility that tuberculosis, leprosy, sarcoidosis and Crohn’s may atypically present in this manner. In 60 patients with oro-facial granulomatosis, 6 (10%) were found to have associated gastrointestinal Crohn’s and 2 were diagnosed to have sarcoidosis based on the presence of lesions suggestive of cutaneous sarcoid and positive Kveim tests.13 It is possible that Crohn’s, sarcoidosis and Melkerrson–Rosenthal syndrome form a spectrum of granulomatous diseases that can have overlapping clinical and histological features and that our patient is an example of such an intermediate variant form.
REFERENCES 1. Mart E. Nonnulla de nervi facialis paralysi. Leipzig 1859. 2. Hubschamann VP. Ueber Recedive und Diplegie bei der sogenannten rheumatischen Facialislaehmung. Neurol Centralbl (Z Neurol Psych) 1894; 13: 815–844. 3. Rossolimo GJ. Rezidivierende Fazialislaehmung bei Migraine. Neurol Centralbl (Z Neurol Psych) 1901; 20: 744–759.
ª 2003 Elsevier Ltd. All rights reserved.
311
4. Melkersson E. Ett fall av recidiverande facial spares: Samband med angioneurotisk Odem. Hygeia (Stockholm) 1928; 90: 737–741. 5. Rosenthal C. Klinisch-erbbiologischer Beitrag zur Konstitutionspathologie. Z Gesamte Neurol Psychiatr 1931; 131: 475–501. 6. Luscher E. Syndrome von Melkersson–Rosenthal. Schweiz Med Wochenschr 1949; 79: 1–3. 7. Meischer G. Ueber essentielle granulomatoese Makrocheilie (cheilitis Granulomatosa). Dermatologica 1945; 91: 57–85. 8. Hornstein OP. Melkersson–Rosenthal syndrome: a neuro-muco-cutaneous disease of complex origin. Curr Probl Dermatol 1973; 5: 117–156. 9. Worsaae N, Christensen KC, Schiodt M, Reibel J. Granulomatous gingival manifestations of Melkersson–Rosenthal syndrome and cheilitis granulomatosa. A clinicopathological study of thirty-three patients with special reference to their oral lesions. Oral Surg Oral Med Oral Pathol 1982; 54: 404–413. 10. Greene RM, Rogers III RS. Melkersson–Rosenthal syndrome: a review of 36 patients. J Am Acad Dermatol 1989; 21: 1263–1270. 11. Zimmer WM, Rogers III RS, Reeve CM, Sheridan PJ. Orofacial manifestations of Melkersson–Rosenthal syndrome. Oral Surg Oral Med Pathol 1992; 74: 610–619. 12. Rogers III RS. Melkersson–Rosenthal syndrome and orofacial granulomatosis. Dermatol Clin 1996; 14: 371–379. 13. Wiesenfeld D, Ferguson MM, Mitchell DN et al. Orofacial granulomatosis – a clinical and pathological analysis. Q J Med 1985; 54: 101–113. 14. Zipes DP. Specific arrhythmias: diagnosis and treatment. In: Braunwald E (ed) Heart Disease: A Textbook of Cardiovascular Medicine. WB Saunders, Philadelphia 1997: 691. 15. Fleming HA. Cardiac Sarcoidosis. In: James DG (ed) Sarcoidosis and Other Granulomatous Disorders. Dekker, New York 1994; 323–334. 16. Rossi L. Sarcoid heart disease. Br Med J 1973; 1: 546–547. 17. Roberts WC, McAllister Jr HA, Ferrans VJ. Sarcoidosis of the heart: a clinicopathologic study of 35 necropsy patients (group 1) and review of 78 previously reported necropsy patients (group 11). Am J Med 1977; 63: 86–108. 18. Uemura A, Morimoto S, Hiramitsu S, Kato Y, Ito T, Hishida H. Histologic diagnostic rate of cardiac sarcoidosis: evaluation of endomyocardial biopsies. Am Heart J 1999; 138: 299–302.
Traumatic acute subdural hygroma mimicking acute subdural hematoma Takao Kamezaki1 MD, Kiyoyuki Yanaka2 MD PHD, Keishi Fujita1 MD, Kazuhiro Nakamura1 MD, Yasushi Nagatomo1 MD, Tadao Nose2 MD PHD 1
Department of Neurosurgery, Ibaraki Seinan Medical Center, Sashima, Ibaraki 306-0433, Japan, 2Department of Neurosurgery, Institute of Clinical Medicine, University of Tsukuba, Tsukuba, Ibaraki 305-8575, Japan
Summary Subdural hygroma is a frequent delayed complication of head trauma. Most hygromas are clinically ‘silent’ and a few cases have shown slow deterioration in the chronic stage. We report a case of subdural hygroma showing unique radiological findings and rapid deterioration. A 74-years-old female presented with a mild headache and consciousness disturbance after head injury. Computed tomography showed a midline shift as a result of two components piling up in the subdural space; the outer components showed low density, the inner components high density. Magnetic resonance imaging demonstrated that these two subdural components were subdural hygroma and subarachnoid hematoma. Simple burr hole irrigation, rather than large craniotomy, was thought to be more appropriate treatment to reduce the mass effect. Simple burr hole irrigation was performed to remove the subdural hygroma and the patient showed an excellent recovery. Careful examination of the radiological findings prevented an unnecessary procedure in this case. A possible mechanism of this phenomenon is discussed. ª 2003 Elsevier Ltd. All rights reserved. Journal of Clinical Neuroscience (2004) 11(3)
312 Kamezaki et al.
Journal of Clinical Neuroscience (2004) 11(3), 311–313 0967-5868/$ - see front matter ª 2003 Elsevier Ltd. All rights reserved. doi:10.1016/j.jocn.2003.10.013
Keywords: subdural hematoma, subdural hygroma, trauma Received 13 March 2003 Accepted 26 October 2003 Correspondence to: Kiyoyuki Yanaka MD PhD, Department of Neurosurgery, Institute of Clinical Medicine, University of Tsukuba, Tsukuba, Ibaraki 305-8575, Japan. Tel.: +81-298-53-7593; Fax: +81-298-53-3214; E-mail: [email protected]
INTRODUCTION Subdural hygroma, an accumulation of cerebrospinal fluid in the subdural space, is a common post-traumatic lesion.1 Typically, the lesion is absent on initial computed tomography (CT) examination and is detectable on repeat CT examination performed 2–14 days later.2 Most subdural hygromas resolve when the brain is well expanded.1 Subdural hygromas commonly occur in patients over 50 or under 5 years of age. Most hygromas are clinically ‘silent’ and a few cases have shown slow deterioration.3;4 Symptoms usually occur in the chronic stage; their occurrence in the acute stage is rare. Surgical treatment is limited to patients who have enlarging subdural hygroma and neurological deterioration.5 In such cases, simple burr hole irrigation is usually performed. Clinical outcome is satisfactory in general, except in the case of patients who have accompanying parenchymal lesions.5 In contrast, patients with acute subdural hematoma have a high mortality and low functional recovery rates. Thus far, early surgical decompression and active intensive care treatment constitute the best approach to these patients.6 Paradoxically, one of the factors contributing to poor outcomes in cases of acute subdural hematoma could be rapid surgical decompression, owing to the severe extrusion of the brain through the craniotomy defect in response to acute brain swelling.7 Thus, it is essential to determine whether simple burr hole procedure or decompressive craniotomy is appropriate in the treatment of patients with subdural mass lesions. Here, we report a case of acute traumatic subdural hygroma showing a similar clinical course to that of an acute subdural hematoma, with unique radiological findings. This patient was successfully treated by simple burr hole procedure within 24 h of injury. The etiology of this rare phenomenon is discussed. CASE REPORT A 74-years-old female presented with a mild headache. Three hours prior to admission, the patient had fallen down while alone in a bathroom and hit her head. The patient had been diagnosed with hepatitis 10 years prior to admission and was otherwise healthy. Physiological examination on admission revealed normal findings. The patient was drowsy, but was easily roused. Glasgow Coma Scale (GCS) score on admission was E3 V4 M6 . The pupils were equal in size, round and reactive to light. No motor abnormalities were noted. Routine laboratory tests revealed normal findings. Computed tomography on admission showed a crescentshaped low-density area in the frontoparietal subdural space, with an underlying high density area. These two components did not form a niveau formation. Slight midline shift was also seen (Fig. 1). Follow-up CT was performed 3 h later and it demonstrated no significant interval changes. Twelve hours later, the patient condition deteriorated and she demonstrated a slight left hemiparesis. At that time, GCS was E2 V2 M5 . On CT, subdural fluid and hematoma, as compared to their prior appearance on CT, did not show a significant change, but Journal of Clinical Neuroscience (2004) 11(3)
Fig. 1 Computed tomography on admission (left), showing a slight midline shift due to subdural fluid and subarachnoid hematoma. These two subdural components do not form a niveau formation. Twelve h later (center), midline shift had worsened. Post-operative computed tomography (right), showing disappearance of the midline shift. Subarachnoid hematoma is still seen.
Fig. 2 Magnetic resonance imaging shows the two components in the subdural space. Outer component shows slight hypointensity on T1-weighted image (left) and high intensity on T2-weighted image (right), suggesting xanthochromic fluid. Inner component shows isointensity on both T1- and T2-weighted images, suggesting a fresh hematoma. Cerebrospinal fluid is also seen beneath the hematoma on the T2-weighted image.
the degree of the midline shift was revealed to be worsening (Fig. 1). Magnetic resonance imaging demonstrated that the subdural space was occupied by two components; subdural hygroma and hematoma. T2-weighted image showed a thin hematoma layer between the CSF and the subdural hygroma, suggesting that the hematoma was in the subarachnoid space. No parenchymal lesion was seen (Fig. 2). Subsequently, simple burr hole irrigation of hygromatous liquid was performed. The subdural fluid collected was not bloody but was xanthochromic. Thin subarachnoid hematoma covering the brain surface was observed through the burr hole. Follow-up CT showed the disappearance of the midline shift though the subarachnoid hematoma was still observed (Fig. 1). The patient demonstrated a prompt recovery and was discharged 2 weeks after surgery without neurological deficit.
DISCUSSION Subdural hygroma is a frequent delayed complication of head trauma and has been reported to represent 5–20% of post-traumatic mass lesions.1 Despite its common occurrence, its pathogenesis and clinical significance are uncertain. An arachnoid tear and flap valve hypothesis is widely accepted as the pathogenic mechanism.1;2;4 The torn arachnoidea mater acts as a ball valve and subsequently traps the cerebrospinal fluid. In addition to this separation of dura-arachnoid interface, a sufficient potential subdural space is a basic requirement for the development of subdural hygroma. In the elderly, brain atrophy leaves potential ª 2003 Elsevier Ltd. All rights reserved.
Normal pressure hydrocephalus
space where fluid may collect.2 Therefore, fluid collection may develop by passive effusion and symptoms may not be evident for quite some time. However, our patient showed relatively acute deterioration. If the tear in the arachnoid is large, the amount of cerebrospinal fluid entering into the subdural space will be large. Asano et al.8 studied 32 patients with subdural hygroma and 6 patients (18.8%) showed symptoms within 24 h of injury. Thus, it is not surprising that some patients show symptoms in the acute stage. In addition, the mass effect in our case was due not only to subdural hygroma but also to concomitant subarachnoid hematoma. These co-factors may have contributed to the exhibition of symptoms in the acute stage in our case. The clinical course of our patient was similar to that of acute subdural hematoma. Subdural lesions causing mass effect are usually treated by large craniotomy, but our patient underwent simple burr hole surgery. MRI demonstrated that the hematoma was covered with the arachnoid membrane and it would have been difficult to remove such subarachnoid hematoma by craniotomy. Simple drainage of the hygroma was sufficient to reduce the mass effect. Subdural hematoma often showed a niveau formation, but that in our case showed a different pattern. Careful examination of the radiological studies enabled us to reach the correct diagnosis and administer appropriate treatment. REFERENCES 1. Lee KS. The pathogenesis and clinical significance of traumatic subdural hygroma. Brain Inj 1998; 12: 595–603. 2. Deltour P, Lemmerling M, Bauters W, Siau B, Kunnen M. Posttraumatic subdural hygroma: CT findings and differential diagnosis. JBR-BTR 1999; 82: 155–156. 3. Borzone M, Capuzzo T, Perria C, Rivano C, Tercero E. Traumatic subdural hygromas: a report of 70 surgically treated cases. J Neurosurg Sci 1983; 27: 161–165. 4. Lee KS, Bae WK, Park YT, Yun IG. The pathogenesis and fate of traumatic subdural hygroma. Br J Neurosurg 1994; 8: 551–558. 5. Ishibashi A, Yokokura Y, Miyagi J. Clinical analysis of nineteen patients with traumatic subdural hygromas. Kurume Med J 1994; 41: 81–85. 6. Yanaka K, Kamezaki T, Yamada T, Takano S, Meguro K, Nose T. Acute subdural hematoma–prediction of outcome with a linear discriminant function. Neurol Med Chir 1993; 33: 552–558. 7. Guilburd JN, Sviri GE. Role of dural fenestrations in acute subdural hematoma. J Neurosurg 2001; 95: 263–267. 8. Asano Y, Hasuo M, Takahashi I, Shimosawa S. Surgical outcome of 32 cases in traumatic subdural hygroma. No To Shinkei 1992; 44: 1127–1131 (in Japanese).
Normal pressure hydrocephalus manifesting as transient prosopagnosia, topographical disorientation, and visual objective agnosia Naoki Otani MD, Hiroshi Nawashiro MD, Shoichiro Ishihara MD, Shinji Fukui MD, Hiroshi Katoh MD, Nobusuke Tsuzuki MD, Akira Ohnuki MD, Takahito Miyazawa MD, Katsuji Shima MD Department of Neurosurgery, National Defense Medical College, Saitama, Japan
Summary Usually, dementia, gait disturbance and urinary incontinence are an integral part of the clinical presentation of normal
ª 2003 Published by Elsevier Ltd.
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pressure hydrocephalus (NPH). However, NPH with transient visual cognitive disorders has not been reported previously. We herein describe an extremely rare case of NPH that presented with transient visual cognitive disorders and long lasting visual memory disturbances that subsided after CSF shunting. A 38years-old man developed transient prosopagnosia, topographical disorientation, color vision disturbance, and visual objective agnosia that progressed over 5 years. Magnetic resonance images showed ventriculomegaly with ischemic lesions in the paraventricular deep white matter. ECD-SPECT showed a reduction of the cerebral blood flow (CBF) in the periventricular deep white matter. Three months after CSF shunting, the radiological findings normalized with dramatic improvement of the extremely rare symptoms. ª 2003 Published by Elsevier Ltd. Journal of Clinical Neuroscience (2004) 11(3), 313–317 0967-5868/$ - see front matter ª 2003 Published by Elsevier Ltd. doi:10.1016/j.jocn.2003.02.014
Keywords: cerebral color vision disturbance, normal pressure hydrocephalus, prosopagnosia, topographical disorientation, visual cognitive agnosia Received 10 December 2002 Accepted 17 February 2003 Correspondence to: Naoki Otani, Department of Neurosurgery, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama 359-8513, Japan. Tel.: +81-42-995-1656; Fax: +81-42-996-5207; E-mail: [email protected]
INTRODUCTION Normal pressure hydrocephalus (NPH) is a well-known clinical entity; however, its etiology varies and its precise pathophysiology is still not fully understood.1 Usually, dementia, gait disturbance, and urinary incontinence are an integral part of the clinical presentation of NPH. However, no case of NPH that manifested as transient visual cognitive disorders has been reported previously. A differential diagnosis of these manifestations may be particularly difficult. Atypical symptoms may be misdiagnosed as transient ischemic attacks,2 epilepsy, migraine aura,3 psychiatric symptoms, or go undiagnosed. Environmental agnosia and prosopagnosia have both been described following focal, right temporo-occipital lesions,4;5 but have not been reported as transient manifestations. We herein describe an extremely rare case of NPH with transient visual cognitive disorders and long lasting visual memory disturbances that subsided after ventriculo-peritoneal shunting.
CASE REPORT A 38-years-old man presented with transient difficulty in finding the way around previously familiar environments with a frequency of about once a month in October 1993. The duration of such episodes ranged from 30 s to several minutes. The frequency and duration of such episodes was gradually reduced. Other episodes such as transient difficulty in recognizing the face of a colleague, the colors and patterns of a paper cover and bedclothes, and in understanding the use of a microwave occurred in October 1997. The patient suffered from these progressive transient neurological deficits and became conscious of recent memory disturbance. There was no previous history of head injury, subarachnoid hemorrhage, or meningitis. A physical examination on admission Journal of Clinical Neuroscience (2004) 11(3)
sedimentation rate, albumin and calcium levels makes the diagnosis of Crohn’s disease unlikely. Likewise, there are no clinicoradiological features of sarcoidosis, and angiotensin converting enzyme levels were normal. Many clinical findings have been reported in association with the MRS. Some are coincidental, but others may be related to MRS in a fundamental manner. Rare associations of MRS that have been described are; hyperhidrosis, hypogeusia, glossodynia, acroparesthesia, hyperacusis, epiphoria and migraine-like headaches.12 Our review of the medical literature could not identify previous description of heart block with MRS. It is rare for complete heart block to occur in a previously healthy 37-year old. In adults, drug toxicity, ischaemic heart disease and degenerative heart diseases cause the majority of heart blocks.14 None of these were present in our patient. Sclerodegenerative disease of the conduction system is unlikely as this usually occurs after the age of 50. We suggest granulomatous infiltration of the conductive tissues as the basis of the heart block in a similar way to cardiac sarcoidosis. Cardiac sarcoidosis commonly presents with complete heart block and typically occurs in a younger age group than with other etiologies.15 Involvement of cardiac muscles, conducting system or atrioventricular node by granulomas has been demonstrated at necropsy in patients dying of complete heart block.16;17 Definitive diagnosis would require positive findings on myocardial biopsy. The procedure has a low sensitivity in cases of cardiac sarcoidosis, approaching 20% in one series of 26 patients18 and the patient declined the procedure. Previously reported ophthalmologic findings in patients with MRS include lag-ophthalmos, exposure keratitis, retrobulbar neuritis, and paralysis of the medial rectus muscle.12 One patient in Greene and Rogers’10 study had diplopia without further characterisation. To our knowledge, limitation of eye abduction was never previously described. MRI scan of the brain and the orbital structures did not provide an explanation. We postulate either abducens nerve palsy or paralysis of the lateral rectus muscle due to periorbital tissue inflammation. The inflammatory cellular infiltration of the nerve bundles with marked interstitial oedema and the lymphocytic cellular infiltration of skeletal muscle fibres with resulting segmental necrosis and myophagia seen in our biopsy specimens could support both possibilities. Wiesenfeld et al.13 introduced the term orofacial granulomatosis to describe, as a group, patients with granulomatous lesions of the mouth and face. The term allows for the possibility of more than one underlying aetiology and gives recognition to the possibility that tuberculosis, leprosy, sarcoidosis and Crohn’s may atypically present in this manner. In 60 patients with oro-facial granulomatosis, 6 (10%) were found to have associated gastrointestinal Crohn’s and 2 were diagnosed to have sarcoidosis based on the presence of lesions suggestive of cutaneous sarcoid and positive Kveim tests.13 It is possible that Crohn’s, sarcoidosis and Melkerrson–Rosenthal syndrome form a spectrum of granulomatous diseases that can have overlapping clinical and histological features and that our patient is an example of such an intermediate variant form.
REFERENCES 1. Mart E. Nonnulla de nervi facialis paralysi. Leipzig 1859. 2. Hubschamann VP. Ueber Recedive und Diplegie bei der sogenannten rheumatischen Facialislaehmung. Neurol Centralbl (Z Neurol Psych) 1894; 13: 815–844. 3. Rossolimo GJ. Rezidivierende Fazialislaehmung bei Migraine. Neurol Centralbl (Z Neurol Psych) 1901; 20: 744–759.
ª 2003 Elsevier Ltd. All rights reserved.
311
4. Melkersson E. Ett fall av recidiverande facial spares: Samband med angioneurotisk Odem. Hygeia (Stockholm) 1928; 90: 737–741. 5. Rosenthal C. Klinisch-erbbiologischer Beitrag zur Konstitutionspathologie. Z Gesamte Neurol Psychiatr 1931; 131: 475–501. 6. Luscher E. Syndrome von Melkersson–Rosenthal. Schweiz Med Wochenschr 1949; 79: 1–3. 7. Meischer G. Ueber essentielle granulomatoese Makrocheilie (cheilitis Granulomatosa). Dermatologica 1945; 91: 57–85. 8. Hornstein OP. Melkersson–Rosenthal syndrome: a neuro-muco-cutaneous disease of complex origin. Curr Probl Dermatol 1973; 5: 117–156. 9. Worsaae N, Christensen KC, Schiodt M, Reibel J. Granulomatous gingival manifestations of Melkersson–Rosenthal syndrome and cheilitis granulomatosa. A clinicopathological study of thirty-three patients with special reference to their oral lesions. Oral Surg Oral Med Oral Pathol 1982; 54: 404–413. 10. Greene RM, Rogers III RS. Melkersson–Rosenthal syndrome: a review of 36 patients. J Am Acad Dermatol 1989; 21: 1263–1270. 11. Zimmer WM, Rogers III RS, Reeve CM, Sheridan PJ. Orofacial manifestations of Melkersson–Rosenthal syndrome. Oral Surg Oral Med Pathol 1992; 74: 610–619. 12. Rogers III RS. Melkersson–Rosenthal syndrome and orofacial granulomatosis. Dermatol Clin 1996; 14: 371–379. 13. Wiesenfeld D, Ferguson MM, Mitchell DN et al. Orofacial granulomatosis – a clinical and pathological analysis. Q J Med 1985; 54: 101–113. 14. Zipes DP. Specific arrhythmias: diagnosis and treatment. In: Braunwald E (ed) Heart Disease: A Textbook of Cardiovascular Medicine. WB Saunders, Philadelphia 1997: 691. 15. Fleming HA. Cardiac Sarcoidosis. In: James DG (ed) Sarcoidosis and Other Granulomatous Disorders. Dekker, New York 1994; 323–334. 16. Rossi L. Sarcoid heart disease. Br Med J 1973; 1: 546–547. 17. Roberts WC, McAllister Jr HA, Ferrans VJ. Sarcoidosis of the heart: a clinicopathologic study of 35 necropsy patients (group 1) and review of 78 previously reported necropsy patients (group 11). Am J Med 1977; 63: 86–108. 18. Uemura A, Morimoto S, Hiramitsu S, Kato Y, Ito T, Hishida H. Histologic diagnostic rate of cardiac sarcoidosis: evaluation of endomyocardial biopsies. Am Heart J 1999; 138: 299–302.
Traumatic acute subdural hygroma mimicking acute subdural hematoma Takao Kamezaki1 MD, Kiyoyuki Yanaka2 MD PHD, Keishi Fujita1 MD, Kazuhiro Nakamura1 MD, Yasushi Nagatomo1 MD, Tadao Nose2 MD PHD 1
Department of Neurosurgery, Ibaraki Seinan Medical Center, Sashima, Ibaraki 306-0433, Japan, 2Department of Neurosurgery, Institute of Clinical Medicine, University of Tsukuba, Tsukuba, Ibaraki 305-8575, Japan
Summary Subdural hygroma is a frequent delayed complication of head trauma. Most hygromas are clinically ‘silent’ and a few cases have shown slow deterioration in the chronic stage. We report a case of subdural hygroma showing unique radiological findings and rapid deterioration. A 74-years-old female presented with a mild headache and consciousness disturbance after head injury. Computed tomography showed a midline shift as a result of two components piling up in the subdural space; the outer components showed low density, the inner components high density. Magnetic resonance imaging demonstrated that these two subdural components were subdural hygroma and subarachnoid hematoma. Simple burr hole irrigation, rather than large craniotomy, was thought to be more appropriate treatment to reduce the mass effect. Simple burr hole irrigation was performed to remove the subdural hygroma and the patient showed an excellent recovery. Careful examination of the radiological findings prevented an unnecessary procedure in this case. A possible mechanism of this phenomenon is discussed. ª 2003 Elsevier Ltd. All rights reserved. Journal of Clinical Neuroscience (2004) 11(3)
312 Kamezaki et al.
Journal of Clinical Neuroscience (2004) 11(3), 311–313 0967-5868/$ - see front matter ª 2003 Elsevier Ltd. All rights reserved. doi:10.1016/j.jocn.2003.10.013
Keywords: subdural hematoma, subdural hygroma, trauma Received 13 March 2003 Accepted 26 October 2003 Correspondence to: Kiyoyuki Yanaka MD PhD, Department of Neurosurgery, Institute of Clinical Medicine, University of Tsukuba, Tsukuba, Ibaraki 305-8575, Japan. Tel.: +81-298-53-7593; Fax: +81-298-53-3214; E-mail: [email protected]
INTRODUCTION Subdural hygroma, an accumulation of cerebrospinal fluid in the subdural space, is a common post-traumatic lesion.1 Typically, the lesion is absent on initial computed tomography (CT) examination and is detectable on repeat CT examination performed 2–14 days later.2 Most subdural hygromas resolve when the brain is well expanded.1 Subdural hygromas commonly occur in patients over 50 or under 5 years of age. Most hygromas are clinically ‘silent’ and a few cases have shown slow deterioration.3;4 Symptoms usually occur in the chronic stage; their occurrence in the acute stage is rare. Surgical treatment is limited to patients who have enlarging subdural hygroma and neurological deterioration.5 In such cases, simple burr hole irrigation is usually performed. Clinical outcome is satisfactory in general, except in the case of patients who have accompanying parenchymal lesions.5 In contrast, patients with acute subdural hematoma have a high mortality and low functional recovery rates. Thus far, early surgical decompression and active intensive care treatment constitute the best approach to these patients.6 Paradoxically, one of the factors contributing to poor outcomes in cases of acute subdural hematoma could be rapid surgical decompression, owing to the severe extrusion of the brain through the craniotomy defect in response to acute brain swelling.7 Thus, it is essential to determine whether simple burr hole procedure or decompressive craniotomy is appropriate in the treatment of patients with subdural mass lesions. Here, we report a case of acute traumatic subdural hygroma showing a similar clinical course to that of an acute subdural hematoma, with unique radiological findings. This patient was successfully treated by simple burr hole procedure within 24 h of injury. The etiology of this rare phenomenon is discussed. CASE REPORT A 74-years-old female presented with a mild headache. Three hours prior to admission, the patient had fallen down while alone in a bathroom and hit her head. The patient had been diagnosed with hepatitis 10 years prior to admission and was otherwise healthy. Physiological examination on admission revealed normal findings. The patient was drowsy, but was easily roused. Glasgow Coma Scale (GCS) score on admission was E3 V4 M6 . The pupils were equal in size, round and reactive to light. No motor abnormalities were noted. Routine laboratory tests revealed normal findings. Computed tomography on admission showed a crescentshaped low-density area in the frontoparietal subdural space, with an underlying high density area. These two components did not form a niveau formation. Slight midline shift was also seen (Fig. 1). Follow-up CT was performed 3 h later and it demonstrated no significant interval changes. Twelve hours later, the patient condition deteriorated and she demonstrated a slight left hemiparesis. At that time, GCS was E2 V2 M5 . On CT, subdural fluid and hematoma, as compared to their prior appearance on CT, did not show a significant change, but Journal of Clinical Neuroscience (2004) 11(3)
Fig. 1 Computed tomography on admission (left), showing a slight midline shift due to subdural fluid and subarachnoid hematoma. These two subdural components do not form a niveau formation. Twelve h later (center), midline shift had worsened. Post-operative computed tomography (right), showing disappearance of the midline shift. Subarachnoid hematoma is still seen.
Fig. 2 Magnetic resonance imaging shows the two components in the subdural space. Outer component shows slight hypointensity on T1-weighted image (left) and high intensity on T2-weighted image (right), suggesting xanthochromic fluid. Inner component shows isointensity on both T1- and T2-weighted images, suggesting a fresh hematoma. Cerebrospinal fluid is also seen beneath the hematoma on the T2-weighted image.
the degree of the midline shift was revealed to be worsening (Fig. 1). Magnetic resonance imaging demonstrated that the subdural space was occupied by two components; subdural hygroma and hematoma. T2-weighted image showed a thin hematoma layer between the CSF and the subdural hygroma, suggesting that the hematoma was in the subarachnoid space. No parenchymal lesion was seen (Fig. 2). Subsequently, simple burr hole irrigation of hygromatous liquid was performed. The subdural fluid collected was not bloody but was xanthochromic. Thin subarachnoid hematoma covering the brain surface was observed through the burr hole. Follow-up CT showed the disappearance of the midline shift though the subarachnoid hematoma was still observed (Fig. 1). The patient demonstrated a prompt recovery and was discharged 2 weeks after surgery without neurological deficit.
DISCUSSION Subdural hygroma is a frequent delayed complication of head trauma and has been reported to represent 5–20% of post-traumatic mass lesions.1 Despite its common occurrence, its pathogenesis and clinical significance are uncertain. An arachnoid tear and flap valve hypothesis is widely accepted as the pathogenic mechanism.1;2;4 The torn arachnoidea mater acts as a ball valve and subsequently traps the cerebrospinal fluid. In addition to this separation of dura-arachnoid interface, a sufficient potential subdural space is a basic requirement for the development of subdural hygroma. In the elderly, brain atrophy leaves potential ª 2003 Elsevier Ltd. All rights reserved.
Normal pressure hydrocephalus
space where fluid may collect.2 Therefore, fluid collection may develop by passive effusion and symptoms may not be evident for quite some time. However, our patient showed relatively acute deterioration. If the tear in the arachnoid is large, the amount of cerebrospinal fluid entering into the subdural space will be large. Asano et al.8 studied 32 patients with subdural hygroma and 6 patients (18.8%) showed symptoms within 24 h of injury. Thus, it is not surprising that some patients show symptoms in the acute stage. In addition, the mass effect in our case was due not only to subdural hygroma but also to concomitant subarachnoid hematoma. These co-factors may have contributed to the exhibition of symptoms in the acute stage in our case. The clinical course of our patient was similar to that of acute subdural hematoma. Subdural lesions causing mass effect are usually treated by large craniotomy, but our patient underwent simple burr hole surgery. MRI demonstrated that the hematoma was covered with the arachnoid membrane and it would have been difficult to remove such subarachnoid hematoma by craniotomy. Simple drainage of the hygroma was sufficient to reduce the mass effect. Subdural hematoma often showed a niveau formation, but that in our case showed a different pattern. Careful examination of the radiological studies enabled us to reach the correct diagnosis and administer appropriate treatment. REFERENCES 1. Lee KS. The pathogenesis and clinical significance of traumatic subdural hygroma. Brain Inj 1998; 12: 595–603. 2. Deltour P, Lemmerling M, Bauters W, Siau B, Kunnen M. Posttraumatic subdural hygroma: CT findings and differential diagnosis. JBR-BTR 1999; 82: 155–156. 3. Borzone M, Capuzzo T, Perria C, Rivano C, Tercero E. Traumatic subdural hygromas: a report of 70 surgically treated cases. J Neurosurg Sci 1983; 27: 161–165. 4. Lee KS, Bae WK, Park YT, Yun IG. The pathogenesis and fate of traumatic subdural hygroma. Br J Neurosurg 1994; 8: 551–558. 5. Ishibashi A, Yokokura Y, Miyagi J. Clinical analysis of nineteen patients with traumatic subdural hygromas. Kurume Med J 1994; 41: 81–85. 6. Yanaka K, Kamezaki T, Yamada T, Takano S, Meguro K, Nose T. Acute subdural hematoma–prediction of outcome with a linear discriminant function. Neurol Med Chir 1993; 33: 552–558. 7. Guilburd JN, Sviri GE. Role of dural fenestrations in acute subdural hematoma. J Neurosurg 2001; 95: 263–267. 8. Asano Y, Hasuo M, Takahashi I, Shimosawa S. Surgical outcome of 32 cases in traumatic subdural hygroma. No To Shinkei 1992; 44: 1127–1131 (in Japanese).
Normal pressure hydrocephalus manifesting as transient prosopagnosia, topographical disorientation, and visual objective agnosia Naoki Otani MD, Hiroshi Nawashiro MD, Shoichiro Ishihara MD, Shinji Fukui MD, Hiroshi Katoh MD, Nobusuke Tsuzuki MD, Akira Ohnuki MD, Takahito Miyazawa MD, Katsuji Shima MD Department of Neurosurgery, National Defense Medical College, Saitama, Japan
Summary Usually, dementia, gait disturbance and urinary incontinence are an integral part of the clinical presentation of normal
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pressure hydrocephalus (NPH). However, NPH with transient visual cognitive disorders has not been reported previously. We herein describe an extremely rare case of NPH that presented with transient visual cognitive disorders and long lasting visual memory disturbances that subsided after CSF shunting. A 38years-old man developed transient prosopagnosia, topographical disorientation, color vision disturbance, and visual objective agnosia that progressed over 5 years. Magnetic resonance images showed ventriculomegaly with ischemic lesions in the paraventricular deep white matter. ECD-SPECT showed a reduction of the cerebral blood flow (CBF) in the periventricular deep white matter. Three months after CSF shunting, the radiological findings normalized with dramatic improvement of the extremely rare symptoms. ª 2003 Published by Elsevier Ltd. Journal of Clinical Neuroscience (2004) 11(3), 313–317 0967-5868/$ - see front matter ª 2003 Published by Elsevier Ltd. doi:10.1016/j.jocn.2003.02.014
Keywords: cerebral color vision disturbance, normal pressure hydrocephalus, prosopagnosia, topographical disorientation, visual cognitive agnosia Received 10 December 2002 Accepted 17 February 2003 Correspondence to: Naoki Otani, Department of Neurosurgery, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama 359-8513, Japan. Tel.: +81-42-995-1656; Fax: +81-42-996-5207; E-mail: [email protected]
INTRODUCTION Normal pressure hydrocephalus (NPH) is a well-known clinical entity; however, its etiology varies and its precise pathophysiology is still not fully understood.1 Usually, dementia, gait disturbance, and urinary incontinence are an integral part of the clinical presentation of NPH. However, no case of NPH that manifested as transient visual cognitive disorders has been reported previously. A differential diagnosis of these manifestations may be particularly difficult. Atypical symptoms may be misdiagnosed as transient ischemic attacks,2 epilepsy, migraine aura,3 psychiatric symptoms, or go undiagnosed. Environmental agnosia and prosopagnosia have both been described following focal, right temporo-occipital lesions,4;5 but have not been reported as transient manifestations. We herein describe an extremely rare case of NPH with transient visual cognitive disorders and long lasting visual memory disturbances that subsided after ventriculo-peritoneal shunting.
CASE REPORT A 38-years-old man presented with transient difficulty in finding the way around previously familiar environments with a frequency of about once a month in October 1993. The duration of such episodes ranged from 30 s to several minutes. The frequency and duration of such episodes was gradually reduced. Other episodes such as transient difficulty in recognizing the face of a colleague, the colors and patterns of a paper cover and bedclothes, and in understanding the use of a microwave occurred in October 1997. The patient suffered from these progressive transient neurological deficits and became conscious of recent memory disturbance. There was no previous history of head injury, subarachnoid hemorrhage, or meningitis. A physical examination on admission Journal of Clinical Neuroscience (2004) 11(3)