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Case Report
Multiple simultaneous intracerebral hemorrhages following
accidental massive lumbar cerebrospinal fluid drainage:
Case report and literature review
Jos L. Ruiz-Sandoval, Ariel Campos, Samuel Romero-Vargas, Mara I. Jimnez-Rodrguez,
Erwin Chiquete
Department of Neurology and Neurosurgery, Hospital Civil de Guadalajara Fray Antonio Alcalde and the Department of
Neurosciences; Centro Universitario de Ciencias de la Salud. Guadalajara, Jalisco; Mxico
Multiple simultaneous intracerebral hemorrhages (ICH) are
uncommon. We report the case of an 80-year-old woman
with previous diagnosis of normal pressure hydrocephalus
and who was brought to our hospital with altered mental
status and uri nary i nconti nence. Medi cal hi story of
hypertension, hematological disorders or severe head
trauma was absent. Platelet count and coagulation profile
were unremarkable. An initial head computed tomography
(CT) showed sulcal enlargement and ventricular dilatation,
but no evidence of ICH. A tap test indicated as a guide to
case selection for shunt surgery accidentally resulted in
cerebrospinal fluid (CSF) overdrainage. The patient
presented sudden neurol ogi cal deteri orati on, wi th
sluggishly responsive pupils and generalized tonic-clonic
seizures. A new head CT demonstrated multiple supra and
infratentorial ICH. The patient became comatose and had
a fatal course. Hence, CSF overdrainage may either cause
or precipitate multiple simultaneous ICHs, affecting both
the infratentorial and supratentorial regions.
Key words: Cerebrospinal fluid, intracranial hemorrhage,
intracranial hypotension, lumbar drainage, neurological
examination
overdrainage resulted in multifocal ICH. To the best of our
knowledge, this is the first report on massive CSF drainage as a
cause of multiple simultaneous ICHs.
Case Report
An 80-year-old woman was brought to our hospital with altered
mental status and urinary incontinence, as her main complaints.
The history revealed that in the previous two months she suffered
from cognitive impairment and gait disturbance, which motivated
her caregivers to seek medical attention in another hospital. After
clinical evaluation and a head computed tomography (CT) scan,
she was given a diagnosis of normal pressure hydrocephalus. She
was not on anticoagulation or antiplatelet therapy. Medical history
of hypertension, hematological disorders or severe head trauma
was absent. The neurological examination at presentation to our
hospital revealed a conscious woman with spatial disorientation
and bilateral hyperreflexia. Focal neurological signs were absent.
Laboratory findings were normal, including platelet count (152 x
10
9
/liter) and coagulation profile (PT: 90% of control, APTT:
27 seconds, fibrinogen: 225 mg/dl). Blood pressure was below
Introduction
Lumbar cerebrospinal fluid (CSF) drainage has several
diagnostic and therapeutic indications, with well documented
hazardous consequences including overdrainage, acute
pneumocephalus, brain collapse and neurological deterioration.
[1-3]
Intracerebral hemorrhage (ICH) has been reported after lumbar
puncture and lumboperitoneal shunts, sometimes related to other
conditions.
[4-6]
We report the case of a woman in whom CSF
130/90 mmHg during her hospital stay. A head CT scan
performed in our center showed ventriculomegaly, sulcal
enlargement and diffuse white matter disease, with chronic
bilateral subcortical infarctions [Figure 1]. No evidence of ICH
was found; nevertheless, a laminar collection of blood in the
posterior interhemispheric fissure was observed, suggestive of
being secondary to previous head trauma for which we had no
knowledge on history-taking. In spite of this finding, a tap test
was indicated as a guide to case selection for shunt surgery, since
no mass effect was observed. The procedure was performed by a
physician in training without supervision. Cerebrospinal fluid was
clear, with opening pressure of 150 mmH
2
O. The catheter was
not withdrawn on time and CSF continued to flow for almost 30
Jos L. Ruiz-Sandoval
Servicio de Neurologa y Neurociruga, Hospital Civil de Guadalajara , Fray Antonio Alcalde Hospital 278. Guadalajara, Jalisco; Mexico 44280.
E-mail: jorusan@mexis.com
Neurology India | December 2006 | Vol 54 | Issue 4 421
422 CMYK
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Ruiz-Sandoval JL, et al.: Multiple simultaneous ICHs after CFS overdrainage
Discussion
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min, until the fluid initiated to drain bloody, with a final CSF
collection of 250 mL, as measured in a graduated flask. After the
procedure the patient presented sudden neurological deterioration,
with pupils sluggishly reacting to light and generalized tonic-clonic
seizures. A new head CT was practiced 18h after CSF
overdrainage, showing multiple infra and supratentorial ICHs
with irruption into the ventricular system [Figure 2]. The patient
became comatose, requiring ventilatory assistance. Replacement
of CSF volume could not be practiced. Two days later the patient
developed pneumonia, which resulted in sepsis and death in one
Figure 1: Head CT at presentation, before CSF overdrainage. Severe
white matter lesions with chronic bilateral subcortical infarctions
(i.e., vascular leukoencephalopathy), as well as sulcal enlargement
(i.e., cortical atrophy) and ventricular dilatation are evident, but
without evidence of ICH. Collections of blood over the left parietal
convexity and posterior interhemispheric fissure are observed
is a strong preponderance for the supratentorial space, especially
affecting the basal ganglia (thus denouncing the hypertensive
nature seen in most cases).
[8]
However, most of the knowledge
regarding multiple simultaneous ICHs is derived from case
reports, which are possibly the type of communications subject to
the strongest reporting bias. Therefore, the clinical picture,
outcome and even the putative causes may vary more than is
reflected in case reports. Since most of the causative factors
previously attributed to multiple simultaneous ICHs were excluded
in the case presented here and given that neurological deterioration
as well as the hemorrhagic findings in the second head CT began
immediately after CSF overdrainage, it seems reasonable to think
that this procedure was the cause or at least, a precipitating factor
of multifocal ICH. To our knowledge, this patient had a cause of
multiple simultaneous ICHs not previously reported [Table 1].
In the present case, the putative pathophysiological mechanism
that led to multiple simultaneous ICHs points to a continuous
and massive lumbar CSF evacuation resulting in a reduction of
CSF volume with the associated lowering in intraspinal and
intracranial pressure, which eventually increased the transmural
pressure gradient of the vessels, leading to a secondary wall stress
rupture.
[1]
Advanced age and the presence of diffuse white matter
disease could be the other important contributing factors.
[9]
The
widespread and prolonged degeneration of the intracerebral
arterioles in older people may also predispose to the development
of multiple ICHs. Unfortunately, amyloid angiopathy or other
age-related cerebrovascular conditions were not completely
Multiple simultaneous ICHs is defined as the presence of two or
more intracerebral hemorrhages affecting different arterial
territories, without continuity between them and with identical
CT density profiles.
[7,8]
This is a rare presentation of the
hemorrhagic cerebrovascular disease, accounting for 0.6 to 2.8%
of the cases of nontraumatic, nonaneurysmal ICH.
[7,8]
The main
causative factors are hypertension, cerebral amyloid angiopathy
and forms of vasculitis, among other conditions [Table 1]. There
week more.
Figure 2: Head CT after CSF overdrainage. (A) A petechial hemorrhage
in pons (arrow). (B) Bilateral ganglionic hemorrhages (arrows) plus
multiple petechial hemorrhages in the right temporal lobe (arrow
head). (C) Ganglionic hemorrhage (arrow) with petechial
hemorrhages in right parietal and occipital lobes (arrow heads).
Ventricular irruption is also evident. (D) The extension of the
ganglionic hemorrhage with its ventricular irruption (arrows) and
petechial hemorrhages in occipital lobe (arrow head).
excluded in our patient because no cerebral biopsy was performed.
Moreover, we were not able to obtain a necropsy. Since amyloid
angiopathy is very common in older people and is also an important
cause of multiple simultaneous ICHs [Table 1], our patient might
have had an underlying susceptibility (e.g., amyloid angiopathy)
of presenting ICH, which in turn was precipitated by CSF
overdrainage. Nevertheless, the association of CSF overdrainage
with ICH in this patient seems clear, either as an independent
causative or precipitating factor.
Indeed, the laminar collection of blood over the left parietal
convexity and the posterior interhemispheric space seen in the
head CT performed at presentation to our hospital [Figure 1]
need comments. We were not told about the antecedent of head
trauma that might explain this abnormality; however, considering
the gait instability that the patient was presenting, falls that might
Neurology India | December 2006 | Vol 54 | Issue 4 422
CMYK423
Ruiz-Sandoval JL, et al.: Multiple simultaneous ICHs after CFS overdrainage
Table 1: Case reports and case series describing nontraumatic, nonaneurysmal multiple simultaneous Intracerebral
hemorrhages and the associated causal factors
Reference Year of publication Number of cases Putative causal factors
Pant SS and Dreyfus PM 1967 1 Amyloid angiopathy
McCormick WF and Rosenfield DB 1973 16 Leukemia, coagulopathy, vasculitis, neoplasms
Brismar J 1980 1 Cerebral vein thrombosis
Tucker WS, et al. 1980 2 Amyloid angiopathy
Beal MF, et al. 1982 1 Cerebral vein thrombosis
Tyler KL, et al. 1982 1 Amyloid angiopathy
Hickey WF, et al. 1983 2 Idiopathic
Tanikake T, et al. 1983 2 Hypertension
Assad F and Lins E 1984 1 Mycotic aneurysm
Gilles C, et al. 1984 11 Amyloid angiopathy
Patel DV, et al. 1984
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2 Amyloid angiopathy
Kobayashi Y, et al. 1987 1 Amyloid angiopathy
Nakamura T, et al. 1988 1 Hypertension
Wakui K, et al. 1988 1 Amyloid angiopathy associated to head injury
Mori H, et al. 1989
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1 Evacuation of chronic subdural hygroma
Tanno H, et al. 1989 5 Hypertension
Green RM, et al. 1990 1 Cocaine abuse
Kase CS, et al. 1990 2 tPA administration
Hasegawa Y, et al. 1991 1 Vasculitis
Nagano N, et al. 1991 2 Anticoagulant therapy
Uno M, et al. 1991 9 Hypertension
Verstichel P, et al. 1991 1 Hypertension
Yanagawa Y, et al. 1994 1 Amyloid angiopathy
Komiyama M, et al. 1995 1 Hypertension
Ozawa T, et al. 1995 1 Vasculitis
Seijo M, et al. 1996 7
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Hypertension, coagulopathy
Dromerick AW, et al. 1997 1 tPA administration
Liou HH, et al. 1997 1 Churg-Strauss syndrome
Nakamura K, et al. 1997 1 Amyloid angiopathyassociated to migraine
Nighoghossian N, et al. 1998 1 Antimigrainous drug abuse
Daloze A, et al. 1999 1 Hypertension associated to renal cell carcinoma
Kimura T, et al. 2000 1 Vasculitis
Kohshi K, et al. 2000 2 Hypertension
Maurio J, et al. 2001 4 Hypertension
Chen CY, et al. 2003 1 Hydrops fetalis
Oide T, et al. 2003
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6 Amyloid angiopathy
Shiomi N, et al. 2004 11 Hypertension
Okuno S and Sakaki T 2005 1 Systemic lupus erythematosus
Yen CP, et al. 2005 10 Hypertension
Ruiz-Sandoval, et al. 2006 1 CSF overdrainage
CSF indicates cerebrospinal fluid; tPA, tissue plasminogen activator.
An up-to-date MEDLINE search (in February 2006) was performed using the terms multiple intracerebral hemorrhage (haemorrhage) (s), multiple simultaneous
intracerebral hemorrhage (haemorrhage) (s), multiple intracranial hemorrhage (haemorrhage) (s) and multiple simultaneous intracranial hemorrhage (haemorrhage)
(s). Only reports available in English or Spanish describing the number of patients and causative factors were referenced; however, information of abstracts
written in other languages were also included in table. The following reports on cases with multiple ICHs were excluded: non-simultaneous, traumatic, aneurysmal
(except mycotic) and arteriovenous malformation ICH.
have caused mild head trauma cannot be discarded. Nevertheless,
even though delayed traumatic ICH exists,
[10,11]
it is mainly
associated with severe head trauma and would hardly cause more
than two ICHs affecting both the infratentorial and supratentorial
regions.
When a tap test is indicated, intermittent lumbar or continuous
CSF drainage at controlled rate are safe strategies in avoiding
overdrainage,
[1,2]
especially because the lower threshold of CSF
volume compatible with life in humans is rather unknown.
[2]
In
our patient, an advanced age, sulcal enlargement and ventricular
dilatation allowing a large CSF volume might have permitted
such drainage of the fluid (250 mL in 30 min). Any time lumbar
CSF drainage is indicated as diagnostic procedure, it is necessary
to be warned about an excessive rate of CSF drainage, which
should not exceed 20-25 mL/h.
[12]
When used as a guide to case
selection for a shunting procedure in normal pressure
hydrocephalus
[2]
or as treatment of CSF fistula,
[12]
lumbar CSF
drainage of 40 to 50 mL per session is considered safe and
effective.
[1,2]
Another concern with respect to the case discussed here is the
medical error that led to this catastrophe. This complication has
the possibility to be repeated, especially in teaching hospitals in
which physicians in training perform without expert supervision.
Appropriate measures were taken in our center to avoid another
accident like this. Excessive work must not be an exception of a
tight supervision to junior doctors.
In conclusion, CSF overdrainage can either cause or precipitate
multiple simultaneous ICHs, affecting both the infratentorial and
Neurology India | December 2006 | Vol 54 | Issue 4 423
424 CMYK
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Ruiz-Sandoval JL, et al.: Multiple simultaneous ICHs after CFS overdrainage
supratentorial regions.
References
1. Bloch J, Regli L. Brain stem and cerebellar dysfunction after lumbar spinal fluid
drainage: Case report. J Neurol Neurosurg Psychiatr 2003;74:992-4.
2. Fishman RA. Cerebrospinal fluid in diseases of the nervous system. 2
nd
ed. WB
Saunders: Philadelphia; 1992.
3. Snow RB, Kuhel W, Martin SB. Prolonged lumbar spinal drainage after the resection
of tumors of the skull base: A cautionary note. Neurosurgery 1991;28:880-3.
4. Adler MD, Comi AE, Walker AR. Acute hemorrhagic complication of diagnostic
lumbar puncture. Pediatr Emerg Care 2001;17:184-8.
5. Stubgen JP. Intraventricular blood after traumatic lumbar puncture: A report
Neurol 2001;58:629-32.
8. Yen CP, Lin CL, Kwan AL, Lieu AS, Hwang SL, Lin CN, et al. Simultaneous
multiple hypertensive intracerebral haemorrhages. Acta Neurochir (Wien)
2005;147:393-9.
9. Smith EE, Gurol ME, Eng JA, Engel CR, Nguyen TN, Rosand J, et al. White matter
lesions, cognition and recurrent hemorrhage in lobar intracerebral hemorrhage.
Neurology 2004;63:1606-12.
10. Cooper PR. Delayed traumatic intracerebral hemorrhage. Neurosurg Clin N Am
1992;3:659-65.
11. Erol FS, Kaplan M, Topsakal C, Ozveren MF, Tiftikci MT. Coexistence of rapidly
resolving acute subdural hematoma and delayed traumatic intracerebral
hemorrhage. Pediatr Neurosurg 2004;40:238-40.
12. Shapiro SA, Scully T. Closed continuous drainage of cerebrospinal fluid via a lumbar
subarachnoid catheter for treatment or prevention of cranial/spinal cerebrospinal
fluid fistula. Neurosurgery 1992;30:241-5.
6.
7.
of two cases. Childs Nerv Syst 1995;11:492-3.
Suri A, Pandey P, Mehta VS. Subarachnoid hemorrhage and intracereebral
hematoma following lumboperitoneal shunt for pseudotumor cerebri: a rare
complication. Neurol India 2002;50:508-10.
Accepted on 29-05-2006
Maurino J, Saposnik G, Lepera S, Rey RC, Sica RE. Multiple simultaneous
Source of Support: Nil, Conflict of Interest: None declared.
intracerebral hemorrhages: Clinical features and outcome. Arch
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