Multiple simultaneous ICH

1. Case Report
Multiple simultaneous intracerebral hemorrhages following
accidental massive lumbar cerebrospinal fluid drainage:
Case report and literature review
José L. Ruiz-Sandoval, Ariel Campos, Samuel Romero-Vargas, María I. Jiménez-Rodríguez,
m
ro
Erwin Chiquete
f
Department of Neurology and Neurosurgery, Hospital Civil de Guadalajara “Fray Antonio Alcalde” and the Department of
d ns
Neurosciences; Centro Universitario de Ciencias de la Salud. Guadalajara, Jalisco; México
a o
overdrainage n
lo intimultifocal ICH. To the best of our
resulted
a
knowledge,w is the first report on massive CSF drainage as a
Multiple simultaneous intracerebral hemorrhages (ICH) are
uncommon. We report the case of an 80-year-old woman
o this blic ICHs.
d u
cause of multiple simultaneous
with previous diagnosis of normal pressure hydrocephalus
e
re w P m).Case Report
and who was brought to our hospital with altered mental
status and urinary incontinence. Medical history of
r f Ano owoman was brought to our hospital with altered
hypertension, hematological disorders or severe head
fo mental statusc urinary incontinence, as her main complaints.
trauma was absent. Platelet count and coagulation profile
n 80-year-old
k w. and
were unremarkable. An initial head computed tomography
ble edThe history revealed that inand gait disturbance, which motivated
(CT) showed sulcal enlargement and ventricular dilatation,
o the previous two months she suffered
n
ila y M herkcaregivers to seek medical attention intomography (CT) After
but no evidence of ICH. A tap test indicated as a guide to
from cognitive impairment
case selection for shunt surgery accidentally resulted in
v a b eclinical evaluation and a head computed another hospital. scan,
d
cerebrospinal fluid (CSF) overdrainage. The patient
a
presented sudden neurological deterioration, with
s ted w.m was not on anticoagulation or antiplatelet therapy. Medicaltrauma
she was given a diagnosis of normal pressure hydrocephalus. She
i
sluggishly responsive pupils and generalized tonic-clonic history
s
seizures. A new head CT demonstrated multiple supra and
DF ho (ww
of hypertension, hematological disorders or severe head
infratentorial ICH. The patient became comatose and had
was absent. The neurological examination at presentation to our
a fatal course. Hence, CSF overdrainage may either cause
P te hospital revealed a conscious woman with spatial disorientation
or precipitate multiple simultaneous ICHs, affecting both
is si
the infratentorial and supratentorial regions.
h a
and bilateral hyperreflexia. Focal neurological signs were absent.
Laboratory findings were normal, including platelet count (152 x
T
Key words: Cerebrospinal fluid, intracranial hemorrhage, 9
10 /liter) and coagulation profile (PT: 90% of control, APTT:
intracranial hypotension, lumbar drainage, neurological
27 seconds, fibrinogen: 225 mg/dl). Blood pressure was below
examination 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
Introduction was found; nevertheless, a laminar collection of blood in the
posterior interhemispheric fissure was observed, suggestive of
Lumbar cerebrospinal fluid (CSF) drainage has several being secondary to previous head trauma for which we had no
diagnostic and therapeutic indications, with well documented knowledge on history-taking. In spite of this finding, a tap test
hazardous consequences including overdrainage, acute was indicated as a guide to case selection for shunt surgery, since
pneumocephalus, brain collapse and neurological deterioration.[1-3] no mass effect was observed. The procedure was performed by a
Intracerebral hemorrhage (ICH) has been reported after lumbar physician in training without supervision. Cerebrospinal fluid was
puncture and lumboperitoneal shunts, sometimes related to other clear, with opening pressure of 150 mmH2O. The catheter was
conditions.[4-6] We report the case of a woman in whom CSF not withdrawn on time and CSF continued to flow for almost 30
José L. Ruiz-Sandoval
Servicio de Neurología y Neurocirugía, 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
CMYK421

2. Ruiz-Sandoval JL, et al. : Multiple simultaneous ICHs after CFS overdrainage
Discussion
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
is a strong preponderance for the supratentorial space, especially
om
affecting the basal ganglia (thus denouncing the hypertensive
nature seen in most cases).[8] However, most of the knowledge
fr
regarding multiple simultaneous ICHs is derived from case
ad ons
reports, which are possibly the type of communications subject to
Figure 1: Head CT at presentation, before CSF overdrainage. Severe
o
the strongest reporting bias. Therefore, the clinical picture,
nl ati
white matter lesions with chronic bilateral subcortical infarctions
(i.e., vascular leukoencephalopathy), as well as sulcal enlargement outcome and even the putative causes may vary more than is
(i.e., cortical atrophy) and ventricular dilatation are evident, but
ow blic
without evidence of ICH. Collections of blood over the left parietal
reflected in case reports. Since most of the causative factors
convexity and posterior interhemispheric fissure are observed previously attributed to multiple simultaneous ICHs were excluded
d u
in the case presented here and given that neurological deterioration
min, until the fluid initiated to drain bloody, with a final CSF
e
re w P m).
as well as the hemorrhagic findings in the second head CT began
collection of 250 mL, as measured in a graduated flask. After the
procedure the patient presented sudden neurological deterioration, f
immediately after CSF overdrainage, it seems reasonable to think
with pupils sluggishly reacting to light and generalized tonic-clonic r
fo kno .co
that this procedure was the cause or at least, a precipitating factor
of multifocal ICH. To our knowledge, this patient had a cause of
overdrainage, showing multiple infra and supratentoriale
seizures. A new head CT was practiced 18h after CSF
bl ICHs d now
multiple simultaneous ICHs not previously reported [Table 1].
with irruption into the ventricular system [Figure 2]. The patient e
In the present case, the putative pathophysiological mechanism
la M dk
became comatose, requiring ventilatory assistance.iReplacement
that led to multiple simultaneous ICHs points to a continuous
of CSF volume could not be practiced. Two days a the patient
vlater by e
and massive lumbar CSF evacuation resulting in a reduction of
CSF volume with the associated lowering in intraspinal and
a
s ted w.m
developed pneumonia, which resulted in sepsis and death in one intracranial pressure, which eventually increased the transmural
week more.
i pressure gradient of the vessels, leading to a secondary wall stress
F os w rupture.[1] Advanced age and the presence of diffuse white matter
PD te h (w
disease could be the other important contributing factors.[9] The
widespread and prolonged degeneration of the intracerebral
s
hi a si
arterioles in older people may also predispose to the development
of multiple ICHs. Unfortunately, amyloid angiopathy or other
T age-related cerebrovascular conditions were not completely
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.
Figure 2: Head CT after CSF overdrainage. (A) A petechial hemorrhage Indeed, the laminar collection of blood over the left parietal
in pons (arrow). (B) Bilateral ganglionic hemorrhages (arrows) plus convexity and the posterior interhemispheric space seen in the
multiple petechial hemorrhages in the right temporal lobe (arrow
head). (C) Ganglionic hemorrhage (arrow) with petechial head CT performed at presentation to our hospital [Figure 1]
hemorrhages in right parietal and occipital lobes (arrow heads). need comments. We were not told about the antecedent of head
Ventricular irruption is also evident. (D) The extension of the
ganglionic hemorrhage with its ventricular irruption (arrows) and
trauma that might explain this abnormality; however, considering
petechial hemorrhages in occipital lobe (arrow head). the gait instability that the patient was presenting, falls that might
422 Neurology India | December 2006 | Vol 54 | Issue 4
422 CMYK

3. 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 2 Amyloid angiopathy
Kobayashi Y, et al. 1987
m 1 Amyloid angiopathy
ro
Nakamura T, et al. 1988 1 Hypertension
Wakui K, et al.
Mori H, et al.
1988
f 1 Amyloid angiopathy associated to head injury
Tanno H, et al.
1989
1989
a d ns 1
5
Evacuation of chronic subdural hygroma
Hypertension
lo tio
Green RM, et al. 1990 1 Cocaine abuse
Kase CS, et al. 1990 2 tPA administration
wn lica
Hasegawa Y, et al . 1991 1 Vasculitis
Nagano N, et al . 1991 2 Anticoagulant therapy
do ub
Uno M, et al. 1991 9 Hypertension
Verstichel P, et al. 1991 1 Hypertension
Yanagawa Y, et al . 1994
e 1 Amyloid angiopathy
re w P m).
Komiyama M, et al. 1995 1 Hypertension
rf o o
Ozawa T, et al. 1995 1 Vasculitis
Seijo M, et al. 1996 7 Hypertension, coagulopathy
Dromerick AW, et al.
Liou HH, et al.
1997
1997 fo kn .c 1
1
tPA administration
Churg-Strauss syndrome
Nakamura K, et al.
Nighoghossian N, et al .
1997
b
1998le ed ow 1
1
Amyloid angiopathyassociated to migraine
Antimigrainous drug abuse
ila y M dkn
Daloze A, et al. 1999 1 Hypertension associated to renal cell carcinoma
Kimura T, et al. 2000 1 Vasculitis
Kohshi K, et al.
Mauriño J, et al.
ava b e
2000
2001
2
4
Hypertension
Hypertension
s ted w.m
Chen CY, et al. 2003 1 Hydrops fetalis
Oide T, et al.
i 2003 6 Amyloid angiopathy
Shiomi N, et al.
Okuno S and Sakaki T F os w 2004
2005
11
1
Hypertension
Systemic lupus erythematosus
PD te h (w
Yen CP, et al. 2005 10 Hypertension
Ruiz-Sandoval, et al. 2006 1 CSF overdrainage
s
hi a si
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
T
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, should not exceed 20-25 mL/h.[12] When used as a guide to case
even though delayed traumatic ICH exists,[10,11] it is mainly selection for a shunting procedure in normal pressure
associated with severe head trauma and would hardly cause more hydrocephalus[2] or as treatment of CSF fistula,[12] lumbar CSF
than two ICHs affecting both the infratentorial and supratentorial drainage of 40 to 50 mL per session is considered safe and
regions. effective.[1,2]
When a tap test is indicated, intermittent lumbar or continuous Another concern with respect to the case discussed here is the
CSF drainage at controlled rate are safe strategies in avoiding medical error that led to this catastrophe. This complication has
overdrainage,[1,2] especially because the lower threshold of CSF the possibility to be repeated, especially in teaching hospitals in
volume compatible with life in humans is rather unknown.[2] In which physicians in training perform without expert supervision.
our patient, an advanced age, sulcal enlargement and ventricular Appropriate measures were taken in our center to avoid another
dilatation allowing a large CSF volume might have permitted accident like this. Excessive work must not be an exception of a
such drainage of the fluid (250 mL in 30 min). Any time lumbar tight supervision to junior doctors.
CSF drainage is indicated as diagnostic procedure, it is necessary In conclusion, CSF overdrainage can either cause or precipitate
to be warned about an excessive rate of CSF drainage, which multiple simultaneous ICHs, affecting both the infratentorial and
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4. Ruiz-Sandoval JL, et al. : Multiple simultaneous ICHs after CFS overdrainage
supratentorial regions. 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)
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lumbar puncture. Pediatr Emerg Care 2001;17:184-8. subarachnoid catheter for treatment or prevention of cranial/spinal cerebrospinal
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fr
Maurino J, Saposnik G, Lepera S, Rey RC, Sica RE. Multiple simultaneous
intracerebral hemorrhages: Clinical features and outcome. Arch
Source of Support: Nil, Conflict of Interest: None declared.
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