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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
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
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


Neurology India | December 2006 | Vol 54 | Issue 4                                                                                                            423
                                                                                                                                                     CMYK423
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)
                                References                                                9.	
                                                                                                 2005;147:393-9.
                                                                                                 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.
1.	   Bloch J, Regli L. Brain stem and cerebellar dysfunction after lumbar spinal fluid          Neurology 2004;63:1606-12.
      drainage: Case report. J Neurol Neurosurg Psychiatr 2003;74:992-4.                  10.	   Cooper PR. Delayed traumatic intracerebral hemorrhage. Neurosurg Clin N Am
2.	   Fishman RA. Cerebrospinal fluid in diseases of the nervous system. 2nd ed. WB              1992;3:659-65.
      Saunders: Philadelphia; 1992.                                                       11.	   Erol FS, Kaplan M, Topsakal C, Ozveren MF, Tiftikci MT. Coexistence of rapidly
3.	   Snow RB, Kuhel W, Martin SB. Prolonged lumbar spinal drainage after the resection          resolving acute subdural hematoma and delayed traumatic intracerebral
      of tumors of the skull base: A cautionary note. Neurosurgery 1991;28:880-3.                hemorrhage. Pediatr Neurosurg 2004;40:238-40.
4.	   Adler MD, Comi AE, Walker AR. Acute hemorrhagic complication of diagnostic          12.	   Shapiro SA, Scully T. Closed continuous drainage of cerebrospinal fluid via a lumbar
      lumbar puncture. Pediatr Emerg Care 2001;17:184-8.                                         subarachnoid catheter for treatment or prevention of cranial/spinal cerebrospinal
5.	   Stubgen JP. Intraventricular blood after “traumatic” lumbar puncture: A report             fluid fistula. Neurosurgery 1992;30:241-5.
      of two cases. Childs Nerv Syst 1995;11:492-3.
6.    Suri A, Pandey P, Mehta VS. Subarachnoid hemorrhage and intracereebral


                                                                    om
      hematoma following lumboperitoneal shunt for pseudotumor cerebri: a rare
      complication. Neurol India 2002;50:508-10.                                           Accepted on 29-05-2006
7.
                                                                  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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424                                                                                                                 Neurology India | December 2006 | Vol 54 | Issue 4
424 CMYK

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5. multiple ich. case report

  • 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 Neurology India | December 2006 | Vol 54 | Issue 4 423 CMYK423
  • 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) References 9. 2005;147:393-9. 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. 1. Bloch J, Regli L. Brain stem and cerebellar dysfunction after lumbar spinal fluid Neurology 2004;63:1606-12. drainage: Case report. J Neurol Neurosurg Psychiatr 2003;74:992-4. 10. Cooper PR. Delayed traumatic intracerebral hemorrhage. Neurosurg Clin N Am 2. Fishman RA. Cerebrospinal fluid in diseases of the nervous system. 2nd ed. WB 1992;3:659-65. Saunders: Philadelphia; 1992. 11. Erol FS, Kaplan M, Topsakal C, Ozveren MF, Tiftikci MT. Coexistence of rapidly 3. Snow RB, Kuhel W, Martin SB. Prolonged lumbar spinal drainage after the resection resolving acute subdural hematoma and delayed traumatic intracerebral of tumors of the skull base: A cautionary note. Neurosurgery 1991;28:880-3. hemorrhage. Pediatr Neurosurg 2004;40:238-40. 4. Adler MD, Comi AE, Walker AR. Acute hemorrhagic complication of diagnostic 12. Shapiro SA, Scully T. Closed continuous drainage of cerebrospinal fluid via a lumbar lumbar puncture. Pediatr Emerg Care 2001;17:184-8. subarachnoid catheter for treatment or prevention of cranial/spinal cerebrospinal 5. Stubgen JP. Intraventricular blood after “traumatic” lumbar puncture: A report fluid fistula. Neurosurgery 1992;30:241-5. of two cases. Childs Nerv Syst 1995;11:492-3. 6. Suri A, Pandey P, Mehta VS. Subarachnoid hemorrhage and intracereebral om hematoma following lumboperitoneal shunt for pseudotumor cerebri: a rare complication. Neurol India 2002;50:508-10. Accepted on 29-05-2006 7. 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. oad ons nl ati ow blic d u e re w P m). rf o o fo kn .c e bl ed now ila y M dk ava b e is ted w.m F os w PD te h (w s Thi a si 424 Neurology India | December 2006 | Vol 54 | Issue 4 424 CMYK