Childs Nerv Syst (2011) 27:1563–1570

DOI 10.1007/s00381-011-1541-7

SPECIAL ANNUAL ISSUE

Guideline for management and treatment of fetal

and congenital hydrocephalus: Center Of Excellence—Fetal
and Congenital Hydrocephalus Top 10 Japan Guideline 2011
Shizuo Oi & Takayuki Inagaki & Masaki Shinoda &
Satoshi Takahashi & Shigeki Ono & Isao Date &
Sadahiro Nomura & Tomoru Miwa & Takashi Araki &
Susumu Ito & Hisaaki Uchikado & Osamu Takemoto &
Reizo Shirane & Hiroshi Nishimoto & Yuzuru Tashiro &
Akira Matsumura &
COE—Fetal and Congenital Hydrocephalus Top 10
Japan Study Group

Received: 24 July 2011 / Accepted: 25 July 2011

# Springer-Verlag 2011

Abstract morphological changes in the central nervous system properly.

Introduction Hydrocephalus does not indicate a single clin-
ical entity, but includes a variety of clinicopathological
conditions caused by excessive cerebrospinal fluid (CSF)
based on the disturbed circulation. Recent progress in prenatal
neuroimagings such as MRI and ultrasound echoencephalog-
raphy on fetus enables to understand clinicopathological
conditions of CSF circulation disorder in conjunction with
It has been revealed that the CSF dynamics develop in the
theory of evolution from the immature brain, as in the animals
with the minor CSF pathway predominance, towards matured
adult human brain together with the completion of the major
CSF pathway: the “Evolution Theory in CSF Dynamics”.
Now, we can analyze CSF circulation dynamically and also
analyze the flow velocity and direction of CSF movement.

S. Oi (*) S. Nomura
Health Sciences Asia Executive Dean Office, Department of Neurosurgery, Yamaguchi University,
Japan International University, 1-1-1 Minamiogushi,
2-18-1 Nakoji, Amagasaki, Ube, Yamaguchi, Japan
Hyogo 661-8530, Japan
e-mail: shizambroi@aol.com T. Miwa
Department of Neurosurgery, Jikei University,
T. Inagaki 3-25-8 Nishi-Shinbashi,
Department of Neurosurgery, Kansai Medical University, Minato-ku, Tokyo, Japan
10-15, Fumizono,
Moriguchi, Osaka, Japan T. Araki
Department of Neurosurgery,
M. Shinoda National Center for Child Health and Development,
Department of Neurosurgery, St. Luke’s International Hospital, 2-10-1 Okura,
9-1 Akashi-cho, Setagaya-ku, Tokyo, Japan
Chuo-ku, Tokyo, Japan
S. Ito
S. Takahashi Department of Neurosurgery,
Department of Neurosurgery, Keio University, Kanagawa Children’s Medical Center,
35 Shinano machi, 2-138-4 Mutsugawa, Minami-ku,
Shinjuku-ku, Tokyo, Japan Yokohama, Kanagawa, Japan

S. Ono : I. Date H. Uchikado

Department of Neurosurgery, Okayama University, Department of Neurosurgery, Kurume University,
1-1-1 Tsushima naka, 67 Asahi,
Kita-ku, Okayama, Japan Kurume, Fukuoka, Japan
1564
Center of Excellence—Fetal Hydrocephalus Top 10 Japan A-
long with this technical improvement, the standards of
clinicopathological evaluation of hydrocephalus as well as
the classification and concept of hydrocephalus shall
undergo a major upgrade. Based on such remarkable
improvement in the recent practical diagnostic evaluation
of fetal hydrocephalus, it is now required to update the
guideline for management and treatment of fetal and
congenital hydrocephalus, and a nationwide study group;
Center of Excellence—Fetal Hydrocephalus Top 10 Japan,
was organized in 2008 in Japan. The retrospective analysis
of 333 cases of congenital hydrocephalus indicated a fact
that 43% of these cases were diagnosed prenatally, and the
majority of cases were treated in these top 10 institutes in
Japan. Now, congenital hydrocephalus diagnosed immedi-
ately after birth is regarded as to be based on embryonic
stage; brain disorder in patients with congenital hydroceph-
alus should be considered in conjunction with neuronal
mature process of embryonic stage. The fact is supported by
the current trends in hydrocephalus research represented by
“Perspective Classification of Congenital Hydrocephalus”
and “Multi-categorical Hydrocephalus Classification”. The
ultimate goal of hydrocephalus treatment remains achieving
arrested hydrocephalus by shunt surgeries. In the future, to
achieve arrested hydrocephalus, minimum quantity of CSF
to be drained should be elucidated. Consideration for
accurate operative indication of ETV along with new neuro-
endoscopic device development and analysis of CSF
circulation is expected in the future. The data in this
prospective multicenter analysis in this guideline are credited
in Oxford Evidence level 2b (Grade II).
O. Takemoto
Department of Neurosurgery,
Osaka Medical Center and Research Institute for Maternal
and Child Health,
840 Murodou,
Izumi, Osaka, Japan
R. Shirane
Department of Neurosurgery, Miyage Children’s Hospital,
4-3-17 Ochiai, Aoba-ku,
Sendai, Miyagi, Japan
H. Nishimoto
Department of Neurosurgery, Saitama Children’s Medical Center,
2100 Ooaza-Magome,
Iwatsuki-ku, Saitama, Japan
Y. Tashiro
Department of Neurosurgery, Shizuoka Children’s Hospital,
860 Urushiyama,
Aoi-ku, Shizuoka, Japan
A. Matsumura
Department of Neurosurgery, University of Tsukuba,
1-1-1 Tennoudai,
Tsukuba, Ibaragi, Japan
Childs Nerv Syst (2011) 27:1563–1570
Keywords Hydrocephalus . Fetal hydrocephalus .
Guideline . Prenatal diagnosis . Postnatal outcome
Introduction
Progress of prenatal radiological diagnosis in the field of
hydrocephalus research gives us a lot of novel insights.
With the modality, now we can understand hydrocephalus
from their embryological aspects along with chronological
change of their pathophysiology. Clinically, it has been also
utilized to make guidelines based on prospective data
collection or to build up a medical team that can manage
patients with hydrocephalus from prenatal periods.
This is the guideline that was built up based on the data
collected by means of prospective, multicenter, and blinded
manner.
Material and method
The aim of this study is to understand the incidence and
type of fetal hydrocephalus in Japan. We sent the
questionnaire to the educational hospitals in Japan to know
the annual number of the patients of hydrocephalus they
diagnosed at year 2008. The retrospective analysis of 333
cases of congenital hydrocephalus indicated a fact that 43%
of these cases were diagnosed prenatally, and the majority
of cases were treated in these top 10 institutes in Japan. We
then selected 10 institutes according to the number of
hydrocephalus patients treated. We collected the data from
those institutes afterwards. We carefully examined the
medical records we collected. Inclusion is the patient
diagnosis in utero and the diagnosis was made until 1 year
of age. Classification of hydrocephalus was made based
upon the neuroradiological findings and obtained informa-
tion. Perspective Classification of Congenital Hydrocepha-
lus (PCCH) and Multi-categorical Hydrocephalus
Classification (McHC) was used. The timing of onset or
diagnosis of hydrocephalus, the classification of the
hydrocephalus, associate anomalies, timing of operation,
and development quotient (DQ) during the follow-up was
analyzed prospectively.
Center of Excellence—Fetal and Congenital
Hydrocephalus Top 10 Guideline 2011
The institutes selected for this study are the following 10
institutes: Osaka Medical Center and Research Institute for
Maternal and Child Health, Kanagawa Children's Medical
Center, Kansai Medical University, Kurume University,
National Center for Child Health and Development,
Childs Nerv Syst (2011) 27:1563–1570
Saitama Children's Medical Center, Sizuoka Children's
Hospital, Tsukuba University, Tokyo Jikei University, and
Miyagi Children's Hospital. The total number of patients
and the detailed information obtained was 107 (Fig. 1).
Fifty-five patients were diagnosed in utero (51.4%). The
period of diagnosis was from 16 to 37 weeks of gestation
(mean, 26.1 week). Remaining 52 patients were diagnosed
after birth. The timing of diagnosis was from 0 day to
12 month after birth (mean, 59.1 day). In one case, father
has the history of hydrocephalus. Three cases had the
chromosomal abnormalities. Two cases had gene abnor-
malities. The cause of hydrocephalus varied (Fig. 2).
Myeloschisis and post-hemorrhagic hydrocephalus are the
most common cause of hydrocephalus in this series. It was
almost the same incidence of previously reported Japanese
congenital hydrocephalus study. Eighty-four patients were
operated with reservoir placement, shunt placement and/or
neuroendoscopic surgery (78.5%). Seventy-four patients
out of 84 required shunt placement. Shunt repair was
performed in 24 out of 74 cases (28.6%), so far (Fig. 3).
The DQ was also measured. There is a tendency that the
patients with epilepsy have low DQ compared with the
patients without epilepsy. Slow progressive or arrested
hydrocephalus patients have high DQ (Figs. 4 and 5).
Cerebrospinal fluid and its circulation in fetus and neonates
In adults, CSF is generated by filtration or secretion from
ventricular colloid plexus or blood vessels, and is absorbed
in venous sinus via pacchionian body or blood vessels in
subarachnoid space. CSF is generated approximately 500 to
600 ml a day. The total amount of CSF in the subarachnoid
space is known to be approximately 10% of cranial volume.
CSF is totally replaced thrice to four times a day.
There are two types of CSF pathway; those are major
CSF pathway (consisted of ventricular system and sub-
arachnoid space) and minor CSF pathway (consisted of
brain and spinal parenchyma, vascular system, nerve root
sheath, and pia matter)
In pediatric populations, circulation velocity of CSF is faster
than adult, and CSF replacement interval is shorter than adult.
Fig. 1 Graph showed the distribution of the patient; approximately
51.4% of the patients were diagnosed in utero
1565
Pacchionian body, a major player in CSF absorption in
major CSF pathway does not appear until infancy. In
infancy, pacchionian body gradually appears and starts to
acquire function of CSF absorption.
CSF pathways of fetus, neonates, and infants are mainly
consisted of minor CSF pathway. Major CSF pathway starts
to develop gradually from infancy. In animals, birds,
rodents, and mammals like Macaca fuscata, the develop-
ment of pacchionian body is not observed and CSF
circulation is mainly consisted of minor CSF pathways.
We herein propose a hypothesis that the CSF dynamics
develop in the theory of evolution from the immature brain,
as in the animals with the minor CSF pathway predomi-
nance, towards matured adult human brain together with
completion of the major CSF pathway: the “evolution
theory in CSF dynamics” [5] (Fig. 6).
We also herein propose a new aspect of classification for
hydrocephalus with special reference to the CSF circulation in
the minor CSF pathway, i.e., “minor pathway hydrocephalus”.
The high incidence of “failure to arrest hydrocephalus” by
neuroendoscopic ventriculostomy in fetal, neonatal, and
infantile periods was explicable by the specific CSF dynam-
ics, in which the major CSF pathway has not developed and
the minor pathway has a significant role [8].
Findings of ventriculo-cisternography in a patient with
non-communicating hydrocephalus due to aqueduct stenosis
treated by endoscopic third ventriculostomy (ETV) in
combination with EAP: after outlet of third ventricle was
released and aqueduct was re-canalized, congestion of
contrast enhanced material inside ventricles and around
ventricular ependymal is observed even 4 days after operation.
Proposal of classification of fetal hydrocephalus and
congenital hydrocephalus Classification of hydrocephalus
is in variety. Congenital and acquired hydrocephalus is
usually classified independently based on the timing of
onset in conjunction with co-existing disease or its
pathophysiology. From their pathophysiological aspects,
hydrocephalus is classified as communicating–non-commu-
nicating or high pressure—normal pressure based on the
changes in CSF dynamics.
Most frequently discussed controversy in the field of
hydrocephalus is controversy in their classification. Classifi-
cation of hydrocephalus such as normal pressure hydroceph-
alus, arrested hydrocephalus, or external hydrocephalus
should be considered from their chronological change.
Hydrocephalus is a pathological state that may change
chronologically.
In the definition of communicating/non-communicating
hydrocephalus by Dandy, communicating hydrocephalus is
the form of hydrocephalus in which the injected dye could
be detected from the spinal subarachnoid space, and in non-
communicating hydrocephalus dye did not reach the spinal
1566
Fig. 2 The graph showed the
number of cases according the
cause of hydrocephalus (since
some cases were classified more
than two categories, one of the
main was shown on the graph)
subarachnoid space [1]. The concept is different from the
concept of obstructive/non-obstructive hydrocephalus by
Russell [10]. In the definition of Russell, the obstruction in
obstructive hydrocephalus can be at any region in the major
CSF pathway including the ventricular system and entire
cistern/subarachnoid space, so that the cause or condition
for non-obstructive hydrocephalus is limited to either CSF
overproduction by choroid plexus papilloma or CSF
malabsorption due to sinus thrombosis. Nowadays, the
two classifications are sometimes used with confusion.
The term “normal pressure hydrocephalus (NPH)” was
firstly proposed by Hakim and Adams in 1964 and 1965.
They defined this type of hydrocephalus as a syndrome
with surgically treatable specific clinical features such as
dementia, urinary incontinence, and gait disturbance [6].
They named the clinical entity as NPH from its pathophys-
iological aspects, but nowadays, recent research revealed
that the intracranial pressure in patients with NPH is not
always uniformly normal pressure. On the other hand,
intracranial pressure in pediatric and neonatal population is
physiologically lower than in adults. It is controversial
whether or not pathophysiological states like NPH exist in
pediatric populations or not.
Fig. 3 Graph indicates the shunt survival
Childs Nerv Syst (2011) 27:1563–1570
Some classifications that describe specific forms of
hydrocephalus such as longstanding overt ventriculomegaly
in adult (LOVA), hydrocephalus-parkinsonism complex, or
hydromyelic hydrocephalus have been proposed. It has not
been elucidated until now whether these types of hydro-
cephalus existed in pediatric populations or not [7, 9].
As for LOVA, it is estimated that the states of “pre-LOVA”
that is presented as merely enlargement of head circumference
without neurological abnormality may exist before it has
become clinically relevant LOVA. Multicenter study of
pediatric hydrocephalus in Japan recently revealed the
existence of pre-LOVA but the pathophysiology of pre-LOVA
has not been fully determined yet. Patients with pre-LOVA are
considered to show normal DQ and intelligence quotient (IQ).
From these findings, to classify hydrocephalus in fetus and
neonates, it is important to pay attention to when hydroceph-
alus occurs and how it would change chronologically. We
recommend using PCCH that focuses on the timing of onset of
hydrocephalus and their chronological change [2].
There exists hydrocephalus which can be classified to
multi-categorical subtypes. To explain the features of
Fig. 4 Difference in DQ between progressive and slow-progressive or
arrested hydrocephalus (based upon McHC classification)
Childs Nerv Syst (2011) 27:1563–1570
without epilepsy with epilepsy
Fig. 5 There is the significant difference in development between the
cases with and without epilepsy
hydrocephalus individually in detailed exposition, Multi-
categorical Hydrocephalus Classification (McHC) is ex-
tremely useful. [3, 4].
McHC classification of hydrocephalus classify hydroceph-
alus by 10 headings consisted of three subjects of “patients
characteristics”, “CSF”, and “treatment.” It is the most detailed
classification of hydrocephalus ever proposed that can classify
hydrocephalus into as many as 72,576,000 types. It involves
many previously proposed classification of hydrocephalus
from classical classification of communicating/non-communi-
cating hydrocephalus and obstructive/non-obstructive hydro-
cephalus to modern classification of evolution theory of CSF
dynamics and hydrocephalus chronology in adults.
It is recommended to perform intrauterine MRI at least
once in 4 weeks to evaluate the progression of hydroceph-
alus. Certainly, there exist some cases with fetal hydro-
cephalus in that the hydrocephalus progresses in rush. In
Fig. 6 Showed the CSF
dynamics maturation, so called
evolution theory of CSF
dynamics (5)
1567
that case, PCCH stage of the patient should be determined
from pathophysiological change of hydrocephalus in the
patient.
A case with PCCH stage II fetal hydrocephalus which showed
rapid progression of progressive hydrocephalus
Proposal concerning about treatment of fetal/congenital
hydrocephalus To change progressive pathophysiology of
hydrocephalus into the state of arrested hydrocephalus is
the aim in the treatment of hydrocephalus. Progressive
hydrocephalus will rarely change into arrested hydroceph-
alus spontaneously; however, in many cases, shunt proce-
dures are required. Many patients with hydrocephalus,
other than patients with non-communicating hydrocephalus,
due to third ventricular brain tumor have brain tumor
removal to cure hydrocephalus and maintain their brain
function relying on shunt system.
There are many types of shunt system nowadays. Most
frequently used shunt system is the three-piece shunt
system that consists of three parts: these are the flushing
device, valve, and chamber.
Shunt system should not be specified in any guideline,
and it should be selected depending on the pathophysio-
logical states of hydrocephalus in each patient. Pathophys-
iological states of hydrocephalus in each patient should be
determined depending on categories VI and VII of McHC
classification of hydrocephalus.
When to operate patients with hydrocephalus varies in
each patient; however, it should be conducted before
1568
irreversible change would be occur to brain parenchyma.
Especially in smaller children, the neurosurgeon should pay
attention not to miss the timing of surgery.
ETV is only effective in patients with non-communicating
hydrocephalus. In pediatric patients who are less than 1 year
old, CSF circulation mainly depends on minor CSF pathway.
ETV is useful only in patients whose CSF circulation mainly
depends on major CSF pathway; however, some specialists do
not recommend performing ETV to this patient population
(Fig. 7).
Main principle of treating patients with hydrocephalus is
aiming to arrest hydrocephalus considering normal physi-
ological function of CSF. In this context, endoscopic
surgical ablation of colloid plexus aiming to the reduction
of CSF production may result in impairment of CSF flow,
thus its usability remains to be controversial for now.
Complication of shunt surgery: concepts and classifica-
tion Complication of shunt surgery can be classified into
four major categories: those are shunt malfunction,
infection, over drainage, and surgical complication.
One of the factors that greatly affects surgical outcome is
shunt management after operation. Chronological change in
physiology of hydrocephalus should be stressed in the
management of shunt system.
A great variety of complications associated with shunt
system including shunt closure or infection have been
reported so for. Complications associated with shunt
systems or surgical procedures have been also documented
so far.
Obstruction of shunt device is known to be caused by the
following reasons. Major causes of obstruction in ventricular
side are connective tissue, inflammatory tissues, necrotic
Fig. 7 Showed the case in which the ETV did not improved the patient's condition
Childs Nerv Syst (2011) 27:1563–1570
tissues, and colloid plexus. On the contrary, major causes of
obstruction in abdominal side are greater omentum and cystic
change of tissues. Clinical manifestation of shunt malfunction
varies depending on pathophysiology of hydrocephalus and
patients' age. To diagnose shunt malfunction with only
flushing device compression findings is difficult. Shunt
malfunction is not always presented with ventricular en-
largement. In those patients continuous ICP evaluation is
extremely useful. Shunt obstruction should be treated by
shunt reconstruction as soon as possible. Number of shunt
reconstructions does not affect prognosis.
Clinical manifestation of shunt infection varies accord-
ing to types of shunt surgery. Typical signs of shunt
infection after VA shunt are fever and cough, and sign of
shunt infection after VP shunt is abdominal pain. When the
signs and symptoms of meningitis presents, a diagnosis of
shunt infection is definitive; however, only one third of all
the patients with shunt infection present the sign of
meningitis while around 80% of the patients present fever.
As many as two thirds of shunt infection occur within
4 months from shunt surgery; however, more than 20% of
shunt infection may occur after more than 1 year from
shunt surgery. A flare or swelling of the skin along with
shunt tube tract should be regarded as the sign of infection.
Increased cell counts and protein level in conjunction with
decreased glucose level in CSF is the typical findings for
shunt infection. Sometimes, CSF culture would be exam-
ined in an attempt of identify pathogenic bacteria. In cases
in which infection is localized at the site of wound or shunt
tube tract, CSF findings can be within normal limit.
Shunt tube in patients suffering from shunt infection
should be removed, and external ventricular drainage
should be placed instead. Keep ICP within normal range
Childs Nerv Syst (2011) 27:1563–1570
with ventricular drainage, administrate antibiotics, and wait
until infection disappears. After infection disappears,
ventricular drainage should be removed and shunt recon-
struction should be done.
Subdural hematoma after shunt surgery: CSF over
drainage after shunt placement sometimes results in slit
ventricle syndrome. In this situation, deterioration of brain
compliance resulted in laceration of brain surface vein and
may cause subdural hematoma. In patients with slit
ventricle syndrome, ICP may raise without ventricular
enlargement. In these situations, surgeries that can decrease
the amount of drained CSF should be considered.
Pathophysiology of isolated fourth ventricle syndrome is
considered to be aqueduct stenosis after shunt placement in
patients with obstruction of outlet of fourth ventricle. There
are various opinions in terms of functional aqueduct
stenosis in this clinical entity (Fig. 8).
Prognosis of fetal/congenital hydrocephalus Both patients
and therapeutic factors define prognosis of hydrocephalus.
Recent data shows that the frequency of super high IQ
patients is statistically higher in patients after hydrocepha-
lus treatment. As for prognosis of hydrocephalus, prognosis
of simple hydrocephalus is usually good. On the contrary,
DQ in patients with syndromic hydrocephalus remains to be
poor. Progressive ventricular enlargement and existence of
convulsion are shown to be negative prognostic factors.
Fig. 8 The heavily-T2-weighted fast-spine-echo MR image demon-
strated severe triventricular hydrocephalus, likely due to aqueductal
stenosis, at 27 weeks in gestational age (a, c, e). The fetal MR images
follow up at 33-weeks gestational age (b, d, f) disclosed more
1569
From these findings, we think that McHC classification
based on PCCH stage can predict prognosis of pediatric
patientswith hydrocephalus (from the result of prospective
study of COE top 10 Japan).
In hydrocephalus patients without brain anomaly, progno-
ses are not always pessimistic, and patients can show DQ over
80 with adequate treatment timing and proper management
after surgery. As noted, our present data shows that there is no
statistical significant difference in the prognosis of patients
with communicating hydrocephalus and those of patients with
non-communicating hydrocephalus. Underlining lesion of
hydrocephalus and existence of convulsion are proved to be
statistically significant prognostic factors.
Shunt malfunction In terms of mechanical obstruction,
major causes of obstruction in ventricular side of VP shunt
are connective tissue, inflammatory tissues, and colloid
plexus. On the contrary, major causes of obstruction in
abdominal side of VP shunt are connective tissue, inflam-
matory tissue, necrotic tissue, and fibrous tissue. Even if
there is no obstruction inside the shunt tube, bending,
laceration, or relative shortening (according to growth) of
shunt tube can cause shunt malfunction. Ventricular end of
shunt tube should be placed apart from colloid plexus.
Factors such as patients' age or underlining lesion for
hydrocephalus also affect shunt closure. In immature brain,
shunt obstruction occurs more frequently. Shunt obstruction
expanded ventriculomegaly and macrocephalus with more severely
comparessed/thinning brain mantle suggesting a fact of rapid
progression of fetal hydrocephalus before birth [4]
1570
frequently occurs within 5 months from shunt placement.
As for location, it occurs more frequently in abdominal end
compared to ventricular end.
Most frequently identified pathogenic bacteria at the
time of shunt infection is known to be Staphylococcus
epidermidis, and when the second frequently identified
pathogenic bacteria, Staphylococcus aureus is added, they
occupy 70% of all the pathogenic bacteria that cause shunt
infection. Breakdowns of remaining 30% are consisted of
mainly gram-negative bacteria and gram-positive bacteria
other than the two above-described bacteria. Shunt infec-
tion rates in pediatric populations is reported to consider-
able variety; 2% to 30%.
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