Accepted Article Anatomical subgroup analysis of the MERIDIAN cohort: Posterior fossa abnormalities Short title: iuMR for the posterior fossa abnormalities Paul D Griffiths PhD FRCR 1 , Karen Brackley FRCOG DM 2 , Michael Bradburn MSc 3 , Daniel JA Connolly BSC MRCP FRCR 4 , Mary L Gawne-Cain MRCP, FRCR, MD 2 , Mark D Kilby DSc MD FRCOG, FRCPI 5 , Laura Mandefield MSc 3 , Cara Mooney MSc 3 , Stephen C Robson MD MRCOG FRCP(Ed) 6 , Brigitte Vollmer MD, PhD 7 , Gerald Mason MD FRCOG 8 1 Academic Unit of Radiology, University of Sheffield, Glossop Road, Sheffield, S10 2JF, England ii) INSIGNEO Institute for in silico Medicine, University of Sheffield, UK, S10 2JF, 2 University Hospital Southampton NHS Foundation Trust, UK SO16 6YD, 3 Clinical Trials Research Unit, School of Health and Related Research, University of Sheffield, S1 4DA, 4 Department of Radiology, Sheffield Children’s Hospital, S10 2TH and the Royal Hallamshire Hospital, Sheffield Teaching Hospitals NHS Foundation Trust, S10 2JF, 5 i) Centre for Women's & Newborn Health, Institute of Metabolism & Systems Research, University of Birmingham, B15 2TT. ii) Fetal Medicine Centre, Birmingham Women's Foundation Trust (Birmingham Health Partners), Birmingham, B15 2TG, 6 Newcastle University, Newcastle upon Tyne NE2 4HH, 7 Clinical and Experimental Sciences University of Southampton and Southampton Children’s Hospital, University Hospital Southampton NHS Foundation Trust SO16 6YD 8 Leeds Teaching Hospitals NHS trust, LS9 7TF This article is protected by copyright. All rights reserved. This article has been accepted for publication and undergone full peer review but has not been through the copyediting, typesetting, pagination and proofreading process, which may lead to differences between this version and the Version of Record. Please cite this article as doi: 10.1002/uog.17485
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Acc
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d A
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eAnatomical subgroup analysis of the MERIDIAN cohort:
Posterior fossa abnormalities
Short title: iuMR for the posterior fossa abnormalities
Paul D Griffiths PhD FRCR1, Karen Brackley FRCOG DM2, Michael Bradburn MSc3,
Daniel JA Connolly BSC MRCP FRCR4, Mary L Gawne-Cain MRCP, FRCR, MD2,
Mark D Kilby DSc MD FRCOG, FRCPI5, Laura Mandefield MSc3, Cara Mooney
MSc3, Stephen C Robson MD MRCOG FRCP(Ed)6, Brigitte Vollmer MD, PhD7,
Gerald Mason MD FRCOG8
1Academic Unit of Radiology, University of Sheffield, Glossop Road, Sheffield, S10
2JF, England ii) INSIGNEO Institute for in silico Medicine, University of Sheffield,
UK, S10 2JF, 2 University Hospital Southampton NHS Foundation Trust, UK SO16
6YD, 3Clinical Trials Research Unit, School of Health and Related Research, University
of Sheffield, S1 4DA, 4Department of Radiology, Sheffield Children’s Hospital, S10
2TH and the Royal Hallamshire Hospital, Sheffield Teaching Hospitals NHS
Foundation Trust, S10 2JF, 5i) Centre for Women's & Newborn Health, Institute of
Metabolism & Systems Research, University of Birmingham, B15 2TT. ii) Fetal
Medicine Centre, Birmingham Women's Foundation Trust (Birmingham Health
Partners), Birmingham, B15 2TG, 6Newcastle University, Newcastle upon Tyne NE2
4HH, 7Clinical and Experimental Sciences University of Southampton and
Southampton Children’s Hospital, University Hospital Southampton NHS Foundation
This article is protected by copyright. All rights reserved.
This article has been accepted for publication and undergone full peer review but has not been through the copyediting, typesetting, pagination and proofreading process, which may lead to differences between this version and the Version of Record. Please cite this article as doi: 10.1002/uog.17485
11. Luna, JA and Goldstein RB. Sonographic visualization of neonatal posterior
fossa abnormalities through the posterolateral fontanelle. AJR Am J Roentgenol
2000; 174(2) : 561-567
12. Steggerda SJ, de Bruïne FT, Smits-Wintjens VE, Walther FJ, van Wezel-Meijler
G. Ultrasound detection of posterior fossa abnormalities in full-term neonates.
Early Human Development 2012; 88(4) : 233-239
CONFLICT OF INTEREST
The authors confirm that there are no conflicts of interest.
This article is protected by copyright. All rights reserved.
Acc
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eLEG
Figur
asses
GENDS
re 1. The al
ss changes i
lgorithm use
in appropria
ed to provid
ate confiden
de the score
nce (modifie
-based weig
ed from Ng
ghted avera
and Palmer
ge data used
r7).
d to
This article is protected by copyright. All rights reserved.
Acc
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eFigur
outco
ultra
utero
is con
space
indic
a sm
large
Figur
outco
ultra
and i
hypo
on th
malfo
the tr
re 2. A case
ome referen
sonography
o MR imagi
nfirmed on
es and disru
cates hydroc
all posterior
e spinal abn
re 3. A case
ome referen
sonography
in utero MR
oplasia of th
he sagittal an
formation. M
rigones of th
e of comple
nce data. Th
y on the basi
ing was perf
the axial iu
uption of the
cephalus. Sa
r fossa and
ormality (m
e of comple
nce data. Th
y on the basi
R imaging w
he cerebellar
nd axial iuM
Mild ventric
he lateral ve
te agreemen
he fetus was
is of a Chia
formed at 2
uMR image
e cavum sep
agittal imag
cerebellar t
myelomenin
te agreemen
he fetus was
is of a Dand
was perform
r vermis and
MR images
culomegaly
entricles (3c
nt between
s recruited in
ari 2 malform
4 weeks ge
(2a) and th
ptum pelluc
ge (2c) show
tissue hernia
gocoeles) is
nt between
s recruited in
dy-Walker m
med at 21 we
d increased
(3a and 3b)
is demonstr
c).
ultrasound,
nto the MER
mation and
stational ag
e effacemen
cidum on th
ws a severe
ating into th
s also show
ultrasound,
nto the MER
malformatio
eeks gestatio
volume of
) in keeping
rated on the
in utero M
RIDIAN stu
ventriculom
ge. Severe v
nt of the ext
e coronal im
Chiari 2 ma
he cervical s
n.
in utero M
RIDIAN stu
on and vent
onal age. Th
the posterio
g with a Dan
e axial imag
R imaging a
udy from
megaly and
ventriculome
ternal CSF
mage (2b)
alformation
spinal canal
R imaging a
udy from
triculomega
here is seve
or fossa pre
ndy-Walker
ge at the lev
and
in
egaly
with
l. A
and
aly
ere
sent
r
el of
This article is protected by copyright. All rights reserved.
Acc
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e
Figur
the c
the M
malfo
week
incre
(4a a
confi
(arro
Figur
the c
dizyg
ultra
utero
cereb
inden
re 4. A case
correct diagn
MERIDIAN
formation an
ks gestation
eased volum
and 4b) in k
firmed on th
owed on 4a a
re 5. A case
correct diagn
gotic twin p
sonography
o MR imagi
bellum (in t
ntation at th
e of disagree
nosis being
N study from
nd ventricul
nal age. Ther
me of the po
eeping with
he axial imag
and 4c and
e of disagree
nosis being
pregnancy a
y on the basi
ing was perf
erms of tran
he site of the
ement betw
made on in
m ultrasonog
lomegaly an
re is severe
sterior fossa
h a Dandy-W
ge (4c) but
confirmed o
ement betw
made on in
and was recr
is of a cereb
formed at 2
nscerebellar
e normal ce
ween ultrasou
n utero MR
graphy on th
nd in utero M
hypoplasia
a present on
Walker malf
there is also
on autopsy)
ween ultrasou
n utero MR
ruited into t
bellar hypop
5 weeks ge
r diameter –
erebellar ver
und and out
imaging. Th
he basis of a
MR imaging
a of the cere
n the sagitta
formation. M
o hypoplasi
).
und and out
imaging. Th
the MERIDI
plasia. The
stational ag
– figure 5a)
rmis (arrow
tcome refer
he fetus wa
a Dandy-W
g was perfo
ebellar verm
al and axial
Mild ventric
a of the cor
tcome refer
his fetus wa
IAN study f
other twin w
ge and confi
but also sho
wed 5a). The
rence data w
s recruited i
Walker
ormed at 21
mis and mild
iuMR imag
culomegaly
rpus callosu
rence data w
as the result
from
was normal
irmed a sma
owed lack o
e inferior
with
into
d
ges
y was
um
with
t of a
l. In
all
of the
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Acc
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d A
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esuper
morp
abno
rhom
5d).
Figur
the c
the M
abno
gesta
confl
centr
to rep
sagit
enlar
diagn
show
rior vermian
phology of t
ormal contin
mbencephalo
re 6. A case
correct diagn
MERIDIAN
ormality in t
ational age.
fluence (6a a
re and high
presented c
ttal sinus, de
rged. A diag
nosis was co
wed partial r
n height app
the cerebell
nuity of the
osynapsis. T
e of disagree
nosis being
N study from
the posterior
There is a l
and 6b) that
signal rim o
lotted blood
eep venous
gnosis of du
onfirmed on
resolution. N
pears to be
lar vermis. T
cerebellar h
These featur
ement betw
made on in
m ultrasonog
r fossa and
large extra-a
t has heterog
on both T2
d at differen
system (6a)
uro-venous
n postnatal
No brain pa
normal (5b)
This indicat
hemispheres
res were con
ween ultrasou
n utero MR
graphy on th
in utero MR
axial mass l
genous sign
(6b) and T1
nt stages of
) and transv
sinus ectasi
MR imagin
arenchymal
) but there i
tes non-form
s over the m
nfirmed on
und and out
imaging. Th
he basis of a
R imaging w
lesion centr
nal on in ute
1-weighted
evolution. I
verse sinuse
ia with throm
ng at 1 day o
abnormality
is none of th
mation of th
mid-line i.e.
postnatal im
tcome refer
he fetus wa
an abnorma
was perform
red on the v
ero MR ima
(6c) images
It extends in
es (6d), all o
mbosis was
of age (6e-6
y was prese
he normal
he vermis an
maging (5c
rence data w
s recruited i
al cystic
med at 29 w
enous
ages - low si
s. This is lik
nto the supe
of which are
s made. The
6g) which
ent. The fron
nd
and
with
into
eeks
ignal
kely
erior
e
e
ntal
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Acc
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eproje
are o
Figur
50%)
the b
refer
ection of the
occluded.
re 7. Bar ch
) diagnoses
bar chart ind
rence data. S
e MR venog
harts of ‘hig
made on ul
dicate those
See text for
graphy study
gh confidenc
ltrasonograp
diagnoses t
details.
y shows tha
ce’ (70 or 9
phy and in u
that were in
at the transv
0%) and ‘lo
utero MR im
ncorrect in c
verse and sig
ow confiden
maging. Th
comparison
gmoid sinus
nce’ (10, 30
e red parts o
with outcom
ses
or
of
me
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Acc
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eFigur
fossa
score
re 8. Chang
a cases (n=8
es (see text
ges in diagn
81). There is
for details a
ostic confid
s a significa
and interpre
dence using
ant increase
etation).
the route sc
e (0.7, p<0.0
core method
0001) in fav
d for Poster
vour of posit
rior
tive
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Acc
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eTable 1 Cases of isolated abnormalities of the posterior fossa diagnosed on USS and the equivalent cases on iuMR compared with ORD.
The subgroup ‘any abnormality of the posterior fossa’ allows any diagnosis of isolated
posterior fossa abnormality to be correct when compared with ORD, whereas subgroups
‘parenchymal’ and ‘CSF’ require the more restrictive diagnoses described in the text to be
correct. * McNemar’s test between USS and iuMR correct diagnoses.
USS Diagnoses iuMR Diagnoses Comparison of diagnostic accuracy
Subgroup n
Number incorrect
Diagnostic accuracy
Number incorrect
Diagnostic accuracy
Difference (95% CI**) p-value*
Any abnormality of the posterior fossa
81 28 65.4% 10 87.7% 22.3 (14.0, 30.5%)
<0⋅0001
Parenchymal abnormality
67 13 80.6% 3 95.5% 14.9 (5.7, 24.1%)
0.0020
CSF abnormality
14 11 21.4% 6 57.1% 35.7 (-1.6, 73.0%)
0.0625
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Acc
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eTable 2 Summary data on the 28 cases in which posterior fossa abnormalities diagnosed on USS were incorrect when compared with outcome reference data from the first line of results in table 1.
Case
USS diagnosis Posterior fossa
abnormality on ORD
Other brain
abnormality on ORD
iuMR
weeks
Posterior fossa
abnormality on iuMR
Other brain
abnormality on iuMR
43 DWS DWS Hypoplasia CC 20 DWS *No
55 Hypoplastic vermis Normal No 29 *Hypoplastic vermis No
148 Hypoplastic vermis Normal No 21 *Hypoplastic vermis No