What is the diagnostic value of the babygram?

Poster No.: C-2448

Congress: ECR 2012
Type: Educational Exhibit
Authors: W. M. Klein, S. Franken, C. Marcelis; Nijmegen/NL
Keywords: Fetus, Dysplasias, Congenital, Diagnostic procedure, Conventional
radiography, Forensic / Necropsy studies, Foetal imaging,
Anatomy
DOI: 10.1594/ecr2012/C-2448

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Learning objectives

To learn what bone length is normal in postmortem fetal radiography ("babygram");

To learn if bone length can be typical for a certain disease.

To learn the diagnostic value of the babygram in diagnosing congenital malformations

and cause of death.

Images for this section:

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Fig. 1: Fetus after 20 weeks gestation; intra-uterine death for unknown reason.

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Background

In case of a deceased fetus, both spontaneously and after termination of pregnancy

with congenital malformations, a definite diagnosis is very important. A definite diagnosis
will indicate the cause of death and the reason for the malformations, helping the
parents understand why their baby could not live. Also a diagnosis is needed for genetic
counseling of the parents and family.

A babygram is a diagnostic tool in the total workup of deceased fetuses, consisting of

antero-posterior and lateral images of the total fetus, and added images on indication.
A babygram is routinely made after prenatal suspicion of serious or lethal congenital
malformations and termination of pregnancy, to confirm prenatal diagnosis made on
ultrasonography. Also, after death in utero, the babygram is made the help find a possible
diagnosis, besides pathological, biochemical and molecular diagnostics.

There is very little literature on postmortem fetal radiography. We have only been able to
find one reference of a study with normal values (thesis W. Scherf 2001) (ref.2), however
the original study is not available on the internet and up to now we were not able to trace
it. The results are used in the book by R. Schumacher ("Fetal radiology, a diagnostic
atlas") (ref.1).

We retrospectively studied the babygrams in our university hospital, to estimate normal

values en to evaluate the diagnostic value of the babygram.

Images for this section:

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Fig. 2: Fetus of 22 weeks after termination of pregnancy because of suspicion of skeletal
dysplasia. Postmortem, the diagnosis was fetal akinesia of unknown cause.

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Fig. 3: intra-uterine death with prenatally diagnosed congenital heart abnormalities and
palatoschisis. The babygram shows abnormal formation of the upper thoracic vertebral
bodies and sacrococcygeal bone, with left and right club feet.

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Imaging findings OR Procedure details

Method:

We retrospectively evaluated the babygrams made in our university hospital from

november 2002 until september 2011 that were available in digital version in Impax 6.4.
We excluded babies who survived more than 48 hours after birth.

We measured the following: skull length en width; crown rump length; vertebral heigth
and interpedicular width of C7, Th12 and L5; length and width of humerus, ulna, radius,
metacarpal 2, femur, tibia, fibula, metatarsal 2 (fig.4) and calculated ratios length to width.
Further we calculated ratios for crown rump length to femur length and to humerus length,
femur to tibia length, humerus to radius length, femur to humerus length. We counted
the number of vertebral bodies with calcification, we noted teeth formation and we noted
calcification of the calcaneus.

We noted gestational age, prenatal diagnosis (ultrasonography), cause of death, findings

on the babygram and final diagnosis.

We estimated normal values using the fetuses who showed no signs of pathology on
prenatal ultrasonography, postmortem or at pathology. These fetuses could have died for
no obvious reason, or maternal reasons or events shortly before death such as asphyxia,
acute cerebral ischemia, maternal or placental infection or acute umbilical complications.

Data were noted in PASW statistics 18.0. We estimated linear regression analysis for
skeletal data and age and made scatter plots with regression line and 95% confidence
interval.

Results:

We collected 212 babygrams of fetuses with gestational age from 12 to 41 weeks, 53 of

which we called 'normal'.

87 fetuses died in utero; 48 fetuses died shortly after birth; 77 fetuses died with
termination of pregnancy.

On prenatal ultrasonography 21 fetuses had growth retardation; 100 showed congenital

malformations (brain or spine 29; thorax 21; cardiovascular 17; abdomen 16; limbs or
skeleton 17).

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All fetuses had postmortem diagnostic workup including a babygram, and all or part of
the following: biochemical, fibroblast and chromosomal analysis, obduction of body, brain
and placenta, postmortem CT and MRI.

A definite diagnosis with absolute certainty was not possible in most cases, especially
after unexpected intra-uterine death. A diagnosis that reasonably explained the death
and / or congenital malformations was available in 109 / 212 cases (51%).

In the majority of cases the babygram showed no or minor malformations.

Only in 13/ 212 cases (6%) the babygram showed malformations important for the
diagnosis:

Osteogenesis imperfecta (3) (fig.5)

Thanatophoric dysplasia (2)

Gracile bone dysplasia (1) (fig.6)

OEIS complex (1)

Femur-fibula-ulna complex (1)

Caudal regression syndrome (1)

Chondrodysplasia punctata (2) (fig.7)

Trisomie 18 with hypoplastic radius (1)

Skeletal deformations probably caused by fetal akinesia (1)

We estimated normal values of the long bones and skull. Normal values of femur and
humerus length for gestational age are presented in the following graphs (fig.8 and fig.9).
These are in accordance with data available on prenatal ultrasonography (ref.3). Also the
other measurements taken were in accordance with literature on ultrasonography and
showed linear growth over time.

We plotted the bone length of the abnormal fetuses against the bone length of normal
fetuses. Fig. 10 and fig.11 are graphs showing femur and humerus length for both
normal and abnormal fetuses, respectively. Most of the bone length of the abnormal
fetuses are within 95% confidence intervals and thus not too small or too tall. The
measurements outside the 95% CI are mostly too short for gestational age and these
represent the growth retarded fetuses as well as patients with osteogenesis imperfecta
and thanatophoric dysplasia or other skeletal dysplasia.

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The other measurements we took were not especially helpful in finding diagnoses. Also
the ratios we calculated were not indicative for certain diagnosis.

We found that the measurements in the babygram were not helpful for diagnostics. Only in
a small minority of the cases (13 /212) the babygram as a whole was helpful in diagnosing
disease. This was especially in cases with prenatal abnormal skeletal formation and
suspicion of skeletal dysplasia. In the other cases (94%) the babygram did not add in
the diagnostic considerations.

Images for this section:

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Fig. 4: Example of measurements in this 17 week old fetus.

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Fig. 5: Termination of pregnancy at gestation age of 23 weeks because of
suspicion of lethal skeletal dysplasia: multiple fractures of ribs and long bones with
undermineralisation, crumpled appearance and shortening and hypoplastic thorax.
Osteogenesis imperfecta type 2.

Fig. 6: A fetus of 23 weeks and 6 days, after termination of pregnancy because of

suspicion of a lethal skeletal dysplasia. There are slender long bones with bowing of

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ulna and radius, and a total fracture of the right femur. The diagnosis was gracile bone
dysplasia.

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Fig. 7: This fetus died in utero in the 35th week of gestation. Prenatal ultrasound
showed growth retardation but no signs of congenital malformation. The babygram
shows stippled epiphyses of the femurs, short humerus, butterfly vertebrae, as well as
hitchhiker's thumbs (maybe because of positioning by technician). This is a rhizomelic
chondrodysplasia punctata.

Fig. 8: Graph showing the length of the femur plotted against the gestational age in the
53 'normal' fetuses.

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Fig. 9: Graph showing the length of the humerus plotted against the gestational age in
the 53 'normal' fetuses.

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Fig. 10: Graph showing the length of the femur plotted against the gestational age in the
53 'normal' fetuses (green circles) as well as the 159 abnormal fetuses (blue stars).

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Fig. 11: Graph showing the length of the humerus plotted against the gestational age in
the 53 'normal' fetuses (green circles) as well as the 159 abnormal fetuses (blue stars).

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Conclusion

We conclude that the babygram is not a valuable diagnostic tool in general. In cases
with suspicion of skeletal malformation or dysplasia the babygram will be helpful in
establishing a final diagnosis; in our study this is only a minority of cases. In cases not
suspected of skeletal dysplasia or major skeletal malformation, a babygram is not useful.

The diagnostic values of postmortem CT and MRI, which have the advantage of better
visualisation of both brain, organs, bone and cartilage, will be investigated next.

Personal Information

Willemijn M. Klein, MD, PhD

pediatric radiologist

University Medical Center Nijmegen St.-Radboud

Geert Grooteplein Zuid 10

6525 GA Nijmegen

the Netherlands

w.klein@rad.umcn.nl

References

1. Fetal Radiology; a diagnostic atlas. R. Schumacher, L. Seaver, J. Spranger. 2nd

edition, 2010. Spranger.

2. Normwerte fetaler Skelettmasse mittels post-mortem-Radiographie. W. Scherf, thesis

2001. Humboldt Universität Berlin.

3. Charts of fetal size: limb bones. L.S. Chitty, D.G. Altman. BJOG 2002;109(8): 919-929.

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