The outcome of trisomy 18 pregnancies following the legalisation of

Ir Med J; November-December 2023; Vol 116; No. 10; P873

December, 2023

The outcome of trisomy 18 pregnancies following the

legalisation of termination of pregnancy

1,2

O'Donoghue ., K

Uribe ., C

Miremberg .H

1. Pregnancy Loss Research Group, Department of Obstetrics and Gynaecology,

Cork University Maternity Hospital, Cork, Ireland

2. INFANT Research Centre, University College Cork, Cork, Ireland

Abstract

Aim

Termination of pregnancy (TOP) became legal in Ireland in 2019, inclusive of

conditions likely to lead to the death of the fetus, making TOP for 'fatal fetal

anomaly' (FFA) an option. Trisomy 18 (T18) is the second most common lethal

trisomy, with associated major structural anomalies. We aimed to study T18

pregnancy decisions and outcomes following the legalisation of TOP for FFA.

Methods

Retrospective study of all pregnancies diagnosed with T18 from 2019-2022,

e.medicine (FM) referral databasidentified through the fetal

Results

We identified forty-seven T18 pregnancies. In 59.6% (28/47), TOP was performed,

with median gestational age (GA) at delivery of 18 weeks (range;17-22 weeks).

Parents choosing to continue pregnancy represented 17% (8/47) of the cohort, with

six women delivering at term. In 11/47 cases, intrauterine death occurred in the

second trimester (range; 13.2-18 weeks). Parents choosing TOP were referred to

fetal medicine services at a significantly earlier GA when compared to those who

chose to continue the pregnancy (14 weeks vs. 21.5 weeks, p<0.010; respectively).

Conclusions

Following a T18 diagnosis, parents may choose to continue or terminate the

pregnancy, with both care options now available in Ireland. Universal access to first-

trimester prenatal aneuploidy screening would facilitate wider and earlier parental

choice and decision-making.

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Introduction

Trisomy 18 (T18) is the second most common autosomal aneuploidy after Trisomy 21.

The main clinical features of T18 include a range of major structural anomalies,

characteristic craniofacial features, distinctive hand posture, and fetal growth

restriction

1,2

. The live birth prevalence of trisomy 18 is estimated as 1/6,000-1/10,000.

However, the overall prevalence is higher (1/2000) due to the high frequency of fetal

loss and pregnancy termination after prenatal diagnosis

3–6

. An older report from our

region (Cork, Ireland) published in 2013 on pregnancies diagnosed with T18

demonstrated the increased risk of spontaneous miscarriage, fetal death, and short

life span (median 1.5 days)

National screening programmes exist in many countries with different combinations

of maternal serum biochemical markers with or without nuchal translucency [NT]

measurement. Non-invasive prenatal testing (NIPT) is now available in over 60

countries and was shown to have higher sensitivity and specificity for T18 compared

to the traditional combined first and second-trimester screening tests. This allows

most T18 pregnancies to be prenatally diagnosed

8–10

Currently, in our settings, pregnant women are offered a first trimester visit, including

an ultrasound to date the pregnancy

with no national screening programme for

aneuploidies

. The mid-trimester anomaly scan is now available to all women in

Ireland with a new clinical guideline published to standardise care

The Eighth Amendment of the Irish Constitution prohibited legal termination of

pregnancy (TOP)

. Following an electoral referendum, legislation led to the

introduction of TOP services in 2019. Section 11 of the legislation describes TOP for a

"condition likely to lead to the death of the fetus in utero or within 28 days of birth"

, including for so-called 'fatal fetal anomaly (FFA),' a non-medical term popularised

by parent groups and the media

Under Irish law and in clinical practice, T18 is considered a FFA therefore, parents may

choose to continue or terminate the pregnancy with no upper gestational age limit. A

delay in prenatal diagnosis of T18 due to the lack of formal first trimester screening

might impact the parents' informed decisions

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Thus, in this current study, we aimed to describe parents' decisions and the outcome

of T18 pregnancies following the legalisation of TOP in the Republic of Ireland.

Methods

We performed a retrospective cohort study in a single tertiary centre between 2019

and 2022. Cases were identified through the fetal medicine referral database. The

institutional ethical review board approved this study.

All pregnancies diagnosed with T18 after referral to a Fetal Medicine specialist clinic

were included in our study. We did not include T18 cases from miscarriages when this

was diagnosed on cytogenetic testing or at a post-mortem examination. We also

excluded births where T18 was diagnosed postnatally. For the purpose of this study,

we compared cases where parents chose to continue pregnancy versus those who

chose TOP.

Computerized files eligible for inclusion were reviewed, and the following data were

obtained from the hospital's electronic maternity record: maternal demographics

characteristics, gestational age (GA) at referral to a Fetal Medicine Specialist,

indication for the referral, gestational age at diagnosis, non-invasive and invasive

genetic testing, mode of delivery, gestational age at delivery or at TOP, birthweight

and length of survival in those who were live born. The parents' decision is

documented in the electronic charts following genetic result confirmation by invasive

test, and approval for TOP requires the input of a multidisciplinary team meeting.

Categorical variables were summarized as frequency and percentage. Continuous

variables were reported as mean, standard deviation, median, and interquartile range.

The Chi-square test and Fisher's exact test were applied to compare categorical

variables, while independent samples T test or Mann-Whitney test were used to

compare continuous variables. P-value less than 0.05 was considered statistically

significant. SPSS software was used for all statistical analyses (IBM SPSS statistic for

Windows, version 28, IBM corp., Armonk, NY USA 2021).

Results

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During the study period, 28,225 births occurred at Cork University Maternity Hospital

(CUMH). Out of them, we identified 47 pregnancies diagnosed with T18 through the

Fetal Medicine database (Figure 1). The demographic characteristics of the cohort are

presented in Table 1. The average maternal age was 37.5±4.2 years, with advanced

maternal age> 35 years in 85% (40/47) of our cohort. Twenty-five percent of the

women were primiparous, and 47.9% had a previous pregnancy loss.

TOP was performed in 59.5% (28/47) pregnancies, with median GA at delivery of 18

weeks (range; 17-22 weeks). The interval from referral to fetal medicine services to

TOP was four weeks (range; 3-5 weeks). This interval encompasses the duration from

the first suspicion of a concern, the referral to the fetal medicine specialist, the process

of genetic testing and awaiting complete test results, the case presentation at the

multidisciplinary meeting, to the approval and certification of the termination of

pregnancy (TOP). Three women chose to undergo chorionic villus sampling (CVS)

at 12

weeks in another institution; others waited for amniocentesis at/after 15 weeks.

Referral indications in the TOP group were: Increased NT/ cystic hygroma in 51.8%

(14/28), high-risk NIPT in 21.4% (6/28), and structural abnormalities in 28.6% (8/28).

In our setting, TOP was performed through medical induction without fetal

monitoring.

In 23.4% (11/47) cases, parents' wishes were not documented since second-trimester

miscarriage occurred before full investigations were completed. The median GA at

diagnosis in this group was 13 weeks (range; 13-18 weeks) and GA at intrauterine fetal

death was 15 weeks (range; 14-16 weeks), with the median interval from diagnosis to

the demise of 2 weeks (range; 1-2.5 weeks

Parents deciding to continue pregnancy represented 17% (8/47) of the cohort. The

average GA at delivery was 38 weeks (range; 33-38.5 weeks), with 75% (6/8) of

pregnancies reaching term.

In Table 2, we compared women who had TOP (n=28) versus those who chose to

continue with pregnancy (n=8). The GA at referral to fetal medicine services occurred

significantly earlier in the TOP group compared to those who decided to continue with

pregnancy (14 vs. 21.5 weeks, p<0.010, respectively). The indication for referral

differed significantly between the groups (p=0.034). In the group that decided to

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continue with the pregnancy, 75% were referred due to structural anomalies detected

at the anatomy ultrasound scan, compared to only 28.6% in the TOP group.

The details of the outcomes of the pregnancies delivered at term are presented in

Table 3.

Table 1- Characteristics of the 47 study participants

Data presented as mean ± standard deviation (SD), or number (rate) as appropriate.

BMI- body mass index; ART- assisted reproductive technology; FMS- fetal medicine

specialist; GA- gestational age; NT- nuchal translucency; NIPT- non-invasive prenatal

testing.

37.5±4.2

Maternal age (years)

40/47 (85.1)

Advanced maternal age>35 years old, n (%)

12/47 (25.5)

Primiparity, n (%)

25.9±3.2

BMI

23/47 (48.9)

Previous pregnancy loss, n (%)

1/47 (2.1)

Smoking, n (%)

2/47 (4.2)

ART, n (%)

14.5 (13-20.5)

GA at 1

FMS clinic (weeks)

Indications for referral

24/47 (51)

Increased NT/ cystic hygroma

8/47 (17)

High Risk NIPT

15/47 (31.9)

Structural abnormalities

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Table 2- Comparison between women choosing to continue pregnancy and women

choosing termination of pregnancy

Data presented as mean ± standard deviation (SD), or median (interquartile range) or

n (%) as appropriate. TOP- termination of pregnancy; AMA- advanced maternal age;

GA- gestational age; FMS- fetal medicine specialist.

Table 3- Outcomes of trisomy 18 term deliveries

Case

Year

Gestational

age at

referral

(weeks)

Indication for

referral

to Fetal

Medicine

Pregnancy

outcome

Neonatal

outcome

P value

Continue pregnancy

n=8

TOP

n=28

0.768

37.1±4.2

37.6±4.2

Maternal age

>.99

7 (87.5)

24 (85.7)

AMA>35 years old, n (%)

0.658

3 (37.5)

7 (25)

Primiparity, n (%)

0.422

3 (37.5)

17 (60.7)

Previous pregnancy loss, n (%)

0.010

21.5 (18.5-21.5)

14 (13-16.5)

GA at 1

FMS clinic (weeks)

0.005

6 (75)

5 (17.8)

Referred from anomaly scan

0.034

6 (75)

8 (28.6)

Structural indication for referral

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2019

22+2

Fetal growth

restriction +

Bilateral choroid

plexus cysts

Induction and

vaginal delivery at

38 weeks

Boy- 2240 grams

Neonatal death at

1 day

2019

30+5

Fetal growth

restriction +

polyhydramnios

Induction and

vaginal delivery at

38 weeks

Pregnancy induced

hypertension

Boy- 1900 grams

Intrapartum

death

2019

21+4

Multiple

anomalies

Induction and

vaginal delivery at

38 weeks

Boy- 1800 grams

Intrapartum

death

2020

22+5

Fetal growth

restriction+

cardiac anomaly

Elective Caesarean

delivery at 38

weeks

Girl- 1840 grams

Neonatal death at

6 days

2021

13+1

Cystic hygroma

(later, multiple

anomalies)

Spontaneous

breech delivery at

39 weeks

Girl- 2280 grams

Intrapartum

death

2021

19+4

Multiple

anomalies

Spontaneous

vaginal delivery at

37 weeks

Girl -1670 grams

Neonatal death at

2 days

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Discussion

In this study, we identified pregnancies diagnosed with Trisomy 18 following the

legalisation of termination of pregnancy in the Republic of Ireland. Our cohort

consisted of forty-seven pregnancies, with 59% of parents choosing TOP, while the

proportion choosing to continue pregnancy represented 17% of the cohort. Of these,

we had six-term deliveries with 50% live births. In contrast to other studies

18,19

that

reported longer lifespans but similar to our previous report

, the most extended

survival was one week.

The American College of Obstetrics and Gynecology (ACOG)

recommends that

prenatal genetic screening options be discussed and offered to all pregnant women

regardless of age or risk for chromosomal abnormality

. However, it is essential to

remember that a screening test also requires extensive pre-and post-test counselling

and informed consent from parents

. Other professional bodies and organisations

provide guidance and recommendations on topics such as patient selection, pre-test

counselling, test performance characteristics, and the integration of NIPT into existing

prenatal screening programmes

21–25

There is no national first trimester aneuploidy screening programme in Ireland

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Women in Ireland can choose to pay for private aneuploidy screening tests

, which is

an apparent inequity in access to care. Moreover, most Irish pregnant women would

choose testing if it was available

28,29

. A recent publication studied the perspectives

and awareness of pregnant women regarding prenatal screening for fetal trisomy in

Ireland. The study demonstrated that while pregnant women possess a reasonable

grasp of how to interpret screening test outcomes, they had limited awareness of the

prenatal screening choices available to them. It is important to prioritise efforts to

ensure that pregnant women receive equal and comprehensive information about

and, ideally, access to NIPT at an early stage in their pregnancy

, so they may make

an informed choice about NIPT. Some will opt out of screening, and this can be

influenced by personal beliefs, risk perceptions, access, affordability and cultural or

religious beliefs. Within our cohort, we do not have full detail on the information given

to women about screening, and whether they had had access to private funding or

declined screening.

The decision to terminate a pregnancy following a FFA diagnosis is a personal and

complex decision influenced by various factors, including the severity of the diagnosis,

cultural and religious beliefs as well as access to prenatal care and genetic diagnosis

31–

. In our study, parents who decided to continue pregnancy were diagnosed later in

gestation, primarily due to structural anomalies identified at the second-trimester

anatomy ultrasound. It is, therefore, reasonable to speculate that the relatively late

GA at diagnosis could influence parents' decisions, and it has been previously shown

how GA at diagnosis substantially affects maternal-fetal attachment

. Further, it is

important to consider the medical consequences of late gestation in cases of TOP

36,37

Our findings contrast with a similar study published recently

that presented the

outcomes of trisomy 18 in a public hospital in South Africa. In their study, no significant

difference in gestational age at diagnosis was demonstrated between those who did

and did not terminate their pregnancies (23.1±5.1 vs. 24.7±5.3 weeks; p = 0.067).

However, in their study, both groups received the diagnosis at a later GA.

A qualitative analysis of parental decision making following the diagnosis of suspected

anomaly identified that parents have a nonlinear three-phase process: "information

seeking," reflecting the way parents-to-be face the uncertainty associated with a fetal

diagnosis and related prognosis; "implications," where consideration is given to future

consequences of the decision; and "decision making," which reflects how the decision

is made (head- or heart-led)

. Another recent qualitative study

described the care

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experiences of Irish parents whose pregnancy was diagnosed with FFA (mainly trisomy

18). This study described parents' need for consistent, well

-communicated, and

comprehensive care, irrespective of decisions made, which encourages a perinatal

palliative care approach that is individual to the parents. Both studies highlight the

need to improve the communication between healthcare professionals and patients

and to allow enough time and provide adequate information for parents facing this

decision.

Our study is not without limitations. The retrospective design and our data collection

are based on cases referred to Fetal Medicine services, thus potentially under-

representing the pregnancies where a diagnosis was made only postnatally. We also

excluded cases where T18 was suspected but confirmed only postnatally. The strength

of the study is in a relatively large cohort over four years from a single referral centre,

as well as the novelty of the study looking at parental decisions following the

legalisation of TOP in Ireland.

In conclusion, following the diagnosis of trisomy 18, parents may choose to continue

or terminate the pregnancy, with both care pathways available in Ireland’s maternity

services since 2019. We should provide the choice to access universal first-trimester

prenatal screening alongside expanded early anatomy ultrasound for all pregnant

women to facilitate wider and earlier parental decision-making.

Declarations of Conflicts of Interest:

None declared.

Corresponding author:

Hadas Miremberg,

Department of Obstetrics and Gynaecology,

Cork University Maternity Hospital,

Wilton,

Cork,

Ireland.

E-Mail: [email protected]

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