20500904, 2021, 6, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ccr3.4368 by Nat Prov Indonesia, Wiley Online Library on [01/02/2023].

See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
Received: 22 January 2021 
|  Revised: 30 April 2021 
|  Accepted: 11 May 2021

DOI: 10.1002/ccr3.4368

CASE REPORT

Successful management of fetal atrial flutter at term pregnancy

with postnatal electrocardioversion

Nnadozie Igbokwe1,2   | Aisha F. Ibrahim2  | Samy Mutalab3  | Oonagh Cleland3

1
Queens University Belfast, Belfast, UK
2
Abstract
Obstetrics and Gynaecology, Royal Jubilee
Maternity Hospital, Belfast, UK Fetal atrial flutter is a lethal tachyarrhythmia with a 10% mortality rate. Diagnosis
3
Royal Jubilee Maternity Hospital, Belfast, is made with echocardiography, and management should be multidisciplinary with
UK obstetricians, fetal cardiologists, and specialist neonatologists.

Correspondence KEYWORDS
Nnadozie Igbokwe, Royal Jubilee Maternity
obstetrics and gynaecology, cardiovascular disorders
Service Belfast, Belfast, UK.
Email: [email protected]

1  |   IN T RO D U C T ION changes. The outcome of fetal tachyarrhythmia is dependent

on the presence of hydrops and cardiac disease, and not the
Fetal atrial flutter (AF) is an uncommon condition account- type of tachyarrhythmia.4 Treatment is individualized with
ing for about 30% of all fetal tachyarrhythmias. It is associ- factors like gestational age, structural heart problems, and
ated with structural heart anomalies and hydrops, with 10% hydrops taken into significant consideration. Early detection
fetal mortality rate. This case demonstrates a successfully and treatment improve clinical outcome significantly.5
managed atrial flutter at term with postnatal electrocardio- We present a case of fetal atrial flutter in a 24-­year-­old
version using a multidisciplinary team (MDT) approach. low-­risk pregnancy at 37 weeks gestation. There were no fetal
It is a serious and threatening form of tachyarrhythmia, hydrops or associated cardiac disease, and she had an urgent
and associated with fetal mortality and neurological com- caesarean section. Neonatal sinus rhythm was restored with
plications. The incidence of fetal tachyarrhythmia in less single electrocardioversion and some course of digitalization.
than 1% of pregnancies, and the two commonest forms are
AF (10%-­ 30%) and supraventricular tachycardia (SVT)
(66%-­90%).1 2  |  CASE REPORT
Fetal AF is characterized by a rapid regular atrial con-
traction (300-­600 beats/minute).2 This makes the ventricles A 24-­year-­old primigravida booked low risk in a different
unable to respond to this rapid speed in a 1:1 fashion leading hospital. Her booking blood results were normal with hemo-
to a 2:1 or variable atrioventricular (AV) block. It commonly globin level of 144 g/L, and she had a normal anomaly scan
develops in the third trimester and should not be confused at 20 weeks gestation. She had regular uneventful antenatal
with fetal tachycardia or distress. The proposed underlying midwife-­led care and had no significant past medical history
mechanism causing fetal AF is the re-­entrant circuit caus- of note. At each clinic visit, the fetal heart rate (FHR) was
ing premature atrial impulses.3 Diagnosis is mainly by fetal normal in keeping with gestational age.
echocardiography which demonstrates the cardiac rhythm At 37 weeks gestation, she attended her routine antenatal
best with M-­mode function, while also assessing any features care, also with first episode of reduced fetal movement. There
of cardiac compromise or associated structural anomalies or was difficulty in detecting the FHR both by the hand-­held

This is an open access article under the terms of the Creative Commons Attribution NonCommercial NoDerivs License, which permits use and distribution in any medium,
provided the original work is properly cited, the use is non commercial and no modifications or adaptations are made.
© 2021 The Authors. Clinical Case Reports published by John Wiley & Sons Ltd.

Clin Case Rep. 2021;9:e04368.  |

wileyonlinelibrary.com/journal/ccr3     1 of 5
https://doi.org/10.1002/ccr3.4368
 |

20500904, 2021, 6, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ccr3.4368 by Nat Prov Indonesia, Wiley Online Library on [01/02/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
2 of 5       IGBOKWE et al.

fetal Doppler and cardiotocographic (CTG) machine as the discussion between the fetal cardiologist, on-­call neonatolo-
heartbeat was too fast. A quick bedside ultrasound scan gist, and the obstetric team. Options of management and their
showed FHR of 220 beats per minute (bpm). The maternal risks and benefits were discussed with the patient including
pulse was 100bpm with blood pressure of 128/76 mmHg. She antenatal medical treatment with drugs like digoxin and so-
was quickly transferred by ambulance to the Obstetric Unit talol, or abdominal delivery with neonatal management. The
of the nearest general hospital. A quick transabdominal ul- high VR of 240bpm and the potential risk of developing im-
trasound scan done by the Obstetric team showed live active minent hydrops were discussed. The patient was not keen on
singleton gestation with ventricular FHR of 260 bpm using medical management, and since the pregnancy was term, the
pulse wave doppler (PW), and an impression of fetal SVT team agreed on abdominal delivery and postnatal treatment
was made. The maternal temperature was normal, and other to which the patient consented. She had an urgent cesarean
noncardiogenic causes of fetal tachycardia were ruled out. section under regional block. The outcome was a live female
There was a discussion to go for emergency cesarean section; neonate who weighed 3530 g at birth with favorable Apgar
however, this was forestalled by specialist advice from the score although with ​initial poor color. The cord gases (PH)
pediatric cardiologist in a tertiary hospital who advised for were normal.
the patient to be transferred by ambulance to the University The baby required some continuous positive airway pres-
teaching hospital for specialist review and to provide ade- sure (CPAP) for support for a few minutes as the oxygen satura-
quate neonatal care after delivery. tion was 70% and was transferred immediately to the neonatal
On arrival at the tertiary hospital 2 hours later, obstetric intensive care unit (NICU). Electrocardiography (ECG) done
ultrasonography showed estimated fetal weight compatible showed ventricular heartbeat of 235bpm (Figure 1), with per-
with her gestation, with normal liquor volume and normal sistent AF. A single synchronized direct-­current cardioversion
anterior placentation. Fetal echocardiography done by the of 4 joules following ketamine sedation reverted the arrhyth-
fetal cardiologist revealed an atrial heart rate of 480bpm and mia to sinus rhythm at 175  bpm. Loading dose of digoxin
ventricular rate (VR) of 240bpm using the M-­mode func- was started (15  mcg/kg) followed later by maintenance dose
tion. There was a mild tricuspid valve regurgitation, but no of 10 mcg twice daily with serum digoxin level monitored. A
obvious cardiac structural anomaly, atrial enlargement, or neonatal echocardiography done on the day of delivery showed
hydrops noted. A clinical impression of fetal AF at 480bpm a patent foramen ovale (PFO) and mild tricuspid regurgitation
with 2:1 nodal AV block was made. There was a quick MDT with otherwise normal ventricular function.

F I G U R E 1   Postnatal ECG precardioversion showing the neonate still in Atrial flutter with the ventricular heartbeat rate of 235 bpm
 |

20500904, 2021, 6, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ccr3.4368 by Nat Prov Indonesia, Wiley Online Library on [01/02/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
IGBOKWE et al.   
    3 of 5

The baby's mother had a good postoperative recovery and features of hydrops.6 In our case, there was only mild tricus-
was well debriefed with follow-­up plan made. There was no pid regurgitation.
neonatal recurrence of AF while on admission, and on day The diagnosis of fetal AF is made by fetal echocardiog-
4 neonatal life, the baby was discharged back to the NICU raphy. Fetal magnetocardiography (fMCG) is a noninvasive
of the general hospital where she was monitored for 3 days. technique used in the diagnosis of complex tachyarrhyth-
She was then discharged home on same dose of digoxin for mia, but it is not available for routine clinical practice.2,6
prophylaxis, and the last serum digoxin level before dis- With echocardiography, both the atria and ventricles should
charge was normal at 1.5  μg/L. Follow-­up echocardiogram be clearly identified in relation to the spine and descend-
done 8  weeks after discharged showed similar findings as ing aorta as landmarks. The focus should not be to get the
before with normal ventricular function. The baby was to be VR only in fetal tachyarrhythmia using the PW button as
followed up every few months with an echocardiogram and was done in the referring hospital. With the M-­mode across
review, and there has been no concern so far. Her develop- respective atrium and ventricle, their respective rates (car-
ment has been normal up to the point of writing this article at diac rhythm) are measured which gives the variable con-
the age of 4 months. duction pattern also. It is very important to make the right
diagnosis before proceeding to treatment especially in low
resource setting or general hospitals without pediatric car-
3  |   D IS C U SSION diologist services. In our case, the initial diagnosis was
thought to be fetal SVT before the patient was referred.
Fetal heart rate monitoring remains an important part of an- This was because of inadequate assessment with no proper
tenatal care, and as seen in our case, the tachycardia was first echocardiography done. This is one of the major reasons
detected by routine fetal heart rate check.5 Fetal AF accounts of writing this report for educational purposes. It is known
for roughly 25-­30 of all fetal tachyarrhythmias and is asso- that up to 20% of fetal tachyarrhythmia is associated with
ciated with variable AV conduction. There are congenital cardiac anomaly, hence the need to also involve a pediatric
structural anomalies that may occur with AF including hy- cardiologist to perform a thorough fetal echocardiography
poplastic left heart syndrome, atrioventricular septal defect, before deciding on management option.3,8 This informs the
pulmonary atresia, and Ebstein's anomaly. Both SVT and need of MDT management with neonatologists, pediatric
fetal AF have similar incidence of hydrops fetalis averaging cardiologist, and obstetricians. At term pregnancies, the
40% and similar overall mortality rate of 10%. The mortality babies should not just be delivered based on fetal distress
rate in fetal AF with hydrops, however, may be up to 30%. without these considerations, as the outcomes may be un-
Studies have shown that hydropic fetuses with fetal AF have toward. This is well exemplified in this case.
higher VR than the nonhydropic ones, but no difference in Based on the points highlighted above, the antenatal man-
the atrial rates.6 agement of fetal AF depends on several factors including
Most cases of fetal AF occur in the third trimester as seen fetal gestational age, presence of fetal hydrops or features of
in our case, with the median age presentation of 32 weeks, al- heart failure, and associated structural heart disease. The risk
though it can occur at midgestation too. The pathophysiology of hydrops in fetal AF is said to be more generally with a
of fetal AF is believed to be due to intra-­atrial macroreentry VR of over 210 bpm, and more rapid onset of hydrops with
circuit which the atria develop a critical capacity to support VR of over 230bpm. There was no hydrops in our case even
at 27-­30 weeks, hence the onset of AF in the third trimester.4 though the ventricular heart rate was 240 bpm.6 The develop-
The ventricles are protected during fetal AF by the AV node ment of associated cardiac anomaly is associated with poorer
which is not part of the intra-­atrial re-­entrant circuit. This is neonatal outcome, hence the advantage of early detection and
achieved by variably blocking the AV conduction, with 2:1 treatment. The major aim of treatment is the prevention or
AV block present in over 80% of patients with fetal AF.4,7 resolution of hydrops either by VR control or conversion to
This finding is in keeping with our patient who had 2:1 block. sinus rhythm. Although prenatal treatment of fetal AF with
Persistent tachyarrhythmia may result in fetal hydrops or transplacental antiarrhythmic medications is the most com-
heart failure, and fetal hydrops develops when the VR above mon documented method of treatment, this is not always
230bpm lasts over 12  hours. There was a potential risk of the case.9 At term or late preterm gestation, delivery of the
hydrops in our case with the VR of 240bpm, and this was fetus with postnatal treatment is often considered as a better
taken into consideration in decision-­making. Early features choice.3,10 This obviates the adverse effects of the medica-
of cardiac compromise include atrioventricular valvular re- tion on the mother and the risk of transplacental treatment on
gurgitation and bilateral atrial enlargement, while decreased the fetus. This explains the basis of our postnatal treatment
systolic function and cardiomegaly indicate later changes. modality.
The findings of subcutaneous oedema, pulmonary effu- The commonly used antiarrhythmic drugs include digoxin
sion, pericardial effusion, and ascites are echocardiographic as first choice and sotalol, flecainide, amiodarone, verapamil,
 |

20500904, 2021, 6, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ccr3.4368 by Nat Prov Indonesia, Wiley Online Library on [01/02/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
4 of 5       IGBOKWE et al.

F I G U R E 2   Postnatal ECG postcardioversion with sinus rhythm achieved with ventricular rate of 176 bpm

procainamide as second choice.2,11 Studies show that there optimal outcome. Postnatal cardioversion is a successful way
is a significantly better response to sinus rhythm prenatally of achieving sinus rhythm, and antiarrhythmic prophylaxis
when digoxin is used in nonhydropic fetuses (80%), com- is often necessary especially for the neonatal period. It is es-
pared with 43% in hydropic fetuses.8,12 sential that fetal AF is not managed as “fetal distress” by gen-
It is important to remember that spontaneous conver- eral obstetricians and midwives. Cases should be managed in
sion to sinus rhythm sometimes happen in fetal AF post- centers with pediatric cardiologist expertise.
natally, although this was not observed in our case.13 The
ECG done postnatally confirmed the neonate was still in ACKNOWLEDGMENTS
AF (Figure  1), hence the use of synchronized cardiover- Special acknowledgment to the patient for writing a very
sion which successfully resulted in sinus rhythm (Figure 2). detailed patient perspective and giving consent for the case
Digoxin was continued as an antiarrhythmic prophylaxis. publication. Published with written consent of the patient.
After sinus rhythm is achieved postnatally, one may monitor
to see whether there is recurrence with AF before institut- CONFLICT OF INTEREST
ing prophylaxis, or electively treat for 6 months to 1 year.10 No conflict of interest declared.
However, the risk of AF recurrence is very rare beyond the
neonatal period. In our case, digoxin was given for the first AUTHOR CONTRIBUTIONS
28 days and to be continued for the first 6 months at least Dr Nnadozie Igbokwe, the lead author: wrote the manuscript,
with follow-­up. did literature review, got patient perspective and consent,
and did the final editing. Dr Aisha F Ibrahim: summarized
the clinical case notes and edited the final script. Dr Samy
4  |   CO NC LU S ION Mutalab: contributed to the discussion and key message. Dr
Oonagh Cleland: suggested the case report and approved the
Fetal AF is a serious and threatening fetal tachyarrhythmia final manuscript.
with an associated general mortality rate of 10%. Adequate
diagnosis, awareness of association including fetal hydrops ETHICAL APPROVAL
and cardiac anomaly, and MDT involvement often ensure A written informed consent was obtained from the patient.
 |

20500904, 2021, 6, Downloaded from https://onlinelibrary.wiley.com/doi/10.1002/ccr3.4368 by Nat Prov Indonesia, Wiley Online Library on [01/02/2023]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
IGBOKWE et al.   
    5 of 5

DATA AVAILABILIT Y STATEMENT 7. Miyoshi T, Maeno Y, Sago H, et al. Antenatal antiarrhythmic treat-
All data used and references available online. ment for fetal tachyarrhythmias: a study protocol for a prospective
multicentre trial. BMJ Open. 2017;7(8):e016597.
8. Krapp M, Kohl T, Simpson JM, et al. Review of diagnosis, treat-
ORCID ment, and outcome of fetal atrial flutter compared with supraven-
Nnadozie Igbokwe  https://orcid. tricular tachycardia. Heart. 2003;89(8):913-­917.
org/0000-0002-7799-6120 9. Wu T-­H, Huang L-­C, Ho M, et al. Fetal atrial flutter: a case re-
port and experience of sotalol treatment. Taiwan J Obstet Gynecol.
R E F E R E NC E S 2006;45(1):79-­82.
1. Choi SH, Lee KH, Sohn CS, et al. A case of fetal atrial flutter with 10. Lisowski LA, Verheijen PM, Benatar AA, et al. Atrial flutter in the
hydrops fetalis. J Korean Pediatr Soc. 1993;36(8):1165-­1170. perinatal age group: diagnosis, management and outcome. J Am
2. Maeno Y, Hirose A, Kanbe T, et al. Fetal arrhythmia: prena- Coll Cardiol. 2000;35(3):771-­777.
tal diagnosis and perinatal management. J Obstet Gynaecol Res. 11. Takei K, Morikawa M, Cho K, et al. Resolution of tachyarrhythmia-­
2009;35(4):623-­629. related fetal hydrops after corticosteroid administration for fetal
3. Rauf M, Sevil E, Ayse B, et al. A case of fetal atrial flutter treated lung maturation. Case Rep. 2015;2015:bcr2015211948.
successfully by cardioversion in the postnatal period. Biomed Res. 12. Api O, Carvalho JS. Fetal dysrhythmias. Best Pract Res Clin Obstet
2017;28(7). Gynaecol. 2008;22(1):31-­48.
4. Wacker-­Gussmann A, Strasburger JF, Srinivasan S, Cuneo BF, 13. Sivakumar S. Atrial flutter in preterm babies. Arch Dis Child -­
Lutter W, Wakai RT. Fetal atrial flutter: electrophysiology and Fetal Neonatal Ed. 2004;89(6):F564.
associations with rhythms involving an accessory pathway. J Am
Heart Assoc. 2016;5(6):e003673.
5. Sengheiser CJ, Channer KC. Recurrent atrial flutter and fibrillation How to cite this article: Igbokwe N, Ibrahim AF,
in pregnancy. Case Rep. 2011;2011:bcr1220103589. Mutalab S, Cleland O. Successful management of
6. Wójtowicz-­ Marzec M, Wysokińska B, Respondek-­ Liberska M. fetal atrial flutter at term pregnancy with postnatal
Successful treatment of neonatal atrial flutter by synchronized electrocardioversion. Clin Case Rep. 2021;9:e04368.
cardioversion: case report and literature review. BMC Pediatr. https://doi.org/10.1002/ccr3.4368
2020;20(1):370.