Transient Non-Autoimmune Fetal Heart Block

Breur, Johannes M.P.J.; Oudijk, Martijn A.; Stoutenbeek, P.; Visser, Gerard H.A.; Meijboom, Erik J.

doi:10.1159/000082427

Cited by 19 publications

(19 citation statements)

References 39 publications

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“…In contrast, spontaneous regression of heart block is quite rare, although it has been reported in 4 fetuses with structurally normal hearts. 14 In our series, 5 fetuses from seronegative mothers showed regression to sinus rhythm (1 with SHD). The first patient in whom remission was observed was seen 10 years ago and had 2:1 AV block alternating with complete AV block.…”

Section: Discussionmentioning

confidence: 52%

“…A possible explanation for this phenomenon is the parasympathetic dominance before 30 weeks' gestation, which could lead to AV block if the stimulation is pronounced in the AV node. 14 Several studies have proposed that treatment with fluorinated steroids, especially dexamethasone, results in benefits to the fetuses affected by seropositive antibody-mediated AV block. 9,[15][16][17][18] The most striking difference between the present study and those previously published concerns the positive impact of steroid therapy.…”

Section: Discussionmentioning

confidence: 99%

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Perinatal Outcome of Fetal Atrioventricular Block

et al. 2008

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Background-Fetal atrioventricular (AV) block is an uncommon lesion with significant mortality. Because of the rarity of this disorder, the natural course, extensive evaluation of untreated fetuses, and late follow-up remain unclear. Methods and Results-Of the 116 consecutive cases of fetal AV block studied from 1988 to 2006, only 1 was terminated, and 75% were live births. Fifty-nine cases of AV block were associated with major structural heart disease, mainly left atrial isomerism (nϭ40), with only 26% of neonatal survivors. Of the 57 fetuses with normal cardiac anatomy, 41 (72%) were positive for maternal antinuclear antibodies, and 32 of these seropositive mothers did not receive any treatment. This untreated group had live-birth and 1-year infant survival rates of 93% and 90%, respectively. Five fetuses from seronegative mothers showed regression to sinus rhythm during pregnancy. The presence of major structural heart disease, hydrops, an atrial rate Յ120 bpm, and a ventricular rate Յ55 bpm were identified as risk factors for mortality. Logistic regression analysis of the whole group showed that the presence of structural heart disease was the only independent predictor of death (PϽ0.001). Conclusions-This long-term study confirms that fetal AV block has a poor outcome when associated with structural heart disease and that spontaneous regression of AV block is possible in seronegative forms. The survival rate of Ͼ90% of our untreated patients with isolated forms of AV block raises concerns about any decision to intervene with immunosuppressive agents. (Circulation. 2008;118:1268-1275.)

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Section: Discussionmentioning

confidence: 52%

Section: Discussionmentioning

confidence: 99%

Perinatal Outcome of Fetal Atrioventricular Block

et al. 2008

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“…There are previous reports of transient and permanent second- and third-degree AVB in fetuses of mothers without clinical or laboratory evidence of connective tissue disorders and negative anti-SSA/Ro–SSB/La antibodies (Breur et al ., 2005; Brucato et al ., 2009). The authors speculated that viral infections may have played a role in some of these cases.…”

Section: Discussionmentioning

confidence: 99%

Doppler fetal mechanical PR interval prolongation with positive maternal anti‐RNP but negative SSA/Ro and SSB/La auto‐antibodies

et al. 2010

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Keywordsfetal atrio-ventriculat block; fetal Doppler; PR interval; systemic lupus erythematosus Cutaneous neonatal lupus can occur with negative SSA/Ro or SSB/La but positive non-SSA/ SSB ribonucleoprotein (RNP) maternal autoantibodies (Provost et al., 1987). No fetalcardiac manifestations, however, have been reported in association with isolated maternal RNP autoantibodies. We report the first association of transient first-degree atrioventricular block (AVB) in a fetus exposed to maternal anti-RNP in the absence of SSA/Ro-SSB/La autoantibodies. CASE REPORTA 27-year-old mother with a history of systemic lupus erythematosus (SLE) underwent an obstetric ultrasound and the 22-week fetus was noted to have hypoplasia of the middle phalanx of the fifth digit and multiple cardiac echogenic foci. The mother was diagnosed with SLE at 20 years old and had received steroids for arthritis in the past. However, for the last 2 years before this pregnancy, she was in remission and on no medications. Because of the findings at 22 weeks of gestation, she was referred for a fetal echocardiogram that demonstrated a normal heart rate with regular rhythm and 1 : 1 atrioventricular conduction. As maternal antibodies were pending, we proceeded to measure a Doppler mechanical PR interval (MPRI) and found it to be normal at 132 ms (normal 120 ± 10 ms with 99th percentile at 150 ms) (Glickstein et al., 2004). Additionally, there were three echogenic intracardiac foci: two in the left ventricle and one in the right ventricle. Otherwise, there was no tricuspid regurgitation or any other intracardiac abnormalities, and there was no pericardial effusion or other fluid collections. NIH-PA Author ManuscriptNIH-PA Author Manuscript NIH-PA Author ManuscriptAt 31 weeks of gestation, a second fetal echocardiogram demonstrated the MPRI to be 146 ms (+2 to +3 SD), and the other cardiac findings remained normal. The maternal antibodies were reported as negative for SSA/Ro and SSB/La.For confirmation, maternal sera were retested and again found to be absent in anti-SSA/Ro or anti-SSB/La reactivity. A third fetal echocardiogram at 32 weeks of gestation demonstrated prolongation of the MPRI at 156 ms (Figures 1 and 2), but otherwise normal echocardiographic findings. A third evaluation of the maternal sera was performed at the New York University School of Medicine's CLIA-approved immunology laboratory and the research laboratory of one of the authors (JPB) using recombinant proteins La48, Ro52 and Ro60 as previously described (Clancy et al., 2005). Although antibodies to all components of the SSA/Ro-SSB/La complex were confirmed to be negative; however, antibodies to RNP were confirmed in high titer (14 848 EU with a negative value <19 EU).Subsequent weekly fetal echocardiograms demonstrated that the MPRI was consistently >150 ms until 36 weeks of gestation when it normalized at 136 ms. The baby had a normal delivery. At 6 h of life, the physical exam, the electrocardiographic PR interval (104 ms), and echocardiogram were normal. The infant continues...

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“…These neonates were also found to exhibit an unstable pattern of AV block, with the blocks changing their degree and often reverting to sinus rhythm. [7] Our case exhibited a similar clinical course with an unstable pattern of AV block alternating with sinus rhythm.…”

Section: Discussionmentioning

confidence: 55%

“…[11] Spontaneous reversal of a heart block and no cardiomyopathy on follow-up has also been documented by a number of reported series on such patients. [71213]…”

Section: Discussionmentioning

confidence: 99%

Remission of congenital complete heart block without anti-Ro/La antibodies: A case report

Mitra

Sardar

et al. 2013

Ann Pediatr Card

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Anti-Ro/La negative congenital heart block (CHB) is uncommon. We report one such case of CHB, with no associated structural heart disease or maternal autoantibodies. The heart block reverted to sinus rhythm spontaneously at two weeks of age, and the patient remains in sinus rhythm at a one year followup. Whether patients with antibody negative complete heart block have a different clinical course is conjectural.

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Transient Non-Autoimmune Fetal Heart Block

Cited by 19 publications

References 39 publications

Perinatal Outcome of Fetal Atrioventricular Block

Perinatal Outcome of Fetal Atrioventricular Block

Doppler fetal mechanical PR interval prolongation with positive maternal anti‐RNP but negative SSA/Ro and SSB/La auto‐antibodies

Remission of congenital complete heart block without anti-Ro/La antibodies: A case report

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