Myocarditis, flail tricuspid valve, and normal rhythm: an exceptional form of neonatal cardiac lupus

González-López, María-Teresa; Pérez-Caballero, Ramón; Gil-Jaurena, Juan-Miguel

doi:10.1017/s1047951117000543

Cited by 4 publications

(19 citation statements)

References 7 publications

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“…Despite the paucity of cases reported, there is some evidence that the most important histopathological finding is the presence of severe atrophy with replacement of the myocytes by fibrosis and dystrophic calcifications of the papillary muscle. 3 In our case, there is no evidence to connect the rupture of the TV with maternal anti-Ro due to the absence of diagnostic histological findings. Hence, a definitive causal association could not be established.…”

Section: Discussioncontrasting

confidence: 53%

“…González 3 described the case of a neonate born to a mother with anti-SSA/Ro and anti-SSB-La antibodies, who underwent surgical repair of the TV at two months of age, when a polytetrafluoroethylene neochord was placed in the anterior leaflet and attached to the papillary muscle. Fleming 4 reported the case of a pregnant woman with positive anti-SSA/Ro whose fetus presented CHB and fibroelastosis.…”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Neonatal Rupture of the Tricuspid Valve and Maternal Lupus: Is There a Correlation?

Bellón

Brunet-Garcia

Toledo

et al. 2021

World J Pediatr Congenit Heart Surg

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Valve dysfunction is not widely recognized as a feature in newborns born to mothers with positive anti-Ro/SSA antibodies, and only scarce reports have suggested an association between rupture of the atrioventricular valve tensor apparatus and these maternal antibodies. We report the follow-up from fetal life to the time of postnatal surgery of a patient with severe tricuspid regurgitation due to a flail of the anterior tricuspid valve leaflet who was born to an anti-Ro/SSA antibodies positive mother.

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Section: Discussioncontrasting

confidence: 53%

Section: Discussionmentioning

confidence: 99%

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Neonatal Rupture of the Tricuspid Valve and Maternal Lupus: Is There a Correlation?

Bellón

Brunet-Garcia

Toledo

et al. 2021

World J Pediatr Congenit Heart Surg

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“…Four articles reported patients with ruptured papillary muscles related to premature constriction of ductus arteriosus, which might be associated with maternal intake of nonsteroidal anti-inflammatory drugs (NSAIDs), structural cardiac lesions such as tetralogy of Fallot, or spontaneous occurrence ( 11 – 14 ). In patients with systemic lupus erythematosus (SLE), the immune reaction within the myocardium extended to the subvalvular apparatus, caused fetal discomfort and hypoxic insult, and finally led to excessive fiber tension, papillary muscle rupture, and critical valve regurgitation ( 1 ). In our case, there was not any evidence of perinatal asphyxia, infection, or maternal intake of NSAIDs.…”

Section: Discussionmentioning

confidence: 99%

“…Severe tricuspid regurgitation (TR) secondary to rupture of the papillary muscles or chordae tendineae in newborns, a rare but potentially lethal condition, is characterized by refractory hypoxemia with typical echocardiography (Echo) findings, including severe TR, compromised antegrade pulmonary flow, and flailed leaflet of the tricuspid valve (TV) ( 1 , 2 ). Rapid deterioration of heart failure may cause death without prompt diagnosis and effective intervention.…”

Section: Introductionmentioning

confidence: 99%

Diagnosis and intervention of severe tricuspid regurgitation secondary to rupture of the chordae tendineae: A case report and literature review

Huang

Du²,

Zhang³

et al. 2023

Front. Pediatr.

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Unguarded severe tricuspid regurgitation caused by rupture of papillary muscle or chordae tendineae is rare but fatal in neonates. The experience in the management of these patients is still limited. A newborn presenting severe cyanosis after delivery was diagnosed with severe tricuspid regurgitation secondary to rupture of chordae tendineae by echocardiography (Echo), then treated by surgical reconstruction of chordae/papillary muscle connection without artificial materials. A takeaway lesson from this case is that Echo is an important method to diagnose a rupture of chordae tendineae or papillary muscle and that prompt diagnosis and timely surgery can be life-saving.

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Case Report: unexpected cause of cyanosis in an infant after acute exposure to high altitude—severe tricuspid regurgitation secondary to tricuspid valve prolapse

Cui,

Yang,

et al. 2024

Front. Cardiovasc. Med.

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BackgroundSevere tricuspid regurgitation (TR) causing cyanosis with patent foramen ovale (PFO) and right-to-left atrial shunting requires a precise diagnosis for optimal therapy. Tricuspid valve prolapse (TVP) can lead to TR and is sometimes overlooked, especially in complex cases with factors like pulmonary hypertension (PH). We present an infant with cyanosis and profound TR after high-altitude exposure, initially misattributed to PH but found to be primarily due to spontaneous chordae tendineae rupture and TVP. This case underscores the challenges in diagnosing TR-induced cyanosis.Case presentationThe 3-month-old infant rapidly developed cyanosis, hypoxemia, right atrial enlargement, severe tricuspid regurgitation (TR), and patent foramen ovale (PFO) shunting after high-altitude exposure. Although echocardiography revealed tricuspid valve prolapse (TVP), initial consideration linked TR and right-to-left shunting to pulmonary hypertension (PH) due to the temporal correlation with rapid altitude exposure. Despite hemodynamic stability and the absence of respiratory distress after respiratory support and combined PH medication therapy, the persistent hypoxemia did not reverse as expected. This treatment outcome and repeated echocardiograms reminded us that TR was primarily caused by TVP rather than PH alone. Intraoperative exploration confirmed that TVP was caused by a rupture of TV chordae tendineae and anterior papillary muscle head, and the chordae tendineae/papillary muscle connection was reconstructed. After surgery, this patient was noncyanotic with an excellent long-term prognosis, a trivial TR with normal TV function being observed echocardiographically.ConclusionsTR-induced cyanosis can be not only a consequence of PH and right-sided heart dilation but also a primary condition. Repetitive reassessment should be undertaken with caution, particularly when patients are not improving on therapy in the setting of conditions known to predisposition to secondary TR. Since TVP caused by rupture of the chordae or papillary muscles is rare but fatal in children, early diagnosis is clinically substantial to proper management and satisfactory long-term outcomes.

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Myocarditis, flail tricuspid valve, and normal rhythm: an exceptional form of neonatal cardiac lupus

Cited by 4 publications

References 7 publications

Neonatal Rupture of the Tricuspid Valve and Maternal Lupus: Is There a Correlation?

Neonatal Rupture of the Tricuspid Valve and Maternal Lupus: Is There a Correlation?

Diagnosis and intervention of severe tricuspid regurgitation secondary to rupture of the chordae tendineae: A case report and literature review

Case Report: unexpected cause of cyanosis in an infant after acute exposure to high altitude—severe tricuspid regurgitation secondary to tricuspid valve prolapse

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