Foetal ductus arteriosus constriction unrelated to non-steroidal anti-Inflammatory drugs: a case report and literature review

Abstract

Foetal ductus arteriosus (DA) constriction can be found in complex foetal heart malformations, but rarely as an isolated defect. Although many cases of DA constriction are usually related to Non-steroidal Anti-Inflammatory Drugs (NSAIDs) maternal intake, other causes remain without an established aetiology and are referred to as idiopathic. Recently, a wide range of risks factors or substances (polyphenol-rich foods intake, naphazoline, fluoxetine, caffeine and pesticides) showed a definitive effect upon the pathway of inflammation, causing DA constriction. We report a case of a premature DA constriction in a woman whose possible risk factor was identified in her maternal occupational exposure to solvents and a comprehensive literature review of 176 cases of NSAID-unrelated DA constriction. A 30-year-old Asian woman was referred to our institution at 33 gestational weeks and 0 days because of suspicion of premature DA constriction. The woman had no history of medication intake, including NSAIDs, alcohol, tobacco or polyphenol-rich-food consumption during pregnancy. A detailed foetal echocardiography revealed a normal cardiac anatomy with hypertrophic, hypokinetic and a dilated right ventricle due to right pressure overload, holosystolic tricuspid regurgitation, and, at the level of the DA, high systolic and diastolic velocities, indicating premature ductal restriction. The right outflow showed dilatation of the pulmonary artery with narrow DA. An urgent caesarean section was performed at 33 gestational weeks and 4 days due to worsening of DA PI and signs of right pressure overload, despite the interruption of exposure to solvents. We assume a relationship exists between premature DA constriction and a maternal occupational exposure to solvents. This hypothesis is reinforced by the presence of associated foetal malformations in in two of the patient's children. Further research is needed to confirm the role of exposure to solvents and toxic chemicals in the pathogenesis of DA constriction, also with experimental animal models. KEY MESSAGES1. Many cases of DA constriction are usually related to Nonsteroidal Anti-Inflammatory Drugs (NSAIDs) maternal intake. 2. A wide range of risks factors or substances (polyphenol-rich foods intake, naphazoline, fluoxetine, caffeine and pesticides) can cause foetal DA constriction. 3. Further investigation are needed to confirm the role of maternal exposure to solvents in the pathogenesis of DA constriction.

Antenatal ultrasound features of isolated recurrent copy number variation in 7q11.23 (Williams syndrome and 7q11.23 duplication syndrome)

“…After birth, inflation of the lungs and the consequent increase of oxygen levels lead to DA closure. PCDA can occur secondary to maternal drug exposure or be idiopathic, most of the time 30 . Only four cases of genetic PCDA were previously reported by Srinivasan et al 12 .…”

“…PCDA can occur secondary to maternal drug exposure or be idiopathic, most of the time. 30 Only four cases of genetic PCDA were previously reported by Srinivasan et al 12 They described 4 patients with a history of PCDA, one of them was diagnosed with WBS and the three other cases were diagnosed with Alagille syndrome. In several patients with 7q11.23 duplication, patent DA has been described.…”

Giant Placental Chorangioma and Severe Ductal Arch Constriction: A Case Report With a Favorable Outcome

“…To our knowledge, this represents the first case of severe DA constriction in the setting of a prenatally detected giant chorangioma. Most cases of non-congenital heart defect and non-NSAID-related DA constriction are idiopathic or associated with a polyphenol-rich diet [ 14 ]. Our patient was counseled to avoid these substances during a prior FE and voiced compliance at diagnosis.…”