A case of adult patent ductus arteriosus‐associated infective endarteritis who was successfully treated with only antibiotics

Abstract

Patent ductus arteriosus‐associated infective endarteritis (PDA‐IE) is an extremely rare complication of PDA in recent years. In this report, we describe a case of PDA‐IE complicated by septic pulmonary embolism who was successfully treated with only antibiotics.

INTRODUCTION

The ductus arteriosus is a structure necessary for fetal circulation which closes after birth via an increase in arterial oxygen concentration and a decrease in circulating prostaglandin E2 concentration. Patent ductus arteriosus (PDA) occurs when it does not close properly and completely. Next to atrial septal defect and ventricular septal defect, PDA is one of the major adult congenital heart disease (ACHD).¹

PDA-associated infective endarteritis (PDA-IE) is one of the serious complications of PDA, and an extremely high mortality rate (approximately 45%) was reported in the 1960s.² In recent years, however, it has become extremely rare due to the significant progress made in surgical and transcatheter closure procedures in combination with the wide use of prophylactic antibiotics in the settings of dental/oral care. In this case report, we describe a case of adult PDA-IE complicated by a septic pulmonary embolism that arose from bacterial vegetation in the PDA between the aorta and pulmonary artery. This patient was treated solely with antibiotics.

CASE PRESENTATION

A 33-year-old woman presented to our outpatient clinic with a prolonged persistent fever of 38–39°C, fatigue, and headache lasting for 2 months. She had a history of PDA, detected during adolescence, which had not been followed by any medical facility. Echocardiography performed in another institute before surgery for an ovarian cyst 4 years prior to this admission did not detect PDA. Upon admission, she presented with sinus tachycardia (110 beats per minute), normal blood pressure (110/56 mmHg), and elevated body temperature (37.9°C), but without oxygen desaturation. Physical examination revealed conjunctival pallor, a moderate continuous murmur on her second left intercostal space of the sternum, poor oral hygiene, and no obvious signs of infective endocarditis such as Osler's nodes and Janeway lesions. Laboratory analysis showed microcytic anemia (Hb 9.3 g/dL), leukocytosis (WBC 9600/μL) with neutrophil dominance of 89.3% and elevated inflammatory markers, an erythrocyte sedimentation rate of 114 mm/h, and C-reactive protein of 8.95 mg/dL. An electrocardiogram revealed sinus tachycardia with inverted T waves in precordial leads, and chest X-rays showed no abnormal signs except slightly increased pulmonary vasculature. Blood cultures revealed Streptococcus oralis with time to positivity of 32 h. Since the antibiotic sensitivity test indicated ceftriaxone was effective, it was started at 2 g daily on the day of admission (Day 0) and was followed by quick defervescence of the fever and improvement of the symptoms within 2 days. Transthoracic echocardiography (TTE) showed preserved left ventricular (LV) function, normal LV size, normal heart valves, and normal tricuspid regurgitation pressure gradient (21 mmHg). Moreover, it revealed no clear signs of vegetation formation. However, in contrast, transesophageal echocardiography (TEE) on Day 3 after initiation of the antibiotic showed blood flow from the descending aorta toward the pulmonary artery suggesting PDA (Figure 1A) (video S1: Blood flow towards the pulmonary artery by transesophageal echocardiography). Furthermore, TEE revealed a multifocal mobile structure 26 × 7 mm in size on the PDA edge on the side of the pulmonary artery indicating a bacterial vegetation (Figure 1C) (video S2: 3D transesophageal echocardiography visualizing septic vegetation). Based on the presence of vegetation with PDA and bacteremia, a diagnosis of PDA-IE was made. Repeated blood culture tests were performed on Days 3 and 6, and they remained negative. However, on Day 6, she experienced a second fever peak (39.2°C) accompanied by left lower back pain, without significant deterioration of her vital signs. Contrast-enhanced computed tomography (CT) showed contrast defects, suggesting septic embolisms in the branches of the posteromedial trunk of the left pulmonary artery (Figure 2). She had been afebrile and asymptomatic since Day 11. Moreover, on Day 11, a reduction in size of the vegetation (17 mm cord-like structure) was demonstrated by a second follow-up TEE (Figure 1D). Following antibiotic treatment for 4 weeks, a third follow-up TEE on Day 33 demonstrated the vegetation had completely disappeared (Figure 1E). A contrast-enhanced 3D-CT on Day 38 visualized the PDA connecting to the aorta via the left interlobular pulmonary artery (Figure 1B). The patient completed antibiotic treatment for 6 weeks without signs of relapse of the PDA-IE and surgical closure of the PDA is pending indefinitely as the patient does not wish to be performed.

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DISCUSSION

The population of ACHD patients is growing at a rate of 4%–5% per year, with half a million people in Japan having CHD as adult.¹ PDA is relatively common defect, and its prevalence has been reported as around 6.5% in ACHD.¹ PDA in adults can lead to a wide range of hemodynamic abnormalities and clinical symptoms including heart failure, pulmonary hypertension, and arrythmia, particularly in cases of a large volume of left to right shunt blood flow through the ductus arteriosus. Infectious endarteritis (IE), one of the most serious complications of PDA, was previously more common,² although it has become extremely rare in recent years (0.24 per 1000 person-years).³ PDA-IE is caused by blood flow turbulence in association with shunt flow through PDA and can occur regardless of its size. It is often difficult to diagnose PDA-IE using TTE, the initial method of diagnosis, as in this case, diagnostic accuracy of TEE is higher than TTE. Similar to the present case, the location of vegetation in PDA-IE has been reported to be inside of pulmonary arteries, which is accordingly prone to induce pulmonary embolism.

Bloodstream infections of streptococci are major causes of infective endocarditis but streptococci are heterogeneous group of bacteria with more than 50 identified species.⁴ Previous study revealed that Streptococcus mutans are most frequent (47.9%) and Streptococcus oralis, detected in the present case, are found in 19.4% in patients with streptococcal infective endocarditis.⁵ Streptococcus oralis, an alpha-hemolytic streptococcus, is a member of the normal human oral microbiota, feasible of opportunistic pathogenicity.⁶ Oral streptococcal infective endocarditis, can also result from not only invasive dental procedure but also daily dental activities. Therefore, oral hygiene, particularly in adult individuals with unclosed PDA, is extremely important and prophylactic antibiotics is needed when performing invasive dental procedure.

Current guidelines recommend the surgical or transcatheter closure of PDA, when left ventricular volume overload is present due to a large amount of shunt flow through the ductus arteriosus.⁷ However, there remains a controversy regarding the closure of “silent” PDA without significant hemodynamic alterations.⁷ With respect to PDA-IE, any closure procedure of PDA solely for its primary prevention has not been recommended.⁸ Moreover, PDA closure for secondary prevention of PDA-IE has been described only sporadically,⁹ but not systematically studied.⁸ Therefore, despite being infrequent, the accumulation of adult PDA-IE cases is important for establishing evidence concerning the optimal treatment in its secondary prevention. In the majority of reported cases thus far, PDA has been surgically closed by ligation or open-heart surgery.⁹ In the present case, surgical closure should be considered in the future, although the patient was successfully treated using only antibiotics and the bacterial vegetation that was complicated by pulmonary embolism was completely diminished. Surgical closure is now pending due to the patient's request.

CONCLUSION

We experienced a 33-year-old woman suffering from persistent fever in combination with continuous murmur who was ultimately diagnosed as PDA-IE by TEE. The patient was successfully treated with only antibiotic therapy. In adult patients with persistent fever, with a history of PDA in particular, IE should be taken in consideration as a complication and early TEE should be performed for appropriate diagnosis.

AUTHOR CONTRIBUTIONS

Akira Kanai: Writing – original draft. Yuichi Chikata: Supervision; writing – review and editing. Hiroshi Iwata: Supervision; writing – review and editing. Tomohi Ajima: Writing – review and editing. Tomohiro Kaneko: Writing – review and editing. Azusa Murata: Writing – review and editing. Nobuyuki Kagiyama: Writing – review and editing. Yoshifumi Fukushima: Writing – review and editing. Sakiko Miyazaki: Writing – review and editing. Tohru Minamino: Supervision; writing – review and editing.

ACKNOWLEDGEMENTS

None.

FUNDING INFORMATION

This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

CONFLICT OF INTEREST STATEMENT

The authors declare that there is no conflict of interest.

DATA AVAILABILITY STATEMENT

The data are not available for public access because of patient privacy concerns but are available from the corresponding author upon reasonable request.

CONSENT

Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.

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