CASE REPORT Open Access

Mitral regurgitation detected during theintraoperative period after atrial septaldefect closure: a case reportJoo Hyun Jun1, Min-Kyung Kang2, Joon-Sang Hyeon1, Eunha Choi1, Youngrok Kim1, Ki Seok Kim3,Mi Hwa Chung1 and In-Jung Jun1*

Abstract

Background: Atrial septal defect (ASD) is a congenital cardiac defect often diagnosed in adult patients. Mitralregurgitation (MR) observed in ASD patients mostly improves after ASD closure. However, a subset of adult ASDpatients present new-onset MR or aggravation of preexisting MR after ASD closure. Intraoperative MR aggravationafter surgical ASD closure is a rare occurrence which has not been reported in the literature to date.

Case presentation: A 54-year-old woman was referred to our center due to large secundum ASD with a diameterof 17 mm which was incidentally detected on pre-operative echocardiography at a local clinic. Surgical repair ofASD under mini-thoracotomy was performed. After completion of the operation, intra-operative transesophagealechocardiography showed newly developed Grade II MR which subsequently deteriorated to severe level onpostoperative day 3. Because the patient was asymptomatic, we decided to observe closely and treat conservativelywith diuretics. Thereafter, echocardiography was evaluated on postoperative day 10 and MR disappeared to triviallevel.

Conclusions: Intraoperative MR aggravation is a rare complication following ASD closure. The possibility of MRaggravation should be evaluated in all ASD patients prior to surgery. This case highlights the importance of mitralleaflet examination after ASD closure for early detection of MR.

Keywords: Mitral regurgitation, Atrial septal defect, Echocardiography

BackgroundAtrial septal defect (ASD) is a congenital cardiac defectoften diagnosed in adult patients [1]. Mild mitral regur-gitation (MR) associated with ASD is often improvedafter shunt closure which reduces right ventricular vol-ume overload and restores ventricular septal configur-ation [2]. However, a subset of adult ASD patients withcoexisting structural changes in the heart chamberspresent new-onset MR or aggravation of preexisting MRafter ASD closure [3]. There are several reports of post-operative MR aggravation after ASD closure [3–5], butto the best of our knowledge, intraoperative detection ofincreased MR has not been reported previously. Here,

we evaluate a rare case of MR aggravation which was de-tected during the intraoperative period directly afterASD closure.

Case presentationA 54-year-old woman was referred to our center due toright ventricular enlargement which was incidentally de-tected on pre-operative echocardiography for ankle sur-gery at a local clinic. The patient was asymptomatic andin normal sinus rhythm. A transthoracic echocardiog-raphy (TTE) showed a large secundum ASD with adiameter of 17 mm. A transesophageal echocardiography(TEE) was performed and showed 20 × 23 mm secun-dum ASD with left to right shunt and right ventricle(RV), right atrium (RA) enlargement (Fig. 1a). The pa-tient had a D-shaped small left ventricle (LV) with a leftventricular ejection fraction of 59%. Mitral valve leaflets

© The Author(s). 2019, Corrected publication: September 2019. Open Access This article is distributed under the terms of theCreative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permitsunrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s)and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative CommonsPublic Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available inthis article, unless otherwise stated.

* Correspondence: christine17@hanmail.net1Department of Anesthesiology and Pain Medicine, Kangnam Sacred HeartHospital, Hallym University College of Medicine, 948-1, Daerim 1-dong,Yeongdeungpo-gu, Seoul 150-950, South KoreaFull list of author information is available at the end of the article

Jun et al. Journal of Cardiothoracic Surgery (2019) 14:140 https://doi.org/10.1186/s13019-019-0964-1

were normal with no MR detected (Fig. 1b). Moderatetricuspid regurgitation (Grade II) due to dilated tricuspidvalve annulus (46 mm) and mild pulmonary hyperten-sion were observed. The rims to both sides of the super-ior vena cava and inferior vena cava were short, thussurgical repair of ASD under mini-thoracotomy wasplanned.In the operating room, standard vital signs (pulse ox-

imetry, end-tidal carbon dioxide, electrocardiogram, andnon-invasive blood pressure) were monitored. The leftradial artery was catheterized for continuous arterialblood pressure monitoring. After 3 min of 100% pre-oxy-genation, general anesthesia was induced withmidazolam (3 mg) followed by continuous infusion ofpropofol with remifentanil, and bolus administration ofrocuronium (50 mg). The patient was intubated with a35 Fr left-sided double-lumen tube for one-lung ventila-tion. A central venous catheter was inserted via the rightsubclavian vein because the right internal jugular veinwas reserved for superior vena cava cannulation for car-diopulmonary bypass (CPB). A TEE probe was insertedto permit close observation.Right anterolateral mini-thoracotomy was done via 4th

Intercostal space. Following full anticoagulation withheparin given at a dose of 300 IU/kg, CPB was institutedusing femoral artery, femoral vein and right internaljugular vein cannulation. Next, the aortic Detachable

Glauber clamp (Cardiomedical GmbH, Germany) wasdeployed for aortic cross-clamp, and 2000mL of Custo-diol® HTK (Koehler Chemie, Bensheim, Germany) solu-tion was infused through aortic root cannula formyocardial protection. Moderate Hypothermia of 31.5 °Cwas permitted as measured by nasopharyngeal and rectalprobes. Subsequently, right atrium was opened and ASDwas closed with a trimmed bovine pericardial patch. Tri-cuspid ring annuloplasty and right atrium reductionplasty were also conducted. After completion of the op-eration, right atrium was closed and CPB was weaned.Intra-operative TEE showed that ASD was closed

with no remnant inter-atrial shunt. There was no tri-cuspid regurgitation and left ventricular ejection frac-tion was 55%. Newly developed Grade II MR withend-diastolic rightward deviated inter-ventricularseptum was detected which was not found in pre-op-erative echocardiography (Fig. 1c, d). We notified thesurgeon of the newly developed MR. Because no ab-normal findings, such as mitral valve prolapse, perfor-ation, or chordae rupture, were observed in the mitralvalve leaflets, it was determined that the surgeryshould proceed. No further adverse surgical eventsoccurred throughout the remainder of the surgicalprocedure. The surgery lasted for 345 min with theCPB time of 190 min and aortic cross-clamp time of140 min. The estimated blood loss of 800 ml. After

Fig. 1 Transesophageal echocardiography was evaluated before (a,b) and after (c,d) ASD closure. (a) Preoperative D-shaped small left ventriclewith dilated right ventricle. (b) No mitral regurgitation noted. (c) Postoperative rightward deviated ventricular septum with dilated mitral valveannulus. (d) Newly developed mitral regurgitation after atrial septal defect closure

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surgery, the patient was transferred to the IntensiveCare Unit. Bilateral lung haziness due to acute MRwas observed in the immediate post-operative chestx-ray. Otherwise, the vital signs were stable withoutcomplaint of any symptoms. The patient was extu-bated after 3 h on arrival of the intensive care unitand transferred to general ward on postoperative day(POD) 1.Transesophageal echocardiography on POD 3 con-

firmed that the ASD patch was intact without shuntflow or remnant tricuspid regurgitation. Both left andright ventricular function was well preserved with leftventricular ejection fraction of 69%. However, LV dia-stolic dysfunction (E/E’ = 26) and aggravated pulmon-ary hypertension which was not observed in thepreoperative TEE was found. The MR was shown tohave deteriorated to severe level without evidence of vege-tation or chordae rupture (Fig. 2). Because the patient wasasymptomatic, conservative treatment using diuretics andclose monitoring was determined to be the best course ofaction. Daily follow-up chest x-ray showed gradual im-provement in pulmonary edema. On POD 6, the patientwas discharged and attend follow-up outpatient appoint-ments. On POD 10, TTE was evaluated. MR disappearedto trivial level and the LV chamber size and deviatedseptum became normalized (Fig. 3).

Discussion and conclusionsMitral regurgitation is observed in less than 10% of adultASD patients caused by mitral valve prolapse related to

longstanding structural changes of the LV secondary toRV volume overload [1]. Generally, ASD closure im-proves MR by reducing the RV volume [4]. However, de-terioration of mitral valve competence is reported inaround 10% of patients undergoing ASD closure [3–5].Previous studies have suggested several mechanisms of

aggravation of MR [3, 4]. Intrinsic MV abnormality andsubsequent poor coaptation of MV may have beenmasked by the abnormal septal configuration in ASD pa-tients [6]. Also, perioperative atrial fibrillation is relatedto aggravation of MR [1]. Atrial fibrillation frequentlycauses left atrium (LA) enlargement and atrial fibrilla-tion patients with normal LA size often have impairedLA function [7, 8]. With LA enlargement or LA impair-ment, LA posterior wall extends beyond LV and resultsin poor coaptation of mitral valve [1, 9].Our patient had no mitral lesion and presented nor-

mal electrocardiography. Thus, the mechanism of in-creased MR of the present case is assumed aslongstanding adaptation of MV annulus geometry tothe abnormal septal configuration and failure of adap-tation to the cardiac geometric changes after ASDclosure [10]. ASD shunt closure increases leftventricular volume and leftward deviated interventric-ular septum becomes normal [11]. Although mitralconfiguration normalizes in most of the patients, fail-ure of mitral competence to acutely improved LVgeometry after ASD closure may cause worsening ofMR [10]. In a subset of adult ASD patients withunderlying age-dependent decline of LV compliance,

Fig. 2 Transthoracic echocardiography showed that mitral regurgitation was deteriorated to a severe level on postoperative day 3 (Effectiveregurgitation orifice area 0.57 cm2)

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the possibility of failure of mitral competence to acutelyimproved LV geometry becomes higher [2, 12].Preoperative echocardiographic features are useful

in predicting postoperative MR. Risk factors for MRinclude D-shaped LV, large ASD, small LV and RVhorizontal diameter to vertical diameter ratio, abnor-mal shape of mitral leaflets [1, 2, 4]. Also, larger pul-monary to systemic flow ratio (Qp/Qs) and high peaktricuspid regurgitation velocity have been identified asrisk factors for MR [13]. As our patient had preopera-tive echocardiographic features of D-shaped LV, largeASD, and small LV horizontal diameter to verticaldiameter ratio, the risk of postoperative MR afterASD was high.The present case is unique in that MR aggravation

was detected during the intraoperative period directlyafter ASD closure and the MV apparatus quicklyadapted LV geometry by POD 10. We believe that in-traoperative MR aggravation after surgical ASD clos-ure is a rare occurrence which, to the best of ourknowledge, has not been reported in the literature todate. Cardiac remodeling usually begins immediatelyafter ASD closure and continues to steadily improveover subsequent months [14, 15]. MR worsening isgenerally detected within 5 years in 50–62.5% of pa-tients and the rest in more than 5 years after ASDclosure [1]. Surgical closure, compared to catheterclosure, may have detrimental effect on diastolicfunction in the early postoperative period from use ofCPB which may result in MR worsening [14]. Weexcluded myocardial stunning as a factor of postoperative

mitral regurgitation aggravation because both left andright ventricular function was well preserved withoutneed of inotropic support. We assumed that acute LVvolume loading after ASD shunt closure in the preex-isting D-shaped small LV configuration influenced thedevelopment of severe MR. Moreover, acute diastolicdysfunction appear to have contributed in MR aggra-vation by increasing LV filling pressure [16]. Acutecompensation may have been possible because the pa-tient did not have left sided heart failure and mitralleaflets were normal. Thus, LV compliance improvedpromptly inducing normalization of cardiac chamberswith postoperative supportive care only.For patients at high-risk of MR deterioration,

prophylactic mitral valve annuloplasty may beconsidered [4]. We decided to observe closely andtreat conservatively with diuretics instead of conduct-ing mitral annuloplasty. We reached the decision be-cause mitral leaflets were normal and we believedthat reopening the heart chamber after the mini-thoracotomy placed an unnecessary burden on thepatient. Mitral leaflet examination through intraopera-tive echocardiography is crucial for early detection ofMR. The American College of Cardiology/AmericanHeart Association guidelines for the management ofadults with congenital heart disease recommend clin-ical and transesophageal echocardiographic follow-upafter ASD closure at 24 h, 1 month, 6 months, and 1year and at regular intervals there after [17]. If severesymptomatic MR develops with mitral leaflet motionabnormality, surgery may be advised.

Fig. 3 On postoperative day 10, transthoracic echocardiography showed that mitral regurgitation disappeared

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AbbreviationsASD: atrial septal defect; CPB: cardiopulmonary bypass; LA: left atrium; LV: leftventricle; MR: mitral regurgitation; POD: postoperative day; RA: right atrium;RV: right ventricle; TEE: transesophageal echocardiography; TTE: transthoracicechocardiography

AcknowledgementsNot applicable.

Available data and materialsNot applicable.

Authors’ contributionsIJJ wrote the manuscript. JJ, MKK participated in manuscript drafting,discussion of the case and revision of the manuscript. JSH participated inmanuscript drafting. EC, YK, KSK, MHC contributed in revision of themanuscript. All authors read and approved the final manuscript.

FundingNone.

Ethics approval and consent to participateNot applicable.

Consent for publicationWritten informed consent for publication of the clinical details was obtainedfrom the patient. A copy of the consent is available for review on request.

Competing interestsThe authors declare that they have no competing interests.

Author details1Department of Anesthesiology and Pain Medicine, Kangnam Sacred HeartHospital, Hallym University College of Medicine, 948-1, Daerim 1-dong,Yeongdeungpo-gu, Seoul 150-950, South Korea. 2Department of Cardiology,Kangnam Sacred Heart Hospital, Hallym University Medical Center, Seoul,South Korea. 3Department of Anesthesiology and Pain Medicine, Fine PainClinic, Seochogu, Seoul, South Korea.

Received: 22 May 2019 Accepted: 15 July 2019

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