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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 5  |  Page : 279-281

A rare cause of pulmonary hemorrhage in the intraoperative period


1 Department of Pediatric Cardiac Sciences, Artemis Hospitals, Gurugram, Haryana, India
2 Medical Services, Apollo Adlux Hospitals, Ernakulum, Kerala, India
3 Department of Pediatric Cardiac Sciences, Aster Hospital, Calicut, Kerala, India
4 Department of Pediatric Cardiac Sciences, Apollo Health City, Jubilee Hills, Hyderabad, Telangana, India

Date of Submission19-May-2020
Date of Decision03-Jul-2020
Date of Acceptance18-Jul-2020
Date of Web Publication14-Sep-2020

Correspondence Address:
Dr. Anurakti Dev Singla
Department of Pediatric Cardiac Sciences, Artemis Hospitals, Sector 51, Gurugram, Haryana, India
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JPCC.JPCC_85_20

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  Abstract 


The perioperative morbidity and mortality in pediatric cardiac surgery can be due to a multitude of factors. Timely identification of the problem helps in altering the treatment strategy and improving the outcome.

Keywords: Cardiac intensive care, central venous catheter, unilateral pulmonary edema


How to cite this article:
Singla AD, Radha AS, Warrier G, Trehan M. A rare cause of pulmonary hemorrhage in the intraoperative period. J Pediatr Crit Care 2020;7:279-81

How to cite this URL:
Singla AD, Radha AS, Warrier G, Trehan M. A rare cause of pulmonary hemorrhage in the intraoperative period. J Pediatr Crit Care [serial online] 2020 [cited 2020 Sep 26];7:279-81. Available from: http://www.jpcc.org.in/text.asp?2020/7/5/279/295026




  Introduction Top


Mitral valve replacement in the younger population is increasingly being done, especially for the mitral valve affected by rheumatic heart disease. The perioperative course can be unusual sometimes, and we should be ready to explore and identify the problem.


  Case Report Top


A 12-year-old girl presented with palpitations and exertional dyspnea, New York Heart Association functional Class III.[1] She was on irregular Benzathine penicillin prophylaxis for the past 5 years after an episode of fever and arthritis. On initial examination, the patient had brisk and bounding pulses with a heart rate of 130/min with respiratory distress and tachypnea and saturation of 98% on room air. She had a hyperdynamic apical impulse felt in the 6th left intercostal space with a late systolic left parasternal thrust and palpable second heart sound. Her first heart sound was normally heard, second heart sound was widely split with a loud pulmonic component; and she had a soft Grade 3/6 pansystolic murmur at the apex, radiating to the axilla and back with a rumbling, and low-pitched flow mid-diastolic murmur. Her systemic examination was noncontributory. There was no evidence of rheumatic activity on blood investigations. On chest radiograph, she had gross cardiomegaly with pulmonary artery enlargement and pulmonary plethora. Her electrocardiogram was suggestive of sinus tachycardia with prominent left ventricular forces and left atrial enlargement. The two-dimensional echocardiogram showed rheumatic involvement of the mitral valve evident from thickened, nodular, and echogenic mitral valve leaflets with severe mitral regurgitation due to prolapse of all segments of the anterior mitral leaflet, lack of mobility of posterior mitral leaflet, and annular dilatation. She had dilated left atrium (LA), left ventricle (LV), and pulmonary artery with pulmonary arterial hypertension. She was stabilized with inhaled oxygen, decongestive therapy, and inotropic support and was planned for elective mitral valve repair (if feasible)/replacement. In the operating room, after initial preoxygenation, she was electively intubated and mechanically ventilated using narcotic-based anesthesia. Neck central line was secured using a posterior approach. After sternotomy followed by adequate heparinization, she was taken under normothermic aortobicaval cardiopulmonary bypass (CPB) with antegrade crystalloid cardioplegic arrest. As seen after left atriotomy, the valve was unsuitable for repair because of severe subvalvular pathology and fixed posterior mitral leaflet. Hence, a 25 mm St. Jude mechanical valve was used to replace the mitral valve. After coming off bypass, there was transient hypotension which did not respond to volume replacement and inotropes. At the same time, there were copious pink frothy secretions from the endotracheal tube. The LA pressure was measured directly and the mean was 5 mmHg. Transesophageal echocardiography (TEE) was done to check LV function, valve position, and function and for residual paravalvular regurgitation, which were normal and were consistent with normal LA pressure. After achieving hemodynamic stability, the patient was shifted to the intensive care unit (ICU). However, the unexplained endotracheal tube bleeding persisted. The chest radiograph revealed a good position of the endotracheal tube, neckline, and chest tubes in the expected position with diffuse haziness of the right lung [Figure 1]. Hence, the causes of unilateral pulmonary edema, and pulmonary hemorrhage were considered. Repeat TEE was done to look for pulmonary vein issues. On TEE, we realized that echo contrast was seen coming into LA, although there was no Patent foramen ovale (PFO). To our surprise, the tip of the neckline was in the right upper pulmonary vein (RUPV) and thus into LA. Due to the side holes in the canula, part of the drugs administered was entering LA and part of it seeping into the lung. This could explain unilateral haziness on chest radiograph and hypotension not responding to inotropes. The neckline was removed, bleeding was stopped, inotropes were decreased, and the patient was extubated the next day. There was a radiological improvement in 48 h [Figure 2], though it took 2 weeks for full recovery. In retrospect, the cannulation was difficult according to the anesthetist.
Figure 1: Chest radiograph taken in anteroposterior view showing unilateral haziness (right side) with neckline tip (black arrow) apparently in expected position

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Figure 2: Chest radiograph taken in anteroposterior view after neckline removal showing reduced lung plethora

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  Discussion Top


Bilateral diffuse pulmonary edema in a patient undergoing mitral valve replacement can be a manifestation of underlying left ventricular dysfunction, prosthetic valve dysfunction, residual paravalvular leak, and extensive chordal resection. LV dysfunction can be due to the inflammatory response syndrome resulting from CPB and also due to increased cross-clamp time, inadequate myocardial protection, and preoperative ventricular dilatation and dysfunction.[2],[3]

Our patient had a refractory unilateral pulmonary edema; volume replacement indeed worsened the hemodynamics. Paradoxically, the common attributable factors were absent. Being unilateral, it made us suspect injury to RUPV, which can rarely happen during LA closure. The abnormal placement of the central venous catheter was realized during a TEE evaluation to check for the RUPV status. In the review of literature of unilateral pulmonary edema (UPE) in postoperative patients, it shows that most reported cases of acute UPE have resulted from severe eccentric MR with the jet predominantly affecting RUPV, leading to a larger increase in mean capillary pressure in the right side.[4],[5]

UPE has also been reported due to variation in the pulmonary venous pressure associated with left heart failure, anomalous vascular distribution, asymmetric pulmonary perfusion due to pulmonary artery agenesis or hypoplasia, local emphysematous changes, and pulmonary inflammatory diseases. UPE following systemic-pulmonary artery shunts can be explained on the basis of preferential flow resulting from such a shunt.[6],[7] UPE can also be reexpansion edema after the rapid evacuation of pneumothorax or a presentation of acute rheumatic fever.[8] Contralateral bronchial obstruction or stenosis causes hypoxia of one lung with resultant pulmonary capillary endothelial damage and lymphatic insufficiency and cause UPE.[9] On rare occasions, compression of a pulmonary vein outlet by a myxoma or atrial wall hematoma or destruction of the pulmonary vascular bed of the right lung can cause UPE.[10] Misplacement of a central venous pressure monitoring catheter into a pulmonary artery can present with UPE as well.[11]

We tried to think about all the possible issues to conclude what went wrong. We present this case to draw attention to an uncommon cause of pulmonary edema in the intraoperative period, where the central venous line put through the jugular access inadvertently reached the RUPV. Anatomically, RUPV courses posterior to the distal end of superior vena cava (SVC) and can be cannulated if our puncture is directed toward the posterior wall of SVC. All the central lines are taken with ultrasound guidance; still, this complication can occur on rare occasions. To prevent this from happening, we should either check the echocardiography to ascertain the position of the neckline by seeing bubble contrast in the right atrium or take venous blood gas samples to be sure that it is not in RUPV. Any unexplained pulmonary edema in operating room or ICU should involve a detailed evaluation of the quantity of volume replacement, intactness of the secured lines for delivery of inotropes, a watch on the CPB time, ventricular dysfunction, residual valvar or paravalvular leak, prosthetic valve dysfunction, increased LA pressures owing to the narrowed mitral orifice or pulmonary venous issues. Logical thinking, stepwise analysis, and an open mind help in identifying uncommon causes for common problems.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Adapted from Dolgin M, Association NYH, Fox AC, Gorlin R, Levin RI, New York Heart Association. Criteria Committee. Nomenclature and Criteria for Diagnosis of Diseases of the Heart and Great Vessels. 9th ed. Boston, MA Lippincott Williams and Wilkins; 1994. Original Source: Criteria Committee, New York Heart Association, Inc. Diseases of the Heart and Blood Vessels. Nomenclature and Criteria for Diagnosis. 6th ed. Boston: Little, Brown and Co; 1964. p. 114.  Back to cited text no. 1
    
2.
Wisenbaugh T, Skudicky D, Sareli P. Prediction of outcome after valve replacement for rheumatic mitral regurgitation in the era of chordal preservation. Circulation 1994;89:191-7.  Back to cited text no. 2
    
3.
Kozik DJ, Tweddell JS. Characterizing the inflammatory response to cardiopulmonary bypass in children. Ann Thorac Surg 2006;81:S2347-54.  Back to cited text no. 3
    
4.
Gamsu G, Peters DR, Hess D, Lehman DH, Amend WJ Jr. Isolated right upper lobe pulmonary edema. West J Med 1981;135:151-4.  Back to cited text no. 4
    
5.
Roach JM, Stajduhar KC, Torrington KG. Right upper lobe pulmonary edema caused by acute mitral regurgitation. Diagnosis by transesophageal echocardiography. Chest 1993;103:1286-8.  Back to cited text no. 5
    
6.
Bahl OP, Oliver GC, Rockoff SD, Parker BM. Localized unilateral pulmonary edema: An unusual presentation of left heart failure. Chest 1971;60:277-80.  Back to cited text no. 6
    
7.
Calenoff L, Kruglik GD, Woodruff A. Unilateral pulmonary edema. Radiology 1978;126:19-24.  Back to cited text no. 7
    
8.
El-Menyar A, Al-Hroob A, Numan MT, Gendi SM, Fawzy IM. Unilateral pulmonary edema: Unusual presentation of acute rheumatic fever. Pediatr Cardiol 2005;26:700-2.  Back to cited text no. 8
    
9.
Shikhani AH, Salman SD, Melhem R. Unilateral pulmonary edema as a complication of contralateral bronchial obstruction. Laryngoscope 1987;97:748-51.  Back to cited text no. 9
    
10.
Saleh M, Miles AI, Lasser RP. Unilateral pulmonary edema in Swyer-James syndrome. Chest 1974;66:594-7.  Back to cited text no. 10
    
11.
Royal HD, Shields JB, Donati RM. Misplacement of central venous pressure catheters and unilateral pulmonary edema. Arch Intern Med 1975;135:1502-5.  Back to cited text no. 11
    


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