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Heliyon
Heliyon
Heliyon
2405-8440
Elsevier

S2405-8440(24)12754-5
10.1016/j.heliyon.2024.e36723
e36723
Case Report
Mechanical support for the treatment of fulminant myocarditis combined with cardiac arrest and MODS in children: A case report and review
Wang Xuan tmuwangxuan1210@163.com

Zou Guangmei zouguangmei2006@126.com
⁎
Department of Cardiac Surgical Intensive Care Unit, Yantai Yuhuangding Hosptial, Qingdao University Affiliated Hospital, No. 20 of Yuhuangding East Road, Yantai, Shandong, 264000, China
⁎ Corresponding author. zouguangmei2006@126.com
22 8 2024
15 9 2024
22 8 2024
10 17 e3672320 10 2023
20 8 2024
21 8 2024
© 2024 The Authors
2024
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
A 13 years and 1 month old child was admitted to the hospital with fulminant myocarditis. After admission, the child's condition deteriorated rapidly, with a rapid drop in blood pressure, alternating malignant arrhythmias, such as ventricular tachycardia and ventricular fibrillation, and then developed rapid cardiac arrest, which lasted for 62 min. Moreover, the child developed multiple organ failure (heart, kidney, brain, lungs, liver, gastrointestinal tract, and inflammatory system) with internal environmental disturbances, indicating critical conditions. After emergency extracorporeal membrane oxygenation (ECMO) and an intra-aortic balloon pump (IABP), continuous renal replacement therapy (CRRT) was administered to dehydrate and maintain homeostasis of the internal environment. The treatment plan was adjusted in a timely manner by performing standardized and detailed comprehensive respiratory, circulatory, anti-infection, volume, anticoagulation, skin, and physical rehabilitation treatments. The patient was transferred to the ICU 15 days later. The patient was successfully discharged from the hospital and resumed normal studies and life without any neurological sequelae. This case is the first study of ECMO combined with IABP and CRRT for the treatment of fulminant myocarditis combined with cardiac arrest and multiple organ failure in children, which has not yet been reported in the literature. The duration of the cardiac arrest in this case was extremely long (62 min), and the patient had a good prognosis for the resumption of normal life and growth. This case suggests the need for early recognition of fulminant myocarditis and early initiation of devices for circulatory function support, such as ECMO and IABP. Additionally, early initiation of CRRT can assist in precise volume management, reduce cerebral edema, stabilize the internal environment, and reduce organ functional damage, which is a strong guarantee of treatment.

Keywords

Child
Fulminant myocarditis
Cardiac arrest
MODS
ECMO
IABP
CRRT
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pmc1 History

The 13 years and 1 month old female child was admitted to the Pediatrics Department at 17:50 due to dizziness, vomiting for 2 days, and fever for 1 day. The patient had experienced dizziness two days prior to hospitalization, accompanied by several episodes of long-standing and non-ejective vomiting. Fever started 1 d ago, with a recorded body temperature of 38.0 °C. She was admitted to our hospital with the diagnosis of “Upper respiratory tract infection, central nervous system infection?”. The patient had been previously healthy.

2 Physical examination

Temperature 36.6 °C, pulse rate (P) 114 times/min, respiratory rate (R) 26 times/min, blood pressure (BP) 81/52 mmHg, height 170 cm, and weight 52 kg. Clear mind, slight congestion in the pharynx, no obvious heart or lung murmur, soft abdomen without tenderness or rebound pain, cold extremities without spots, and no neurological abnormalities were noted.

3 Status and diagnosis

Supplementary examination after admission immediately: Blood gas analysis showed lactic acid 2.2 mmol/L; ALT 109 U/L; BNP 938.16 pg/ml; TnI 39393.50 pg/ml, CK-MB 65.3 ng/ml, MyO 221.4 μg/L; CRP 18.66 mg/L, PCT 0.071 ng/ml; blood routine, DIC series, and cytokines were normal. Electrocardiography revealed sinus tachycardia, intraventricular conduction block, low voltage in the limb leads, abnormal Q waves, and ST-T changes. The patient was admitted with persistent dull pain under the raphe and had a low systolic BP (70–80 mmHg). Acute myocarditis was diagnosed, and mepredrone, gamma globulin, dobutamine, and cefazoxime were administered as anti-inflammatory, anti-hypertensive, and anti-infection therapies, respectively. Five hours after admission, the patient experienced recurrent loss of consciousness and convulsions in the limbs. Considering fulminant myocarditis, cardiogenic shock, and Asperger's syndrome, the patient was transferred to the pediatric ICU at 23:55. A physical examination after transfer revealed: P 128 times/min, R 45 times/min, BP 59/44 mmHg, clear mind, and faint heart sounds. Adrenaline failed to increase BP. At 01:13 on day 2 of hospitalization, the patient experienced sudden ventricular fibrillation and was immediately treated with defibrillation, continuous chest compressions, tracheal intubation, and mechanical ventilation. Because of the persistent ventricular fibrillation, the Cardiac Surgical Department was consulted and they immediately initiated extracorporeal membrane oxygenation (ECMO)-assisted treatment with chest compressions. After the successful placement of ECMO at 02:15, the systolic blood pressure increased to 63 mmHg, and electrocardiographic monitoring demonstrated ventricular ectopic rhythm. The patient was then transferred to the cardiac surgical ICU. The patient was in critical condition due to the following reasons: 1) Circulation: Electrocardiogram monitoring showed sustained ventricular tachycardia with a heart rate of 190 beats/min (Fig. 1). Invasive arterial blood pressure monitoring revealed a flat line with no pulsatile blood flow, indicating ECMO-assisted advection. Bedside echocardiography revealed a severe reduction in cardiac systolic function with an ejection fraction (EF) of 26 %. 2) Blood test results: BNP: 1179.46 pg/ml, hsTnI: >50,000 pg/ml, CK-MB: 100.3 ng/ml, MyO: >1200 μg/L. 3) Neurological aspects: She was in a coma, accompanied by bulbar conjunctiva edema and intermittent tetanic convulsions. 4) Respiration: Pink foamy sputum was observed. Chest radiography revealed exudation in both lungs, and bedside ultrasonography showed an abundant B-line (Fig. 2), suggesting acute pulmonary edema. 5) Internal environment: Blood gas analysis: PH: 7.039, BE -21.84, HCO3-7.5 mmol/L, K+ 5.51 mmol/L, and Lac 15.7 mmol/L. These results suggested metabolic acidosis, hyperkalemia, and hyperlactemia. In terms of abdominal organs, the patient experienced oliguria with urine output less than 10 ml/h, creatinine levels 125 μmol/L, ALT 1177 U/L, and albumin 26.48 g/L. Bedside ultrasonography revealed intestinal wall edema and weak gastrointestinal peristalsis.Fig. 1 Electrocardiogram after ECMO assisted on day 2 of hospitalization.

Fig. 1

Fig. 2 Chest radiograph on day 2 of hospitalization.

Fig. 2

4 Treatment

The general treatment received by the patient included electrocardiogram monitoring, measurement of inputs and outputs, control of infusion volume and speed, and fentanyl combined with midazolam for sedation and analgesia. For nerve management, target temperature management, brain protection with an ice bag, sodium valproate for anti-epilepsy, mannitol to reduce cranial pressure, volume and electrolyte management (blood sodium at 140–145 mmol/L), colloidal supplementation (albumin, plasma) to reduce brain edema were administered. Circulatory support involved ECMO for cardiopulmonary assistance, administration of dopamine and epinephrine to enhance cardiac function, and codarone to control heart rhythm. Because of high ventricular tension, intra-aortic balloon pump (IABP) adjuvant therapy was initiated urgently on day 2 to reduce cardiac afterload. Respiratory support included mechanical ventilation with high PS and PEEP to reduce pulmonary edema, along with albumin supplementation and volume control to reduce lung exudation. Liver and kidney functions were managed with hepatoprotective drugs and albumin supplementation and continuous renal replacement therapy (CRRT) initiated on day 2 to dehydrate and maintain homeostasis of the internal environment, respectively. Anti-infection and immunotherapy measures consisted of vancomycin (0.5 g q12h) combined with cefoperazone sulbactam (2.0g q8h) for infection treatment, gamma globulin 30g/day for three days and high-dose methylprednisolone shock treatment (750 mg/day for 3 days, 500 mg/day for 2 days, 80 mg q12h for 3 days, 80 mg/day for 2 days, gradually reduced to stop), and high-dose vitamin C (5g/day) treatment.

5 Treatment effect

The treatment effect was observed in various aspects of the patient's health. Circulation: Sinus rhythm was restored on day 3 (Fig. 3), and blood pressure increased gradually. Cardiac ultrasound showed continued improvement in the left heart systolic function, with the EF reverting to 51 % on day 8. ECMO and IABP were discontinued on days 9 and 10, respectively. Respiration: The pulmonary edema resolved significantly. Tracheal intubation was removed on day 12; Neurological function: Consciousness was regained on day 8, and extremities showed progressive improvement in movement. Sodium valproate dosage was gradually reduced and eventually stopped as convulsions subsided. Abdominal organs: Creatinine levels decreased gradually, and urine volume recovered. CRRT was discontinued on day 9. The liver enzyme levels gradually returned to normal. Gastrointestinal congestion was improved with a flat and soft abdomen and normal stool passage every 1–2 days. Daily nasogastric nutrition was transferred to oral feeding on day 16, and after administering vancomycin combined with cefoperazone sulbactam for treating infection, the body temperature was regulated with decreased leukocyte and CRP levels. The patient was transferred to the ICU on day 16. Her condition was stable and she was discharged from the hospital without any complications. She has now resumed her normal life activities and academic studies.Fig. 3 Electrocardiogram on day 3 of hospitalization.

Fig. 3

6 Discussion

Fulminant myocarditis is defined as sudden and severe disseminated inflammation of the heart [1] , typically caused by viral infections [2]. It has a rapid onset and progression, leading to hemodynamic disturbances that can result in fatal ventricular arrhythmias or multiple organ or system failures. It has an extremely high early mortality rate. Fulminant myocarditis is sporadic and has been reported globally. The disease is relatively uncommon, but not rare, and exact morbidity data are currently lacking owing to diagnostic differences. The definition of the World Health Organization/International Society and Federation of Cardiology (WHO/ISFC) recommends diagnosis based on established histological (Dallas criteria), immunological, and immunohistochemical criteria [3]. However, several pediatric patients with clinical manifestations of myocarditis do not undergo Endomyocardial Biopsy (EMB), making it challenging to arrive at a definitive diagnosis that cannot be established according to the WHO/ISFC criteria. The annual incidence of acute myocarditis is approximately 0.022 % of the population [4], translating to approximately 1–22 cases per 100, 000 children [5]. Preliminary estimates indicate that fulminant myocarditis accounts for 38 % of all pediatric myocarditis cases [2,5]. Generally, patients with hemodynamic disorders have been treated with vasoactive and cardiotonic drugs, with the mortality rate reported abroad being up to 50%–70 % [6]. However, recent advancements in treatment have significantly reduced the mortality rates from >50 % [6] to <5 % [7,8] in specialized clinical centers. In 2017, the “Chinese Expert Consensus on the Diagnosis and Treatment of Fulminant Myocarditis in Chinese Adults” was published to guide clinical management [9]. Several prominent medical/cardiac centers have actively implemented and adhered to this program, resulting in a 50 %–3.7 % reduction in the inpatient mortality risk. The basic principles include (1) relieving the cardiac burden and allowing the heart to rest from extreme failure through mechanical circulatory support, including the use of IABP and ECMO; (2) reasonable immunomodulatory therapy, including the use of appropriate doses of glucocorticoids and immunoglobulin; and (3) the combined use of neuraminidase inhibitors [10].

In the last two decades, significant advancements in treating pediatric fulminant myocarditis have been achieved through updated equipment and consumables and accumulated clinical experience in ECMO therapy. Numerous domestic and international have suggested the notable effect of ECMO as an adjuvant therapy in pediatric fulminant myocarditis, with the survival rates ranging from 52.9 % to 85.7 % after ECMO treatment [[11], [12], [13], [14], [15]]. Based on domestic and international references, a Chinese expert group developed an expert consensus for the ECMO treatment of fulminant myocarditis in children in 2020 [16]. However, most children with fulminant myocarditis exhibit hemodynamic instability without cardiac arrest. Although there were a few cases of cardiac arrest, they had a short duration, and there were few reports of multiple organ failure at home and abroad. Xiaolong's team [17] reported a case of cardiogenic shock and cardiac arrest combined with multiple organ failure that was successfully resuscitated. Survival rates after cardiopulmonary resuscitation (CPR) for cardiac arrest in children have been reported to be 51.0 %–62.5 % [18,19]. Most affected children have complete or partial recovery of left ventricular function, which may take years. An outcome study from the American Pediatric Cardiomyopathy Registry in the US showed that nearly half of the patients had normal echocardiography findings three years after presentation [20]. Those who do not fully recover their heart function may develop dilated cardiomyopathy [21,22]. For prolonged CPR, a case of acute fulminant myocarditis with CPR 110 min after cardiac arrest for ECMO establishment was reported by Chew and Than [23]. The patient's neurological function was well evaluated after discharge. A similar result was observed in a child in Japan who underwent CPR for 67 min from cardiac arrest to ECMO establishment [24]. The combination of ECMO and CRRT in treating acute fulminant myocarditis in children is rarely reported internationally; only two children with cardiac arrest were treated with ECMO combined with CRRT in China [25].

The only effective treatment for fulminant myocarditis with refractory cardiogenic shock, which is often fatal, is mechanical circulatory support as a bridge to the recovery of cardiac function. Except for ECMO, the use of IABP in pediatric patients was first reported by Pollock [26] in 1981. The IABP inflates at the beginning of diastole, thus increasing diastolic pressure and deflating prior to systole, thereby reducing left ventricular (LV) afterload, which can increase coronary blood flow and cardiac output by 10–40 %. IABP is primarily associated with improved left ventricular performance. However, it may also have positive effects on right ventricular (RV) function through complex mechanisms, including increased myocardial flow in the RV and reduced vascular pressure in the left atrium, lung, and RV afterload due to unloading LV [[27]]. The ultimate goal is to increase myocardial oxygen supply and decrease myocardial oxygen demand. However, owing to the limitations of balloon size, pediatric research on the application of IABP lags far behind that in adults. Until the 1990s, many scholars attempted to apply the IABP to children with varying degrees of success [28]. Although the number of patients was small, the IABP has been successfully used to support children with acute myocarditis and cardiac trauma, which can cause cardiac failure. Compared with the 80 % survival rate of children with myocarditis supported by ECMO, the combined application of IABP can improve survival [29]. Pual et al. [30] used IABP support in two children with myocarditis, and their survival rate was 100 %. Sachdev et al. [31] reported the case of a 13-year-old child with drug-refractory cardiogenic shock who was discharged from the hospital after the timely application of an IABP with eventual improvement. To date, IABP has been reported less frequently in pediatric patients than in adults, mainly in children with low cardiac output after surgery for congenital heart disease, and to a lesser extent in children undergoing cardiac transplantation or awaiting further mechanical cardiac assistance [32]. This may be related to the increased use of ECMO and assist devices. Compared with these two technologies, the indications for IABP are too limited, and cases of IABP application in children with myocarditis are even less well-documented. In our case, the combination of ECMO with IABP to successfully resuscitate a child with fulminant myocarditis is relatively rare internationally.

In this case, COVID-19, influenza A virus, adenovirus, Mycoplasma pneumoniae, parainfluenza virus, and respiratory syncytial virus were tested. Blood culture and G/GM tests were also conducted, but the results were all negative. Unfortunately, due to the urgency of the condition and limited technology, virus detection was not comprehensive, lacking MRI, PET, or endomyocardial biopsy. Although the diagnosis of this case did not conform to the (WHO/ISFC) specified diagnosis, according to Chinese and international expert consensus [16,33], fulminant myocarditis is generally defined as an inflammatory disease of the myocardium with acute onset and severe hemodynamic disturbances. Therefore, fulminant myocarditis is more of a clinical diagnosis than a histologic or pathological diagnosis. This is because the diagnosis needs to be analyzed in conjunction with clinical manifestations, laboratory, and imaging studies. A clinical diagnosis of fulminant myocarditis can be made when there is a sudden onset of severe hemodynamic disturbances with obvious prodromal symptoms of viral infection, especially generalized malaise, poor appetite, and diffuse ventricular wall motion loss on echocardiography. Therefore, we consider that this case could be diagnosed as fulminant myocarditis even though there was no specific pathology, MRI/PET, or myocardial biopsy. After admission, the child's condition deteriorated rapidly, with a rapid drop in blood pressure, alternating malignant arrhythmias such as ventricular tachycardia and ventricular fibrillation, and then rapid cardiac arrest, which lasted for 62 min in total. Although she was transferred to the Cardiac Surgical ICU with emergency ECMO, the blood pressure remained extremely unstable, and electrocardiogram monitoring showed ventricular rhythm, which lasted for a prolonged period. Emergency IABP was used to reduce cardiac afterload. The child developed multiple organ failure (heart, kidney, brain, lungs, liver, gastrointestinal tract) with internal environmental disturbances, sustained ventricular tachycardia with prolonged periods of shock, and very poor cardiac function suggested by ultrasound. Moreover, after cardiopulmonary resuscitation, the systemic inflammatory reaction was severe with a paralyzed immune mechanism, indicating an extremely critical condition. The cardiac surgical ICU team strictly monitored the patient's vital signs and changes in the internal environment, coagulation indexes, blood glucose, and urine output. The treatment plan was timely adjusted by performing standardized and detailed comprehensive treatment of respiratory, circulatory, anti-infection, volume, anticoagulation, skin, and physical rehabilitation. The treatment was standardized through multidisciplinary cooperation including the department of pediatrics, cardiothoracic surgery, infectious diseases, clinical pharmacy, and ultrasound medicine. In this case, the patient experienced poor surgical incision healing after ECMO withdrawal. Minor bleeding was observed around the incision, and the bacterial culture of secretions was negative. The incision gradually healed after 2 months of daily sterilization, dressing changes, and application of growth factors. The poor incision healing was attributed to thick ECMO lines, lower extremity hemodynamic disorders, poor asepsis in emergencies, and fatty liquefaction of the patient's skin [34]. After 15 days of ICU treatment, the patient was successfully discharged to resume normal life and studies without any neurological sequelae. This case is the first study that reported the use of ECMO combined with IABP and CRRT for the treatment of fulminant myocarditis combined with cardiac arrest and multiple organ failure in children. There are no clear guidelines for the use of ECMO combined with IABP in pediatric fulminant myocarditis. However, the positive outcome of this case may be attributed to the appropriate application of mechanical support. Therefore, publishing more successful cases is necessary to support these findings and to compare outcomes with different mechanical supports. Notably, the duration of cardiac arrest in this case was extremely long (62 minutes), and the patient had a good prognosis for resuming normal life and growth, which far exceeded the average resuscitation time for both domestic and international pediatric cardiac arrests (44 minutes) [35].

This case highlights the importance of early recognition of fulminant myocarditis and prompt initiation of circulatory support devices. Combining ECMO and IABP may allow the heart to rest effectively and gain time for cardiac function recovery, especially in children. Early CRRT initiation can assist in precise volume management, reduce cerebral edema, stabilize the internal environment, and reduce organ function damage, ensuring effective treatment. However, further studies are required to confirm these findings.

Funding disclosure

We have no funding. All the authors have no financial interests to declare.

Code availability

The authors had access to data and analysis and that no sponsors participates in the case.

Consent for publication

All authors read, contributed, and approved the final version of the manuscript.

Ethics declarations

For ethics approval: This study was reviewed and approved by ethics committee of Yantai Yuhuangding Hospital with the approval number: 2022–5, dated Feb 24th, 2022. The patient's and her guardian's provided written informed consent for the publication of their anonymised case details and images.

Data availability statement

Data sharing is not applicable to this article as no new data were created or analyzed in this case. The information of the case can be obtained on request by contacting the corresponding author.

CRediT authorship contribution statement

Xuan Wang: Writing – original draft, Investigation, Data curation. Guangmei Zou: Writing – review & editing, Resources, Project administration, Conceptualization.

Declaration of competing interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
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