Orthotopic heart transplantation in a child with histiocytoid cardiomyopathy

Zangwill, Steven; Trost, Beth A.; Zlotocha, Jane; Tweddell, James S.; Jaquiss, Robert D.B.; Berger, Stuart

doi:10.1016/j.healun.2003.07.021

Cited by 16 publications

(7 citation statements)

References 12 publications

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“…Although resection of histiocytic lesions has been tried when discrete lesions exist, heart transplant has been a common final pathway. 6 Cardiac evaluation of our patient, including echocardiography, electrocardiography, magnetic resonance imaging, cardiac catheterization, angiography, invasive electrophysiology testing, and ultimately ICD telemetry, was not diagnostic for histiocytic cardiomyopathy and did not identify a discrete abnormality that was a candidate for surgical or catheter-based intervention.…”

Section: E94 Circ Arrhythm Electrophysiolmentioning

confidence: 74%

Percutaneous, Ultrasound-Guided Stellate Ganglion Nerve Block Suppresses Recurrent Ventricular Fibrillation in an Infant Awaiting Heart Transplant

Boe

Webster

Asher

et al. 2012

Circ: Arrhythmia and Electrophysiology

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e93 e93P ercutaneous stellate ganglion nerve block has been used to treat chronic pain syndromes. It has been described for refractory ventricular arrhythmias.1,2 However, it has rarely been described in an infant. 3,4 To the best of our knowledge, this block has not been used at this age as a diagnostic challenge and therapeutic bridge to surgical left cardiac sympathetic denervation (LCSD) and cardiac transplant.This patient presented shortly after birth with orthodromic reciprocating tachycardia and ventricular preexcitation. She was treated with propranolol and discharged. Three months later, she became rigid and cyanotic at home, requiring cardiopulmonary resuscitation, cardioversion, and amiodarone for ventricular tachycardia (VT). She underwent electrophysiology study with successful ablation of her accessory connection, but in the following days she developed recurrent ventricular fibrillation. Magnetic resonance imaging revealed asymmetric left ventricular hypertrophy, otherwise normal anatomy and no focal lesions; catheterization was nondiagnostic. An epicardial implantable cardioverter-defibrillator (ICD) was placed 13 days after presentation, and she was discharged home on amiodarone and propranolol.At 7 months of age, she had 28 appropriate shocks for monomorphic VT, polymorphic VT, and ventricular fibrillation over 3 days, despite amiodarone and lidocaine drips. She was transferred to our facility for a transplant evaluation. She was listed for heart transplantation 1 day after arrival. We prolonged her ICD detection to allow spontaneous termination, but she continued to have appropriate shocks 2 to 3 times/day. Esmolol, fosphenytoin, and verapamil were tried, but 3 days after presentation she was still having intractable arrhythmia.A percutaneous ultrasound-guided left-sided stellate ganglion block was performed to test the hypothesis that sympathetic innervation to the heart might be contributing to the intractable arrhythmia. In the cardiac intensive care unit setting, the patient was placed on nasal canula in the supine position, and intravenous sedation with midazolam was administered. Under sterile conditions (gloves, gel, sheath), ultrasound imaging (Sonosite M-Turbo, L25 × 13-6 MHz transducer) was used to identify the anatomy of the lower neck, showing the left stellate ganglion in its typical position surrounded by pretracheal fascia overlying the longus colli muscle (Figure 1). A 27-gauge, 1.25-inch needle was inserted lateral to the left stellate ganglion, and a single dose of 2 mL of 0.2% ropivacaine was injected after negative aspiration. Within 20 minutes of injection, she was predominantly in normal sinus rhythm with occasional ventricular premature beats ( Figure 2) and had transient ipsilateral Horner syndrome (Figure 3). The block was repeated daily; she maintained sinus rhythm with <1% ventricular ectopy. Over the next 10 days, mexiletine and propranolol were tried to obtain control of her rhythm with oral medications. The transition to oral medications was unsuccessful because o...

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Section: E94 Circ Arrhythm Electrophysiolmentioning

confidence: 74%

Percutaneous, Ultrasound-Guided Stellate Ganglion Nerve Block Suppresses Recurrent Ventricular Fibrillation in an Infant Awaiting Heart Transplant

Boe

Webster

Asher

et al. 2012

Circ: Arrhythmia and Electrophysiology

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“…8 In some cases, it can also present as heart failure requiring heart transplantation. 9 HC is also known to be associated with left ventricular noncompaction 10 and rarely endocardial fibroelastosis. 11 In one case series of 6 patients who suffered SCD due to HC, the cascade of lethal arrhythmias was preceded by fever, similar to our patient.…”

Section: Discussionmentioning

confidence: 99%

A Unique Case of Sudden Cardiac Death in an Infant

Samuel

Fountain

Start

et al. 2018

Clin Pediatr (Phila)

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“…Chest X‐ray revealed cardiomegaly, prompting echocardiogram that revealed dilated cardiomyopathy with LVNC with an LV ejection fraction of 25%. After initiation of milrinone therapy, she was transferred to LPCH pediatric intensive care unit for heart transplant evaluation .…”

Section: Casementioning

confidence: 99%

Orthotopic heart transplantation in two infants with histiocytoid cardiomyopathy and left ventricular non‐compaction

Siehr

Bernstein

Yeh

et al. 2013

Pediatric Transplantation

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HC is a rare cause of congestive heart failure that typically presents with malignant ventricular arrhythmias in infants, often requiring urgent intervention. Successful heart transplantation in a patient with HC has only been reported once (J Heart Lung Transplant 2004: 23: 902). The combination of HC with concurrent LVNC has only been described three times (Int J Legal Med 2009: 123: 47; Hum Pathol 2005: 36: 403; Pediatr Dev Pathol 2012: 15: 397). We report two rare cases of HC with LVNC in two infants presenting with cardiogenic shock, one requiring ECMO support who was successfully bridged to orthotopic heart transplantation with a Berlin Heart LVAD.

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Orthotopic heart transplantation in a child with histiocytoid cardiomyopathy

Cited by 16 publications

References 12 publications

Percutaneous, Ultrasound-Guided Stellate Ganglion Nerve Block Suppresses Recurrent Ventricular Fibrillation in an Infant Awaiting Heart Transplant

Percutaneous, Ultrasound-Guided Stellate Ganglion Nerve Block Suppresses Recurrent Ventricular Fibrillation in an Infant Awaiting Heart Transplant

A Unique Case of Sudden Cardiac Death in an Infant

Orthotopic heart transplantation in two infants with histiocytoid cardiomyopathy and left ventricular non‐compaction

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