Neonatal cardiogenic shock revealing obstructive cardiac Hibernoma: case report.


Journal

Journal of cardiothoracic surgery
ISSN: 1749-8090
Titre abrégé: J Cardiothorac Surg
Pays: England
ID NLM: 101265113

Informations de publication

Date de publication:
04 Aug 2021
Historique:
received: 03 05 2021
accepted: 09 07 2021
entrez: 5 8 2021
pubmed: 6 8 2021
medline: 6 10 2021
Statut: epublish

Résumé

Cardiac Hibernomas are very rare benign tumors and usually remain asymptomatic. Neonatal cardiogenic shock due to cardiac tumors is extremely very rare. Until this date a few cases of cardiac hibernoma have been reported in the literature. Transthoracic echocardiography help in the differential diagnosis, but the definitive diagnosis is histological. The management strategy is not clearly codified. The Aim is to report and discuss the clinical features of a cardiac Hibernoma and review the relevant literature. We describe a case of a 2-day-old Caucasian full-term male neonate admitted in neonate intensive care with cardiogenic shock, having fluid resuscitation and inotropic drugs. Ventilatory support was started immediately with the subsequent reestablishment of normal blood pressure. Then he was transferred to the echocardiography laboratory. Transthoracic echocardiography showed two echogenic masses in the right atrium and right ventricle. The masses were extended to the pulmonary trunk. Pulmonary artery flow measurements showed the presence of pulmonary and tricuspid obstruction. Surgery was rapidly considered since the baby was hemodynamically unstable. Intraoperative evaluation showed a mass embedded in the interventricular septum that occupy the right ventricular cavity and the right atrium. The tumor involved also the chordae of the tricuspid. Partial resection was done. Tricuspid valve repair was performed by construction of new chordae from the autologous pericardium. The specimen was sent for histopathological analysis. The baby died immediately after surgery. Histological examination of the surgical specimen revealed clear multivacuolated cells filled with lipid droplets and granular intense eosinophilic cytoplasm which confirms the diagnosis of Hibernoma. Cardiac Hibernomas are rare benign tumors. The prognosis and treatment strategy is closely dependent on the location, initial clinical presentation and possible complications. The prognosis can be unfavorable if the tumor was obstructive and infiltrate the myocardium.

Sections du résumé

BACKGROUND BACKGROUND
Cardiac Hibernomas are very rare benign tumors and usually remain asymptomatic. Neonatal cardiogenic shock due to cardiac tumors is extremely very rare. Until this date a few cases of cardiac hibernoma have been reported in the literature. Transthoracic echocardiography help in the differential diagnosis, but the definitive diagnosis is histological. The management strategy is not clearly codified. The Aim is to report and discuss the clinical features of a cardiac Hibernoma and review the relevant literature.
CASE PRESENTATION METHODS
We describe a case of a 2-day-old Caucasian full-term male neonate admitted in neonate intensive care with cardiogenic shock, having fluid resuscitation and inotropic drugs. Ventilatory support was started immediately with the subsequent reestablishment of normal blood pressure. Then he was transferred to the echocardiography laboratory. Transthoracic echocardiography showed two echogenic masses in the right atrium and right ventricle. The masses were extended to the pulmonary trunk. Pulmonary artery flow measurements showed the presence of pulmonary and tricuspid obstruction. Surgery was rapidly considered since the baby was hemodynamically unstable. Intraoperative evaluation showed a mass embedded in the interventricular septum that occupy the right ventricular cavity and the right atrium. The tumor involved also the chordae of the tricuspid. Partial resection was done. Tricuspid valve repair was performed by construction of new chordae from the autologous pericardium. The specimen was sent for histopathological analysis. The baby died immediately after surgery. Histological examination of the surgical specimen revealed clear multivacuolated cells filled with lipid droplets and granular intense eosinophilic cytoplasm which confirms the diagnosis of Hibernoma.
CONCLUSION CONCLUSIONS
Cardiac Hibernomas are rare benign tumors. The prognosis and treatment strategy is closely dependent on the location, initial clinical presentation and possible complications. The prognosis can be unfavorable if the tumor was obstructive and infiltrate the myocardium.

Identifiants

pubmed: 34348738
doi: 10.1186/s13019-021-01582-z
pii: 10.1186/s13019-021-01582-z
pmc: PMC8340373
doi:

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

222

Informations de copyright

© 2021. The Author(s).

Références

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pubmed: 8253487
Pediatr Cardiol. 2004 May-Jun;25(3):252-73
pubmed: 15360117
Heart Surg Forum. 2006;9(3):E623-5
pubmed: 16753934
Radiographics. 2004 Sep-Oct;24(5):1433-66
pubmed: 15371618
Lancet Oncol. 2005 Apr;6(4):219-28
pubmed: 15811617
Histopathology. 2012 Nov;61(5):985-7
pubmed: 22747461
Interact Cardiovasc Thorac Surg. 2009 Oct;9(4):717-9
pubmed: 19596706

Auteurs

Rym Gribaa (R)

Cardiology Department, Sahloul University Hospital, Sousse, Tunisia. rym_gribaa@yahoo.fr.

Marwen Kacem (M)

Cardiology Department, Sahloul University Hospital, Sousse, Tunisia.

Sami Ouannes (S)

Cardiology Department, Sahloul University Hospital, Sousse, Tunisia.

Wiem Majdoub (W)

Anatomic and cytopathologic laboratory, Sahloul University Hospital, Sousse, Tunisia.

Houssem Thabet (H)

Cardiology Department, Sahloul University Hospital, Sousse, Tunisia.

Imen Ben Ali (IB)

Cardiology Department, Sahloul University Hospital, Sousse, Tunisia.

Aymen Elheraiche (A)

Cardiology Department, Sahloul University Hospital, Sousse, Tunisia.

Mehdi Slim (M)

Cardiology Department, Sahloul University Hospital, Sousse, Tunisia.

Sihem Hmissa (S)

Anatomic and cytopathologic laboratory, Sahloul University Hospital, Sousse, Tunisia.

Elyes Neffati (E)

Cardiology Department, Sahloul University Hospital, Sousse, Tunisia.

Taieb Cherif (T)

Cardiovascular surgery department, Sahloul University Hospital, Sousse, Tunisia.

Chokri Kortas (C)

Cardiovascular surgery department, Sahloul University Hospital, Sousse, Tunisia.

Jamli Marah (J)

Cardiovascular surgery department, Sahloul University Hospital, Sousse, Tunisia.

Sofiene Jerbi (S)

Cardiovascular surgery department, Sahloul University Hospital, Sousse, Tunisia.

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