Isolated Thoracoschisis with Rib Agenesis and Liver Herniation: A Case Report

BACKGROUND Thoracoschisis is a very rare congenital birth defect defined by the herniation of intra-abdominal organs through a defect in the thoracic wall. Though often associated with other birth defects as a part of the "limb-body wall complex" deformities, thoracoschisis has very rarely...

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Bibliographic Details
Published in:	The American journal of case reports Vol. 20; pp. 1915 - 1919
Main Authors:	Masden, Tyler B, Taela, Atenasio, da Rocha, Massada, Moores, Donald C, Radulescu, Andrei
Format:	Journal Article
Language:	English
Published:	United States International Scientific Literature, Inc 21-12-2019
Subjects:	Congenital Abnormalities - diagnostic imaging Congenital Abnormalities - surgery Hernias, Diaphragmatic, Congenital - diagnostic imaging Hernias, Diaphragmatic, Congenital - surgery Humans Infant, Newborn Liver Diseases - congenital Liver Diseases - diagnostic imaging Liver Diseases - surgery Male Rare Diseases Ribs - abnormalities Thoracic Wall - abnormalities Thoracic Wall - diagnostic imaging Thoracic Wall - surgery Tomography, X-Ray Computed
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Summary:	BACKGROUND Thoracoschisis is a very rare congenital birth defect defined by the herniation of intra-abdominal organs through a defect in the thoracic wall. Though often associated with other birth defects as a part of the "limb-body wall complex" deformities, thoracoschisis has very rarely been reported as an isolated finding. CASE REPORT Here we present the case of a 30-day-old term male infant with an isolated left thoracoschisis managed successfully by primary closure. The patient was monitored postnatally in the Neonatal Intensive Care Unit (NICU) of Maputo Central Hospital because of the presence of a herniated mass through a left chest wall defect below the left nipple. Computed tomography (CT) scans suggested the presence of a left diaphragmatic hernia, left rib agenesis, and herniation of an unidentifiable intra-abdominal organ through the anterior left chest wall. On day of life (DOL) 30, when global health outreach pediatric surgeons arrived at the hospital, the decision was made to operate on the child. The mass was found to be of liver origin, the exposed tissue was excised, and primary closure of the chest wall was accomplished. The patient's postoperative course involved a wound infection that resolved favorably with treatment, allowing for discharged home on postoperative day (POD) 17 in stable condition. CONCLUSIONS Our case report highlights the importance of recognizing this rare condition and directing appropriate surgical care.
Bibliography:	ObjectType-Case Study-2 SourceType-Scholarly Journals-1 ObjectType-Feature-4 content type line 23 ObjectType-Report-1 ObjectType-Article-3 Funds Collection Authors’ Contribution Conflict of interest: None declared Data Interpretation Literature Search Data Collection Study Design Manuscript Preparation Statistical Analysis
ISSN:	1941-5923 1941-5923
DOI:	10.12659/AJCR.919125