Thoracoschisis with limb agenesis

Bhattacharyya, Nirmal C.; Gogoi, Manoj; Deuri, Pradip Kumar

doi:10.4103/0971-9261.93972

Cited by 11 publications

(4 citation statements)

References 5 publications

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“…These included limb body wall complex and cardiac defects, neither of which were present in our case [4][5][6][7]. Our case demonstrates a less severe form of isolated thoracoschisis without diaphragmatic hernia.…”

Section: Discussionmentioning

confidence: 76%

A Rare Case of Thoracoschisis

et al. 2017

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A term male baby, after delivery, was found to have a 3-centimeter beefy-red mass protruding from the left chest wall, adjacent to the left nipple. Radiological imaging suggested it's origin from the left lateral liver segment. A diagnostic laparoscopy confirmed the isolated connection to the liver, elevated left hemidiaphragm, and protrusion between the ribs. The mass was excised using electrocautery, and pathologic examination showed normal liver tissue.

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Section: Discussionmentioning

confidence: 76%

A Rare Case of Thoracoschisis

et al. 2017

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“…Current survival rates of all forms of thoracoschisis are about 66%, and are mainly dependent on the associated malformations (Table 1). Of the 5 reported cases of thoracoschisis that resulted in death, 1 was intrauterine with undiagnosed associated malformation, 3 were associated with limb malformations, and 1 was isolated [2,5,6,8,14]. For those that survived reduction of the herniated contents, no post-operational complications were noted.…”

Section: Discussionmentioning

confidence: 99%

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

et al. 2019

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Patient: Male, 30-day-oldFinal Diagnosis: ThoracoschisisSymptoms: Respiratory distressMedication: —Clinical Procedure: —Specialty: SurgeryObjective:Congenital defects/diseasesBackground: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.

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“…Van Allen et al described a LBWC deformity that consists of a poly-malformation syndrome with the presence of two out of three fetal anomalies: thoraco/abdomino-schisis associated with eventration of internal organs, craniofacial malformation and limbs deformities [5]. A literature search revealed only 11 reported cases of thoracoschisis with LBWC [1,2,[6][7][8][9][10][11][12][13][14]. Almost all had an associated diaphragmatic hernia and limb abnormality, except the case of the only male patient with the left diaphragmatic eventration [8].…”

Section: Discussionmentioning

confidence: 99%

Thoracoschisis associated with Limb Body Wall Complex

et al. 2017

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Thoracoschisis is a rare condition. A female newborn presented with right-sided thoracoschisis, associated with diaphragmatic hernia and protrusion of an accessory liver lobe through the chest wall defect along with deformity of the right forearm and hand duplication. Diagnosed as part of the limb-body wall complex (LBWC), management included resection of the exteriorized liver lobe followed by right hemidiaphragm and thoracic wall reconstruction.

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Thoracoschisis with limb agenesis

Abstract: A rare case of thoracoschisis, diaphragmatic and supraumbilical abdominal wall defect with right upper limb agenesis in a neonate is reported.

Cited by 11 publications

References 5 publications

A Rare Case of Thoracoschisis

A Rare Case of Thoracoschisis

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

Thoracoschisis associated with Limb Body Wall Complex

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