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Published on: June 16, 2023
Hepatic exstrophy complicating Poland's anomaly
Mark Puder1, Arin Greene, David Mooney
1Department of Pediatric Surgery, Children's Hospital, Boston, Massachusetts 02115, USA.
Insights
Poland anomaly is a rare congenital condition affecting chest muscles and limbs. This report details a unique case involving hepatic exstrophy, highlighting the syndrome's complex presentation.
Area of Science:
- Congenital abnormalities
- Pediatric surgery
- Clinical case reports
Background:
- Poland anomaly is a rare congenital condition characterized by chest muscle underdevelopment and limb abnormalities.
- The syndrome typically involves pectoralis major and minor muscle deficiency on one side of the body.
- Associated anomalies can include syndactyly, costal aplasia, and adactyly.
Observation:
- A case report describes an infant diagnosed with Poland anomaly.
- The infant presented with a rare and severe complication: hepatic exstrophy.
- Hepatic exstrophy occurred through a full-thickness chest wall defect.
Findings:
- This case highlights the potential for severe intrathoracic and extrathoracic anomalies in Poland anomaly.
- The co-occurrence of hepatic exstrophy with Poland anomaly is exceptionally rare.
- The chest wall defect facilitated the herniation of abdominal organs.
Implications:
- This case underscores the importance of comprehensive evaluation in infants with Poland anomaly.
- Understanding the spectrum of Poland anomaly aids in surgical planning and management.
- Further research is needed to elucidate the embryological mechanisms linking these anomalies.
Abstract:
Poland's anomaly is comprised of a constellation of anomalies. To be included in the syndrome, a child must have a deficiency of the pectoralis major and minor muscles and an associated anomaly of either the ipsilateral breast or hand. Associated defects may include syndactyly osseous and cartilagenous costal aplasia and adactyly. A case of hepatic exstrophy through a full-thickness chest wall defect in an infant with Poland's anomaly is reported.
