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Syndactyly and Risk of Cancer: A Multi-institutional Study
Kira L Smith, MD
1, Matthew V Abola, MD
2, Logan Good, MD
3, Raymond W Liu, MD
4, Apurva S. Shah, MD, MBA
5, Samir K. Trehan, MD
6; Aaron Daluiski, MD
7(1)University Hospitals/Case Western Reserve University, Cleveland, OH, (2)Hospital for Special Surgery, New York, NY, (3)University Hospitals Cleveland Medical Center, Cleveland, OH, (4)Rainbow Babies and Children's Hospital, Cleveland, OH, (5)Children's Hospital of Philadelphia, Philadelphia, PA, (6)Orthopedic Surgery, Hospital for Special Surgery, New York, NY, (7)Hand and Upper Extremity Surgery, Hospital for Special Surgery, New York, NY
Introduction: Syndactyly is the most common upper extremity congenital condition and cases can be associated with genetic syndromes or arise sporadically (non-syndromic). The purpose of this investigation was to determine rates of both malignant and benign neoplasms among pediatric patients diagnosed with non-syndromic syndactyly compared to matched controls.
Materials and Methods: The TriNetX US Collaborative database was queried using ICD-10 codes to identify patients aged 10 years or younger diagnosed with syndactyly. Patient history of syndactyly was utilized to categorize patients into two cohorts. These cohorts were propensity-matched by age, sex, race, ethnicity, and congenital malformation syndromes. Subsequent rate of malignancy was compared between patients with and without syndactyly.
Results: A total of 7,478 patients aged 10 years or younger diagnosed with syndactyly were identified. After 1:1 propensity matching, both cohorts included 5,461 patients. The rate of benign neoplasms was significant at >5 years follow-up duration (p=0.016, 95% CI 0.001-0.005) but was not significant at 1-year or 1-5 years. Similarly, rate of any neoplasm was significant at >5 years follow-up duration (p=0.013, 95% CI 0.001-0.005) but not at 1-year or 1-5 years. Before controlling for follow-up duration, digestive neoplasms were significantly higher in the syndactyly cohort compared to the control cohort. This did not remain significant after sub-analysis by follow-up duration.
Conclusion: Non-syndromic syndactyly is associated with an increased rate of benign neoplasms and digestive malignancies. Further investigation is needed to better understand the relationship between the syndactyly phenotype and risk of malignancy.
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