Polydactyly and Risk of Cancer.

IF 1.5 3区 医学 Q3 ORTHOPEDICS
Matthew V Abola, Logan M Good, Kira L Smith, Alexander N Berk, Robert J Burkhart, Raymond W Liu, Apurva S Shah, Samir K Trehan, Aaron Daluiski
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引用次数: 0

Abstract

Introduction: Syndromic polydactyly is often associated with an increased cancer risk. However, limited data exist regarding nonsyndromic polydactyly and its associated risk of subsequent malignancy. Therefore, our purpose was to investigate differences in rates of both malignant and benign neoplasms among pediatric patients diagnosed with nonsyndromic polydactyly compared with matched controls.

Methods: The TriNetX US Collaborative Network database was queried using ICD-9 and ICD-10 codes to identify patients aged 10 and younger diagnosed with any form of polydactyly. Patients were then divided into 2 cohorts depending on their history of polydactyly. These cohorts were propensity-matched based on age, sex, race, ethnicity, and congenital malformation syndromes associated with both polydactyly and malignancy. Subsequent rates of cancer were compared between patients with and without polydactyly.

Results: A total of 9,266,353 patients aged 10 and younger were identified, of which 16,478 (0.18%) were diagnosed with polydactyly. After 1:1 propensity matching, both cohorts included 14,361 patients. Mean follow-up was 73 months for the polydactyly cohort and 78 months for the control cohort. Overall, the polydactyly cohort had a significantly increased rate of any neoplasm (3.3% vs. 2.6%) and benign neoplasms (2.6% vs. 2.2%) compared with the control cohort. There was no significant difference between rates of malignant neoplasms between cohorts. In contrast, when analyzing site-specific cancer risk between cohorts, malignant neoplasms of bone or cartilage (0% vs. 0.1%) as well as breast (0% vs. 0.1%) were significantly higher in the control cohort compared with the polydactyly cohort. However, there were no significant differences between cohorts among rates of alternative site-specific cancers, including digestive, respiratory or intrathoracic, skin, mesothelial or soft tissue, urinary tract, central nervous system (CNS), thyroid or endocrine, neuroendocrine, lymphoid, and lip, oral cavity, or pharynx. Furthermore, after stratifying follow-up time periods, no significant differences were appreciated between polydactyly and control cohorts in rates of all neoplasms, malignant neoplasms, and benign neoplasms at follow-up <1 year, between 1 and 5 years, and >5 years.

Conclusions: Nonsyndromic polydactyly seems to be associated with increased rates of neoplasms, particularly benign neoplasms. However, site-specific cancers of bone, cartilage, and breast were significantly decreased in polydactyly patients. Although our large study further investigates this complex relationship, further studies are needed to elucidate polydactyly and its cancer implications.

Level of evidence: Retrospective cohort study, level of evidence III.

多指畸形与癌症风险
导言:综合征型多指畸形通常与癌症风险增加有关。然而,关于非综合征性多指畸形及其相关恶性肿瘤风险的数据有限。因此,我们的目的是研究诊断为非综合征性多指畸形的儿童患者与匹配对照组相比,恶性和良性肿瘤发生率的差异。方法:使用ICD-9和ICD-10代码查询TriNetX美国协作网络数据库,以识别诊断为任何形式的多指畸形的10岁及以下患者。然后根据多指畸形病史将患者分为2组。这些队列根据年龄、性别、种族、民族和与多指畸形和恶性肿瘤相关的先天性畸形综合征进行倾向匹配。比较有多指畸形和无多指畸形患者的后续癌症发生率。结果:共发现9266353例10岁及以下患者,其中16478例(0.18%)诊断为多指畸形。经过1:1的倾向匹配,两个队列都包括14361例患者。多指畸形组的平均随访时间为73个月,对照组为78个月。总体而言,与对照组相比,多指畸形组任何肿瘤(3.3% vs. 2.6%)和良性肿瘤(2.6% vs. 2.2%)的发生率均显著增加。各组间恶性肿瘤发生率无显著差异。相比之下,当分析队列之间的部位特异性癌症风险时,骨或软骨恶性肿瘤(0% vs. 0.1%)以及乳房恶性肿瘤(0% vs. 0.1%)在对照队列中明显高于多指畸形队列。然而,在不同的队列中,包括消化、呼吸或胸内、皮肤、间皮或软组织、泌尿道、中枢神经系统(CNS)、甲状腺或内分泌、神经内分泌、淋巴、唇部、口腔或咽部在内的其他部位特异性癌症的发生率没有显著差异。此外,在对随访时间进行分层后,在随访5年期间,多指组和对照组在所有肿瘤、恶性肿瘤和良性肿瘤的发生率方面没有明显差异。结论:非综合征性多指畸形似乎与肿瘤发病率增加有关,尤其是良性肿瘤。然而,在多指畸形患者中,骨、软骨和乳腺癌的部位特异性癌症明显减少。虽然我们的大型研究进一步探讨了这种复杂的关系,但需要进一步的研究来阐明多指畸形及其癌症意义。证据水平:回顾性队列研究,证据水平为III。
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来源期刊
CiteScore
3.30
自引率
17.60%
发文量
512
审稿时长
6 months
期刊介绍: ​Journal of Pediatric Orthopaedics is a leading journal that focuses specifically on traumatic injuries to give you hands-on on coverage of a fast-growing field. You''ll get articles that cover everything from the nature of injury to the effects of new drug therapies; everything from recommendations for more effective surgical approaches to the latest laboratory findings.
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