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. 2018 Sep 15;124(18):3715-3723.
doi: 10.1002/cncr.31641. Epub 2018 Sep 12.

Pediatric melanoma in melanoma-prone families

Alisa M Goldstein  1 , Kelsey C Stidd  1 , Xiaohong R Yang  1 , Mary C Fraser  1 , Margaret A Tucker  1
Affiliations

Pediatric melanoma in melanoma-prone families

Alisa M Goldstein et al. Cancer. .

Abstract

Background: In the United States, only approximately 0.4% of all melanomas are diagnosed in patients aged <20 years. To the authors' knowledge, melanoma in pediatric members of melanoma-prone families has not been fully investigated to date. The objective of the current study was to evaluate pediatric patients with melanoma with extensive follow-up in melanoma-prone families with and without cyclin-dependent kinase inhibitor 2A (CDKN2A) mutations.

Methods: For this non-population-based study, families were followed prospectively for up to 40 years. A total of 60 families with ≥ 3 patients with melanoma were included for analysis: 30 CDKN2A mutation-positive (CDKN2A+) and 30 CDKN2A mutation-negative (CDKN2A-) families. Age at the time of first melanoma and number of melanomas were obtained for each patient and summarized by family or sets (CDKN2A + vs CDKN2A-). For set comparisons and categorical variables (occurrence of melanoma in pediatric patients, number of melanomas, number of patients with single or multiple melanomas), the Pearson chi-square or Fisher exact test was used.

Results: Regardless of CDKN2A status, melanoma-prone families were found to have 6-fold to 28-fold higher percentages of patients with pediatric melanoma compared with the general population of patients with melanoma in the United States. Within CDKN2A + families, pediatric patients with melanoma were significantly more likely to have multiple melanomas compared with their relatives who were diagnosed at age >20 years (71% vs 38%, respectively; P = .004). CDKN2A + families had significantly higher percentages of pediatric patients with melanoma compared with CDKN2A- families (11.1% vs 2.5%; P = .004).

Conclusions: These observations have implications for the prevention of melanoma as well as clinical care for its early detection. Children in melanoma-prone families should have careful sun protection from an early age and skin surveillance to reduce their risk of melanoma.

Keywords: cyclin-dependent kinase inhibitor 2A (CDKN2A); family research; genetics; melanoma; pediatrics; sun protection.

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Conflict of interest statement

Conflict of Interest: The authors have no conflicts of interest to disclose.

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