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This is a Case Report study (also classified as: Literature Review).

May 6, 2026Open Access

Co‐Occurrence of Agminated Lentigines, Café‐Au‐Lait Macules, and Vitiligo: A Case Report and Literature Review

XLXinXin LeiZhejiang Chinese Medical University BXBo XieZhejiang Chinese Medical University

Key Points

This research examines the co-occurrence of agminated lentigines, café-au-lait macules, and vitiligo and its implications for potential genetic syndromes.
Presented case of an 18-year-old male with CALMs, agminated lentigines, and vitiligo.
Conducted comprehensive examinations to exclude genetic syndromes like Peutz-Jeghers, Leopard, Carney complex, and Neurofibromatosis type 1.
Followed the patient for eight years to observe the natural history of the conditions.
Patient maintained stable vitiligo during the follow-up period.
Lentigines reached a plateau in early adulthood, indicating a benign natural history.
Negative autoimmune screening points to localized immune dysregulation.

Abstract

ABSTRACT Lentigines, café‐au‐lait macules (CALMs), and vitiligo are pigmentary disorders that seldom occur together in a single individual. Their co‐occurrence may indicate underlying genetic syndromes requiring differential diagnosis. We report an 18‐year‐old male who developed CALMs at age 11, agminated lentigines at age 13, and vitiligo on the right chest six months thereafter. Comprehensive examinations excluded Peutz‐Jeghers syndrome, Leopard syndrome, Carney complex, and Neurofibromatosis type 1. Eight‐year follow‐up—the longest reported for this combination—demonstrated a benign natural history: Stable vitiligo throughout and lentigines reaching a plateau by early adulthood. Negative autoimmune screening suggests localized immune dysregulation, supporting the Immunocompromised Cutaneous District hypothesis. A staged therapeutic approach prioritizing vitiligo stabilization is recommended.

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