Familial oral lichen planus in a 3-year-old boy: a case report with eight years of follow-up
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CASE REPORT
Familial oral lichen planus in a 3-year-old boy: a case report with eight years of follow-up Fang Wang1,2, Ya‑Qin Tan1,2, Jing Zhang1,2 and Gang Zhou1,2*
Abstract Background: Oral lichen planus (OLP) is a chronic mucocutaneous disease characterized by adult predominance and a prolonged course. However, it is rare in the pediatric population with familial aggregation. Case presentation: A 3-year-old boy presented with pain and irritation on the oral mucosa while contacting spicy food for 2 months. Oral examination showed widespread whitish reticular and papular lesions on the lips, the dor‑ sum of the tongue, and bilateral buccal mucosa, with diffuse erosions covered with pseudomembrane on the buccal mucosa. The boy’s parents were examined to exhibit white reticular and plaque-like lesions on their oral mucosa. The three patients were clinically diagnosed as affected by OLP and histopathologically confirmed. The boy underwent topical treatment with recombinant bovine basic fibroblast growth factor (rb-bFGF) gel, and oral lesions gradually resolved and healed. Neither of his parents received treatment. During the subsequent follow-ups, none of three patients underwent any medical treatment. Fortunately, their lesions had almost faded over 8 years. Conclusions: Our case emphasizes that pediatric OLP should be recorded with family history. Besides, long-term periodic follow-up is recommended in pediatric patients with OLP for monitoring any changes in lesions. Keywords: Case report, Family history, Oral lichen planus, Pediatric population, Rb-bFGF Background Oral lichen planus (OLP) is a chronic T-cell-mediated mucocutaneous disease of unknown etiology [1]. The potential triggers, with a possible role in the pathogenesis, include autoimmunity, infective agents, microorganisms, vitamin deficiencies, psychological stress, and genetic predisposition [1, 2]. OLP is clinically characterized by well-defined and slightly raised white striae in the oral mucosa. Oral lesions are categorized as reticular, atrophic, erosive, papular, plaque, and bullous forms [1]. The typical distribution is almost symmetrical, frequently affecting the buccal mucosa, tongue, and gingiva. OLP’s histopathological features are epithelial hyperparakeratosis, basal cell liquefactive degeneration, and a band-like *Correspondence: [email protected] 2 Department of Oral Medicine, School and Hospital of Stomatology, Wuhan University, Luoyu Road, Wuhan 237, China Full list of author information is available at the end of the article
lymphocytic infiltrate in the lamina propria [1, 3]. Additional microscopic findings include atrophy, acanthosis, saw-tooth rete ridges, and colloid bodies. According to the World Health Organization (WHO), OLP is defined as an oral potentially malignant disorder (OPMD) [1]. A regular follow-up is essential to screen for changes that might indicate malignant transformation in OLP lesions [2]. OLP is uncommon in pediatric patients, and the etiology of pediatric OLP has not been well established
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