Tsaqilah Laila, Nastiti Hedwika Advina, Usman Hermin Aminah, Avriyanti Erda, Dharmadji Hartati Purbo, Hidayah Risa Miliawati Nurul
Department of Dermatology and Venereology, Faculty of Medicine, Universitas Padjadjaran - Dr. Hasan Sadikin General Hospital, Bandung, West Java, Indonesia.
Department of Anatomical Pathology, Faculty of Medicine, Universitas Padjadjaran - Dr. Hasan Sadikin General Hospital, Bandung, West Java, Indonesia.
Clin Cosmet Investig Dermatol. 2024 Aug 23;17:1895-1904. doi: 10.2147/CCID.S468181. eCollection 2024.
Familial Reactive Perforating Collagenosis (FRPC) is a very rare form of benign dermatosis frequently presented during early childhood and not associated with systemic diseases. Less than 50 FRPC patients have been reported in the literature. Due to the limited number of cases, the pathophysiology of this unique entity remains elusive; moreover, no standard treatment has been agreed upon. Here, we report a case of FRPC in a 20-year-old male who was presented with generalized multiple discrete papules covered with central keratotic plugs in all regions of his body, particularly in the facial area, neck, abdominal, and extensor region of the extremities for more than 7 years. Similar symptoms were acknowledged in the patient's family members. Histopathological analyses identified the crateriform shape invagination in the epidermis filled with inflammatory lymphocytes and basophilic debris and perforated by basophilic collagen bundles from the underlying dermis. Based on the clinical and histopathological findings, the patient was diagnosed with FRPC. He was treated with topical desoximetasone 0.25% cream applied 2-3 times daily. A follow-up evaluation after 4 weeks revealed a near-complete resolution of skin papules. To our knowledge, this is the first report of FRPC case from Indonesia. Unlike the majority of FRPC patients who had their disease onsets during infancy or early childhood, FRPC skin manifestations in our patient started during the adolescence period. The resolution of skin manifestations after daily application of topical desoximetasone suggests that topical corticosteroids are a potential treatment option for FRPC patients.
家族性反应性穿孔性胶原病(FRPC)是一种非常罕见的良性皮肤病,常在儿童早期出现,且与全身性疾病无关。文献报道的FRPC患者不足50例。由于病例数量有限,这种独特疾病的病理生理学仍不清楚;此外,尚未达成标准治疗方案。在此,我们报告一例20岁男性FRPC病例,该患者全身各部位,特别是面部、颈部、腹部和四肢伸侧出现泛发性多个离散丘疹,中央有角质栓,病程超过7年。患者家庭成员也有类似症状。组织病理学分析发现表皮呈火山口状内陷,充满炎性淋巴细胞和嗜碱性碎屑,并被来自真皮下层的嗜碱性胶原束贯穿。根据临床和组织病理学表现,该患者被诊断为FRPC。给予其外用0.25%地索奈德乳膏,每日涂抹2 - 3次。4周后的随访评估显示皮肤丘疹几乎完全消退。据我们所知,这是印度尼西亚首例FRPC病例报告。与大多数在婴儿期或儿童早期发病的FRPC患者不同,我们的患者FRPC皮肤表现始于青春期。每日外用糖皮质激素后皮肤表现消退,提示外用糖皮质激素是FRPC患者的一种潜在治疗选择。