Pinto Christopher J, Nayyar Rajesh, Asvita Dandamudi, Chirumamilla Avinash, Patel Prachi
Medical Research, Karnataka Insititute of Medical Sciences, Hubballi, IND.
Internal Medicine, Washington University School of Medicine, Washington, USA.
Cureus. 2022 Jun 15;14(6):e25967. doi: 10.7759/cureus.25967. eCollection 2022 Jun.
Xeroderma pigmentosum (XP) is a rare autosomal recessive pathology affecting nucleotide excision repair against ultraviolet radiation. This leads to an increased predisposition to developing ophthalmological, neurological, and cutaneous conditions with an increased cell turnover. This case reports a late presentation of XP presenting with metastatic squamous cell carcinoma (SCC) and septic shock in an eight-year-old Indian male. Emergency management with IV fluid boluses and broad-spectrum antibiotics showed no improvement in vitals. Urgent surgical debridement and tumor debulking failed to improve laboratory values. Postoperative leukocytosis with fever spikes warranted the need to transfer the patient to a super-specialty oncology unit. Such an adverse presentation is commonly seen in XP-related invasive squamous cell carcinoma. Preventive management requires early identification and a multidisciplinary approach involving dermatologists, ophthalmologists, and surgeons. Late presentations revolve around control of the disease process by sharp debridement and chemotherapy with regular surveillance as the lesions tend to reoccur even after excision and chemotherapy.
着色性干皮病(XP)是一种罕见的常染色体隐性疾病,会影响针对紫外线辐射的核苷酸切除修复。这会导致患眼科、神经科和皮肤疾病的易感性增加,细胞更新加快。本病例报告了一名8岁印度男性患者,其着色性干皮病晚期表现为转移性鳞状细胞癌(SCC)和感染性休克。通过静脉推注液体和使用广谱抗生素进行紧急处理,生命体征未见改善。紧急手术清创和肿瘤减容未能改善实验室检查结果。术后白细胞增多伴发热高峰,因此有必要将患者转至超专科肿瘤病房。这种不良表现常见于与着色性干皮病相关的侵袭性鳞状细胞癌。预防性管理需要早期识别,并采用皮肤科医生、眼科医生和外科医生参与的多学科方法。晚期表现围绕通过锐利清创和化疗控制疾病进程展开,同时要定期监测,因为即使在切除和化疗后,病变仍容易复发。