Kassira Sama, Jaleel Tarannum, Pavlidakey Peter, Sami Naveed
Department of Dermatology, University of Alabama at Birmingham, EFH 414, 1530 3rd Avenue S, Birmingham, AL 35294, USA.
Case Rep Dermatol Med. 2015;2015:635481. doi: 10.1155/2015/635481. Epub 2015 Nov 30.
Objective. We report a rare case of keloidal scleroderma and provide an analysis of similar cases. Results. A 41 year-old woman presented with dark brown, indurated, exophytic nodules over the chest along with smaller hyperpigmented plaques scattered over the abdomen, with concomitant sclerodactyly. The clinical, laboratory, and pathological findings were consistent with a diagnosis of keloidal scleroderma. The patient was treated with methotrexate, resulting in reduced firmness of her plaques and no new lesions. A literature review of previously reported cases was performed using keywords including keloidal morphea, keloidal scleroderma, nodular morphea, and nodular scleroderma. In our review, the majority of patients were African American and female. 91% of cases had nodular lesions with distribution on the trunk. The majority of patients exhibited sclerodactyly and pulmonary involvement was reported in 28%1. The majority of patients were ANA positive (63%) and only 10% demonstrated anti-SCL-70 positivity. Conclusion. Keloidal scleroderma is a rare presentation, which can often be clinically confused with keloid and scar formation. Due to this being a rare variant, our knowledge of treatment options and efficacy is limited. Methotrexate could be considered as an initial treatment option for patients with progressive keloidal scleroderma.
目的。我们报告一例罕见的瘢痕疙瘩性硬皮病病例，并对类似病例进行分析。结果。一名41岁女性，胸部出现深褐色、硬结性、外生性结节，腹部散在较小的色素沉着斑，同时伴有指（趾）硬皮病。临床、实验室及病理检查结果均符合瘢痕疙瘩性硬皮病的诊断。该患者接受甲氨蝶呤治疗后，斑块硬度降低，未出现新病灶。我们使用瘢痕疙瘩性局限性硬皮病、瘢痕疙瘩性硬皮病、结节性局限性硬皮病和结节性硬皮病等关键词，对既往报道的病例进行了文献综述。在我们的综述中，大多数患者为非裔美国女性。91%的病例有结节性病变，分布于躯干。大多数患者表现为指（趾）硬皮病，28%的病例报告有肺部受累。大多数患者抗核抗体阳性（63%），仅10%的患者抗SCL - 70阳性。结论。瘢痕疙瘩性硬皮病是一种罕见的表现形式，临床上常与瘢痕疙瘩和瘢痕形成相混淆。由于这是一种罕见的变异型，我们对治疗选择和疗效的了解有限。对于进行性瘢痕疙瘩性硬皮病患者，可考虑将甲氨蝶呤作为初始治疗选择。
