Aljehani Wael, Antlee Shaheed, Aljohani Reeman, Tharwat Taif, Alrohaily Lujain, Aljohani Badr
Department of Dermatology, King Salman bin Abdulaziz Medical City, Madinah, SAU.
Department of General Medicine, King Fahad Hospital, Madinah, SAU.
Cureus. 2025 Mar 1;17(3):e79884. doi: 10.7759/cureus.79884. eCollection 2025 Mar.
Lymphangioma circumscriptum (LC) is a rare benign lymphatic anomaly that often manifests during childhood. We report an atypical instance of enormous hyperkeratotic LC in an older patient, augmenting the little literature on severe manifestations of this illness. This is a case of a 79-year-old woman with a medical history notable for deep vein thrombosis (DVT) and morbid obesity who presented with a developing skin lesion on her left thigh. During the clinical examination, a well-defined, verrucous lesion measuring approximately 15 cm × 2 cm and resembling a cauliflower was identified. The excisional samples obtained for histological analysis to verify the diagnosis revealed an edematous fibro-adipose stroma with dilated lymphatic channels, hyperkeratosis, and moderate papillomatosis, confirming the diagnosis of hyperkeratotic LC. To our knowledge, this is the first reported case involving LC late in life, occurring in an unusual site with gigantic proportions and being hyperkeratotic, which are all regarded as atypical features for LC. The patient, having a history of DVT and being obese, would most likely have lymphatic dysfunction, predisposing her to the development of LC. The form of hyperkeratotic LC seen in this case is even less common than the more familiar presentation of LC, which typically forms large, translucent bullae. This case highlights the different clinical presentations of LC and emphasizes that it should be regarded as a differential diagnosis for verrucous lesions, especially in older people. It also emphasizes the significance of a biopsy in validating instances with unusual manifestations, such as in older age groups, bigger lesion sizes, and atypical sites. Further research is necessary to elucidate the etiopathogenetic causes of acquired LC in adults and develop therapeutic procedures for large lesions.
局限性淋巴管瘤(LC)是一种罕见的良性淋巴管异常,常在儿童期出现。我们报告了一例老年患者的巨大角化过度性LC非典型病例,补充了关于该疾病严重表现的少量文献。这是一名79岁女性患者,有深静脉血栓形成(DVT)病史且病态肥胖,其左大腿出现了一个不断发展的皮肤病变。在临床检查中,发现了一个边界清晰、疣状的病变,大小约为15厘米×2厘米,类似菜花。为进行组织学分析以确诊而获取的切除样本显示,有一个水肿的纤维脂肪基质,伴有扩张的淋巴管、角化过度和中度乳头瘤样增生,证实为角化过度性LC。据我们所知,这是首例报道的发生在老年期的LC病例,发生在一个不寻常的部位,体积巨大且角化过度,这些都被视为LC的非典型特征。该患者有DVT病史且肥胖,很可能存在淋巴功能障碍,使其易患LC。本例中所见的角化过度性LC形式比更常见的LC表现(通常形成大的半透明水疱)更为少见。该病例突出了LC的不同临床表现,并强调应将其视为疣状病变的鉴别诊断,尤其是在老年人中。它还强调了活检在验证具有不寻常表现的病例(如老年人群、更大的病变尺寸和非典型部位)中的重要性。有必要进一步研究以阐明成人获得性LC的病因,并开发针对大病变的治疗方法。