Nahm Walter K, Moise Sharon, Eichenfield Lawrence F, Paller Amy S, Nathanson Laura, Malicki Denise M, Friedlander Sheila Fallon
Division of Dermatology, University of California-San Diego School of Medicine, San Diego, CA 92113, USA.
J Am Acad Dermatol. 2004 May;50(5 Suppl):S101-6. doi: 10.1016/s0190-9622(03)02468-x.
Blue rubber bleb nevus syndrome (BRBNS) is a rare disorder characterized by discrete venous malformations of varying size and appearance that are present on the skin and within the gastrointestinal tract. The characteristic cutaneous lesions consist of deep-blue, soft, rubbery blebs, which are easily compressible. A serious complication is gastrointestinal bleeding. Because venous malformations were described historically as cavernous hemangiomas, the lesions of BRBNS were also inappropriately called hemangiomas in the literature. We describe 3 cases to delineate the venous malformations of BRBNS and to highlight their variable onset of presentation and progression. In one case, a venous malformation was noted during a prenatal ultrasound evaluation at 5 months gestation. The other 2 cases demonstrated a lack of the classic cutaneous lesions at birth. BRBNS consists of multiple venous malformations, rather than hemangiomas as described. Subcutaneous venous malformations may occasionally be the sole presenting finding in patients with this unusual syndrome, and may be evident even in the prenatal period.
蓝色橡皮疱痣综合征(BRBNS)是一种罕见的疾病,其特征为皮肤和胃肠道出现大小及外观各异的离散性静脉畸形。典型的皮肤损害由深蓝色、柔软、橡皮样的疱组成,易于压缩。严重的并发症是胃肠道出血。由于静脉畸形在历史上被描述为海绵状血管瘤,BRBNS的损害在文献中也被不恰当地称为血管瘤。我们描述3例病例,以描绘BRBNS的静脉畸形,并强调其表现和进展的可变起始情况。在1例病例中,妊娠5个月时产前超声检查发现静脉畸形。另外2例病例出生时未表现出典型的皮肤损害。BRBNS由多个静脉畸形组成,而非如所述的血管瘤。皮下静脉畸形偶尔可能是这种不寻常综合征患者的唯一表现,甚至在孕期就可能明显。