Colmant Caroline, Franck Deborah, Marot Liliane, Matthijs Gert, Sznajer Yves, Blomme Sandrine, Tromme Isabelle
Department of Dermatology, Cliniques Universitaires Saint-Luc, Université Catholique de Louvain, Brussels, Belgium.
Centrum Menselijke Erfelekheid, Universitaire Ziekenhuizen Leuven, Leuven, Belgium.
Dermatol Pract Concept. 2018 Jan 31;8(1):59-62. doi: 10.5826/dpc.0801a14. eCollection 2018 Jan.
LEOPARD syndrome, also known as Gorlin syndrome II, cardiocutaneous syndrome, lentiginosis profusa syndrome, Moynahan syndrome, was more recently coined as Noonan syndrome with multiple lentigines (NSML), inside the RASopathies. Historically, the acronym LEOPARD refers to the presence of distinctive clinical features such as: lentigines (L), electrocardiographic/conduction abnormalities (E), ocular hypertelorism (O), pulmonary stenosis (P), genital abnormalities (A), retardation of growth (R), and sensorineural deafness (D). This condition is identified in 85% of patients with phenotype hallmarks caused by presence a germline point mutation in PTPN11 gene. Association of melanoma to NSML seems to be rare: to our knowledge, two patients so far were reported in the literature. We herein present a patient diagnosed with LEOPARD syndrome, in whom molecular investigation confirmed the presence of the c.1403C>T mutation in exon 12 of the PTPN11 gene, who developed four superficial spreading melanomas and three atypical lentiginous hyperplasias. Three of the melanomas were achromic or hypochromic, three were in situ, and one had a Breslow index under 0.5 mm. Dermoscopic examination showed some characteristic white structures in most of the lesions, which were a signature pattern and a key for the diagnosis.
豹皮综合征,也称为戈尔林综合征II、心脏皮肤综合征、泛发性雀斑综合征、莫伊纳汉综合征,最近在RAS病范畴内被重新命名为伴有多发雀斑的努南综合征(NSML)。从历史上看,首字母缩写词LEOPARD指的是存在以下独特临床特征:雀斑(L)、心电图/传导异常(E)、眼距增宽(O)、肺动脉狭窄(P)、生殖器异常(A)、生长发育迟缓(R)和感音神经性耳聋(D)。85%具有由PTPN11基因种系点突变导致的表型特征的患者被诊断为此病。黑色素瘤与NSML的关联似乎很罕见:据我们所知,迄今为止文献中仅报道了两例患者。我们在此报告一例被诊断为豹皮综合征的患者,分子检测证实其PTPN11基因第12外显子存在c.1403C>T突变,该患者发生了4例浅表扩散性黑色素瘤和3例非典型雀斑样增生。其中3例黑色素瘤为无色素或色素减退型,3例为原位癌,1例 Breslow厚度指数小于0.5 mm。皮肤镜检查显示大多数病变中有一些特征性白色结构,这是一种标志性图案,也是诊断的关键。