Velasco-Oses A, Alonso-Alvaro A, Blanco-Pozo A, Nogales F F
Departments of Pathology, Hospital General Yagüe, Burgos, Spain.
Cancer. 1988 Jul 1;62(1):222-5. doi: 10.1002/1097-0142(19880701)62:1<222::aid-cncr2820620134>3.0.co;2-w.
A six-year-old girl had Ollier's disease (OD) associated with juvenile granulosa cell tumor (JGCT). The clinical manifestations were right hemicorporal deformity due to multiple skeletal enchondromas and precocious pseudopuberty. After the removal of a Stage Iai JGCT, hormonal symptoms disappeared. Neither the ovarian tumor recurred nor the enchondromas underwent sarcomatous change after a follow-up period of 7 years. A review of the literature showed five previous cases, three associated with OD and two with Mafucci's syndrome (MS). In these cases, patients were young and the ovarian tumors were homolateral to the hemicorporal side involved by enchondromatosis. Data provided from these cases emphasize the notion of a generalized mesodermal dysplasia. JGCT behave in association with the OD in its usual fashion of hormonal production and low-degree aggressiveness.
一名六岁女孩患有骨软骨瘤病(OD)并伴有青少年颗粒细胞瘤(JGCT)。临床表现为因多发性骨内生软骨瘤导致的右侧身体畸形和性早熟假性青春期。切除Ⅰa期JGCT后,激素症状消失。随访7年后,卵巢肿瘤未复发,骨内生软骨瘤也未发生肉瘤样变。文献回顾显示此前有5例病例,3例与OD相关,2例与马富西综合征(MS)相关。在这些病例中,患者均为青少年,卵巢肿瘤与发生内生软骨瘤病的身体同侧。这些病例提供的数据强调了广义中胚层发育异常的概念。JGCT与OD相关时,其激素产生方式及侵袭性较低,表现与OD通常的情况一致。