Zhang Jin,Wan Naijun,Ma Lishuang,et al.Ovarian juvenile granulosa cell tumors with Ollier’s disease in children: A Case report and literature review[J].Journal of Clinical Pediatric Surgery,,22():576-581.[doi:10.3760/cma.j.cn101785-202112038-015]
Ovarian juvenile granulosa cell tumors with Ollier’s disease in children: A Case report and literature review
- Keywords:
- Ovary; Granular Cell Tumor; Enchondromatosis; Child
- Abstract:
- Objective To explore the clinical manifestations, treatments, pathological features and pathogenesis of ovarian juvenile granulosa cell tumors (JGCT) with Ollier’s disease (OD) in children.Methods From October 2019 to October 2020, clinical data were retrospectively reviewed for one case of ovarian JGCT with OD.And the relevant literature reports were retrieved through the databases of PubMed and Wanfang.Results One 4-year-old girl showed multiple skeletal deformities, bilateral breast development with chromatosis and vulvar secretion.Radiography of extremities hinted at enchondromatosis.Pelvic ultrasound and abdominal computed tomography (CT) revealed right ovarian solid mass.Wide resection of soft tissue tumor was performed in distal right femur along with artificial bone substitution and right salpingooophorectomy.Pathologic examination of bone mass confirmed enchondromatosis and right ovarian solid mass was JGCT.A c.394C>T chr2-209113113 p.R132C mutation of IDH1 gene was detected in both ovarian JGCT and enchondromatosis.During a follow-up period of 1 year, there was no sign of recurrent enchondromatosis.There was no evidence of recurrence of ovarian JGCT over 3 months.A total of 11 cases of JGCT with OD were reported.The onset age ranged 0.5 to 36 years.The major manifestations of OD are skeletal deformities and JGCT had peripheral precocious puberty, abnormal menstruation, abdominal distension and abdominal masses.Surgery was a mainstay for two diseases.The overall prognosis was decent.Conclusion Ovarian JGCT with OD is rare and it may be caused by IDH1 gene mutation.Surgery is a first option.Regular postoperative follow-ups are essential.
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Memo
收稿日期:2021-12-23。
基金项目:吴阶平医学基金会项目（320.6750.2020—06—86）
通讯作者:万乃君,Email:wann6971@163.com