腹腔镜下卵泡穿刺加来曲唑治疗卵巢功能自主性卵巢囊肿合并外周性性早熟1例五年随访分析
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摘要
目的探讨腹腔镜卵泡穿刺术加来曲唑治疗合并雌激素水平持续显著增高的功能自主性卵巢囊肿的外周性性早熟的可行性。方法回顾分析1例功能自主性卵巢囊肿合并外周性性早熟患儿的临床资料。结果 3.5岁女性患儿,乳房发育伴间断少量阴道出血1年,骨龄和身高明显高于同年龄同性别儿童,雌激素水平持续增高。为明确病因及快速降低雌激素水平,行腹腔镜探查术,术中卵巢病理结果为纤维性囊壁,内衬少许颗粒样细胞,考虑卵巢多发性滤泡囊肿,遂行滤泡穿刺术。术后雌激素水平快速下降,术后20天复查雌激素E2 121 pg/mL,给予口服来曲唑1.25 mg/d。随访50个月,患儿雌激素水平在6～51pg/mL之间,身高增长速度逐渐恢复正常水平,骨龄发育较前延缓,肝肾功能正常。结论腹腔镜卵泡穿刺术加来曲唑治疗合并雌激素水平持续显著增高的功能自主性卵巢囊肿的外周性性早熟是一种可供选择的方法。
Objective To investigate the feasibility of laparoscopic follicular puncture plus letrozole in the treatment of precocious pseudopuberty caused by functional autonomous ovarian cysts with a significantly higher estrogen level.MethodThe clinical data of functional autonomic ovarian cysts combined with precocious pseudopuberty in a child were retrospective analyzed.Results A 3.5-year-old girl suffered from early breast development and intermittent small amount of vaginal bleeding for one year.Her bone age and height were significantly higher than those of normal children of the same age and gender,and estrogen level was continuously increased.In order to clarify the etiology and reduce the estrogen level rapidly,laparoscopic exploration was performed.During the operation,the pathological results of the ovary showed fibrous cyst wall with a few granular cells lining.Then follicle puncture was performed because multiple follicular cysts of the ovary were considered.The estrogen level decreased rapidly after operation.The estrogen E2 level was 121 pg/mL 20 days after the operation and letrozole was given orally at 1.25 mg/d.During the 50-month follow-up,the estrogen level fluctuated between 6～51 pg/mL,and the growth rate of height gradually returned to normal level,with delayed bone age development and normal function of liver and kidney.Conclusion Laparoscopic follicular puncture plus letrozole is an alternative method for the treatment of precocious pseudopuberty caused by functional ovarian cysts with a significantly higher estrogen level.
Objective To investigate the feasibility of laparoscopic follicular puncture plus letrozole in the treatment of precocious pseudopuberty caused by functional autonomous ovarian cysts with a significantly higher estrogen level.MethodThe clinical data of functional autonomic ovarian cysts combined with precocious pseudopuberty in a child were retrospective analyzed.Results A 3.5-year-old girl suffered from early breast development and intermittent small amount of vaginal bleeding for one year.Her bone age and height were significantly higher than those of normal children of the same age and gender,and estrogen level was continuously increased.In order to clarify the etiology and reduce the estrogen level rapidly,laparoscopic exploration was performed.During the operation,the pathological results of the ovary showed fibrous cyst wall with a few granular cells lining.Then follicle puncture was performed because multiple follicular cysts of the ovary were considered.The estrogen level decreased rapidly after operation.The estrogen E2 level was 121 pg/mL 20 days after the operation and letrozole was given orally at 1.25 mg/d.During the 50-month follow-up,the estrogen level fluctuated between 6～51 pg/mL,and the growth rate of height gradually returned to normal level,with delayed bone age development and normal function of liver and kidney.Conclusion Laparoscopic follicular puncture plus letrozole is an alternative method for the treatment of precocious pseudopuberty caused by functional ovarian cysts with a significantly higher estrogen level.
引文
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[14]Kang E,Cho JH,Choi JH,et al.Etiology and therapeutic outcomes of children with gonadotropin-independent precocious puberty[J].Ann Pediatr Endocrinol Metab,2016,21(3):136-142.
[15]Bercaw-Pratt JL,Moorjani TP,Santos XM,et al.Diagnosis and management of precocious puberty in atypical presentations of McCune-Albright syndrome:a case series review[J].J Pediatr Adolesc Gynecol,2012,25(1):e9-e13.
[16]Gucev Z,Tasic V,Jancevska A,et al.McCune-Albright syndrome(MAS):early and extensive bone fibrous dysplasia involvement and"mistaken identity"oophorectomy[J].JPediatr Endocrinol Metab,2010,23(8):837-842.
[2]Chae HS,Rheu CH.Precocious pseudopuberty due to an autonomous ovarian follicular cyst:case report with a review of literatures[J].BMC Res Notes,2013,6:319.
[3]Tessiatore P,Guana R,Mussa A,et al.When to operate on ovarian cysts in children[J].J Pediatr Endocrinol Metab,2012,25(5-6):427-433.
[4]Sinnecker G,Willig RP,Stahnke N,et al.Precocious pseudopuberty associated with multiple ovarian follicular cysts and low plasma oestradiol concentrations[J].Eur JPediatr,1989,148(7):600-602.
[5]Rodriguez-Macias KA,Thibaud E,Houang M,et al.Follow up of precocious pseudopuberty associated with isolated ovarian follicular cysts[J].Arch Dis Child,1999,81(1):53-56.
[6]Spinelli C,Di GM,Cei M,et al.Functional ovarian lesions in children and adolescents:when to remove them[J].Gynecol Endocrinol,2009,25(5):294-298.
[7]Stocco C.Tissue physiology and pathology of aromatase[J].Steroids,2012,77(1-2):27-35.
[8]Geisler J.Breast cancer tissue estrogens and their manipulation with aromatase inhibitors and inactivators[J].JSteroid Biochem Mol Biol,2003,86(3-5):245-253.
[9]Folkestad L,Bjarnason NH,Bjerregaard JK,et al.The effect of aromatase inhibitors on bone metabolism[J].Basic Clin Pharmacol Toxicol,2009,104(1):3-10.
[10]Wit JM,Hero M,Nunez SB.Aromatase inhibitors in pediatrics[J].Nat Rev Endocrinol,2012,8(3):135-147.
[11]Diaz-Thomas A,Shulman D.Use of aromatase inhibitors in children and adolescents:what's new[J].Curr Opin Pediatr,2010,22(4):501-507.
[12]Feuillan P,Calis K,Hill S,et al.Collins MT.Letrozole treatment of precocious puberty in girls with the McCuneAlbright syndrome:a pilot study[J].J Clin Endocrinol Metab,2007,92(6):2100-2106.
[13]Lenz AM,Shulman D,Eugster EA,et al.Bicalutamide and third-generation aromatase inhibitors in testotoxicosis[J].Pediatrics,2010,126(3):e728-e733.
[14]Kang E,Cho JH,Choi JH,et al.Etiology and therapeutic outcomes of children with gonadotropin-independent precocious puberty[J].Ann Pediatr Endocrinol Metab,2016,21(3):136-142.
[15]Bercaw-Pratt JL,Moorjani TP,Santos XM,et al.Diagnosis and management of precocious puberty in atypical presentations of McCune-Albright syndrome:a case series review[J].J Pediatr Adolesc Gynecol,2012,25(1):e9-e13.
[16]Gucev Z,Tasic V,Jancevska A,et al.McCune-Albright syndrome(MAS):early and extensive bone fibrous dysplasia involvement and"mistaken identity"oophorectomy[J].JPediatr Endocrinol Metab,2010,23(8):837-842.