Advances in Clinical Medicine
Vol. 14  No. 01 ( 2024 ), Article ID: 78772 , 7 pages
10.12677/ACM.2024.141028

儿童卵巢畸胎瘤临床特征分析

张鹏1,刘琳2,朱荣坤2,郝希伟2*

1青岛大学青岛医学院,山东 青岛

2青岛大学附属医院小儿外科,山东 青岛

收稿日期:2023年12月4日;录用日期:2023年12月28日;发布日期:2024年1月8日

摘要

目的:了解儿童卵巢畸胎瘤临床表现、辅助检查、诊断及治疗方式的特点,提高对本病的认识。方法:回顾性分析85例儿童卵巢畸胎瘤患儿的临床诊疗资料分析,归纳总结儿童卵巢畸胎瘤的临床特征。结果:85例卵巢畸胎瘤患儿中,56例(65.88%)以腹痛为就诊原因,腹部包块与查体发现均为9例,以月经不规律、腹胀、排尿困难等就诊11例。部分患儿的甲胎蛋白、CA199、CA125及癌胚抗原均有不同程度升高。CT (86.54%)检出率明显高于超声(68.83%)检查。本组研究65例(76.47%)患儿行腹腔镜手术,其术中出血量、切口长度以及术后住院时间均少于开放手术。随访3例患儿复发,2例为成熟性畸胎瘤,1例2级IV期未成熟畸胎瘤,并伴有胸腔转移。结论:儿童卵巢畸胎瘤的临床表现多样,需要结合相关辅助检查进行综合分析,早期诊断,早期诊断,多可获得较好的预后。腹腔镜手术具有创伤小、恢复快的优势,为临床上首选的治疗方式。

关键词

卵巢畸胎瘤,儿童,诊断,手术

Clinical Characteristics Analysis of Ovarian Teratoma in Children

Peng Zhang1, Lin Liu2, Rongkun Zhu2, Xiwei Hao2*

1Qingdao Medical College, Qingdao University, Qingdao Shandong

2Pediatric Surgery Department of Qingdao University Affiliated Hospital, Qingdao Shandong

Received: Dec. 4th, 2023; accepted: Dec. 28th, 2023; published: Jan. 8th, 2024

ABSTRACT

Objective: To understand the characteristics of clinical manifestations, auxiliary examination, diagnosis and treatment of ovarian teratoma in children, and to improve the understanding of this disease. Methods: The clinical diagnosis and treatment data of 85 children with ovarian teratoma were analyzed retrospectively, and the clinical characteristics of children with ovarian teratoma were summarized. Results: Of the 85 children with ovarian teratoma, 56 cases (65.88%) were treated with abdominal pain, 9 cases were found with abdominal mass and only physical examination, and 11 cases were treated with irregular menstruation, abdominal distension, dysuria and so on. The levels of alpha-fetoprotein, CA199, CA125 and carcinoembryonic antigen were increased in some children. The detection rate of CT (86.54%) was significantly higher than that of ultrasound (68.83%). In this study, 65 patients (76.47%) underwent laparoscopic surgery, and the intraoperative blood loss, incision length and postoperative hospital stay were less than those of open surgery. Three patients were followed up with recurrent teratoma, 2 with mature teratoma and 1 with immature teratoma of grade 2 stage IV, accompanied by thoracic metastasis. Conclusion: The clinical manifestations of ovarian teratoma in children are varied, and it is necessary to give related auxiliary examination for comprehensive analysis. Early diagnosis and early diagnosis can obtain better prognosis. Laparoscopic surgery has the advantages of less trauma and faster recovery, and is the preferred treatment in clinical practice.

Keywords:Ovarian Teratoma, Children, Diagnosis, Surgery

Copyright © 2024 by author(s) and Hans Publishers Inc.

This work is licensed under the Creative Commons Attribution International License (CC BY 4.0).

http://creativecommons.org/licenses/by/4.0/

1. 引言

卵巢畸胎瘤是最常见的卵巢肿瘤,约占成人卵巢肿瘤的10%~20%,而在儿童卵巢肿瘤中约占50% [1] [2] 。卵巢畸胎瘤可分为成熟性畸胎瘤(mature teratoma, MT)、未成熟性畸胎瘤(immature teratoma, IT)和卵巢甲状腺肿(struma ovarii, SO),其中95%为MT [3] 。对于卵巢畸胎瘤的发病机制现在尚未明确,有相关研究认为肿瘤的发生与生殖细胞减数分裂异常有关 [4] 。儿童卵巢畸胎瘤表现多样,多数以急腹症前来就诊,部分患儿起病隐匿,无任何不适,临床上往往存在误诊、漏诊等问题。为进一步提高儿童卵巢畸胎瘤的诊断,本研究回顾性分析我院2018年~2022年收治的85例儿童卵巢畸胎瘤患儿的临床诊治特点,提高对本病的认识。

2. 资料与方法

2.1. 研究对象

2018年至2022年于青岛大学附属医院收治的病理诊断为卵巢畸胎瘤且病历资料完整的患儿共85例,最小2岁3个月,最大14岁,平均127.1 ± 37.7个月。

2.2. 方法

回顾性分析85例患儿的临床表现、实验室检查、影像学检查、组织病理学检查及治疗,了解儿童卵巢畸胎瘤临床特点及诊治情况。

2.3. 统计学方法

利用SPSS 25.0软件对数据进行统计学的处理。计量资料采用均数( x ¯ ± s )表示,采用独立样本t检验对比各数据间的差异。计数资料采用例或百分率表示。当P < 0.05时表示差异具有统计学意义。

3. 结果

3.1. 临床表现

85例卵巢畸胎瘤患儿中,就诊原因为腹痛56例,其中24例术中探查见肿瘤蒂扭转,腹部包块与仅查体时发现均为9例,以月经不规律、停经、腹胀、排尿困难等就诊11例。详见表1

Table 1. Clinical manifestations of 85 children with ovarian teratoma

表1. 85例儿童卵巢畸胎瘤患儿临床表现

3.2. 血清肿瘤标志物

大部分患儿术前完善甲胎蛋白(AFP)、肿瘤糖类抗原199 (CA199)、肿瘤糖类抗原125 (CA125)及癌胚抗原(CEA)。11例(17.71%)患儿CA199升高(48.2~394.3 U/mL);13例(21.67%)患儿CA125升高(44~413 U/mL);6例(10.00%)患儿CEA升高,其中5例轻度升高(6.45~12.18 ng/mL),1例升高显著为408 ng/mL;AFP仅4例(5.00%)患儿轻度升高。

3.3. 影像学检查

77例患儿术前完善超声检查,52例患儿行CT检查,4例患儿行MRI检查。影像学检查结果详见表2。其中超声及CT检查结果考虑恶性可能分别为2例和1例,术后组织病理学检查结果均为良性。CT (86.54%)检出率明显高于超声(68.83%)检查,完善MRI检查的患儿较少,其中4例患儿中3例检查结果考虑卵巢畸胎瘤,1例肿瘤性质未确定。

Table 2. Preoperative imaging diagnosis of 85 children

表2. 85例患儿术前影像学检查诊断

3.4. 肿瘤位置及大小

85例患儿中,左侧30例,右侧45例,双侧10例。肿瘤最小直径2 cm,最大直径22 cm,平均8.25 ± 4.01 cm,其中肿瘤直径5~10 cm者54例(56.84%),其余直径分布大致均匀。术中探查见肿瘤蒂扭转共25例,其中单侧22例,双侧3例,肿瘤直径均5~15 cm。详见表3

Table 3. Tumor diameter distribution and tumor torsion diameter distribution

表3. 肿瘤直径分布及肿瘤扭转直径分布

3.5. 组织病理学检查

85例患儿中,78例MT,7例IT。IT中,病理组织学分级1级3例、2级2例、3级1例,临床分期I期5例、III期1例、IV期1例。

3.6. 治疗及随访

85例患儿均行手术治疗,20例患儿行开放手术,65例患儿行腹腔镜手术,其中67例行肿瘤剥除,18例行同侧附件切除术。二者比较详见表4。6例IT术后行BEP (博来霉素 + 依托泊苷 + 顺铂)化疗方案。随访3例患儿复发,2例为MT,1例2级IV期IT,并伴有胸腔转移。

Table 4. Comparison of open and laparoscopic surgeries

表4. 开放与腹腔镜手术情况比较

4. 讨论

儿童卵巢畸胎瘤临床表现多样,大多数患儿为非特异性盆腔肿物症状,腹痛、腹部包块、腹胀等较为多见,部分患儿也以月经异常等妇科疾病表现而就诊。腹痛是儿童卵巢畸胎瘤最常见的症状,在本组实验中65.88%的患儿因此就诊,其中有42.86%的患儿出现卵巢肿瘤扭转。儿童急腹症的原因众多,但鉴别广泛,卵巢畸胎瘤误诊为急性阑尾炎的病例报道多见 [5] [6] [7] ,由于右侧卵巢肿瘤扭转坏死的症状与急性阑尾炎相似,同时扭转在儿童卵巢肿瘤并发症发生率仅为3%~4%,所以在临床上易被忽视 [5] 。若不及时进行手术治疗可导致卵巢坏死,所以对于体征不典型的女孩急腹症,要考虑到卵巢畸胎瘤扭转的可能。部分患儿无特异性症状,多为体检或查体时发现。少数患儿因月经不规律、停经等就诊时,常常完善妇科超声而检出。

卵巢畸胎瘤的实验室检查多无特异性,且大部分结果大致正常。少数卵巢肿瘤扭转的患儿存在电解质紊乱,需术前及时纠正。血清肿瘤标志物对卵巢畸胎瘤的诊断存在一定意义 [8] ,部分患儿可出现CA199、CA125、CEA及AFP升高,但检查的特异性较低。血清肿瘤标志物结果的异常对于区分MT与IT具有提示作用,上述肿瘤标志物在血清中的表达升高,提示体内肿瘤的发生可能,且数值越高提示恶性可能性越大 [9] [10] 。

超声检查由于价格低廉、简便,所以在卵巢畸胎瘤诊断中常作为首选检查方法,但对检查医生的能力具有一定要求。卵巢畸胎瘤内容物多为来源于外胚层组织和皮脂腺物质,肿瘤内部可有皮肤、神经、毛发、骨骼等一种或多种成分 [11] ,在超声征象上以混合回声为主,内可含钙化影,多数肿瘤轮廓规整,边缘清晰,内部无明显血流信号探及 [12] 。如肿块直径 ≥ 10 cm,边缘欠清,有实性不均回声与厚薄不均分割,且肿块可探及血流信号,则需要警惕IT可能 [13] 。CT检查相较超声,对于肿瘤的位置与周围毗邻关系有较为明显的优势,有利于术前手术方式的制定及手术风险的评估。在CT图像上,MT多以囊性包块为主,内部可见局限的散在钙化影。有研究发现,当肿块中钙化数量 > 5处,且钙化分布多在肿块中央时,对IT诊断的特异度及灵敏度分别为85.7%、70.0%,对于鉴别MT与IT具有一定意义 [14] 。增强扫描时,MT表现为轻度均匀强化,且各期变化不明显,而IT具有典型的“网格样”、“地图样”强化,成渐进性持续强化特点 [14] 。虽然超声检查简便易行,但在本组实验中检出率明显低于CT,所以对卵巢畸胎瘤的诊断,需结合超声与CT检查进行综合分析,制定治疗方案。

肿瘤扭转是卵巢畸胎瘤主要的并发症。本组实验中,单侧肿瘤发生扭转多见,发生扭转的肿瘤直径全部 > 5 cm,且随着肿瘤直径的增加,发生扭转的可能性降低,当肿瘤直径 ≥ 15 cm时,无扭转发生。这也与一项814例卵巢畸胎瘤患者临床资料的回顾性分析结论一致 [15] 。因此,当检查发现卵巢肿物大于5 cm时,应考虑早期行手术治疗,以降低卵巢肿瘤发生扭转、破裂的可能。

对于儿童卵巢畸胎瘤的治疗,多数主张积极早期手术治疗 [16] 。近些年来随着微创技术的不断发展,腹腔镜手术逐渐成为首先治疗方式,并且广泛应用。在本组研究中,腹腔镜手术的术中出血量、切口长度以及术后住院时间均少于开放手术(P < 0.05)。相较开放手术,腹腔镜手术具有创伤小、恢复快的优势。而对于直径较大的肿瘤,由于腹腔镜手术操作空间有限,开放手术仍具有优势。由于肿瘤多为囊实性,术中易发生破裂,若肿瘤出现破裂,需及时吸净溢出物,同时使用大量的生理盐水进行彻底地冲洗,减少腹腔内的粘连,降低术后并发症发生的可能。有回顾性分析显示 [17] ,术中经过良好的处理,肿瘤破裂并不对增加复发发生率。对于儿童卵巢畸胎瘤手术方式的选择,当术前考虑MT时,应尽可能保留卵巢功能,保留卵巢的手术方式作为首选 [18] 。而对于术前无法明确诊断为MT,术中应送快速病理学检查,当考虑恶性肿瘤可能,或术后病理学检查诊断为恶性肿瘤时,才可选择同侧附件切除 [19] [20] 。而对于扭转的卵巢肿瘤,有研究认为,不论卵巢缺血程度或肿瘤大小,扭转复位并保留卵巢都作为首选的手术方式,但术后需要密切随访监测卵巢血流及体积 [21] ,如有异常则需手术切除,以达到最大程度保留生育功能的目的。对于IT术后是否需要辅助化疗,Pashankar等人的研究认为 [22] ,病理组织学分级为1级肿瘤患儿无复发,2级肿瘤患儿复发率较低,对于不同临床分期复发率无明显差异,大多数复发发生在3级肿瘤的患儿中,III/IV期复发率明显增加,所以他们对于1级肿瘤及I/II期的2级肿瘤,主张单纯手术治疗,III/IV期的2级肿瘤及3级肿瘤,术后常需辅助长春新碱 + 博来霉素 + 顺铂(VBP)或博来霉素 + 依托泊苷 + 顺铂(BEP)化疗方案 [23] ,但辅助化疗并没有明显降低肿瘤复发率 [24] 。Yousef等人 [17] 认为,肿瘤不完整切除有增加肿瘤复发的可能,术中应在尽可能保留卵巢皮质的同时完整地切除肿瘤,降低复发风险。

卵巢畸胎瘤是儿童常见的卵巢肿瘤,其临床表现多样,需要对实验室及影像学检查进行综合分析,早期诊断,早期手术治疗,可以获得较好的预后。腹腔镜手术具有创伤小、恢复快的优势,为临床上首选的治疗方式。由于本研究为单中心,小样本资料回顾性分析,存在选择偏倚的可能,我们期待未来有多中心、大样本数据的研究进一步证明我们的结论。

文章引用

张 鹏,刘 琳,朱荣坤,郝希伟. 儿童卵巢畸胎瘤临床特征分析
Clinical Characteristics Analysis of Ovarian Teratoma in Children[J]. 临床医学进展, 2024, 14(01): 190-196. https://doi.org/10.12677/ACM.2024.141028

参考文献

  1. 1. He, X., Zhao, X., Wang, X., et al. (2021) Distinctive Pattern of Left—Right Asymmetry of Ovarian Benign Teratomas in Chinese Population: A 12-Year-Long Cross-Sectional Study. Archives of Gynecology and Obstetrics, 303, 729-737. https://doi.org/10.1007/s00404-020-05864-0

  2. 2. Trabzonlu, L., Durmaz, G., Vural, C., Muezzinoglu, B. and Co-rakci, A. (2017) Malignant Tumors Associated with Ovarian Mature Teratoma: A Single Institution Experience. Pathol-ogy—Research and Practice, 213, 518-521. https://doi.org/10.1016/j.prp.2017.01.016

  3. 3. Saleh, M., Bhosale, P., Menias, C.O., et al. (2021) Ovarian Terato-mas: Clinical Features, Imaging Findings and Management. Abdominal Radiology, 46, 2293-2307. https://doi.org/10.1007/s00261-020-02873-0

  4. 4. Heskett, M.B., Sanborn, J.Z., Boniface, C., et al. (2020) Mul-tiregion Exome Sequencing of Ovarian Immature Teratomas Reveals 2N Near-Diploid Genomes, Paucity of Somatic Mutations, and Extensive Allelic Imbalances Shared across Mature, Immature, and Disseminated Components. Modern Pathology, 33, 1193-1206. https://doi.org/10.1038/s41379-019-0446-y

  5. 5. Tsai, T.C., Wong, L.Y. and Wu, H.P. (2011) Ovarian Torsion Caused by Teratoma Masquerading as Perforated Appendicitis in a 5-Year-Old Girl. Pediatrics & Neonatology, 52, 51-54. https://doi.org/10.1016/j.pedneo.2010.12.002

  6. 6. Kokoska, E.R., Keller, M.S. and Weber, T.R. (2000) Acute Ovarian Torsion in Children. The American Journal of Surgery, 180, 462-465. https://doi.org/10.1016/S0002-9610(00)00503-1

  7. 7. Pomeranz, A.J. and Sabnis, S. (2004) Misdiagnoses of Ovarian Masses in Children and Adolescents. Pediatric Emergency Care, 20, 172-174. https://doi.org/10.1097/01.pec.0000117925.65522.e8

  8. 8. Gkrozou, F., Tsonis, O., Vatopoulou, A., Galaziou, G. and Paschopoulos, M. (2022) Ovarian Teratomas in Children and Adolescents: Our Own Experience and Review of Lit-erature. Children, 9, Article 1571. https://doi.org/10.3390/children9101571

  9. 9. 段拽林, 赵瑞霞, 魏丽军, 等. CEA、CA125、CA199、HE4和AFP在185例卵巢畸胎瘤的表达分析[J]. 肿瘤学杂志, 2021, 27(8): 686-689.

  10. 10. Loh, A.H.P., Gee, K.W.C. and Chua, J.H.Y. (2013) Diagnostic Accuracy of Preoperative α-Fetoprotein as an Ovarian Tumor Marker in Children and Adolescents: Not as Good as We Thought? Pediatric Surgery International, 29, 709-713. https://doi.org/10.1007/s00383-013-3316-6

  11. 11. Cong, L., Wang, S., Yeung, S.Y., et al. (2023) Mature Cystic Teratoma: An Integrated Review. International Journal of Molecular Sciences, 24, Article 6141. https://doi.org/10.3390/ijms24076141

  12. 12. 杨琼宇, 李天刚, 王艺璇, 等. 超声诊断儿童卵巢畸胎瘤伴输卵管扭转1例[J]. 临床超声医学杂志, 2021, 23(9): 654.

  13. 13. 郑园园, 潘姣娥, 王军梅. 卵巢恶性畸胎瘤的超声诊断价值[J]. 影像研究与医学应用, 2021, 5(14): 12-14.

  14. 14. 陆宽, 金丹, 徐亮, 等. 卵巢未成熟性畸胎瘤与成熟性畸胎瘤的CT定量与征象分析[J]. 临床放射学杂志, 2019, 38(12): 2357-2360.

  15. 15. Lawrence, A.E., Fallat, M.E., Hewitt, G., et al. (2021) Factors Associated with Torsion in Pediatric Patients with Ovarian Masses. Journal of Surgical Re-search, 263, 110-115. https://doi.org/10.1016/j.jss.2020.12.058

  16. 16. 童婷婷. 腹腔镜手术治疗卵巢畸胎瘤72例临床分析[J]. 中国妇幼保健, 2014, 29(20): 3236-3238.

  17. 17. Yousef, Y., Pucci, V. and Emil, S. (2016) The Relation-ship between Intraoperative Rupture and Recurrence of Pediatric Ovarian Neoplasms: Preliminary Observations. Journal of Pediatric and Adolescent Gynecology, 29, 111-116. https://doi.org/10.1016/j.jpag.2015.08.002

  18. 18. Tielli, A., Scala, A., Alison, M., et al. (2022) Ovarian Torsion: Di-agnosis, Surgery, and Fertility Preservation in the Pediatric Population. European Journal of Pediatrics, 181, 1405-1411. https://doi.org/10.1007/s00431-021-04352-0

  19. 19. Spinelli, C., Strambi, S., Masoni, B., et al. (2021) Surgical Management of Ovarian Teratomas in Childhood: A Multicentric Study on 110 Cases and a Literature Review. Gyneco-logical Endocrinology, 37, 950-954. https://doi.org/10.1080/09513590.2021.1948527

  20. 20. Özcan, R., Kuruoğlu, S., Dervişoğlu, S., et al. (2012) Ova-ry-Sparing Surgery for Teratomas in Children. Pediatric Surgery International, 29, 233-237. https://doi.org/10.1007/s00383-012-3228-x

  21. 21. Geimanaite, L. and Trainavicius, K. (2019) Pediatric Ovarian Tor-sion: Follow-Up after Preservation of Ovarian Tissue. Journal of Pediatric Surgery, 54, 1453-1456. https://doi.org/10.1016/j.jpedsurg.2019.02.004

  22. 22. Pashankar, F., Hale, J.P., Dang, H., et al. (2015) Is Adjuvant Chemotherapy Indicated in Ovarian Immature Teratomas? A Combined Data Analysis from the Malignant Germ Cell Tumor International Collaborative. Cancer, 122, 230-237.

  23. 23. Pavone, R., Dijoud, F., Galmiche, L., et al. (2020) Pure Pediatric Ovarian Immature Teratomas: The French Experience. Pediatric Blood & Cancer, 67, e28186. https://doi.org/10.1002/pbc.28186

  24. 24. Vieira, A.G.S., Puthenpura, V., Krailo, M., et al. (2023) Adjuvant Chemo-therapy Does Not Improve Outcome in Children with Ovarian Immature Teratoma: A Comparative Analysis of Clinical Trial Data from the Malignant Germ Cell International Consortium. Pediatric Blood & Cancer, 70, e30426. https://doi.org/10.1002/pbc.30426

  25. NOTES

    *通讯作者。

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