成年骶尾部畸胎瘤的诊治:单中心8年经验回顾（英文）
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摘要
目的:探讨成人骶尾部畸胎瘤的临床表现、影像学特点、组织学特征、手术方式及预后。方法:回顾分析2010年8月至2018年8月在我院收治经组织病理学诊断为骶尾部畸胎瘤患者的临床资料。结果:本组患者病例8例(男2例,女6例),中位年龄34岁。6例患者表现为骶尾部疼痛,4例表现为骶尾部肿块和溃疡。采用计算机断层扫描(CT)和磁共振成像(MRI)相结合的方法进行评估,所有患者均进行手术治疗。7例经病理诊断为良性畸胎瘤,术后无复发。1例为恶性畸胎瘤,术后84个月复发,二次手术后随访6月无复发。结论:(1)由于缺乏典型的临床表现,成人骶尾部畸胎瘤难以诊断;(2)CT和MRI检查相结合有利于术前诊断;(3)手术入路和手术切除方式的选择取决于肿瘤的大小、位置和组成,可以通过术前的CT和MRI来确定;(4)大多数成年骶尾部畸胎瘤是良性的,恶性畸胎瘤手术切除效果良好。即使对于复发性恶性畸胎瘤,再次手术效果良好。
Objective: To determine the clinical, imaging, and histological features, and surgical resection modalities and outcomes of adult sacrococcygeal teratoma(SCT). Methods: Adult patients with histopathologically diagnosed SCT were enrolled in our hospital between August 2010 and August 2018. Each patient's characteristics and clinical information were reviewed. Results: There were 8 patients in the study(2 males, 6 females) with a median age of 34 years(range, 18–67 years). The time to clinical symptoms was 14 d to 35 years, with a median time of 4 years. Six patients presented with symptoms of sacrococcygeal pain, and four with signs of sacrococcygeal mass and ulceration in the sacrococcygeal region. Six patients were evaluated using a combination of computed tomography(CT) and magnetic resonance imaging(MRI). All patients showed a presacral tumor with heterogeneous intensity on CT images. All patients underwent surgical treatment, including 6 parasacral, 1 transabdominal, and 1 combined anteriorposterior surgery cases. Seven patients were histopathologically diagnosed with benign mature SCT, and have shown no recurrence. One patient had malignant SCT, with recurrence at 84 months after surgery. After a second surgery, the patient had no recurrence within 6 months follow-up after re-resection. Conclusions: Our retrospective study demonstrated:(1) adult SCT is difficult to diagnose because of a lack of typical clinical symptoms and signs;(2) a combination of CT and MRI examination is beneficial for preoperative diagnosis;(3) the choice of surgical approach and surgical resection modality depends on the size, location, and components of the tumor, which can be defined from preoperative CT and MRI evaluation;(4) most adult SCTs are benign; the surgical outcome for the malignant SCT patient was good after complete resection. Even for the patient with recurrent malignant SCT, the surgical outcome was good after re-resection.
Objective: To determine the clinical, imaging, and histological features, and surgical resection modalities and outcomes of adult sacrococcygeal teratoma(SCT). Methods: Adult patients with histopathologically diagnosed SCT were enrolled in our hospital between August 2010 and August 2018. Each patient's characteristics and clinical information were reviewed. Results: There were 8 patients in the study(2 males, 6 females) with a median age of 34 years(range, 18–67 years). The time to clinical symptoms was 14 d to 35 years, with a median time of 4 years. Six patients presented with symptoms of sacrococcygeal pain, and four with signs of sacrococcygeal mass and ulceration in the sacrococcygeal region. Six patients were evaluated using a combination of computed tomography(CT) and magnetic resonance imaging(MRI). All patients showed a presacral tumor with heterogeneous intensity on CT images. All patients underwent surgical treatment, including 6 parasacral, 1 transabdominal, and 1 combined anteriorposterior surgery cases. Seven patients were histopathologically diagnosed with benign mature SCT, and have shown no recurrence. One patient had malignant SCT, with recurrence at 84 months after surgery. After a second surgery, the patient had no recurrence within 6 months follow-up after re-resection. Conclusions: Our retrospective study demonstrated:(1) adult SCT is difficult to diagnose because of a lack of typical clinical symptoms and signs;(2) a combination of CT and MRI examination is beneficial for preoperative diagnosis;(3) the choice of surgical approach and surgical resection modality depends on the size, location, and components of the tumor, which can be defined from preoperative CT and MRI evaluation;(4) most adult SCTs are benign; the surgical outcome for the malignant SCT patient was good after complete resection. Even for the patient with recurrent malignant SCT, the surgical outcome was good after re-resection.
引文
Emoto S,Kaneko M,Murono K,et al.,2018.Surgical management for a huge presacral teratoma and a meningocele in an adult with Currarino triad:a case report.Surg Case Rep,4(1):9.https://doi.org/10.1186/s40792-018-0419-2
Firszt OP,Myga-Porosi?o J,Po?pieszny K,et al.,2018.Radiological features of sacrococcygeal teratomas in fetal magnetic resonance imaging and computed tomographya case report.Pol J Radiol,83:e19-e23.https://doi.org/10.5114/pjr.2018.74861
Hambraeus M,Al-Mashhadi A,Wester T,et al.,2018a.Functional outcome and health-related quality of life in patients with sacrococcygeal teratoma-a Swedish multicenter study.J Pediatr Surg,in press.https://doi.org/10.1016/j.jpedsurg.2018.10.044
Hambraeus M,Hagander L,Stenstrom P,et al.,2018b.Long-term outcome of sacrococcygeal teratoma:a controlled cohort study of urinary tract and bowel dysfunction and predictors of poor outcome.J Pediatr,198:131-136.e2.https://doi.org/10.1016/j.jpeds.2018.02.031
Hiller DJ,Waters GS,Bohl JL,2015.Incidence and operative excision of presacral masses:an institutional analysis.Am Surg,81(12):1237-1239.
Hunter CJ,Ford HR,Estrada JJ,et al.,2009.Alpha-fetoprotein levels correlate with the pathologic grade and surgical outcomes of pediatric retroperitoneal teratomas.Pediatr Surg Int,25(4):331-336.https://doi.org/10.1007/s00383-009-2321-2
Kremer MEB,Dirix M,Koeneman MM,et al.,2015.Quality of life in adulthood after resection of a sacrococcygeal teratoma in childhood:a Dutch multicentre study.Arch Dis Child Fetal Neonatal Ed,100(3):F229-F232.https://doi.org/10.1136/archdischild-2014-307589
Luk SY,Tsang YP,Chan TS,et al.,2011.Sacrococcygeal teratoma in adults:case report and literature review.Hong Kong Med J,17(5):417-420.
Mengual-Ballester M,Pellicer-Franco E,Valero-Navarro G,et al.,2014.Presacral tumor as a differential diagnosis of recurrent pilonidal sinus.Cir Cir,82(5):567-572.
Patel N,Maturen KE,Kaza RK,et al.,2016.Imaging of presacral masses-a multidisciplinary approach.Br JRadiol,89(1061):20150698.https://doi.org/10.1259/bjr.20150698
Saba L,Fellini F,Greco FG,et al.,2014.MRI evaluation of not complicated Tailgut cyst:case report.Int J Surg Case Rep,5(10):761-764.https://doi.org/10.1016/j.ijscr.2014.02.014
Saxena D,Pandey A,Bugalia RP,et al.,2015.Management of presacral tumors:our experience with posterior approach.Int J Surg Case Rep,12:37-40.https://doi.org/10.1016/j.ijscr.2015.05.015
Shatnawi NJ,Khammash MR,Omari AH,2019.A giant sacrococcygeal teratoma in adult female:a case report.Int JSurg Case Rep,54:47-50.https://doi.org/10.1016/j.ijscr.2018.11.039
Sheng QS,Xu XM,Cheng XB,et al.,2015.Multi-stage resection and repair for the treatment of adult giant sacrococcygeal teratoma:a case report and literature review.Oncol Lett,10(1):425-429.https://doi.org/10.3892/ol.2015.3249
Simpson PJ,Wise KB,Merchea A,et al.,2014.Surgical outcomes in adults with benign and malignant sacrococcygeal teratoma:a single-institution experience of 26cases.Dis Colon Rectum,57(7):851-857.https://doi.org/10.1097/DCR.0000000000000117
Sukhadiya MV,Das U,2015.Laparoscopic approach to type IV sacrococcygeal teratoma in an adult.Indian J Surg,77(Suppl 1):62-63.https://doi.org/10.1007/s12262-014-1130-6
Szyllo K,Lesnik N,2013.Sacrococcygeal teratoma-case report and review of the literature.Am J Case Rep,14:1-5.https://doi.org/10.12659/AJCR.883727
Varma AV,Malpani G,Agrawal P,et al.,2017.Clinicopathological spectrum of teratomas:an 8-year retrospective study from a tertiary care institute.Indian J Cancer,54(3):576-579.
Wessell A,Hersh DS,Ho CY,et al.,2018.Surgical treatment of a type IV cystic sacrococcygeal teratoma with intraspinal extension utilizing a posterior-anterior-posterior approach:a case report.Childs Nerv Syst,34(5):977-982.https://doi.org/10.1007/s00381-018-3718-9
Firszt OP,Myga-Porosi?o J,Po?pieszny K,et al.,2018.Radiological features of sacrococcygeal teratomas in fetal magnetic resonance imaging and computed tomographya case report.Pol J Radiol,83:e19-e23.https://doi.org/10.5114/pjr.2018.74861
Hambraeus M,Al-Mashhadi A,Wester T,et al.,2018a.Functional outcome and health-related quality of life in patients with sacrococcygeal teratoma-a Swedish multicenter study.J Pediatr Surg,in press.https://doi.org/10.1016/j.jpedsurg.2018.10.044
Hambraeus M,Hagander L,Stenstrom P,et al.,2018b.Long-term outcome of sacrococcygeal teratoma:a controlled cohort study of urinary tract and bowel dysfunction and predictors of poor outcome.J Pediatr,198:131-136.e2.https://doi.org/10.1016/j.jpeds.2018.02.031
Hiller DJ,Waters GS,Bohl JL,2015.Incidence and operative excision of presacral masses:an institutional analysis.Am Surg,81(12):1237-1239.
Hunter CJ,Ford HR,Estrada JJ,et al.,2009.Alpha-fetoprotein levels correlate with the pathologic grade and surgical outcomes of pediatric retroperitoneal teratomas.Pediatr Surg Int,25(4):331-336.https://doi.org/10.1007/s00383-009-2321-2
Kremer MEB,Dirix M,Koeneman MM,et al.,2015.Quality of life in adulthood after resection of a sacrococcygeal teratoma in childhood:a Dutch multicentre study.Arch Dis Child Fetal Neonatal Ed,100(3):F229-F232.https://doi.org/10.1136/archdischild-2014-307589
Luk SY,Tsang YP,Chan TS,et al.,2011.Sacrococcygeal teratoma in adults:case report and literature review.Hong Kong Med J,17(5):417-420.
Mengual-Ballester M,Pellicer-Franco E,Valero-Navarro G,et al.,2014.Presacral tumor as a differential diagnosis of recurrent pilonidal sinus.Cir Cir,82(5):567-572.
Patel N,Maturen KE,Kaza RK,et al.,2016.Imaging of presacral masses-a multidisciplinary approach.Br JRadiol,89(1061):20150698.https://doi.org/10.1259/bjr.20150698
Saba L,Fellini F,Greco FG,et al.,2014.MRI evaluation of not complicated Tailgut cyst:case report.Int J Surg Case Rep,5(10):761-764.https://doi.org/10.1016/j.ijscr.2014.02.014
Saxena D,Pandey A,Bugalia RP,et al.,2015.Management of presacral tumors:our experience with posterior approach.Int J Surg Case Rep,12:37-40.https://doi.org/10.1016/j.ijscr.2015.05.015
Shatnawi NJ,Khammash MR,Omari AH,2019.A giant sacrococcygeal teratoma in adult female:a case report.Int JSurg Case Rep,54:47-50.https://doi.org/10.1016/j.ijscr.2018.11.039
Sheng QS,Xu XM,Cheng XB,et al.,2015.Multi-stage resection and repair for the treatment of adult giant sacrococcygeal teratoma:a case report and literature review.Oncol Lett,10(1):425-429.https://doi.org/10.3892/ol.2015.3249
Simpson PJ,Wise KB,Merchea A,et al.,2014.Surgical outcomes in adults with benign and malignant sacrococcygeal teratoma:a single-institution experience of 26cases.Dis Colon Rectum,57(7):851-857.https://doi.org/10.1097/DCR.0000000000000117
Sukhadiya MV,Das U,2015.Laparoscopic approach to type IV sacrococcygeal teratoma in an adult.Indian J Surg,77(Suppl 1):62-63.https://doi.org/10.1007/s12262-014-1130-6
Szyllo K,Lesnik N,2013.Sacrococcygeal teratoma-case report and review of the literature.Am J Case Rep,14:1-5.https://doi.org/10.12659/AJCR.883727
Varma AV,Malpani G,Agrawal P,et al.,2017.Clinicopathological spectrum of teratomas:an 8-year retrospective study from a tertiary care institute.Indian J Cancer,54(3):576-579.
Wessell A,Hersh DS,Ho CY,et al.,2018.Surgical treatment of a type IV cystic sacrococcygeal teratoma with intraspinal extension utilizing a posterior-anterior-posterior approach:a case report.Childs Nerv Syst,34(5):977-982.https://doi.org/10.1007/s00381-018-3718-9