Abstract

BackgroundThe incidence of children developing recurrent sacrococcygeal teratoma (SCT) is 2–35%. Serum alpha-fetoprotein (AFP) is often used as a tumor marker for (malignant) recurrences of SCT and could potentially be used during routine follow-up after SCT resection. However, the diagnostic accuracy of serum AFP levels during follow-up has not been well established. Therefore, we aimed to systematically review the diagnostic accuracy of serum AFP levels in recurrent SCT. MethodsWe queried Search Premier, COCHRANE Library, EMCARE, EMBASE, PubMed, ScienceDirect and Web of Science databases to identify studies regarding patients with SCT with follow-up using serum AFP levels postoperative. We estimated sensitivity and specificity of serum AFP levels. ResultsFifteen studies (613 patients, 121 recurrences) were included and these mainly described serum AFP levels in patients with recurrent SCT (n = 111); 83 (75%) patients with recurrent SCT had elevated serum AFP levels. A subgroup analysis of articles that measured serum AFP levels in all patients (n = 6, 136 patients, 14 recurrences) showed a sensitivity and specificity of 79% and 95%, respectively. The sensitivity of AFP levels to detect malignant recurrence was 96%. ConclusionDiagnostic accuracy of serum AFP levels to detect recurrent SCT seems promising, though sensitivity could be overestimated since serum AFP levels are mainly described in patients with elevated AFP levels or at recurrent SCT. Furthermore, serum AFP levels could be helpful to detect malignant recurrences. Type of studySystematic review of level 2–4 studies. Level of evidenceLevel 2–4 (mostly level 2).

Highlights

  • The incidence of children developing recurrent sacrococcygeal teratoma (SCT) is 2–35%

  • Diagnostic accuracy of serum AFP levels to detect recurrent SCT seems promising, though sensitivity could be overestimated since serum AFP levels are mainly described in patients with elevated AFP levels or at recurrent SCT

  • Oncological follow-up of patients treated for SCT is recommended for at least three years and includes physical examination, radiological imaging and serum alpha-fetoprotein (AFP) levels [4]

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Summary

Background

The incidence of children developing recurrent sacrococcygeal teratoma (SCT) is 2–35%. Serum alpha-fetoprotein (AFP) is often used as a tumor marker for (malignant) recurrences of SCT and could potentially be used during routine follow-up after SCT resection. We aimed to systematically review the diagnostic accuracy of serum AFP levels in recurrent SCT. Conclusion: Diagnostic accuracy of serum AFP levels to detect recurrent SCT seems promising, though sensitivity could be overestimated since serum AFP levels are mainly described in patients with elevated AFP levels or at recurrent SCT. The diagnostic accuracy of serum AFP levels in order to detect recurrent SCT after resection has not been well established. The aim of this systematic review is to assess the diagnostic accuracy of serum alpha-fetoprotein levels in detecting a recurrent SCT. Studies which were (a) written in another language than English, (b) case reports, and (c) publications before 1980 were excluded from data analysis

Study selection and methodological quality assessment
Data extraction and analysis
Literature search
Protocol
Study characteristics
Methodological quality
Patients with recurrent SCT
AFP levels in recurrent SCT
Study design
Diagnostic accuracy
AFP normative values
AFP levels and histology
Discussion
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