The long-term outcome of patients diagnosed with sacrococcygeal teratoma in childhood. A study of a national cohort

Shalaby, Mohamed Sameh; Walker, Gregor; O’Toole, Stuart; Hammond, Philip; Carachi, Robert

doi:10.1136/archdischild-2014-306414

Cited by 30 publications

(19 citation statements)

References 20 publications

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“…In previous studies, fecal incontinence was reported to affect 13% to 27% of the patients in all age groups, and constipation was reported to affect a wide range of 8% to 39% [5,16]. The percentage of patients in our study who developed functional sequelae is comparable to that in previous reports.…”

Section: Discussionsupporting

confidence: 86%

Analysis of Recurrent Sacrococcygeal Teratoma in Children: Clinical Features, Relapse Risks, and Anorectal Functional Sequelae

Wang

et al. 2017

Med Sci Monit

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BackgroundSacrococcygeal teratoma (SCT) is a relatively uncommon tumor. Recurrence with poor survival and anorectal dysfunction are the 2 leading problems for patients. Here, we would review the clinic features of patients with SCTs in our hospital to identify risk factors of recurrent SCTs and to analyze anorectal functional sequelae.Material/MethodsA retrospective review of all patients with SCTs in our center between 2007 and 2013 was performed. We analyzed the recorded data on each patient and performed follow-up through phone calls.ResultsOur study included 105 inpatients (78 girls and 27 boys); 104 cases underwent surgical resection, and 62.5% cases had a mature histopathology. The proportion of malignant teratomas rose with increasing age. Fifteen children developed recurrent SCTs with a median of 11.5 months, and most of them had an elevation of AFP levels. Four recurrent children experienced a second tumor relapse. We observed a statistically significant difference in survival rate through Kaplan-Meier method between relapsed (66.7%) and non-relapsed (94.4%) patients. In univariate analysis, incomplete primary resection and malignant histology were proven to increase recurrence risks. Nearly half of patients had at least 1 of the parameters reflecting abnormal bowel function (e.g., involuntary bowel movements, fecal incontinence, and constipation). For those recurrent SCTs patients, difficulty defecating was a major problem.ConclusionsTumor recurrence affected the prognosis of children with SCT. In our research, incomplete resection and malignant histology were considered risk factors. Constipation was the main problem in anorectal functional sequelae for children who had recurrence.

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Section: Discussionsupporting

confidence: 86%

Analysis of Recurrent Sacrococcygeal Teratoma in Children: Clinical Features, Relapse Risks, and Anorectal Functional Sequelae

Wang

et al. 2017

Med Sci Monit

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“…Two studies that evaluated anorectal function at adult age after SCT resection found improvement of soiling but an increasing incidence of constipation . Other studies confirmed these findings, however the majority of patients included in these studies were children …”

Section: Introductionmentioning

confidence: 90%

Patient‐Reported Defecation and Micturition Problems Among Adults Treated for Sacrococcygeal Teratoma During Childhood—The Need for New Surveillance Strategies

Kremer

Derikx

Baren

et al. 2016

Pediatric Blood & Cancer

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Background. To evaluate defecation and micturition complaints in adults treated for sacrococcygeal teratoma (SCT) during childhood and to identify risk factors for soiling, urinary incontinence, and constipation beyond childhood. Procedure. Records of patients aged ࣙ18 treated for SCT during infancy in the Netherlands were retrospectively reviewed. Frequency and severity of soiling, constipation, and urinary incontinence were evaluated using questionnaires designed in line with the Krickenbeck classification. Problems during childhood were compared to outcomes at adult age in part of the cohort. Associations between patient-and disease-related factors with complaints beyond childhood were analyzed with the chisquare test or Fisher's exact test, when appropriate. Results. Of 47 included patients (mean age 26.2 years, SD ±6.5), 49% reported at least one defecation or micturition complaint. Urinary incontinence was present in 30% and had a greater negative impact than soiling (24%). Ten patients (21%) reported constipation; five found this severely bothering. Three patients reported social restrictions due to defecation or micturition complaints (6.4%). While sex and tumor histology were not identified as risk factors, a tumor diameter of >10 cm and Altman type I or type II SCT were associated with constipation during adulthood. Conclusions. One-third of the patients treated for SCT during childhood reported urinary and defecation problems beyond childhood. In only a minority of cases, these led to social restrictions. A greater tumor diameter was associated with a higher risk of constipation during adulthood. Prolonged surveillance strategies are advised for all patients with SCT.

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“…Impaired bowel function and urinary incontinence are common long‐term complications after surgery for SCT. The occurrence of such complications ranges from 19% to 38% for bowel function, and around 50% for urinary incontinence, with both occurring in 11–45.2% of cases . Risk factors include obstruction of the urinary tract and intestinal tract at prenatal imaging diagnosis, tumor recurrence, types other than Altman type I and abdominosacroperineal resection for Altman type III and IV .…”

Section: Recommendationsmentioning

confidence: 99%

“…Regarding pregnancy and delivery, 38.5% of patients were able to give birth without requiring intervention . According to a report in a Scottish national cohort, 22.2% of women >16 years of age had gone through pregnancy and given birth …”

Section: Recommendationsmentioning

confidence: 99%

“…The occurrence of such complications ranges from 19% to 38% for bowel function, and around 50% for urinary incontinence, with both occurring in 11-45.2% of cases. [44][45][46][47] Risk factors include obstruction of the urinary tract and intestinal tract at prenatal imaging diagnosis, tumor recurrence, types other than Altman type I and abdominosacroperineal resection for Altman type III and IV. 46,47 Cozzi et al, however, reported that there was no significant difference between adult patients with SCT and the control group.…”

Section: Cq6: What Long-term Complications Might Occur After Treatment?mentioning

confidence: 99%

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Japanese clinical practice guidelines for sacrococcygeal teratoma, 2017

Fumino

Tajiri

Usui

et al. 2019

Pediatrics International

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Background Sacrococcygeal teratoma (SCT) is the most common extragonadal germ cell tumor in neonates and infants. Although most cases of infantile SCT are benign tumors by nature, some develop into extremely large lesions, leading to massive bleeding, high‐output heart failure, disseminated intravascular coagulation, and even fatal outcomes during the neonatal period. In addition, some patients may present with tumor recurrence, malignant transformation, long‐term sequelae (including bladder and bowel dysfunction) and lower leg palsy during the long‐term follow up. SCT, however, is very rare, and there are few opportunities to encounter this disease, therefore general physicians without expert credentials currently lack information relevant to clinical practice. For this reason, the research project committee has compiled guidelines concerning SCT. Methods The purpose of these guidelines was to share information concerning the treatment and follow up of infantile SCT. The guidelines were developed using the methodologies in the Medical Information Network Distribution System. A comprehensive search of the English‐ and Japanese‐language articles in PubMed and Ichu‐Shi Web identified only case reports or case series, and the recommendations were developed through a process of informal consensus. Results The clinical questions addressed the risk factors, the efficacy of cesarean section, the initial devascularization of tumor feeding vessels, interventional radiology, recommended clinical studies for follow up and possible long‐term complications. Conclusions These are the first guidelines for SCT to be established in Japan, and they may have huge clinical value and significance in terms of developing therapeutic strategies and follow up, potentially contributing to the improvement of the prognosis and quality of life of SCT patients.

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The long-term outcome of patients diagnosed with sacrococcygeal teratoma in childhood. A study of a national cohort

Abstract: This is one of the largest national studies assessing the long-term outcome of patients with SCT. It highlights the significant gastrointestinal and urological long-term morbidities of SCT patients, which is useful for counselling families.

Cited by 30 publications

References 20 publications

Analysis of Recurrent Sacrococcygeal Teratoma in Children: Clinical Features, Relapse Risks, and Anorectal Functional Sequelae

Analysis of Recurrent Sacrococcygeal Teratoma in Children: Clinical Features, Relapse Risks, and Anorectal Functional Sequelae

Patient‐Reported Defecation and Micturition Problems Among Adults Treated for Sacrococcygeal Teratoma During Childhood—The Need for New Surveillance Strategies

Japanese clinical practice guidelines for sacrococcygeal teratoma, 2017

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