The purpose of this systematic review by the American Pediatric Surgical Cancer Committee was to summarize evidence from the current medical literature regarding fertility restoration and hormone ...replacement for female children and adolescents treated with gonadotoxic treatments.
Using PRISMA guidelines, questions were addressed by searching Medline, Cochrane, Embase Central and National clearing house databases using relevant search terms. Eligible studies included those that addressed ovarian tissue cryopreservation (OTC), oocyte harvest, ovarian transposition, and ovarian tissue auto-transplantation for females under the age of 20. Four reviewers independently screened studies for eligibility, extracted data and assessed the risk of bias. Study outcomes were summarized in a narrative synthesis.
Two thousand two hundred seventy-six studies were identified by database search and manual review and 2185 were eliminated based on defined exclusion criteria. Ninety-one studies served as the basis for the systematic review. There were 1019 patients who underwent OTC with ages ranging from 0.4 to 20.4 years old, with 298 under the age of 13. Twenty patients aged 13–20 years old underwent successful oocyte harvest. Thirty-seven children underwent ovarian transposition as a means of fertility preservation. Eighteen patients underwent auto-transplantation of thawed ovarian cortical tissue that was harvested before the age of 21 years resulting in 10 live births.
Clinically accepted and experimental fertility preservation options such as OTC, oocyte cryopreservation, and ovarian transposition are available to females aged 20 years and younger who are at risk for premature ovarian insufficiency and infertility due to gonadotoxic treatments. There is a large cohort of pediatric-aged patients, with a wide variety of diagnoses and treatments, who have undergone fertility preservation. Currently, fertility and hormone restoration experience for patients who were 20- years of age or younger at the time of fertility preservation remains limited.
IV.
Many survivors of childhood cancer will experience premature gonadal insufficiency or infertility as a consequence of their medical treatments. Ovarian tissue cryopreservation (OTC) remains an ...experimental means of fertility preservation with few reports focused on the surgical technique and postoperative outcomes for OTC in children.
This is a single institution, retrospective review of OTC cases from January 2011 to December 2017. Children were eligible for OTC if they had a greater than 80% risk of premature ovarian insufficiency or infertility owing to their anticipated gonadotoxic medical treatment.
OTC was performed in 64 patients. Median age was 12 years old (range: 5 months–23 years). Nearly half (48%) of the patients were premenarchal. Laparoscopic unilateral oophorectomy was performed in 84% of patients. There were no surgical complications. In 76% of patients, OTC was performed in conjunction with an ancillary procedure. The majority (96%) of patients were discharged within 24 hours. Median time from operation to medical therapy was six days, with no unanticipated treatments delays attributable to OTC.
Laparoscopic unilateral oophorectomy for OTC can be performed safely, in combination with other ancillary procedures, as an outpatient procedure without delaying medical therapy for children facing a fertility-threatening diagnosis or treatment.
IV.
Ovarian tissue cryopreservation is increasing as a preferred option for fertility preservation for prepubertal and young adolescent females facing a fertility threatening diagnosis or treatment.
Ovid ...MEDLINE and PubMed searches for terms related to ovarian tissue removal for fertility preservation revealed there is no current consensus on operative technique for surgical ovarian cortical tissue removal in adult females. Additionally, there are limited published reports of surgical approach and outcomes in the pediatric population. In total, 22 publications were reviewed for their operative approach, ovarian tissue harvesting techniques, complications and outcomes.
Reported operative approaches and techniques for ovarian tissue cryopreservation for pediatric and adolescent patients are variable. Further investigations into operative technique and tissue harvesting that maintains healthy ovarian follicles for transplant will help establish standard technical principles for surgery in pediatric and adolescent females undergoing fertility preservation.
•Ovarian tissue cryopreservation is increasing as a preferred option for fertility preservation for prepubertal and young adolescent females facing a fertility threatening diagnosis or treatment.•Reported operative approaches for ovarian tissue cryopreservation are variable.•Further investigation into operative technique and tissue harvesting that maintains healthy ovarian follicles for transplant will help establish standard technical principles for surgery in pediatric and adolescent females undergoing fertility preservation.
The only pre-treatment fertility preservation option for prepubertal girls, who are at risk for infertility due to their diagnosis or treatment, is surgical removal of ovarian tissue for ...cryopreservation (OTC). We investigated ovarian tissue health following isolation with an ultrasonic advanced energy device (UAED), that has a previously reported thermal spread of ≤2 cm.
The ovaries of eight Yucatan minipigs were isolated by laparoscopy (1) close dissection with the UAED located up to 2 mm away from the ovarian capsule, (2) far dissection with the UAED located >2 cm away, or by (3) laparotomy for control ovaries using cold scissors. Ovarian cortex tissues were cultured for 4 days to assess tissue health.
Ovarian cortex tissue isolated using a UAED produced an altered metabolic ratio in both the far and close dissection compared to control (p < 0.001). There was an increase in folliculogenesis in the control samples over samples isolated with far and close dissection (p < 0.0001), and a reduction in estradiol production in experimental groups (p < 0.0001).
This model defines differences in ovarian tissue health among different isolation techniques. Ongoing work will further define the standard of care surgical technique for OTC.
Adrenocortical carcinoma (ACC) is a rare but aggressive pediatric endocrine tumor. However, there is no recent US national report on the management or outcomes of pediatric ACC. We aimed to examine ...the clinical characteristics, current management strategies, and outcomes of pediatric ACC.
In this retrospective National Cancer Database study between 2004 and 2019, children (<18 y) with ACC were included. Overall survival was examined by means of Kaplan–Meier method, log-rank tests, and Cox regression modeling.
Seventy-eight children with ACC were included. The median age was 10 y, the median tumor size was 10.2 cm, and 35.9% had metastasis at diagnosis. Most patients underwent surgical treatment (84.6%), 56.4% received chemotherapy, and 7.7% received radiation. The 1-, 3-, and 5-y overall survival rates were 87.0%, 62.0%, and 60.1%, respectively. In unadjusted analysis, surgical treatment was associated with improved overall survival (log-rank test, P < 0.001). In multivariable Cox regression, metastasis at diagnosis was associated with inferior overall survival (hazard ratio: 2.72, 95% confidence interval: 1.15-6.40, P = 0.02), when adjusting for age, tumor size, receipt of surgical treatment, and chemotherapy. In patients with nonmetastatic ACC, increasing age was associated with inferior overall survival (hazard ratio: 1.12, 95% confidence interval: 1.00-1.24, P = 0.04), when adjusting for tumor size, receipt of surgical treatment, and chemotherapy.
Most children with ACC in the USA undergo surgical treatment with about half of these also receiving chemotherapy. Metastasis at diagnosis was independently associated with inferior overall survival; in patients with nonmetastatic ACC, increasing age was independently associated with inferior overall survival.
Three large national data sets are commonly used to assess operative outcomes in pediatric surgery; National Surgical Quality Improvement Program Pediatric (NSQIP-P), Pediatric Health Information ...System (PHIS), and Kids' Inpatient Data set (KID). Hepatectomy and nephrectomy are rare pediatric surgical procedures, which may benefit from large administrative data sets for the assessment of short-term complications.
A retrospective review of NSQIP-P (2012-2015), KID (2012), and PHIS (2012-2015) was performed for hepatectomy or nephrectomy cases for children aged 0 to 18 y. Thirty-day perioperative outcomes were collected, analyzed, and compared across data sets and surgical cohorts.
Rates of surgical site infection, wound dehiscence, central line infection, sepsis, and venous thromboembolism were similar across NSQIP-P, PHIS, and KID in both cohorts. Rates of pneumonia and renal insufficiency were higher in PHIS and KID versus NSQIP-P in both cohorts. Blood transfusions in NSQIP-P were higher than PHIS and KID in the hepatectomy group (50.9% versus 43.0% versus 32.4%, P < 0.001), but similar across data sets in the nephrectomy cohorts (12.0% versus 14.0% versus 13.0%, P = 0.15). PHIS reported higher readmission rates than NSQIP-P for both the hepatectomy (56.5% versus 17.9%, P < 0.001) and nephrectomy (32.6% versus 7.6%,P < 0.001) cohorts. Thirty-day mortality rates were similar between NSQIP-P and PHIS, but higher in KID as compared with NSQIP-P for hepatectomy (6.4% versus 0.4%, P < 0.001) and nephrectomy (2.0% versus 0.3%, P < 0.001) cases.
Administrative data sets provide large sample sizes for the study of low-volume procedures in children, but there are significant variations in the reported rates of perioperative outcomes between NSQIP-P, PHIS, and KID. Therefore, surgical outcomes should be interpreted within the context of the strengths and limitations of each data set.
To evaluate for disparities in surgical care among US children with hepatoblastoma (HB) and hepatocellular carcinoma (HCC).
In this retrospective National Cancer Database study (2004-2015), children ...aged <18 years with HB or HCC were included. Multivariable mixed-effects logistic regression was used to evaluate the association of sociodemographic factors (age, sex, race and ethnicity, insurance status, income, proximity to treating hospital) with the odds of undergoing surgical treatment after adjusting for disease-related factors (tumor size, metastasis, comorbidities) and hospital-level effects. Subgroup analyses by tumor histology were performed.
A total of 811 children were included (HB: 80.9%; HCC: 19.1%), of which 610 (75.2%) underwent surgical treatment. Following adjustment, decreased odds of undergoing surgical treatment were associated with Black race (OR: 0.46 vs White, 95% CI 95% CI: 0.26-0.80, P = .01), and having Medicaid (OR: 0.58 vs private, 95% CI: 0.38-0.88, P = .01) or no insurance (OR: 0.33 vs private, 95% CI: 0.13-0.80, P = .02). In children with HB, Black race was associated with decreased odds of undergoing surgical treatment (OR: 0.47 vs White, 95% CI: 0.25-0.89, P = .02). In children with HCC, Medicaid (OR: 0.10 vs private, 95% CI: 0.03-0.35, P < .001), or no insurance status (OR: 0.10 vs private, 95% CI: 0.01-0.83, P = .03) were associated with decreased odds of undergoing surgical treatment. Other than metastatic disease, no additional factors were associated with likelihood of surgical treatment in any group.
Black race and having Medicaid or no insurance are independently associated with decreased odds of surgical treatment in children with HB and HCC, respectively. These children may be less likely to undergo curative surgery for their liver cancer.
Predicted 1-year survival of children with trisomy 18 (T18) has increased to 59.3%. We aimed to systematically review the characteristics, management, and outcomes of children with T18 and ...hepatoblastoma.
A systematic literature review of the PubMed, Embase, Scopus, Web of Science, and Cochrane Library databases was performed according to the PRISMA 2020 statement (end-of-search date: 03/03/2024).
Fifty studies reporting on 70 patients were included. The median age at diagnosis was 11.5 months, 85.9% were female (n = 55/64), and 15.0% had mosaic T18 (n = 6/40). Diagnosis was made during symptom evaluation (most commonly hepatomegaly or abdominal mass) in 45.5% (n = 15/33), incidentally in 24.2% (n = 8/33), during surveillance with abdominal ultrasound in 18.2% (n = 6/33), and at autopsy in 12.1% (n = 4/33). The median tumor size was 6.4 cm, 33.3% had multiple tumors (n = 14/42), and metastasis was present in one patient (3.8%; n = 1/26). Neoadjuvant chemotherapy was administered in 42.6% (n = 26/61) and adjuvant chemotherapy in 31.6% (n = 18/57). Surgical treatment was performed in 64.2% (n = 43/67). Of the patients not diagnosed on autopsy, overall mortality was 35.5% (n = 22/62) over a median follow-up of 11.0 months. Among the 26 deceased patients (including those diagnosed on autopsy), the most common causes of death were cardiopulmonary disease (38.5%, n = 10/26) and tumor progression (30.8%, n = 8/26).
T18 does not preclude resection with curative intent for hepatoblastoma. Combination of surgery and chemotherapy should be considered in children on an individualized basis depending on tumor characteristics and underlying cardiopulmonary comorbidities. Locoregional modalities may have a role in the setting of severe comorbidities.
Level IV evidence.
•What is currently known about this topic?•Due to improvements in intensive care and treatment of underlying abnormalities, especially cardiac disease, the survival of patients with trisomy 18 has increased.•Although it is now known that trisomy 18 is associated with an increased risk for hepatoblastoma, no systematic review has clearly defined the presentation, optimal management, and outcomes for these patients.•What new information is contained in this article?•Trisomy 18 does not preclude resection with curative intent for hepatoblastoma if aligned with the family's goals and wishes.•Combination of surgery and chemotherapy should be considered in children with trisomy 18 and hepatoblastoma on an individualized basis depending on tumor characteristics and underlying cardiopulmonary comorbidities.•Locoregional modalities may have a role in the setting of severe cardiopulmonary comorbidities.
Purpose
Thoracic outlet syndrome (TOS) is a rare disorder involving compression of the brachial plexus, subclavian artery, and subclavian vein. There is a paucity of data for this pathology’s ...surgical treatment within pediatrics. The objective of this study is to explore the presentation, management, and outcome of pediatric TOS.
Methods
A retrospective chart review was conducted for 44 patients at a single institution undergoing surgery for TOS. Data was collected on demographics, pre- and postoperative factors, and outcomes.
Results
Forty-four patients underwent 50 surgeries (8 bilaterally). The average age was 15.5 years with 72% female. The most common symptoms were numbness (72%) and pain (66%), with a normal exam in 58%. The average symptom duration prior to surgery was 35.2 months. A supraclavicular approach was performed in all patients, with anterior scalene section (90%), rib resection (72%), neurolysis (92%), and intraoperative EMG (84%) commonly used. Two patients had a lymphatic leak. All patients reported subjective improvement of preoperative symptoms of numbness (26%), pain (22%), and weakness (6%). Differences between vTOS (
n
= 9) and nTOS (
n
= 35) included higher preop swelling (
p
< 0.012), decreased symptom duration (
p
< 0.022), higher venogram usage (
p
< 0.0030), and higher preoperative thrombolytics/angioplasty (
p
< 0.001) in vTOS compared to nTOS. A comparison of soft tissue and soft tissue with bone decompression did not reveal any outcome differences.
Conclusion
Pediatric TOS benefits from a multidisciplinary approach, showing good outcomes in postoperative symptom resolution. In our cohort, a supraclavicular approach provided an effective window for decompression with a low complication rate.
Early postoperative fever is common. Adult data indicate that workup is unnecessary in the early postoperative period, but comparable data in children is limited. The objectives are to determine the ...incidence of fever and the utilization and yield of tests ordered in children.
Single-institution, retrospective analysis of surgical patients undergoing an elective inpatient/observational surgery between 2011 and 2015 was performed. Early fever was defined >38.0°C within two days post-procedure. Encounters were queried for all blood cultures (BC), urinalysis (UA), urine cultures (UC), chest radiographs (CXR), and respiratory viral panels (RVP) obtained.
We identified 6943 patients, of whom 30.6% developed fever. UA was positive in 19.8% of patients tested. UC was positive in 15.7% of patients and 92.0% had a urinary catheter during surgery. BC was positive in 0.69% of patients, all with a central venous catheter. CXRs were considered infectious in 3.0% of patients tested. Patients with PICU stay and/or fever ≥38.9°C were more likely to undergo BC and UC, but no more likely to have a positive result compared those without PICU stay and/or fever <38.9°.
Early postoperative fever is common in pediatric surgical populations and rarely associated with an infectious source. Workup should be applied selectively.
Level IV.
PDF
