Mark W. Kieran, Liliana C. Goumnerova, Michael Prados and Nalin Gupta
Liliana Goumnerova, Dan Drzymalski, Mark Kieran, Scott Pomeroy, R. Michael Scott and Nancy Tarbell
Brain tumors are the most common solid tumors of childhood. As treatment options such as surgery, chemotherapy, and radiotherapy improve, and affected children's survival increases, the focus has shifted toward understanding long-term outcomes. We hypothesized that tumor recurrence and long-term neurological outcomes can be correlated with age at diagnosis, site and size of tumor, histology, extent of surgery, and presence or absence of hydrocephalus. Thus, the aims of this study were to determine predictive factors for tumor recurrence and long-term morbidity in children who undergo surgery only for low-grade brain tumors. The study was approved by the institutional review board.
Medical records were reviewed for each child and information collected regarding sex, age at surgery, histology site, size of tumor, presence of hydrocephalus, extent of surgery, and date of tumor recurrence. Preoperative, postoperative, and last follow-up neurological exams were reviewed and patients were evaluated for ataxia, hemiparesis, impaired vision, cranial nerve deficits, presence of seizures or headaches, dysmetria, nystagmus, and depression.
Forty-nine children (29 male, 20 female) ranging from 2.2 to 16.9 years of age at first operation were included; 31% had recurrence/progression. This was significantly correlated with tumors that were subtotally resected (p = 0.002) located in the supratentorial region (p = 0.017) and not astrocytoma or craniopharyngioma (p = 0.041). At last follow-up, 42% of children had no neurological sequelae, 53% had mild neurological sequelae, and 5% had moderate neurological sequelae. No children had experienced depression before or immediately after their initial surgery. However, 6 patients (16% of 38 patients) had depression at long-term follow-up.
These data indicate that we may be able to predict recurrence/progression of low-grade brain tumors based on extent of surgery location and histology. We also see that more than half of these children will experience a variety of surgical and neurological burdens throughout their lives.
Albert H. Kim, Elizabeth A. Thompson, Lance S. Governale, Catalina Santa, Kevin Cahll, Mark W. Kieran, Susan N. Chi, Nicole J. Ullrich, R. Michael Scott and Liliana C. Goumnerova
Low-grade glial and glioneuronal brain tumors are frequently encountered in the pediatric population and can be effectively treated by resection. The authors aimed to use imaging to evaluate how often tumors recurred and to determine if recurrences were associated with any clinical symptoms, along with the financial costs of imaging, in patients with radiographically proven gross-total resection (GTR) at Boston Children's Hospital. These data were assessed to propose guidelines regarding postoperative surveillance.
The authors performed a retrospective cohort analysis of the Pediatric Brain Tumor Program database from 1993 to 2003 to identify patients with glial or glioneuronal tumors initially evaluated at Boston Children's Hospital. Among the 888 patients evaluated for any type of brain tumor during this period, 67 patients had WHO Grade I glial or glioneuronal lesions with radiographically proven GTR and available follow-up data. The frequency and timing of postoperative imaging was compared with the institutional protocol. Recurrence-free survival was calculated using the Kaplan-Meier method. Financial costs of imaging were available from 2001 to 2009 and were averaged to extrapolate the postoperative surveillance costs.
Among the 67 patients with GTR, 13 recurrences were detected radiographically with a mean time to recurrence of 32.4 months (range 2.9–128.5 months). The mean duration of follow-up after surgery was 6.6 years. The recurrence-free survival at 2 and 5 years after GTR for all low-grade glial and glioneuronal tumors was 0.90 (95% CI 0.82–0.97) and 0.82 (95% CI 0.73–0.92), respectively. No clinical symptoms were associated with any of the recurrences, and no deaths occurred. Under the institutional protocol of surveillance imaging, the estimated cost per recurrence at 5 years was $104,094 per patient. The proposed protocol would reduce the number of MR scans in the first 5 years from 10 to 5, providing a potential cost savings of $52,047 per recurrence.
Given the slow-growing, clinically asymptomatic nature of low-grade glial and glioneuronal tumors coupled with the financial and psychological costs of repeated imaging, the authors propose a postoperative surveillance MRI schedule that is less intensive than current institutional practice.
Maria-Jesus Lobon-Iglesias, Vicente Santa-Maria Lopez, Patricia Puerta Roldan, Santiago Candela-Cantó, Monica Ramos-Albiac, Marta Gomez-Chiari, Stephanie Puget, Stephanie Bolle, Liliana Goumnerova, Mark W. Kieran, Ofelia Cruz, Jacques Grill and Andres Morales La Madrid
Diffuse intrinsic pontine glioma (DIPG) is a highly aggressive and lethal brainstem tumor in children. In the 1980s, routine biopsy at presentation was abandoned since it was claimed “unnecessary” for diagnosis. In the last decade, however, several groups have reincorporated this procedure as standard of care or in the context of clinical trials. Expert neurosurgical teams report no mortality and acceptable morbidity, and no relevant complications have been previously described. The aim of this study was to review needle tract dissemination as a potential complication in DIPG.
The authors retrospectively analyzed the incidence of dissemination through surgical tracts in DIPG patients who underwent biopsy procedures at diagnosis in 3 dedicated centers. Clinical records and images as well as radiation dosimetry from diagnosis to relapse were reviewed.
Four patients (2 boys and 2 girls, age range 6–12 years) had surgical tract dissemination: in 3 cases in the needle tract and in 1 case in the Ommaya catheter tract. The median time from biopsy to identification of dissemination was 5 months (range 4–6 months). The median overall survival was 11 months (range 7–12 months). Disseminated lesions were in the marginal radiotherapy field (n = 2), out of the field (n = 1), and in the radiotherapy field (n = 1).
Although surgical tract dissemination in DIPG is a rare complication (associated with 2.4% of procedures in this study), it should be mentioned to patients and family when procedures involving a surgical tract are proposed. The inclusion of the needle tract in the radiotherapy field may have only limited benefit. Future studies are warranted to explore the benefit of larger radiotherapy fields in patients with DIPG.