Abstract:These findings and previously published cases show that cavernous hemangiomas may occur after irradiation of the brain several years after the end of therapy irrespective of the radiation dose and type of malignancy. Particularly children < 10 years of age at the time of irradiation are at higher risk. Since patients with RICH frequently do not show symptoms but hemorrhage is a possible severe complication, imaging of the central nervous system should be performed routinely for longer follow- ups, particularly… Show more
“…16,20 Strenger and colleagues, who reported 8 RICMs in a series of 171 pediatric patients with cancer, found RICM to be more likely and have shorter latency if radiation therapy occurred in the first 10 years of life. 36 Taking together patients of all ages with RICM, our finding of a median latency of 12 years from radiation therapy to CM diagnosis is consistent with data from Keezer and Del Maestro, who gathered the majority of previously reported RICM cases (n = 84) and added 1 of their own in a 2009 report in which the mean and median latencies were 10.3 and 8 years, respectively. 20 In a stratified comparison of pediatric and adult RICM, we found that radiation treatment at a younger age makes multiple RICMs more likely.…”
abbreviatioNs CM = cavernous malformation; GRE = gradient echo; mRS = modified Rankin Scale; RICM = radiation-induced CM; RIT = radiation-induced telangiectasia; SWI = susceptibility-weighted imaging. results Thirty-two patients with RICMs were identified (56.2% men), with a median age of 31.1 years at RICM diagnosis. The median latency from radiation treatment to RICM diagnosis was 12.0 years (interquartile range 5.0-19.6 years). RICMs were always within the previous radiation port. RICMs were symptomatic at diagnosis in 46.9%, and were associated with symptomatic intracranial hemorrhage at any time in 43.8%. Older age at the time of radiation treatment and higher radiation dose were associated with shorter latency. RICMs tended to be diagnosed at a younger age than nonradiation CMs (median 31.1 vs 42.4 years, respectively; p = 0.054) but were significantly less likely to be symptomatic at the time of diagnosis (46.9% vs 65.8%, respectively; p = 0.036). RICMs were more likely to be multiple CMs than nonradiation CMs (p = 0.0002). Prospectively, the risk of symptomatic hemorrhage was 4.2% for RICMs and 2.3% for nonradiation CMs per person-year (p = 0.556). In the absence of symptoms at presentation, the risk of hemorrhage for RICMs was higher than for nonradiation CMs (4.2% vs 0.35%, respectively; p = 0.118). coNclusioNs In this patient population, RICMs occurred within the radiation port approximately 12 years after radiation treatment. Compared with nonradiation CMs, RICMs were more likely to occur as multiple CMs, to present at a younger age, and were at least as likely to cause symptomatic hemorrhage.
“…16,20 Strenger and colleagues, who reported 8 RICMs in a series of 171 pediatric patients with cancer, found RICM to be more likely and have shorter latency if radiation therapy occurred in the first 10 years of life. 36 Taking together patients of all ages with RICM, our finding of a median latency of 12 years from radiation therapy to CM diagnosis is consistent with data from Keezer and Del Maestro, who gathered the majority of previously reported RICM cases (n = 84) and added 1 of their own in a 2009 report in which the mean and median latencies were 10.3 and 8 years, respectively. 20 In a stratified comparison of pediatric and adult RICM, we found that radiation treatment at a younger age makes multiple RICMs more likely.…”
abbreviatioNs CM = cavernous malformation; GRE = gradient echo; mRS = modified Rankin Scale; RICM = radiation-induced CM; RIT = radiation-induced telangiectasia; SWI = susceptibility-weighted imaging. results Thirty-two patients with RICMs were identified (56.2% men), with a median age of 31.1 years at RICM diagnosis. The median latency from radiation treatment to RICM diagnosis was 12.0 years (interquartile range 5.0-19.6 years). RICMs were always within the previous radiation port. RICMs were symptomatic at diagnosis in 46.9%, and were associated with symptomatic intracranial hemorrhage at any time in 43.8%. Older age at the time of radiation treatment and higher radiation dose were associated with shorter latency. RICMs tended to be diagnosed at a younger age than nonradiation CMs (median 31.1 vs 42.4 years, respectively; p = 0.054) but were significantly less likely to be symptomatic at the time of diagnosis (46.9% vs 65.8%, respectively; p = 0.036). RICMs were more likely to be multiple CMs than nonradiation CMs (p = 0.0002). Prospectively, the risk of symptomatic hemorrhage was 4.2% for RICMs and 2.3% for nonradiation CMs per person-year (p = 0.556). In the absence of symptoms at presentation, the risk of hemorrhage for RICMs was higher than for nonradiation CMs (4.2% vs 0.35%, respectively; p = 0.118). coNclusioNs In this patient population, RICMs occurred within the radiation port approximately 12 years after radiation treatment. Compared with nonradiation CMs, RICMs were more likely to occur as multiple CMs, to present at a younger age, and were at least as likely to cause symptomatic hemorrhage.
“…The cumulative incidence increased up to 5% at 15 years after radiation exposure. In children less than 10 years old at the time of radiation, the cumulative incidence has been reported to be as high as 8.5% at 15 years, suggesting that age is truly a key risk factor for the development of RICH [3]. This is in sharp contrast to a prevalence of only 0.3–0.7% among the general population without any radiation exposure [5].…”
Section: Introductioncontrasting
confidence: 43%
“…The best time to operate varies from patient to patient according to the opinion of the treating team; however, the major criteria for intervention include a change in size, hemorrhage or symptom development. In a series of 8 patients previously reported who developed RICH, 3 underwent surgery, all secondary to an increase in size of the lesions [3]. The risk of spontaneous bleeding has been reported from 0.5 to 3% each year but it is most likely influenced by other factors such as location and ongoing morbidities.…”
Section: Discussionmentioning
confidence: 99%
“…Typically, the diagnosis of RICH is made at a median of 8.9 years from radiation exposure [2]. Some data suggest that younger age (children less than 10 years old) at the time of radiation exposure is a risk factor for developing RICH [3]. Radiation doses greater than 3,000 cGy have demonstrated a shorter latency period compared to doses less than 3,000 cGy [4].…”
Radiation-induced cavernous hemangiomas (RICH) are a known complication of radiation exposure, especially in young children. The current treatment approaches to these lesions include observation and surgical resection. We report the case of a 4-year-old male with recurrent medulloblastoma who had resolution of an incidental RICH lesion while being treated with bevacizumab for his recurrent brain tumor. There was no evidence of worsening hemorrhage with this therapy and the RICH did not recur upon discontinuation of the chemotherapy regimen. This is the first documented case of a RICH lesion responding to antiangiogenic therapy, suggesting the possible use of this class of agents in the treatment of symptomatic patients who are not considered appropriate candidates for surgical resection. Although the risk of bleeding must be taken into consideration, antiangiogenic therapies have the potential to be a novel treatment modality for symptomatic RICH lesions.
“…Many of the existing retrospective analyses are limited in validity due to little information about actual organ dose levels and older radiation techniques that can not be compared to modern therapy approaches [4,12]. However, detailed data regarding dose-volume-effect relationships of radiotherapy for organs in children and adolescents are indispensable for exact planning of combined modalities in tumor therapy.…”
BackgroundDue to low cure rates in former times therapy optimization approaches have mainly been focused on survival improvement for long times. Nowadays, with the enormous increases in cure rates over the last 20 to 30 years also a higher frequency of therapy-associated late effects among the rising amount of survivors occurs. Therefore the characterization of late effects after cancer therapy in childhood is of rising interest [1,5]. In Germany, several study groups like the "Late Effects Surveillance System" (LESS) [9], the German Childhood Cancer Registry [8], the "Hodgkin Late Effects Study Group" [11] or the working group "Quality of life" [7] examine different aspects of late effects [6]. In the United States of America, the "Childhood Cancer Survivor Study" has been established to characterize retrospectively the health status of 5-year-survivors of childhood cancer [10]. For radiotherapy, this study is not able to give detailed information about late side effects due to rare data about radiation doses and organ dose levels. Undoubtedly, radiotherapy is an important treatment strategy for many malignancies in paediatric oncology. However, sufficient data about late side effects of radiotherapy do not exist.There is a lack of information about radiation dose-effect relationships in view of late side effects in childhood and adolescence. Many of the existing retrospective analyses are limited in validity due to little information about actual organ dose levels and older radiation techniques that can not be compared to modern therapy approaches [4,12]. However, detailed data regarding dose-volume-effect relationships of radiotherapy for organs in children and adolescents are indispensable for exact planning of combined modalities in tumor therapy. The German Group of Paediatric Radiation Oncology (APRO) started to establish a prospective multi-centre registry study called "Registry for the evaluation of late side effects after radiation in childhood and adolescence" (RiSK) in 1998. This project has successfully developed and has now begun to expand internationally. Aim of this prospective multicentric register study is to evaluate irradiation dose effect relationships of organs and part of organs with respect to late effects. The study protocol [3] and first results have already been published [2,5]. To our knowledge, "RiSK" is the only multicentric study that evaluates radiation associated side effects prospectively with detailed information about organ dose levels. This analysis presents the first preliminary results regarding dose-volume-based characterizations of late sequelae in children and adolescents.
Patients and MethodsThe structure and the study protocol of "RiSK" have already been published [3]. In short, documentation forms including evaluation of treatment period, fractionation schedule, target volume, radiation technique and dose including detailed information about organ dose levels were established. Beside documentation of maximum doses for some organs, dose-volume histograms are to be chara...
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