Clinical outcome of pediatric choroid plexus tumors: retrospective analysis from a single institute

Koh, Eun Jung; Wang, Kyu Chang; Phi, Ji Hoon; Lee, Ji Yeoun; Choi, Jung Won; Park, Shin Young; Park, Kyung Duk; Kim, Il Han; Cho, Byung Kyu; Kim, Seung Ki

doi:10.1007/s00381-013-2223-4

Cited by 41 publications

(22 citation statements)

References 36 publications

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“…The extent of surgical resection was divided into 4 groups: subtotal resection (STR), gross total resection (GTR), surgery-not otherwise specified (NOS), and no surgery. The STR group included patients with surgical codes 20 (Local excision of tumor, lesion, or mass, excisional biopsy, or stereotactic biopsy of the brain) and 21 (Subtotal resection of tumor, lesion or mass in brain), while the GTR group included patients with surgical codes 30 (Radical, total, gross resection of tumor, lesion or mass in brain), 40 (Partial resection of lobe of brain, when the surgery cannot be coded as [20][21][22][23][24][25][26][27][28][29][30], and 55 (Gross total resection of lobe of brain (lobectomy)). ''Surgery, NOS'' included patients with surgical code 90 (Surgery, NOS).…”

Section: Methodsmentioning

confidence: 99%

Pediatric choroid plexus tumors: epidemiology, treatments, and outcome analysis on 202 children from the SEER database

et al. 2014

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Choroid plexus papillomas (CPPs) and carcinomas (CPCs) are rare neoplasms that affect mostly children. Due to their rarity, their epidemiology and outcomes are incompletely understood. The National Cancer Institute's Surveillance, Epidemiology and End Results (SEER) Program is a well-established population-based group of registries that collects and publishes cancer incidence and survival data representing approximately 28 % of the US population. SEER-STAT v8.1.2 was used to identify patients with ICD-O-3 codes for choroid plexus tumors in patients aged 0-19. Demographics, initial treatment, and follow-up data were collected. Statistical methods including Kaplan-Meier curves, log rank tests, and Cox proportional hazards regression were used to estimate associations between independent variables and survival. The SEER registries contained 107 CPPs (2004-2010) and 95 CPCs (1978-2010). Median follow-up was 38 and 40 months, respectively. More than 75 % of CPCs were diagnosed before the age of 5 years, versus 48 % for CPPs. Sixty-five percent of CPCs and 57 % of CPPs occurred in males. In both groups at least 90 % of children underwent surgical resection. Gross total resection (GTR) was achieved in 67.0 % of CPCs and 63.6 % of CPPs. Almost 17 % of CPCs were treated with radiation versus only 0.9 % of CPPs. More than 98 % of patients with CPP were alive at the last follow-up, versus 62 % of CPC patients. For CPC, surgery was significantly associated with increased overall survival, but contrary to previous reports, extent of surgical resection was not associated with survival. Age, sex, race, and radiation treatment also had no effect on survival. This report, using the SEER datasets, corroborates many findings of previous smaller studies on CPTs. CPC occurs in younger children, with a male predominance, and a much worse prognosis than CPP. As such, these tumors have been treated aggressively with high rates of GTR and radiation treatment. Despite these treatments, overall survival for CPC remains poor.

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Section: Methodsmentioning

confidence: 99%

Pediatric choroid plexus tumors: epidemiology, treatments, and outcome analysis on 202 children from the SEER database

et al. 2014

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“…Based on our data and previous publications, the achievement of a gross-total resection appears to be prognostically favorable. 2,5,[8][9][10]12,16,17 In fact, all of the long-term survivors in our series underwent a near-total or gross-total resection of their tumor. As CPCs show a close relation to the ventricular system, the anatomical localization is commonly less of a concern when planning resection.…”

Section: Discussionmentioning

confidence: 99%

Neoadjuvant chemotherapy reduces blood loss during the resection of pediatric choroid plexus carcinomas

Schneider¹,

Kamaly-Asl

Ramaswamy

et al. 2015

PED

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OBJECT Choroid plexus carcinomas (CPCs) are rare brain tumors originating from the ventricular choroid plexus. They account for 2%–4% of all pediatric brain tumors and are most frequently seen in very young children. This pediatric proclivity, in combination with a marked vascularity, renders an aggressive resection a difficult and often dangerous endeavor. Blood losses of several total blood volumes in small children are not uncommon, sometimes forcing the neurosurgeon to abort the procedure, often leaving residual tumor. Great extent of tumor resection is an accepted beneficial factor for overall survival. Therefore, a second resection usually follows the administration of adjuvant chemotherapy. Second-look surgery appears to be associated with markedly decreased blood loss. Histological examination of specimens obtained at a second intervention shows decreased vascularity and fibrotic changes in tumor tissue. At the Hospital for Sick Children in Toronto, this empirical finding led to the strategy of neoadjuvant chemotherapy to minimize blood loss and maximize cytoreduction. The authors undertook this study to assess the potentially beneficial effect of neoadjuvant chemotherapy on blood loss during surgery for CPCs. METHODS In this retrospective cohort review, the demographic, clinical, and treatment parameters of 22 consecutive patients diagnosed with CPC are presented. All underwent surgical treatment at the Hospital for Sick Children from 1982 to 2013. Special attention was given to the impact of neoadjuvant chemotherapy on extent of resection and intraoperative blood loss. Extent of resection was calculated based on perioperative neuroimaging, and amount of blood loss was estimated based on transfusion parameters and perioperative changes in hematocrit. RESULTS Ten patients did not receive neoadjuvant chemotherapy, and 12 were treated with 2–5 cycles of ICE (ifosfamide, carboplatin, etoposide) chemotherapy in a neoadjuvant fashion. The 22 patients included in the study underwent a total of 37 tumor resection surgeries. In all of the cases in which neoadjuvant chemotherapy was used, at least a near-total resection (> 95% of tumor volume) was achieved. Patients who underwent gross-total resection had prolonged overall survival. Of the 37 resections, 18 were performed after chemotherapy. Mean blood loss in the neoadjuvant chemotherapy group was 22% of total estimated blood volume as opposed to 96% in patients without preoperative chemotherapy. CONCLUSIONS In children with CPC, the administration of neoadjuvant chemotherapy decreases intraoperative blood loss and increases extent of resection with a significant positive effect on overall survival.

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“…The lesions are commonly located in the lateral and the fourth ventricles, and rarely in the cerebellopontine angle and the third ventricle (3,4). The majority of CPTs arise with clinical symptoms that are associated with hydrocephalus as a result of direct mechanical obstruction of the flow of cerebrospinal fluid (CSF) and arachnoid granulation blockage from hemorrhage and overproduction of CSF (1,5).…”

Section: Introductionmentioning

confidence: 99%

“…According to the World Health Organization (WHO) classification scheme, CPTs are diagnosed as choroid plexus papilloma (CPP, WHO grade I), atypical choroid plexus papilloma (ACPP, WHO grade II) and choroid plexus carcinoma (CPC, WHO grade III) (3). For all classifications, the general treatment strategy is surgical resection unless this is not possible due to tumor location, for example.…”

Section: Introductionmentioning

confidence: 99%

“…Gross total resection (GTR) is the indicated course of treatment for CPP as well as for ACPP. CPP is a benign neoplasm and recurrence following GTR is rare, therefore adjuvant radiotherapy and chemotherapy are not generally recommended (2,3,6,7). ACPP cases exhibit atypical histological features along with increased mitotic activity; however, patients with this type of tumor also exhibit a good prognosis following GTR.…”

Section: Introductionmentioning

confidence: 99%

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Increased NG2 and SOX2 expression is associated with high-grade choroid plexus tumors

Zhao

Feng

et al. 2017

Oncology Letters

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Abstract. The World Health Organization classification of choroid plexus tumors (CPT) includes three distinct grades:Choroid plexus papilloma (CPP), atypical choroid plexus papilloma (ACPP) and choroid plexus carcinoma (CPC). The molecular basis for these pathological distinctions may help to stratify tumors and provide an insight into the clinical behavior of CPTs. In the present study, the progenitor and stem cell markers neuron glia antigen-2 (NG2) and sex-determining region Y-box 2 (SOX2) were investigated as potential biomarkers that may distinguish between distinct CPT grades. Immunohistochemistry was used to determine the expression of NG2 and SOX2 in CPTs (n=34) from Chinese patients (21 males and 13 females) with a mean age of 31.1 years (range, 1-63 years). The proportion of cells stained were scored using a scale between 0 and 3+, where 0 represents no staining and 3+ represents strong staining, and mean scores for each marker were determined on the basis of tumor grade. Pathological diagnosis revealed a distribution of cases as follows: CPP, 25; ACPP, 5; and CPC, 4. NG2 and SOX2 were expressed in CPTs of all grades. The mean labeling indices for CPP, ACPP and CPC were 1.12, 1.80 and 2.75 for NG2, respectively, and 1.20, 2.00 and 3.00 for SOX2, respectively. Statistical analysis of the mean labeling indices revealed a significant association between the expression of NG2 and SOX2 and CPT grade (P= 0.001 and <0.001 for CPP/ACPP and CPP/CPC, respectively). The results of the present study indicated that increased expression of NG2 and SOX2 was associated with higher-grade tumors and that these markers may be useful in determining CPT grade. IntroductionChoroid plexus tumors (CPTs) are rare intracranial neoplasms that derive from the choroid plexus epithelium of the ventricles (1,2). The lesions are commonly located in the lateral and the fourth ventricles, and rarely in the cerebellopontine angle and the third ventricle (3,4). The majority of CPTs arise with clinical symptoms that are associated with hydrocephalus as a result of direct mechanical obstruction of the flow of cerebrospinal fluid (CSF) and arachnoid granulation blockage from hemorrhage and overproduction of CSF (1,5).Treatment of CPTs is currently based on histological diagnosis. According to the World Health Organization (WHO) classification scheme, CPTs are diagnosed as choroid plexus papilloma (CPP, WHO grade I), atypical choroid plexus papilloma (ACPP, WHO grade II) and choroid plexus carcinoma (CPC, WHO grade III) (3). For all classifications, the general treatment strategy is surgical resection unless this is not possible due to tumor location, for example. Gross total resection (GTR) is the indicated course of treatment for CPP as well as for ACPP. CPP is a benign neoplasm and recurrence following GTR is rare, therefore adjuvant radiotherapy and chemotherapy are not generally recommended (2,3,6,7). ACPP cases exhibit atypical histological features along with increased mitotic activity; however, patients with this type of tumor also e...

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Clinical outcome of pediatric choroid plexus tumors: retrospective analysis from a single institute

Abstract: CPP and ACPP were surgically curable. Multi-modal treatments are necessary in the management of CPC with poor prognosis. HDCT and aPBSCT may be important to treat infants for whom radiotherapy is limited. Hydrocephalus and subdural effusion should be resolved with appropriate management.

Cited by 41 publications

References 36 publications

Pediatric choroid plexus tumors: epidemiology, treatments, and outcome analysis on 202 children from the SEER database

Pediatric choroid plexus tumors: epidemiology, treatments, and outcome analysis on 202 children from the SEER database

Neoadjuvant chemotherapy reduces blood loss during the resection of pediatric choroid plexus carcinomas

Increased NG2 and SOX2 expression is associated with high-grade choroid plexus tumors

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