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Strahlentherapie und Onkologie

, Volume 189, Issue 9, pp 759–764 | Cite as

Incidence and clinical course of radionecrosis in children with brain tumors

A 20-year longitudinal observational study
  • V. StrengerEmail author
  • H. Lackner
  • R. Mayer
  • P. Sminia
  • P. Sovinz
  • M. Mokry
  • A. Pilhatsch
  • M. Benesch
  • W. Schwinger
  • M. Seidel
  • D. Sperl
  • S. Schmidt
  • C. Urban
Original article

Abstract

Radionecrosis (RN) in children treated for brain tumors represents a potentially severe long-term complication. Its diagnosis is challenging, since magnetic resonance imaging (MRI) cannot clearly discriminate between RN and tumor recurrence. A retrospective single-center study was undertaken to describe the incidence and clinical course of RN in a cohort of 107 children treated with external radiotherapy (RT) for various brain tumors between 1992 and 2012. During a median follow-up of 4.6 years (range 0.29–20.1 years), RN was implied by suspicious MRI findings in in 5 children (4.7 %), 5–131 months after RT. Suspicion was confirmed histologically (1 patient) or substantiated by FDG positron-emission tomography (FDG-PET, 2 patients) or by FDG-PET and MR spectroscopy (1 patient). Before developing RN, all 5 patients had received cytotoxic chemotherapy in addition to RT. In addition to standard treatment protocols, 2 patients had received further chemotherapy for progression or relapse. Median radiation dose expressed as the biologically equivalent total dose applied in 2 Gy fractions (EQD2) was 51.7 Gy (range 51.0–60.0 Gy). At RN onset, 4 children presented with neurological symptoms. Treatment of RN included resection (n = 1), corticosteroids (n = 2) and a combination of corticosteroids, hyperbaric oxygen (HBO) and bevacizumab (n = 1). One patient with asymptomatic RN was not treated. Complete radiological regression of the lesions was observed in all patients. Clinical symptoms normalized in 3 patients, whereas 2 developed permanent severe neurological deficits. RN represents a severe long-term treatment complication in children with brain tumors. The spectrum of clinical presentation is wide; ranging from asymptomatic lesions to progressive neurological deterioration. FDG-PET and MR spectroscopy may be useful for distinguishing between RN and tumor recurrence. Treatment options in patients with symptomatic RN include conservative management (steroids, HBO, bevacizumab) and surgical resection.

Keywords

Paediatric oncology Radiotherapy Chemotherapy Late effects Cerebral radionecrosis 

Inzidenz und klinischer Verlauf von Radionekrosen bei Kindern mit Schädeltumoren

Eine 20-Jahres-Langzeit-Beobachtungsstudie

Zusammenfassung

Radionekrosen (RN) bei Kindern nach Behandlung von Schädeltumoren stellen eine schwerwiegende Komplikation mit teilweise lebenslangen Spätfolgen dar. Die Unterscheidung zwischen RN und Tumorrezidiv oder -progression ist mittels Magnetresonanztomographie (MRT) nicht immer eindeutig möglich. In einer retrospektiven Single-Center-Studie beschreiben wir Inzidenz und klinischen Verlauf der RN. Bei 5 (4,7 %) von 107 Kindern, die in den Jahren 1992 bis 2012 wegen unterschiedlicher Schädeltumoren eine Strahlentherapie erhalten hatten, erhärtete sich während des medianen Nachbeobachtungszeitraums von 4,6 (0,29–20,1) Jahren 5–131 Monate nach Bestrahlung der Verdacht auf eine RN. Dieser wurde entweder histologisch (1 Patient) oder mittels FDG-Positronenemissionstomographie (FDG-PET; 2 Patienten) oder mittels MR-Spektroskopie und FDG-PET (1 Patient) bestätigt. Alle 5 Patienten waren vor Auftreten der RN mit einer zytotoxischen Chemotherapie behandelt worden. Wegen Relaps bzw. Progression wurden bei 2 Patienten weitere Chemotherapeutika – zusätzlich zum jeweiligen Standardtherapieprotokoll – verabreicht. Die mediane Strahlendosis, ausgedrückt als Bioequivalenzdosis EQD2, betrug 51,7 Gy (51,0–60,0 Gy). Neurologische Symptome beim Auftreten der RN zeigten 4 Kinder. Die Behandlung bestand aus Resektion (n = 1), Kortikosteroiden (n = 2) oder einer Kombination aus Kortikosteroiden, hyperbarer Oxigenierung und Bevacizumab (n = 1). Ein asymptomatischer Patienten erhielt keine Therapie. Bei allen Patienten kam es zu einer kompletten radiologischen Rückbildung der Läsionen. Bei 3 Patienten reduzierte sich die Symptomatik; 2 Patienten leiden weiterhin an schweren neurologischen Defiziten. RN stellen eine schwerwiegende Langzeitkomplikation bei Kindern nach Behandlung eines Schädeltumors dar. Die Bandbreite der klinischen Symptomatik reicht von asymptomatischen Läsionen bis hin zu fortschreitender neurologischer Symptomatik. FDG-PET und MR-Spektroskopie helfen, RN von Tumorrezidiv oder -progression zu unterscheiden. Die Behandlungsmöglichkeiten bei symptomatischen Patienten umfassen konservatives Management (Kortikosteroide, hyperbare Oxygenierung, Bevacizumab) sowie die chirurgische Resektion.

Schlüsselwörter

Pädiatrische Onkologie Strahlentherapie Chemotherapie Spätkomplikation Zerebrale Radionekrose 

Notes

Compliance with ethical guidelines

Conflict of interest. V. Strenger, H. Lackner, R. Mayer, P. Sminia, P. Sovinz, M. Mokry, A. Pilhatsch, M. Benesch, W. Schwinger, M. Seidel, D. Sperl, S. Schmidt and C. Urban state that there are no conflicts of interest.

All studies on humans described in the present manuscript were carried out with the approval of the responsible ethics committee and in accordance with national law and the Helsinki Declaration of 1975 (in its current, revised form). Informed consent was obtained from all patients included in studies.

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Copyright information

© Springer Heidelberg Berlin 2013

Authors and Affiliations

  • V. Strenger
    • 1
    Email author
  • H. Lackner
    • 1
  • R. Mayer
    • 2
  • P. Sminia
    • 3
  • P. Sovinz
    • 1
  • M. Mokry
    • 4
  • A. Pilhatsch
    • 5
  • M. Benesch
    • 1
  • W. Schwinger
    • 1
  • M. Seidel
    • 1
  • D. Sperl
    • 1
  • S. Schmidt
    • 1
  • C. Urban
    • 1
  1. 1.Division of Pediatric Hematology/OncologyMedical University of GrazGrazAustria
  2. 2.Department of RadiotherapyEBG MedAustron GmbHWiener NeustadtAustria
  3. 3.Department of Radiation OncologyVU University Medical CenterAmsterdamNetherlands
  4. 4.Department of NeurosurgeryMedical University of GrazGrazAustria
  5. 5.Division of Pediatric RadiologyMedical University of GrazGrazAustria

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