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Outcomes After Gamma Knife Stereotactic Radiosurgery in Pediatric Patients with Cushing Disease or Acromegaly: A Multi-Institutional Study

A. Shrivastava, N. Mohammed, Z. Xu, R. Liščák, M. Kosak, M. Krsek, KA. Karim, CC. Lee, N. Martínez-Moreno, M. Lee Vance, LD. Lunsford, JP. Sheehan,

. 2019 ; 125 (-) : e1104-e1113. [pub] 20190218

Jazyk angličtina Země Spojené státy americké

Typ dokumentu časopisecké články, multicentrická studie

Perzistentní odkaz   https://www.medvik.cz/link/bmc20006579

OBJECTIVE: Pituitary adenomas comprise about 3% of all intracranial tumors in pediatric patients. This study examines the role of stereotactic radiosurgery in the management of pediatric acromegaly or patients with Cushing disease (CD). METHODS: From an international consortium, we retrospectively collected treatment and outcome data on pediatric adrenocorticotrophic hormone and growth hormone-secreting pituitary adenomas treated with Gamma Knife radiosurgery (GKRS). There were a total of 36 patients including 24 with CD and 12 with acromegaly. The data were analyzed to assess outcomes including tumor control, endocrine remission, and adverse effects. Statistical analysis was performed to determine correlation between clinical/treatment parameters and outcomes. RESULTS: At the last follow-up after GKRS, endocrine remission rates for CD and acromegaly were 80% and 42%, respectively. Tumor control was achieved in 87.5% of patients with CD and in 42% of patients with acromegaly. New pituitary hormone deficiency occurred in 7 of the 36 patients at a median time of 18 months after GKRS (range, 12-81 months). The predictive factors for endocrine remission were age <15 years (P = 0.015) and margin dose (P = 0.042). The median endocrine follow-up was 63.7 months (range, 7-246 months). CONCLUSIONS: GKRS affords reasonable rates of endocrine remission and tumor control in most pediatric patients with functioning adenomas. The most common post-GKRS complication was hypopituitarism, although this occurred in only a few patients. Given the larger at-risk period for pediatric patients, further study is required to evaluate for delayed recurrences and hypopituitarism.

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$a OBJECTIVE: Pituitary adenomas comprise about 3% of all intracranial tumors in pediatric patients. This study examines the role of stereotactic radiosurgery in the management of pediatric acromegaly or patients with Cushing disease (CD). METHODS: From an international consortium, we retrospectively collected treatment and outcome data on pediatric adrenocorticotrophic hormone and growth hormone-secreting pituitary adenomas treated with Gamma Knife radiosurgery (GKRS). There were a total of 36 patients including 24 with CD and 12 with acromegaly. The data were analyzed to assess outcomes including tumor control, endocrine remission, and adverse effects. Statistical analysis was performed to determine correlation between clinical/treatment parameters and outcomes. RESULTS: At the last follow-up after GKRS, endocrine remission rates for CD and acromegaly were 80% and 42%, respectively. Tumor control was achieved in 87.5% of patients with CD and in 42% of patients with acromegaly. New pituitary hormone deficiency occurred in 7 of the 36 patients at a median time of 18 months after GKRS (range, 12-81 months). The predictive factors for endocrine remission were age <15 years (P = 0.015) and margin dose (P = 0.042). The median endocrine follow-up was 63.7 months (range, 7-246 months). CONCLUSIONS: GKRS affords reasonable rates of endocrine remission and tumor control in most pediatric patients with functioning adenomas. The most common post-GKRS complication was hypopituitarism, although this occurred in only a few patients. Given the larger at-risk period for pediatric patients, further study is required to evaluate for delayed recurrences and hypopituitarism.
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$a Mohammed, Nasser $u Department of Neurosurgery, University of Virginia, Charlottesville, Virginia, USA.
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$a Xu, Zhiyuan $u Department of Neurosurgery, University of Virginia, Charlottesville, Virginia, USA.
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$a Liščák, Roman $u Department of Stereotactic and Radiation Neurosurgery, Na Homolce Hospital, Prague, USA.
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$a Kosak, Mikulas $u Department of Stereotactic and Radiation Neurosurgery, Na Homolce Hospital, Prague, USA.
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$a Karim, Khaled Abdel $u Department of Clinical Oncology, Ain-Shams University, Cairo, USA.
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$a Lee, Cheng-Chia $u Department of Neurosurgery, Taipei Veteran General Hospital, Taiwan, USA.
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$a Martínez-Moreno, Nuria $u Department of Neurological Surgery and Gamma Knife Radiosurgery, Ruber International Hospital, Madrid, USA.
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$a Lee Vance, Mary $u Department of Medicine, University of Virginia Health System, Charlottesville, Virginia, USA.
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$a Lunsford, L Dade $u Department of Neurosurgery, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania, USA.
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$a Sheehan, Jason P $u Department of Neurosurgery, University of Virginia, Charlottesville, Virginia, USA. Electronic address: jps2f@virginia.edu.
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