Radiotherapy for pediatric adrenocortical carcinoma – Review of the literature

Wiegering, Verena; Riedmeier, Maria; Thompson, Lester D.�R.; Virgone, Calogero; Redlich, Antje; Kuhlen, Michaela; Gültekin, M.; Yalçın, Bilgehan; Decarolis, Boris; Härtel, Christoph; Schlegel, Paul‐Gerhardt; Fassnacht, Martin; Timmermann, Beate

doi:10.1016/j.ctro.2022.05.003

Cited by 3 publications

(3 citation statements)

References 59 publications

(69 reference statements)

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“…Unfortunately, new therapeutic strategies targeting tumor-specific aberrant pathways have not been studied in pediatric adrenocortical carcinoma. And Radiation therapy in pediatric adrenocortical carcinoma needs further exploration ( 50 ).…”

Section: Discussionmentioning

confidence: 99%

Long-term survival outcomes of pediatric adrenal malignancies: An analysis with the upstaged SEER registry during 2000-2019

Luo

et al. 2022

Front. Endocrinol.

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ObjectiveTo investigate the clinicopathological characteristics and long-term survival outcomes of pediatric adrenal malignancies.MethodThis study retrospectively analyzed children with pathologically confirmed pediatric adrenal malignancies from Surveillance, Epidemiology, and End Results Database from 2000 to 2019. Kaplan-Meier curve was used to assess the overall survival (OS) and cancer-special survival (CSS), and the Log-Rank method was used to calculate statistical differences. Cox proportional hazards model and Fine-and-Grey model were used to calculate the hazard ratio (HR) of all-cause mortality risk and the sub-distribution HR (sHR) of disease-specific mortality risk, respectively, and their corresponding 95% confidence intervals (CI).Results1601 children were included in the study in which 1335 (83.4%) neuroblastoma, 151 (9.4%) ganglioneuroblastoma, 89 (5.6%) adrenocortical carcinoma, and 26 (1.6%) were diagnosed with other types malignancies. Metastatic disease accounted for the largest proportion (69.3%), and the proportion of metastases diagnosed by neuroblastoma was higher than that of adrenocortical carcinoma and ganglioneuroblastoma (73.9% vs. 45.7% vs. 47.2%). The 5-year OS and CSS of all cohort were 69.5% and 70.5%, respectively. Adrenal cortical carcinoma had the worst prognosis, with 5-year OS and CSS of 52.5% and 53.1%, respectively. Patients in recent years had no better OS and CSS than in previous years at diagnosis. The tumor stage remained the main prognostic predictor. Compared to metastatic adrenal tumors, the risk of all-cause mortality (adjusted HR: 0.12, 95% CI: 0.06-0.25, P < 0.001) and the risk of disease-specific mortality (adjusted sHR: 0.11, 95% CI: 0.05-0.25, P<0.001) was significantly lower for patients with localized diseases. Additionally, higher age, adrenal cortical carcinoma, and lack of complete tumor resection are independent risk factors for poor prognosis. Furthermore, it was found that the prognosis of patients who received chemotherapy was worse than those who did not, mainly because the former mostly had metastasis at the presentation and complete resection of the tumor cannot be achieved.ConclusionThe clinicopathological characteristics of pediatric adrenal malignancies have not changed significantly in the past two decades, while the prognosis of patients has improved. Early diagnosis of disease and complete resection of local tumors are the keys to improving prognosis.

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Section: Discussionmentioning

confidence: 99%

Long-term survival outcomes of pediatric adrenal malignancies: An analysis with the upstaged SEER registry during 2000-2019

Luo

et al. 2022

Front. Endocrinol.

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“…It should not go unmentioned that radiotherapy to the tumour bed and CNS metastases, respectively, was administered in 2 of the surviving patients. The role of radiotherapy in the treatment of paediatric ACC, however, has yet not been determined [26].…”

Section: Tablementioning

confidence: 99%

Refractory and relapsed paediatric ACC in the MET studies – A challenging situation necessitating novel diagnostic and therapeutic concepts

Kuhlen,

Kunstreich,

Lessel

et al. 2023

EJC Paediatric Oncology

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“…Adult patients who received RT as an adjuvant treatment had higher overall survival (p=0.004) than patients treated with surgery alone (50). Wiegering at al (51). analysed cases of children with ACC, in whom RT was administered.…”

Section: Treatmentmentioning

confidence: 99%

Case Report: Adrenocortical carcinoma in children—symptoms, diagnosis, and treatment

Zagojska,

Malka,

Gorecka

et al. 2023

Front. Endocrinol.

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Adrenocortical carcinomas are extremely rare in the paediatric population. Most of them are hormone-secretive lesions; therefore, they should be taken into consideration in a child with signs of precocious puberty and/or Cushing’s syndrome symptoms. Nonetheless, differentiation from benign adrenal tumours is necessary. We report a rare case of adrenocortical carcinoma in a girl and a literature review using the PubMed database. A four-year-old girl presented with rapidly progressing precocious puberty and signs of Cushing’s syndrome. Imaging of the abdomen revealed a large heterogeneous solid mass. Histopathologic evaluation confirmed adrenocortical carcinoma with high mitotic activity, atypical mitoses, pleomorphism, necrosis, and vascular invasion. After tumourectomy, a decrease of previously elevated hormonal blood parameters was observed. Genetic tests confirmed Li Fraumeni syndrome. Adrenocortical carcinoma should be suspected in children with premature pubarche and signs of Cushing’s syndrome. Diagnosis must be based on clinical presentation, hormonal tests, imaging, and histopathological evaluation. Complete surgical resection of the tumour is the gold standard. Oncological treatment in children is not yet well-studied and should be individually considered, especially in advanced, inoperable carcinomas with metastases. Genetic investigations are useful for determining the prognosis in patients and their siblings.

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Radiotherapy for pediatric adrenocortical carcinoma – Review of the literature

Cited by 3 publications

References 59 publications

Long-term survival outcomes of pediatric adrenal malignancies: An analysis with the upstaged SEER registry during 2000-2019

Long-term survival outcomes of pediatric adrenal malignancies: An analysis with the upstaged SEER registry during 2000-2019

Refractory and relapsed paediatric ACC in the MET studies – A challenging situation necessitating novel diagnostic and therapeutic concepts

Case Report: Adrenocortical carcinoma in children—symptoms, diagnosis, and treatment

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