Rapidly Fatal Ectopic Adrenocorticotropic Hormone Syndrome in a 9-Year-Old Girl With Ewing Sarcoma

Varghese, Johann; Mythili, A.; Reddy, Sagar S.L.; Mandapati, Navya Sruthi; Subrahmanyam, K

doi:10.1016/j.aace.2021.09.003

AACE Clinical Case Reports

2022

DOI: 10.1016/j.aace.2021.09.003

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Rapidly Fatal Ectopic Adrenocorticotropic Hormone Syndrome in a 9-Year-Old Girl With Ewing Sarcoma

Johann Varghese

A. Mythili

Sagar S.L. Reddy

et al.

Abstract: This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, a… Show more

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Cited by 3 publications

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“… 8 Authors also reported an interesting case of “vanishing bone disease” in a 22-year-old man, 9 and another case of ectopic ACTH secretion due to Ewing sarcoma in a 9-year-old girl is reported. 10 …”

mentioning

confidence: 99%

Editorial for March/April Issue of AACE Clinical Case Reports

Jasim¹

2022

AACE Clinical Case Reports

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mentioning

confidence: 99%

Editorial for March/April Issue of AACE Clinical Case Reports

Jasim¹

2022

AACE Clinical Case Reports

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Cushing Syndrome in an Infant Due to Paraneoplastic Syndrome Associated with Ewing Sarcoma

Priyadarshini,

Haldorai,

Verma

et al. 2024

Indian J Pediatr

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Paediatric and adolescent ectopic Cushing's syndrome: systematic review

Yami Channaiah,

Karlekar,

Sarathi

et al. 2023

European Journal of Endocrinology

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Objective The data on clinical, biochemical, radiological characteristics and outcomes in pediatric ectopic adrenocorticotropic hormone syndrome (EAS) is limited owing to rarity of the condition. We report three new cases and perform a systematic review of pediatric EAS. Design and Method Case records of pediatric and adolescent EAS patient’s ≤20 years presenting at our center between 1997-2021 were retrospectively reviewed and a systematic review of the literature published between January 1970 to December 2022 was performed. Results A total of 161 patients including three new patients from our center were identified. Bronchial neuroendocrine tumors (NET) (28.5%), thymic NET (22.9%), primitive cell-derived tumors (PCDT, 18.6%), and gastro-entero-pancreatic-NET (13.7%) were the common causes. PCDT were the most common in the first decade (24/45, 53.4%) and were the largest [82 (60-100) mm], whereas bronchial NETs predominated during the second decade (42/116, 36.2%) and were the smallest [15 (10-25) mm]. Computed tomography localized 92.9% (118/127) of pediatric EAS patients. Immediate postoperative remission was attained in 77.9% (88/113) patients whereas 30.4% (24/79) relapsed over a median (IQR) period of 13 (8-36) months. Over a median (IQR) follow-up of 2 (0.6-4.6) years, 31.4% of patients died. The median survival was higher in bronchial NET than in other tumor groups. Distant metastasis and tumor size were independent negative predictors of survival. Conclusions Etiological profile of pediatric and adolescent EAS is distinct from that of adults. Bronchial NETs have the best long-term survival whereas distant metastasis and tumor size predict poor survival.

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Rapidly Fatal Ectopic Adrenocorticotropic Hormone Syndrome in a 9-Year-Old Girl With Ewing Sarcoma

Cited by 3 publications

References 15 publications

Editorial for March/April Issue of AACE Clinical Case Reports

Editorial for March/April Issue of AACE Clinical Case Reports

Cushing Syndrome in an Infant Due to Paraneoplastic Syndrome Associated with Ewing Sarcoma

Paediatric and adolescent ectopic Cushing's syndrome: systematic review

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