{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,5,14]],"date-time":"2025-05-14T04:49:22Z","timestamp":1747198162671,"version":"3.40.5"},"reference-count":15,"publisher":"Walter de Gruyter GmbH","issue":"7","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,7,26]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec id=\"j_jpem-2024-0138_abs_001\">\n                  <jats:title>Objectives<\/jats:title>\n                  <jats:p>Cushing\u2019s disease (CD) is a diagnostic and therapeutic challenge, especially in pediatric patients. CD, primarily caused by adrenocorticotropic hormone-secreting pituitary adenomas, manifests typically with growth retardation and weight gain. There are no published guidelines for pediatric patients.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2024-0138_abs_002\">\n                  <jats:title>Case presentation<\/jats:title>\n                  <jats:p>We report three pediatric patients diagnosed with CD in a Portuguese tertiary hospital. All patients presented with hypercortisolism features. All patients underwent transsphenoidal pituitary surgery (TSS) as a first-choice treatment; however, it was unsuccessful in one patient and the other patients experienced recurrence. Patients were submitted to different approaches so basal serum cortisol levels could be achieved. Two of three patients achieved remission.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2024-0138_abs_003\">\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>TSS remains the first-line treatment yet challenging due to microadenomas and technical complexities. Medical therapy with agents like metyrapone or ketoconazole, pituitary radiotherapy, or bilateral adrenalectomy are, usually, second-line interventions, unless there is a contraindication to surgery. Our findings support the finding that a shorter hypothalamic\u2013pituitary\u2013adrenal axis recovery time increases the risk of recurrence of CD. Our cases illustrate the intricate management and variable outcomes of pediatric CD, underscoring the importance of multidisciplinary care and continuous surveillance.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1515\/jpem-2024-0138","type":"journal-article","created":{"date-parts":[[2024,5,24]],"date-time":"2024-05-24T10:40:35Z","timestamp":1716547235000},"page":"663-667","source":"Crossref","is-referenced-by-count":0,"title":["Treatment modalities and outcomes in pediatric Cushing\u2019s disease\u00a0\u2013 report of three cases and literature review"],"prefix":"10.1515","volume":"37","author":[{"given":"Viviana","family":"Marques","sequence":"first","affiliation":[{"name":"Faculdade de Medicina da Universidade do Porto (FMUP) , Alameda Prof. Hern\u00e2ni Monteiro , Porto , Portugal"}]},{"given":"Sofia","family":"Ferreira","sequence":"additional","affiliation":[{"name":"Servi\u00e7o de Pediatria, ULS S\u00e3o Jo\u00e3o , Alameda Prof. Hern\u00e2ni Monteiro , Porto , Portugal"},{"name":"Departamento de Ginecologia-Obstetr\u00edcia e Pediatria , Faculdade de Medicina da Universidade do Porto (FMUP) , Porto , Portugal"}]},{"given":"Carla","family":"Costa","sequence":"additional","affiliation":[{"name":"Servi\u00e7o de Pediatria, ULS S\u00e3o Jo\u00e3o , Alameda Prof. Hern\u00e2ni Monteiro , Porto , Portugal"},{"name":"Departamento de Ginecologia-Obstetr\u00edcia e Pediatria , Faculdade de Medicina da Universidade do Porto (FMUP) , Porto , Portugal"}]},{"given":"C\u00edntia","family":"Castro-Correia","sequence":"additional","affiliation":[{"name":"Servi\u00e7o de Pediatria, ULS S\u00e3o Jo\u00e3o , Alameda Prof. Hern\u00e2ni Monteiro , Porto , Portugal"},{"name":"Departamento de Ginecologia-Obstetr\u00edcia e Pediatria , Faculdade de Medicina da Universidade do Porto (FMUP) , Porto , Portugal"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0338-3399","authenticated-orcid":false,"given":"Rita","family":"Santos Silva","sequence":"additional","affiliation":[{"name":"Servi\u00e7o de Pediatria, ULS S\u00e3o Jo\u00e3o , Alameda Prof. Hern\u00e2ni Monteiro , Porto , Portugal"},{"name":"Departamento de Ginecologia-Obstetr\u00edcia e Pediatria , Faculdade de Medicina da Universidade do Porto (FMUP) , Porto , Portugal"}]}],"member":"374","published-online":{"date-parts":[[2024,5,27]]},"reference":[{"key":"2024071118565843293_j_jpem-2024-0138_ref_001","doi-asserted-by":"crossref","unstructured":"Storr, HL, Savage, MO. 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Ann Pediatr Endocrinol Metab 2023;28:87\u201397. https:\/\/doi.org\/10.6065\/apem.2346074.037 [Internet].","DOI":"10.6065\/apem.2346074.037"},{"key":"2024071118565843293_j_jpem-2024-0138_ref_006","doi-asserted-by":"crossref","unstructured":"Storr, HL, Chan, LF, Grossman, AB, Savage, MO. Paediatric Cushing\u2019s syndrome: epidemiology, investigation and therapeutic advances. Trends Endocrinol Metab 2007;18:167\u201374. https:\/\/doi.org\/10.1016\/j.tem.2007.03.005.","DOI":"10.1016\/j.tem.2007.03.005"},{"key":"2024071118565843293_j_jpem-2024-0138_ref_007","doi-asserted-by":"crossref","unstructured":"Pomahacova, R, Paterova, P, Nykodymova, E, Sykora, J, Krsek, M. Pediatric Cushing\u2019s disease: case reports and retrospective review. Biomed Pap Med Fac Univ Palacky Olomouc Czech Repub 2022;166:20 [Internet].","DOI":"10.5507\/bp.2021.051"},{"key":"2024071118565843293_j_jpem-2024-0138_ref_008","doi-asserted-by":"crossref","unstructured":"Savage, MO, Storr, HL. Pediatric Cushing\u2019s disease: management issues. Indian J Endocrinol Metab 2012;16:S171\u20135. https:\/\/doi.org\/10.4103\/2230-8210.104032 [Internet].","DOI":"10.4103\/2230-8210.104032"},{"key":"2024071118565843293_j_jpem-2024-0138_ref_009","doi-asserted-by":"crossref","unstructured":"Savage, MO, Chan, LF, Afshar, F, Plowman, PN, Grossman, AB, Storr, HL. Advances in the management of paediatric Cushing\u2019s disease. Horm Res 2008;69:327\u201333. https:\/\/doi.org\/10.1159\/000117388.","DOI":"10.1159\/000117388"},{"key":"2024071118565843293_j_jpem-2024-0138_ref_010","doi-asserted-by":"crossref","unstructured":"Savage, MO, Ferrigno, R. Paediatric Cushing\u2019s disease: long-term outcome and predictors of recurrence. 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Arq Bras Endocrinol Metabol 2010;54:17\u201323. https:\/\/doi.org\/10.1590\/s0004-27302010000100004.","DOI":"10.1590\/S0004-27302010000100004"},{"key":"2024071118565843293_j_jpem-2024-0138_ref_013","doi-asserted-by":"crossref","unstructured":"Joshi, SM, Hewitt, RJ, Storr, HL, Rezajooi, K, Ellamushi, H, Grossman, AB, et al.. Cushing\u2019s disease in children and adolescents: 20 years of experience in a single neurosurgical center. Neurosurgery 2005;57:281\u20135. https:\/\/doi.org\/10.1227\/01.neu.0000166580.94215.53.","DOI":"10.1227\/01.NEU.0000166580.94215.53"},{"key":"2024071118565843293_j_jpem-2024-0138_ref_014","doi-asserted-by":"crossref","unstructured":"Tatsi, C, Neely, M, Flippo, C, Bompou, M, Keil, M, Stratakis, CA. Recovery of hypothalamic-pituitary-adrenal axis in paediatric Cushing disease. 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