Pediatric pituitary adenomas are more aggressive, more likely to be hormone producing and are more difficult to cure than adult pituitary adenomas: case series and systematic literature review

Kelly, Alexander; Greenfield, Jeffrey P.; Dobri, Georgiana; Schwartz, Theodore H.

doi:10.1007/s00381-022-05445-3

Cited by 10 publications

(8 citation statements)

References 48 publications

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“…There were 11 (25.6%) patients with high-risk adenomas. Data from multiple centers have revealed that lactotroph adenoma is predominant among pediatric PAs, accounting for 39%–72.7% of all cases ( 25 – 27 ). In the published series of pediatric PAs patients who underwent surgery, lactotroph adenoma also accounts for the vast majority, approximately 47.1%–62% of all cases ( 27 – 29 ).…”

Section: Resultsmentioning

confidence: 99%

PD-L1 and tumor-infiltrating CD8+ lymphocytes are correlated with clinical characteristics in pediatric and adolescent pituitary adenomas

et al. 2023

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ObjectiveTo investigate the levels of tumor-infiltrating CD8+ lymphocytes (CD8+ TILs) and the expression of programmed cell death receptor ligand 1 (PD-L1) in the tumor microenvironment (TME) of pediatric and adolescent pituitary adenomas (PAPAs) and analyze the correlation between their levels and the clinical characteristics.MethodsA series of 43 PAPAs cases were enrolled over a period of 5 years. To compare the TME of PAPAs and adult PAs, 43 PAPAs cases were matched with 60 adult PAs cases (30 cases were between 20 and 40 years old, and 30 cases were older than 40 years) for main clinical characteristics. The expression of immune markers in PAPAs was detected by immunohistochemistry, and their correlation with the clinical outcomes was analyzed using statistical methods.ResultsIn the PAPAs group, CD8+ TILs level was significantly lower (3.4 (5.7) vs. 6.1 (8.5), p = 0.001), and PD-L1 expression (0.040 (0.022) vs. 0.024 (0.024), p < 0.0001) was significantly higher as compared with the older group. The level of CD8+ TILs was negatively correlated with the expression of PD-L1 (r = −0.312, p = 0.042). Moreover, CD8+ TILs and PD-L1 levels were associated with Hardy (CD8, p = 0.014; PD-L1, p = 0.018) and Knosp (CD8, p = 0.02; PD-L1, p = 0.017) classification. CD8+ TILs level was associated with high-risk adenomas (p = 0.015), and it was associated with the recurrence of PAPAs (HR = 0.047, 95% CI 0.003–0.632, p = 0.021).ConclusionCompared with the TME in adult PAs, the TME in PAPAs was found to express a significantly altered level of CD8+ TILs and PD-L1. In PAPAs, CD8+ TILs and PD-L1 levels were associated with clinical characteristics.

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Section: Resultsmentioning

confidence: 99%

PD-L1 and tumor-infiltrating CD8+ lymphocytes are correlated with clinical characteristics in pediatric and adolescent pituitary adenomas

et al. 2023

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“…A paediatric pituitary adenoma surgical series found that 55% (28/51) of patients had Ki-67 of ≥3% 5 . Furthermore, data on 42 paediatric pituitary tumours suggested that the combination of ≥3% Ki-67 and local invasion on imaging predicts a 25% recurrence rate after surgery 63 .…”

Section: Histopathologymentioning

confidence: 99%

“…Pituitary adenomas are neoplasms (usually non-malignant) arising from the hormone-secreting cells of the anterior pituitary. In CYP, they often secrete hormones in excess but the resulting characteristic signs and symptoms, such as pubertal delay, amenorrhoea, features of Cushing disease or rapid growth velocity, might be occult or missed during development, leading to late diagnoses 5,6 . Pituitary adenomas are defined as macroadenomas if they measure ≥1 cm and microadenomas if they measure <1 cm, whereas neoplasms of >4 cm in size are referred to as giant adenomas.…”

Section: Introductionmentioning

confidence: 99%

Consensus guideline for the diagnosis and management of pituitary adenomas in childhood and adolescence: Part 1, general recommendations

Korbonits,

Blair,

Boguslawska

et al. 2024

Nat Rev Endocrinol

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Tumours of the anterior part of the pituitary gland represent just 1% of all childhood (aged <15 years) intracranial neoplasms, yet they can confer high morbidity and little evidence and guidance is in place for their management. Between 2014 and 2022, a multidisciplinary expert group systematically developed the first comprehensive clinical practice consensus guideline for children and young people under the age 19 years (hereafter referred to as CYP) presenting with a suspected pituitary adenoma to inform specialist care and improve health outcomes. Through robust literature searches and a Delphi consensus exercise with an international Delphi consensus panel of experts, the available scientific evidence and expert opinions were consolidated into 74 recommendations. Part 1 of this consensus guideline includes 17 pragmatic management recommendations related to clinical care, neuroimaging, visual assessment, histopathology, genetics, pituitary surgery and radiotherapy. While in many aspects the care for CYP is similar to that of adults, key differences exist, particularly in aetiology and presentation. CYP with suspected pituitary adenomas require careful clinical examination, appropriate hormonal work-up, dedicated pituitary imaging and visual assessment. Consideration should be given to the potential for syndromic disease and genetic assessment. Multidisciplinary discussion at both the local and national levels can be key for management. Surgery should be performed in specialist centres. The collection of outcome data on novel modalities of medical treatment, surgical intervention and radiotherapy is essential for optimal future treatment. Sections Developing recommendationsWhere there was sufficient moderate-quality evidence to answer the PICO questions, the GDG originally made 54 recommendations whose strength was determined using the GRADE criteria 20 by a trade-off between benefits and harms given the quality of the underpinning evidence. Where the evidence base was lacking, of low quality, conflicting or largely extrapolated from adult experiences, the GDG formulated consensus recommendations and submitted these to experts outside the GDG (the international Delphi consensus panel, Supplementary Table 2) in three rounds of a Delphi consensus process (first round,

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“…During the pediatric age, they are more frequent in adolescents than in younger age groups, and while in the former they are more often seen in girls than boys, it is noteworthy that no gender predilection is found among pre-pubertal patients ( 78 ). In contrast to adult counterparts, the vast majority (about 90% of cases) of pediatric PitNETs requiring surgery are mainly prolactin- or ACTH-producing tumors ( 79 , 80 ).…”

Section: Sellar and Suprasellar Tumorsmentioning

confidence: 99%

Neuroimaging of pediatric tumors of the sellar region—A review in light of the 2021 WHO classification of tumors of the central nervous system

et al. 2023

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Sellar/suprasellar tumors comprise about 10% of all pediatric Central Nervous System (CNS) tumors and include a wide variety of entities, with different cellular origins and distinctive histological and radiological findings, demanding customized neuroimaging protocols for appropriate diagnosis and management. The 5th edition of the World Health Organization (WHO) classification of CNS tumors unprecedently incorporated both histologic and molecular alterations into a common diagnostic framework, with a great impact in tumor classification and grading. Based on the current understanding of the clinical, molecular, and morphological features of CNS neoplasms, there have been additions of new tumor types and modifications of existing ones in the latest WHO tumor classification. In the specific case of sellar/suprasellar tumors, changes include for example separation of adamantinomatous and papillary craniopharyngiomas, now classified as distinct tumor types. Nevertheless, although the current molecular landscape is the fundamental driving force to the new WHO CNS tumor classification, the imaging profile of sellar/suprasellar tumors remains largely unexplored, particularly in the pediatric population. In this review, we aim to provide an essential pathological update to better understand the way sellar/suprasellar tumors are currently classified, with a focus on the pediatric population. Furthermore, we intend to present the neuroimaging features that may assist in the differential diagnosis, surgical planning, adjuvant/neoadjuvant therapy, and follow-up of this group of tumors in children.

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Pediatric pituitary adenomas are more aggressive, more likely to be hormone producing and are more difficult to cure than adult pituitary adenomas: case series and systematic literature review

Cited by 10 publications

References 48 publications

PD-L1 and tumor-infiltrating CD8+ lymphocytes are correlated with clinical characteristics in pediatric and adolescent pituitary adenomas

PD-L1 and tumor-infiltrating CD8+ lymphocytes are correlated with clinical characteristics in pediatric and adolescent pituitary adenomas

Consensus guideline for the diagnosis and management of pituitary adenomas in childhood and adolescence: Part 1, general recommendations

Neuroimaging of pediatric tumors of the sellar region—A review in light of the 2021 WHO classification of tumors of the central nervous system

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