Descriptive epidemiology of craniopharyngiomas in the United States

Momin, Arbaz; Recinos, Miguel A.; Cioffi, Gino; Patil, Nirav; Soni, Pranay; Almeida, João Paulo; Kruchko, Carol; Barnholtz‐Sloan, Jill S.; Recinos, Pablo F.; Kshettry, Varun R.

doi:10.1007/s11102-021-01127-6

Cited by 49 publications

(37 citation statements)

References 11 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Craniopharyngiomas are embryological tumors arising from the remnants of Rathke’s pouch along the craniopharyngeal canal. They are mainly located in the sellar/parasellar region, accounting for 2–5% of all the primary intracranial neoplasms ( 1 ). They can be detected at any age, and a bimodal age distribution has been reported, with one peak during 5–9 years in children and another during 55–69 years in adults ( 1 ).…”

Section: Introductionmentioning

confidence: 99%

“…They are mainly located in the sellar/parasellar region, accounting for 2–5% of all the primary intracranial neoplasms ( 1 ). They can be detected at any age, and a bimodal age distribution has been reported, with one peak during 5–9 years in children and another during 55–69 years in adults ( 1 ). Although histologically benign, the tumor often grows aggressively, causing damage to surrounding vital structures, such as the optic apparatus, the pituitary gland, and the hypothalamus.…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

Risk Factors for Hypothalamic Obesity in Patients With Adult-Onset Craniopharyngioma: A Consecutive Series of 120 Cases

Sun

Zhang

et al. 2021

Front. Endocrinol.

View full text Add to dashboard Cite

ContextHypothalamic obesity (HO) is a severe complication following craniopharyngioma, but studies regarding the sequelae in adult-onset patients with craniopharyngioma are sparse.ObjectiveThe objective of the study was to describe weight changes after surgical treatment in adult-onset craniopharyngioma patients and to analyze risk factors for postoperative weight gain and HO.Subjects and MethodA retrospective analysis was conducted of 120 adult-onset patients who underwent surgery for craniopharyngioma and follow-up at the institution of the authors between January 2018 and September 2020. Clinical characteristics, anthropometric data, image features, treatment modalities, and endocrine indices were collected. Multivariable logistic regression analysis was used to identify independent risk factors for postoperative weight gain and HO.ResultsForty-nine (40.8%) patients had clinically meaningful weight gain (≥5%) in a median follow-up time of 12.0 months (range 1.0–41.0 months) after surgery. The mean postoperative weight gain in this subgroup was 17.59 ± 12.28 (%). Weight gain continued in the first year following surgery. Patients with lower preoperative BMI [OR 0.78, 95% CI (0.67–0.90), P = 0.001] and the adamantinomatous subtype [OR 3.46, 95% CI (1.02–11.76), P = 0.047] were more likely to experience postoperative weight gain ≥5%. The prevalence of HO was 19.2% preoperatively and increased to 29.2% at last follow-up postoperatively. Only preoperative BMI [OR 2.51, 95% CI (1.64–3.85), P < 0.001] was identified as an independent risk factor for postoperative HO.ConclusionsHO is a common complication in patients with adult-onset craniopharyngioma. Patients with higher preoperative BMI had a greater risk for developing HO postoperatively.

show abstract

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Risk Factors for Hypothalamic Obesity in Patients With Adult-Onset Craniopharyngioma: A Consecutive Series of 120 Cases

Sun

Zhang

et al. 2021

Front. Endocrinol.

View full text Add to dashboard Cite

show abstract

“…From October 2018 to October 2020, a total of 118 adult patients underwent EEES for primary craniopharyngiomas at Beijing Tiantan Hospital of Capital Medical University. Inclusion criteria were as follows: (1) adult patients aged ≥18 years, (2) newly confirmed diagnosis of craniopharyngioma, (3) computed tomography (CT), magnetic resonance imaging (MRI), and ophthalmologic tests before and after surgery. The exclusion criteria were (1) past medical history of treatment including radiotherapy and surgery, (2) any ophthalmic condition other than compressive optic neuropathy caused by craniopharyngiomas, (3) any medical illness (including glaucoma, diabetes mellitus) known to affect optic apparatus, (4) ineligible OCT parameters, (5) unreliable visual field (VF) and best-corrected visual acuity (BCVA) testing (fixation losses more than 20%, false-negative error more than 20%, and false-positive error more than 20%), ( 6) myopia greater than -6.00 diopters, (7) and papilledema on fundoscopy.…”

Section: Patient Populationmentioning

confidence: 99%

“…Craniopharyngiomas are rare brain tumors originating from any point along with the pituitary-hypothalamic axis, accounting for 1.2%-4.6% of all intracranial tumors (1,2). Because of the close vicinity of optic chiasma, visual deterioration is known as a common complication following surgery for craniopharyngiomas (3)(4)(5)(6)(7).…”

Section: Introductionmentioning

confidence: 99%

Prognostic Utility of Optical Coherence Tomography for Visual Outcome After Extended Endoscopic Endonasal Surgery for Adult Craniopharyngiomas

Qiao

et al. 2022

Front. Oncol.

View full text Add to dashboard Cite

IntroductionOwing to the close vicinity of the optic chiasma, visual dysfunction is known as one of the most common surgical indications and postoperative complications in adult patients with craniopharyngiomas, probably leading to poor quality of life. Historically, very few consistent predictive factors associated with the visual outcome are identified, which may not be helpful for patient counseling and preoperative decision making. Recently, optical coherence tomography (OCT) serving as a novel high-resolution imaging technique can assess the retinal morphology by measuring the circumpapillary retinal nerve fiber layer (cpRNFL) and macular ganglion cell complex thickness. However, few studies have examined the prognostic utility of OCT parameters for visual outcome after surgery for craniopharyngiomas. This study aims to use the largest series to evaluate the association between OCT parameters and visual outcome after extended endoscopic endonasal surgery (EEES) for primary craniopharyngiomas in adults.Material and MethodsFrom October 2018 to October 2020, one hundred and seventy eyes in 88 adult patients with newly confirmed craniopharyngiomas were retrospectively reviewed and pertinent prognostic factors were analyzed.ResultsGross total resection was performed in 82 (93.2%) patients. The median postoperative follow-up time was 10.9 months. Multiple logistic regression analysis showed that increased temporal cpRNFL thickness was associated with higher odds of visual acuity (VA) improvement and maintenance (OR = 1.070; 95% CI, 1.005–1.140; p = 0.035), and greater inferior cpRNFL thickness was significantly associated with visual field (VF) improvement and maintenance (OR = 1.034; 95% CI, 1.001–1.068; p = 0.046). Furthermore, tight adhesion between optic nerves and craniopharyngiomas was demonstrated as an independent adverse factor for either postoperative VA or VF (p = 0.048, p = 0.030, respectively). The ROC results further verified the robustness of the prediction model either in VA (AUC = 0.843; 95% CI, 0.734–0.952; p < 0.001) or VF (AUC = 0.849; 95% CI, 0.741–0.958; p < 0.001).ConclusionPreoperative OCT can effectively predict visual outcome after EEES for adult craniopharyngiomas. It can also serve as a reliable alternative to evaluate preoperative visual field defects, especially for patients with lower compliance. Tight adhesion was confirmed as an independent risk factor for postoperative visual outcome. The OCT-based multivariable prediction models developed in the present study may contribute to patient counseling on visual prognosis.

show abstract

“…Craniopharyngioma (CP) is a rare primary type of benign brain tumor with incidence rate of 0.16 per 100,000 [ 1 , 2 ]. CP develops in the sellar region adjacent to many important functional structures, such as optic nerve, optic chiasm, pituitary stalk, hypothalamus and internal carotid artery [ 3 ].…”

Section: Introductionmentioning

confidence: 99%

Characterization of novel CTNNB1 mutation in Craniopharyngioma by whole-genome sequencing

Zeng

Wang

et al. 2021

Mol Cancer

View full text Add to dashboard Cite

Background Craniopharyngioma (CP) is rare histologically benign but clinically challenging tumor because of its intimate relationship with the critical structure in the central brain. CP can be divided into two major histologic subtypes: adamantinomatous-type CP (ACP) and papillary-type CP (PCP). Although some genetic aberrations for both categories have been revealed in previous studies, the complete spectrum of genetic changes of this tumor remains unknown. Methods In this study, we conducted whole genome sequencing (WGS) on twenty-six CPs including 16 ACPs and 10 PCPs together with their matched blood samples. Somatic variants (SNVs, InDels, SVs and CNVs) were identified and mutational signatures were characterized for each patient. We investigated the impact of a novel CTNNB1 mutant on its protein stability, ubiquitination and Wnt pathway activity. Cell proliferation ability of the CTNNB1 mutant in ACP primary cells was additionally analyzed by CCK8 and colony formation assays. Results We found that CPs had showed less complexity with fewer somatic mutations compared with malignant tumors. Moreover, mutations in CTNNB1 (68.75% of ACP) and BRAF V600E (70.00% of PCP) are mutually exclusive in ACP and PCP, consolidating that the driving roles of these two genes in ACP and PCP, respectively. A novel mutation in the exon 3 of CTNNB1 which compromised both a transversion and in-frame deletion was identified in ACP. This mutation was experimentally validated to confer β-catenin increased stability by inhibiting its ubiquitination, thus activating Wnt-signaling pathway and promoting cell proliferation. Conclusions Whole genome landscape for CP was revealed by WGS analysis, and a novel mutation in the exon 3 of CTNNB1 was identified. This novel mutation activates Wnt-signaling pathway through increasing the stability of β-catenin. Our findings provided us with more comprehensive insight into the spectrum of genetic alterations in CP.

show abstract

Descriptive epidemiology of craniopharyngiomas in the United States

Cited by 49 publications

References 11 publications

Risk Factors for Hypothalamic Obesity in Patients With Adult-Onset Craniopharyngioma: A Consecutive Series of 120 Cases

Risk Factors for Hypothalamic Obesity in Patients With Adult-Onset Craniopharyngioma: A Consecutive Series of 120 Cases

Prognostic Utility of Optical Coherence Tomography for Visual Outcome After Extended Endoscopic Endonasal Surgery for Adult Craniopharyngiomas

Characterization of novel CTNNB1 mutation in Craniopharyngioma by whole-genome sequencing

Contact Info

Product

Resources

About