Coexistence of GH-Producing Pituitary Macroadenoma and Meningioma in a Patient with Multiple Endocrine Neoplasia Type 1 with Hyperglycemia and Ketosis as First Clinical Sign

Herrero-Ruiz, A.; Villanueva-Alvarado, H. S.; Corrales, J. J.; Higueruela-Mínguez, Cecilia; Feito, Jorge; Recio-Córdova, José María

doi:10.1155/2017/2390797

Cited by 7 publications

(6 citation statements)

References 14 publications

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“…Pituitary adenoma and meningioma are the most common benign tumors in the CNS; there were only 33 PAMs described before 2017 [7]. We summarized 8197 pituitary adenomas in our hospital, and there were 57 PAMs, 5 pituitary adenoma associated with glioma and 5 pituitary adenoma associated with schwannoglioma in the present study.…”

Section: Discussionmentioning

confidence: 73%

“…Meningiomas are generally slow-growing tumors derived from the arachnoid membrane surrounding the central nervous system and they are among the most common intracranial tumors, with an overall incidence of 6 per 100,000 (15–25% of all brain tumors) and a 2:1 female to male ratio [3–6]. PAM is a rare clinical situation, and there were only 33 cases described before 2017 [7]. The precise cause for the development of PAM remains unknown.…”

Section: Introductionmentioning

confidence: 99%

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The clinical characteristics and molecular mechanism of pituitary adenoma associated with meningioma

et al. 2019

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BackgroundPituitary adenoma and meningioma are the most common benign tumors in the central nervous system. Pituitary adenoma associated with meningioma (PAM) is a rare disease and the clinical features and mechanisms of PAM are unclear.MethodsWe summarized the clinical data of 57 PAM patients and compared with sporadic pituitary adenoma (SPA) and sporadic meningioma (SM). 5 pituitary adenomas of PAM and 5 SPAs were performed ceRNA microarray. qRT-PCR, Western Blot, siMEN1 and rapamycin inhibition experiment were validated for ceRNA microarray.ResultsClinical variable analyses revealed that significant correlations between PAM and female sex as well as older age when compared with SPA and significant correlations between PAM and transitional meningioma as well as older age when compared with SM. Additionally, the characteristics of PAM were significantly different for MEN1 patients. Functional experiments showed lower expression of MEN1 can upregulate mTOR signaling, in accordance with the result of ceRNA microarray. Rapamycin treatment promotes apoptosis in primary pituitary adenoma and meningioma cells of PAM.ConclusionsMEN1 plays an important role in PAM by upregulating mTOR signaling pathway. Rapamycin represents a potential therapeutic strategy for PAM in the future.

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Section: Discussionmentioning

confidence: 73%

Section: Introductionmentioning

confidence: 99%

The clinical characteristics and molecular mechanism of pituitary adenoma associated with meningioma

et al. 2019

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“…Pituitary adenoma and meningioma are the most common benign tumors in the CNS (9). A total of 57 novel PAM cases were identified from 8,197 pituitary adenoma cases in the present study.…”

Section: Discussionmentioning

confidence: 99%

“…Meningioma is one of the most common intracranial tumor types, with an estimated incidence of 7.86 cases per 100,000 people per year, ~80% of which are benign and World Health Organization classification grade I (5)(6)(7)(8). In contrast, pituitary adenoma associated with meningioma (PAM) is a rare disease, with only 33 cases described before 2017 (9). The tumorigenesis of PAM remains unknown and there are currently no known epidemiological or well-characterized genetic associations between meningioma and pituitary adenoma.…”

Section: Introductionmentioning

confidence: 99%

Germline mutations in MEN1 are associated with the tumorigenesis of pituitary adenoma associated with meningioma

et al. 2020

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Pituitary adenoma and meningioma are two of the most common benign tumors in the central nervous system. Pituitary adenoma associated with meningioma (PAM) is a rare disease, the tumorigenesis of which remains unclear. Therefore, the aim of the present study was to investigate the tumorigenesis of PAM. A total of 8,197 patients with pituitary adenoma were analyzed. Furthermore, the clinical data of 57 patients with PAM were compared with patients with multiple endocrine neoplasia 1 (MEN-1) syndrome. Whole exome sequencing (WES) was performed on 23 samples from patients with PAM and the germline mutation was verified by Sanger sequencing. The age of tumor penetrance (age of patients at diagnosis) for PAM was significantly higher than that for patients with MEN-1. Compared with MEN-1 patients, there was a significant association between PAM and female sex (P=0.004). Clonal analysis and phylogenetic tree construction suggested that the pituitary adenoma and meningioma in PAM don't originate from a common progenitor. WES revealed that 5/23 PAM samples had the recurrent germline mutation MEN1 c.1523G>A; p.G508D, which may be a genetic risk factor for PAM. Compared with patients with sporadic pituitary adenoma, the difference was statistically significant (P=0.0004). Compared with wild-type MEN1, there was a significant association between the MEN1 mutation and recurrence of pituitary adenoma, young age and larger diameter of the meningioma. The present study indicated that germline mutations in MEN1 may be associated with the tumorigenesis of PAM.

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“…We had identi ed 27 acromegalic patients presented with DKA [17,7,[18][19][20][21][22][23][24][25][26][27][28][29][30][31][32][33][34]. For the majority of the cases (25 out of 27) DKA was the initial presentation of unrecognized active acromegaly in patients harboring macroadenoma with signi cantly high levels of GH and IGF-1.…”

Section: Discussionmentioning

confidence: 99%

Sodium glucose cotransporter 2 inhibitors treatment in acromegalic patients with diabetes—a case series and literature review

et al. 2021

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PurposeDiabetes mellitus (DM) represents one of the most frequent comorbidities in patients with acromegaly. Sodium glucose cotransporter 2 inhibitors (SGLT2i) represent an important class for diabetes management. However, limited data is reported regarding the use of this class in patients with acromegaly and diabetes. MethodsReporting data regarding patients with acromegaly and diabetes under treatment with SGLT2i. Results34 acromegalic patients with diabetes were identi ed. Treatment with SGLT-2i was documented in nine patients, out of them 5 females and 4 males with a mean age (SD) of 61± 12 yr. The mean (SD) duration of treatment with SGLT2i was 27.5 ± 7.3 months. Mean HbA1c before and after SGLT-2i initiation was 8.1 ± 1.1% and 7.0 ± 0.9% respectively. Mean IGF-1 level (SD) before SGLT-2i initiation was 177 ± 68 ng/mL and the mean GH level (SD) was 0.7 ± 0.5 µg/L. All nine patients are still under treatment with SGLT2i and none of them had reported any adverse reaction related to SGLT2i. ConclusionsThe present article provides us for the rst time with new data regarding the use of SGLT2i among acromegalic patients with diabetes.

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Coexistence of GH-Producing Pituitary Macroadenoma and Meningioma in a Patient with Multiple Endocrine Neoplasia Type 1 with Hyperglycemia and Ketosis as First Clinical Sign

Cited by 7 publications

References 14 publications

The clinical characteristics and molecular mechanism of pituitary adenoma associated with meningioma

The clinical characteristics and molecular mechanism of pituitary adenoma associated with meningioma

Germline mutations in MEN1 are associated with the tumorigenesis of pituitary adenoma associated with meningioma

Sodium glucose cotransporter 2 inhibitors treatment in acromegalic patients with diabetes—a case series and literature review

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