Case Report Open Access
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. May 6, 2022; 10(13): 4249-4263
Published online May 6, 2022. doi: 10.12998/wjcc.v10.i13.4249
Coexistence of meningioma and other intracranial benign tumors in non-neurofibromatosis type 2 patients: A case report and review of literature
Tian-Hao Hu, Run Wang, Hai-Yun Wang, Yi-Fu Song, Zi-Xun Wang, Yu-Zhou Duan, Sheng Han, Department of Neurosurgery, The First Hospital of China Medical University, Shenyang 110001, Liaoning Province, China
Juan-Han Yu, Department of Pathology, China Medical University, Shenyang 110001, Liaoning Province, China
Ting Liu, Department of Radiology, The First Hospital of China Medical University, Shenyang 110001, Liaoning Province, China
ORCID number: Tian-Hao Hu (0000-0002-3865-9746); Run Wang (0000-0001-6127-4257); Hai-Yun Wang (0000-0003-3476-5848); Yi-Fu Song (0000-0002-0122-540X); Juan-Han Yu (0000-0001-7570-6284); Zi-Xun Wang (0000-0002-5652-9996); Yu-Zhou Duan (0000-0001-6476-2248); Ting Liu (0000-0001-6827-6028); Sheng Han (0000-0001-6991-0529).
Author contributions: Hu TH, Wang R, Duan YZ, Liu T and Han S performed the data acquisition and prepared the figures; Hu TH, Wang HY and Song YF prepared the manuscript and followed up the patients; Yu JH and Wang ZX performed histopathology examination of the patients; Hu TH and Han S designed the report; All authors contributed to the article and approved the submitted version.
Supported by the LiaoNing Revitalization Talents Program, No. XLYC1807253; and the National Natural Science Foundation of China, No. 81772653.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Sheng Han, Doctor, MD, PhD, Academic Research, Surgeon, Surgical Oncologist, Department of Neurosurgery, The First Hospital of China Medical University, No. 155 Nanjing Street, Heping District, Shenyang 110001, Liaoning Province, China. hansheng@cmu.edu.cn
Received: October 2, 2021
Peer-review started: October 2, 2021
First decision: October 22, 2021
Revised: October 29, 2021
Accepted: March 15, 2022
Article in press: March 15, 2022
Published online: May 6, 2022
Processing time: 210 Days and 1.4 Hours

Abstract
BACKGROUND

The coexistence of meningioma and other intracranial primary benign tumors is rare, especially in non-neurofibromatosis type 2, and there is limited guidance for the management of such patients. Here, we report a series of 5 patients with concomitant meningioma and other intracranial benign tumors, including subependymoma and pituitary adenoma.

CASE SUMMARY

Five non-neurofibromatosis type 2 patients with simultaneous occurrence of meningioma and other intracranial benign tumors were retrospectively reviewed. The patients had no history of previous irradiation. The clinical features, pre- and postoperative imaging, surgical procedure and pathological findings were extracted from electronic medical records. There were 4 female patients (80%) and 1 male patient (20%). The mean age was 42.8 years (range: 29-52 years). The coexisting tumors included subependymoma in 1 case (20%) and pituitary adenoma in 4 cases (80%). The most common clinical symptom was headache (3/5, 60%). Four patients (80%) underwent craniotomy. One patient (20%) underwent transsphenoidal surgery followed by transcranial operation. All tumor diagnoses were confirmed by histopathological examination. The mean follow-up was 38.8 mo (range: 23-96 mo), and all 5 patients were in a stable condition at the last follow-up.

CONCLUSION

The simultaneous occurrence of meningioma and other intracranial benign tumors is a rare clinical event. Histological examination is necessary for the accurate diagnosis. Neurosurgeons should select the appropriate surgical strategy according to the clinical features of each patient, which may provide a more favorable prognosis for individual patients.

Key Words: Meningioma, Sub-ependymoma, Pituitary adenoma, Coexisting tumors, Case report

Core Tip: The simultaneous occurrence of meningioma and other intracranial primary benign tumors is rare, especially in non-neurofibromatosis type 2, and there is limited guidance for the management of such patients. In this study, we report a series of 5 patients with coexistence of meningioma and other intracranial benign tumors, including subependymoma and pituitary adenoma.



INTRODUCTION

Meningioma is the most frequent intracranial benign tumor, accounting for 37.1% of tumors overall[1]. However, the concomitant occurrence of meningioma and other intracranial benign tumors is extremely rare[2]. Only a few coexisting meningioma and other intracranial benign tumor cases have been reported in previous publications, such as pituitary adenoma[3,4], craniopharyngioma[5] and vestibular schwannoma[6-9]. It is well-recognized that the occurrence of multiple nervous system tumors in the same patient is a characteristic of neurofibromatosis type 2 (NF2)[8]. Patients are diagnosed with NF2 when they meet the Manchester criteria (Supplementary Table 1)[10,11]. However, clinical and pathological features of non-NF2 patients with collision-tumors remain unclear. Here we report the clinical presentation, radiological features, surgical management and outcomes in our series of 5 non-NF2 patients with concomitant meningioma and other intracranial benign tumors, including one subependymoma, which was reported for the first time, and four pituitary adenomas. In addition, we also reviewed the available literature.

CASE PRESENTATION
Chief complaints

Case 1: A 45-year-old female patient presented with a sudden night epileptic seizure 10 d prior.

Case 2: A 40-year-old female patient presented with dizziness and headache for 1 mo.

Case 3: A 48-year-old male patient presented with frontal and bilateral temporal headache for 1 mo.

Case 4: A 52-year-old female patient presented with left progressive blurred vision for 6 mo and headaches for 2 mo.

Case 5: A 29-year-old female patient presented with menstrual disorder for 2 years and intermittent headache for 3 mo.

History of present illness

Case 1: The patient suffered a sudden epileptic seizure 10 d ago at night, which lasted for a few minutes.

Case 2: The patient had dizziness and headaches for 1 mo.

Case 3: The patient had a headache for 1 mo. The headache took place at night and affected his sleep.

Case 4: The patient had progressive vision loss in the left eye for 6 mo. She suffered from headache from 2 mo.

Case 5: The patient had menstrual disorder for 2 years. She developed amenorrhea and intermittent headaches 3 mo ago.

History of past illness

All patients had no history of specific illnesses.

Personal and family history

All patients had no special personal and family history.

Physical examination

Case 1, 2, 3 and 5: Neurological examination of the patient found no positive signs.

Case 4: There was only slight sensation in her left eye when she was admitted.

Laboratory examinations

Case 1, 3 and 4: Preoperative endocrine examination showed no abnormal changes.

Case 2: Endocrine examination indicated that prolactin levels were moderately elevated (61.19 ng/mL).

Case 5: Preoperative endocrine examination showed that prolactin levels were slightly elevated (59.17 ng/mL).

Imaging examinations

Case 1: Brain computed tomography at the local hospital suggested an intracranial space-occupying lesion in the left parietal lobe. Brain contrast-enhanced magnetic resonance imaging (MRI) showed a well-circumscribed mass (3.6 cm × 2.7 cm × 2.7 cm) in the left parietal parafalcine and a mass (1.9 cm × 1.2 cm × 1.1 cm) in the left lateral ventricle.

Case 2: MRI showed a mass (3.0 cm × 2.3 cm × 2.5 cm) in the right middle cranial fossa and a mass (0.5 cm × 0.5 cm × 0.5 cm) in the sellar region.

Case 3: Brain MRI showed lesions located in the planum sphenoidale and sellar regions.

Case 4: There was a well-circumscribed mass (3.5 cm × 3.2 cm × 2.7 cm) surrounded with brain edema in the left sphenoid ridge and a mass (2.8 cm × 2.5 cm × 2.5 cm) encasing the internal carotid artery in the sellar and suprasellar regions, as determined by radiological examination.

Case 5: MRI showed a mass (5.7 cm × 3.3 cm) encasing the internal carotid artery located in the left petroclival region and a mass (maximum diameter 1.0 cm) located in the sellar region.

FINAL DIAGNOSIS

Case 1: Postoperative histopathological examination showed meningioma and subependymoma (Figure 1).

Figure 1
Figure 1 Histopathological examination showed a meningioma and subependymoma. A-C: Preoperative axial and coronal contrast-enhanced magnetic resonance imaging (MRI) showed a mass in the left lateral ventricle (yellow arrow) and a mass in the left parietal parafalcine (orange arrow); D-F: Postoperative axial and coronal MRI showed that gross total resection of the tumors was achieved; G: Histological examination revealed subependymoma; H: Histological examination revealed meningioma.

Case 2: Histopathological examination showed a meningioma and non-functioning pituitary adenoma (Figure 2).

Figure 2
Figure 2 Histopathological examination showed a meningioma and non-functioning pituitary adenoma. A: Sagittal contrast magnetic resonance imaging showed an intrasellar mass (yellow arrow); B and C: Sagittal coronal contrast magnetic resonance imaging showed an intrasellar mass (yellow arrow) and a mass in the right middle cranial fossa (orange arrow); D: Histological examination revealed pituitary adenoma; E: Histological examination revealed meningioma.

Case 3: Histopathological examination showed a meningioma and non-functioning pituitary adenoma (Figure 3).

Figure 3
Figure 3 Histopathological examination showed a meningioma and non-functioning pituitary adenoma. A and B: Mid-sagittal contrast magnetic resonance imaging showed space-occupying lesions located in the planum sphenoidale and sellar region; C: Histological examination revealed pituitary adenoma; D: Histological examination revealed meningioma.

Case 4: Histopathological examination showed a meningioma and non-functioning pituitary adenoma (Figure 4).

Figure 4
Figure 4 Histopathological examination showed a meningioma and non-functioning pituitary adenoma. A and C: Axial contrast magnetic resonance imaging showed a mass (yellow arrow) in the sellar and suprasellar region and a well-circumscribed mass (orange arrow) in the left sphenoid ridge; B and D: Postoperative histological examination revealed pituitary adenoma and meningioma.

Case 5: Postoperative histopathological examination showed a meningioma and non-functioning pituitary adenoma (Figure 5).

Figure 5
Figure 5 Postoperative histopathological examination showed a meningioma and non-functioning pituitary adenoma. A and B: Histological examination revealed pituitary adenoma and meningioma.
TREATMENT

Case 1: After this discovery, she underwent craniotomy, and the two neoplasms were removed in one session.

Case 2: First, endonasal transsphenoidal surgery was performed for resection of the intrasellar mass. Then, the right middle cranial fossa mass was removed (Simpson grade II) by transcranial surgery.

Case 3: The patient underwent transcranial resection for the two tumors through the right transpterional approach.

Case 4: A single transcranial procedure was performed for removal of the sphenoid ridge mass (Simpson grade II) and sellar region mass. The pituitary tumor underwent subtotal resection.

Case 5: The patient underwent craniotomy for the two tumors. The petroclival tumor was hard in consistency and rich in blood supply with internal carotid artery encasement. The tumor was also close to cranial nerves II-VI and compressed the brain stem. A subtotal resection was performed (Simpson grade IV) for the petroclival tumor. Then the intrasellar tumor was removed.

OUTCOME AND FOLLOW-UP

Between January 2011 and January 2019, 2922 consecutive patients were diagnosed with meningioma in our institution. There were 5 meningioma patients (0.17%) with different intracranial benign tumors, and none of them were diagnosed with NF2. They had no history of previous irradiation. Clinical data were obtained and analyzed through retrospective medical history reviews, neuroimaging information, histopathological examination and follow-up. This retrospective study was approved by the institutional review board. Written consent was obtained from each patient for the use of their clinical data for research.

There were 4 female patients (80%) and 1 male patient (20%) with concomitant meningioma and other intracranial benign tumors. The mean age was 42.8 years (range: 29-79 years). The coexisting tumors included subependymoma in 1 case (20%) and pituitary adenoma in 4 cases (80%, four non-functional pituitary adenomas). The mean follow-up time was 38.8 mo (range: 23-96 mo). The clinical information of these cases is summarized in Table 1.

Table 1 Clinical features of 5 meningioma patients in the study.
Case No.
Sex/age
Symptom
Meningioma location
WHO grade of meningioma
Second primary tumor
Operation
Surgical approach
Simpson grade
Recurrence
Postoperative treatment
Follow-up, mo
1F/45SeizureLeft frontal parafalcine ISubependymomaSynchronousTCIINoneNone27
2F/40HeadacheRight middle cranial fossaIPituitary adenomaSynchronousTS + TCIINoneNone24
3M/48Headache Tuberculum sellaeIPituitary adenomaSynchronousTCIINoneNone23
4F/52Blurred vision, headacheLeft sphenoid ridgeIPituitary adenomaSynchronousTCIIGrowth of residual pituitary tumor after 4 mo Radiotherapy24
5F/29Amenorrhea and headacheLeft petroclivalIPituitary adenomaSynchronousTCIVNoneNone96

Case 1: The patient was followed up for 27 mo with no evidence of recurrent disease.

Case 2: The patient was followed up for 24 mo, and she was in good health.

Case 3: He was treated by hormone replacement therapy because of postoperative hypopituitarism. The patient was followed up for 23 mo. He took LT4 regularly and is currently in good health.

Case 4: Four months after the surgery, MRI reexamination revealed the growth of residual pituitary tumor. Subsequently, she underwent radiotherapy. The patient was in good health at the 24-mo follow-up.

Case 5: As of this writing, the patient had been in stable condition for 96 mo.

DISCUSSION

The coexistence of meningioma and other intracranial benign tumors is a rare phenomenon that nevertheless deserves our attention. The most frequent coexistence of simultaneous benign tumors is pituitary adenoma with meningioma and schwannoma with meningioma[12]. The co-occurrence of meningioma and schwannoma is more likely to occur in patients with NF2, which has been well described previously. However, the clinical characteristics of non-NF2 patients with coexisting tumors are largely unknown.

To the best of our knowledge, we present the first case of concomitant meningioma and intracranial subependymoma. Subependymomas are rare, benign, slow-growing tumors and represent only 0.2% to 0.7% of intracranial tumors[13-16]. These tumors most often arise in the fourth ventricle (50%-60%) and the lateral ventricles (30%-40%)[14,15]. Most patients present with hydrocephalus as a consequence of ventricular obstruction or less commonly focal neurological dysfunction and seizures caused by mass effects[14,17]. The main purpose of surgery is to maximize the removal of the tumor[18]. In the published case series, there were satisfactory postoperative mortality and morbidity rates from supratentorial subependymomas[13-18]. In our case 1, resection of the left parietal meningioma and supratentorial subependymoma was accomplished via a single procedure. In a recent long-term outcome study of subependymoma, no patients exhibited a deterioration of performance status or tumor recurrence at medium to long-term follow-up[16]. As with the single subependymoma, the patient in our case also had a good prognosis after operation, and there was no evidence of recurrence at the last follow-up. Although the extremely rare coexistence of meningioma and subependymoma in our case may be an incidental event, the intrinsic relationship of these two tumors might require future investigation.

Until now, our understanding of the coexistence of meningioma and pituitary adenoma is based on occasional case reports[2-4,12,19-54]. We reviewed all the reported cases that were available to us, and the information was summarized in Table 2. The mean age of patients was 54.6 years (range: 26-82 years), and there were 39 women and 14 men among the published cases (female:male = 2.79:1). Our 4 cases also showed a female tendency (female:male = 3:1), and the mean age was 42.8 years (range: 29-52 years). A preference for parasellar, suprasellar and sphenoid ridge localization was found with 27 reported cases (50.94%)[2-4,20-23,25,26,30,32-37,42,47-49,51,54]. Consistently, the meningioma of our cases was in the petroclival, sellar and sphenoid wing regions.

Table 2 Summary of coexisting meningioma and pituitary adenoma.
No.
Ref.
Age
Sex
Symptom
Size of meningioma, cm
Type of meningioma
Location of meningioma
Type of pituitary adenoma
History of radiation
Treatment
Postoperative therapy
1Love et al[19], 195565FNANAMeningothelialSylvian fissureNon-secretingNANANA
2O’Connell[20], 196147FFailing vision in right eyeNAMeningothelialTuberculum sellaeNon-secretingNoTC (MN + PA)No
3Kitamura et al[21], 196566FHeadache and impaired visionNAMeningothelialSphenoid wingNon-secretingNoTC (MN + PA)No
4Probst[22], 197148FNANAMeningothelialSuprasellarACTH-producingNANANA
5Brennan et al[23], 197736MBlurred vision of right eyeNATransitionalSphenoid wingNon-secretingNoTC (MN + PA)Radiotherapy
6Bunick et al[24], 197857MIntermittent right-sided headache, acromegaly6 × 6FibrousRight frontal lobeGH-producingNoTC (MN + PA)No
7Hainer et al[25], 197872MNANANASuprasellarGH-producingNANANA
8Deen et al[26], 198175FChronic dementia, only had postmortem examinationNANARight sphenoid ridgeNANAAutopsyNo
9Hyodo et al[27], 198252FAcromegaly, diabetes mellitus, headache, right hemiparesis and right impaired visual acuityNAFibrous and meningothelialLeft parietal regionGH-producingNoTC (MN) + TS (PA, 4 mo later)No
10Irsy et al[28], 198559FNANANACentro-parietalGH-producingNANANA
11Ohata[29], 198550FAcromegaly, visual disturbance, headache, vomiting and a floating sensation 4 × 4 × 4TransitionalFalcotentorial junctionGH-producingNoTS (PA) + TC (MN, 2 mo later)Bromocriptine, acetylcortisone, desiccated thyroid
12Yamada et al[30], 198652FHeadache, disturbance of visual acuity and galactorrheaNAMeningothelialSphenoid ridge (parasellar)Non-secretingNoTC (PA + MN)Bromocriptine
13Honegger et al[31], 198937FMarked alopecia2.5MeningothelialRight temporal polePRL-producingNoTC (MN)Bromocriptine
14Honegger et al[31], 198949FAcromegaly persisted after radiotherapy3MeningothelialLeft parasagittalGH-producingYesTS (PA) + TC (MN, 2 mo later)No
15Honegger et al[31], 198974MRecurrence of pituitary adenoma1.5NALeft parietalNon-secretingYesTS (PA) + TC (PA, 5 yr later)No
16Zentner et al[32], 198946MCT demonstration of a large intrasellar and suprasellar space-occupying lesion1.5TransitionalPlanum sphenoidalePRL-producingNoTC (PA + MN)No
17Zentner et al[32], 198963FAtaxiaNAMeningothelialSphenoid wingNon-secretingNoTS (PA) + TC (MN, 1 mo later)No
18Zentner et al[32], 198961FFrontal headacheNAMeningothelialInfradiaphragmaticNon-secretingNoTS + TS (PA + MN) + TC (PA, 1 d later)No
19Partington et al[2], 198926MEvaluation of persistent symptom of Cushing’s disease; left temporal hemianopsia (10 yr later)NAMeningothelialTuberculum sellaeACTH-secretingYesTS (PA) + TS (PA, 8 yr later) + TC (1 yr later)No
20Uno et al[33], 199170FHeadache and acromegalyNAMeningothelialSphenoid ridgeGH-producingNoTC (MN + PA)No
21Cannavo et al[34], 199347FAcromegaly, diminished visual acuity, weakness and headacheNAMeningothelialRight latero- and retrosellarGH-producingNoTC (PA + MN)No
22Abs et al[35], 199347FAphasia and temporary right-sided hemiparesisNAMeningothelialTuberculum sellaePRL-producingNoTC (MN) + TS (PA, 3 mo later)No
23Abs et al[35], 199361FA toxic thyroid adenoma, Cushing’s diseaseNAMeningothelial; transitionalFrontal convexity; occipital convexityACTH-producingNoTC (MN)No
24Abs et al[35], 199345FAmenorrhea and galactorrheaNANATemporal fossaPRL-producingNoNo surgeryNo
25Abs et al[35], 199345FA toxic multinodular goiter and suspicion of acromegalyNANAParietal convexityGH-producingNoTC (Schwannoma) + TS (PA)No
26Abs et al[35], 199382FBitemporal hemianopsiaNANASphenoid ridge; parasellarNon-secretingNoTS (PA)No
27Abs et al[35], 199361FHeadache2.6NAChoroid plexus (right lateral ventricle)PRL-producingNoNo surgeryNo
28Abs et al[35], 199351FUnilateral palpebral edema and exophthalmosNANASphenoid wingNon-secretingNoEmbolization (MN)No
29Gorge et al[36], 199353MProgressive impotence, decrease of libido and left-sided defective visionNANAPara- and suprasellar PRL-producingNATC (PA + MN)No
30Laun et al[37], 199361FDeteriorating vision of the left eye and bitemporal hemianopiaNAMeningothelialTuberculum sellaeNon-secretingNANANo
31Mathuriya et al[38], 200058FAcromegalyNANAParasagittalGH-producingNoTC (PA + MN)Radiotherapy
32Maiuri et al[39], 200549MAcromegaly, right hemiparesis3.5NANAGH-producingNoTC (MN) + TS (PA, 3 mo later)No
33Maiuri et al[39], 200563FLeft hemiparesis, bitemporal visual field defect4NANANon-secretingNoTC (MN) + TS (PA, 1 mo later)No
34Curto et al[40], 200761FAcromegaly and visual field impairment3.5NARight frontal GH-producingNoNANo
35da Costa et al[41], 200745MGeneralized malaise and weight loss for a few months. Progressive headache, vomiting and gait ataxia for 2 wkNANAThe fourth ventriclePRL-producingNoTC (MN)Bromocriptine
36Prevedello et al[42], 200752FUnremitting headache and profound right temporal visual field loss1.0 × 0.6NARight planum sphenoidaleNon-secretingNoTS (PA + MN)No
37Basu et al[43], 201039MFrontal headaches and erectile dysfunction with loss of libidoNAMeningothelialLeft cavernous sinusPRL-secretingNoTC (MN)No
38Furtado et al[12], 201053MHeadache for 2 yr and altered sensorium for 2 wk6.0 × 5.5 × 5.0MeningothelialParasagittalNon-secretingNoTS (PA) + TC (MN)No
39Guaraldi et al[44], 201246FAcromegaly2.3 × 2.6 × 2.6NALeft parasagittalGH-producingNoTS (PA) + TC (MN, 10 yr later) + TS (Recurrence PA, 8 mo later)No
40Ramirez et al[45], 201261FHeadache and acute hydrocephalus for 2 mo1.0 × 1.0PsammomatousLeft anterior clinoidalSilent corticotroph adenoma subtype IINoTC (MN) + TS (PA, 1 mo later)No
41Masoodi et al[46], 201365MAcromegalyNANAFrontal parasagittalGH-producingNoTS (PA) + TC (MN)No
42Mahvash et al[47], 201436FFrontal headache and extended right visual field loss2.0 × 2.0 × 2.5NATuberculum sellaeNon-secretingNoTS (PA + MN)No
43Karsy et al[48], 201470FAltered mental status, mutism and incontinence3.6 × 4.1 × 4.5FibroepithelialTuberculum sellaeNon-secretingNoTS (PA + MN)No
44Ruiz-Juretschke et al[49], 201561MProgressive visual loss and bitemporal hemianopia for 6 mo2.0 × 2.2 × 2.2MeningothelialTuberculum sellae and planum sphenoidaleNon-secretingNoTS (PA + MN) + TS (residual tumor)No
45Ben Nsir et al[50], 201661FLoss of vision for 3 yr and occasional episode headacheNANAForamen magnumNon-secretingNoTC (MN) + [TS + TC] (PA)No
46Lim et al[51], 201665FVisual symptoms and an episode of self-resolving vertigo1.5 × 1.3 × 1.3; 0.6 × 0.5NATuberculum sellae and olfactory grooveNon-secretingNoTS (PA + MN)No
47Amirjamshidi et al[3], 201737FOligomenorrhea for 8 mo, headache, diplopia and progressive visual impairment3.0 × 2.5 × 2.0NASuprasellarPRL-secretingNoTC (MN)No
48Amirjamshidi et al[3], 201742MAcromegaly, visual acuity decreased and bitemporal hemianopia3.0 × 3.0 × 2.0NASuprasellarNon-secretingNoTS + TC (coexisting tumors)No
49Herrero-Ruiz et al[52], 201735FChagas disease and acromegaly2.0 × 2.0 × 1.2NALeft parietal parasagittalGH-producing NoTS (Only PA)No
50Kumaria et al[53], 201746FLethargy, sleep disturbance, personality change and mild daily right-sided headache for 18 moNATransitional; AtypicalRight frontal and left temporal regionsMammosomatotroph cell adenoma (PRL- and GH-secreting)YesTC (MN) + TC (MN) + TS (PA)No
51Zhao et al[54], 201758FAcromegaly and headacheNANASellarGH-producingNoTS (PA) + TC (MN, 3 mo later)No
52Zhao et al[54], 201758FAcromegalyNANASellarGH-producingNoTS (PA) + TC (MN, 4 mo later)No
53de Vries et al[4], 201975FDepression, fatigue and unintended weight lossNAMeningothelialSuprasellarNon-secretingNoTS (PA + MN)No

The most common type of pituitary adenoma with coexisting meningioma among reported cases was a non-secreting tumor (21/53, 39.62%) followed by growth hormone-producing tumor (17/53, 32.08%). In this paper, all 4 cases were non-functioning pituitary adenoma, representing the most common type. Although prolactinomas are the most frequent pituitary adenomas in general, the higher prevalence of acromegaly in patients with coexisting meningioma has led some authors to propose an association between growth hormone-producing adenomas and meningioma[29,34,40,42,49]. They suggested that persistently elevated growth hormone might stimulate arachnoid cap cells to play a role in the development of meningioma[24,39,51]. A recent study suggested that patients with acromegaly were at increased risk of meningioma[55].

Some researchers have tried to explore the association between meningiomas and pituitary tumors. The early reports suggested that this phenomenon was related to history of irradiation for pituitary tumors[2]. However, many cases with no history of radiotherapy were reported as well[24,27,29,48,49], including our cases. Therefore, Curto et al[40] suggested that the coexistence of meningioma and pituitary adenoma was a coincidental phenomenon. However, there was a higher proportion of involvement of chromosome 14 and 22 in estrogen receptor positive de novo meningiomas[56]. Similar genetic changes shared by two unrelated tumors found on the same chromosome may explain their coexistence[12].

Moreover, due to the indolence of benign tumors, a significant portion of this coexisting tumor population may remain undiagnosed[12]. MRI is useful for the diagnosis of the coexistence of two intracranial tumors but has limited significance for adjacent pituitary adenoma and meningioma[4]. Histological results are necessary for diagnosis because other preoperative findings cannot support accurate diagnosis[54]. For example, some reported cases were coexisting sellar meningioma and pituitary adenoma[4,42,47-49,54]. Because of the close location of the two tumors, it is difficult to produce an accurate diagnosis by preoperative imaging, as in our case 3. It deserves special attention because the two different types of tumors were not definitely diagnosed before surgery but later when the pathologist’s results were obtained. The patient in case 3 developed hypopituitarism after the operation, and this complication was also reported in other similar cases[47,49].

Traditionally, the treatment of these two coexisting tumors required one craniotomy[30,31,33] or two separate operations using two different approaches[2,32,35,39]. Prevedello et al[42] performed a single endoscopic expanded endonasal approach in patients with coexisting tuberculum sellae meningiomas and pituitary adenoma. In our opinion, surgical strategies should be decided according to the characteristics of the coexisting tumors (e.g., location, size and adjacent neurovascular structures) and the clinical features of individual patients (e.g., symptoms and systemic conditions). Like the single pituitary adenoma, the patient with coexisting meningioma and pituitary adenoma had a favorable prognosis using the retrospective case reports. Postoperative endocrine reexamination should be periodically monitored at the endocrinology outpatient department, especially in patients with postoperative hormone imbalance after surgery. However, as this study is a retrospective analysis and the case numbers are limited, we cannot draw strong conclusions.

CONCLUSION

The simultaneous occurrence of meningioma and other intracranial benign tumors is a rare clinical event, and histological examination is necessary for their accurate diagnosis. Neurosurgeons should select the appropriate surgical strategy according to the clinical features of individual patients, which may provide the patient with a more favorable prognosis.

Footnotes

Provenance and peer review: Unsolicited article; Externally peer reviewed.

Peer-review model: Single blind

Corresponding Author's Membership in Professional Societies: Neurosurgery Society of Chinese Medical Association, Youth committee.

Specialty type: Oncology

Country/Territory of origin: China

Peer-review report’s scientific quality classification

Grade A (Excellent): A

Grade B (Very good): 0

Grade C (Good): 0

Grade D (Fair): D

Grade E (Poor): 0

P-Reviewer: Nuño JSZ, Mexico; Yelamanchi R, India S-Editor: Liu JH L-Editor: Filipodia P-Editor: Liu JH

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