
==== Front
J Neurosurg Case Lessons
J Neurosurg Case Lessons
J Neurosurg Case Lessons
Journal of Neurosurgery: Case Lessons
2694-1902
American Association of Neurological Surgeons

39186831
10.3171/CASE24247
CASE24247
OncologyOncologyCase Lesson
Intraventricular pituicytoma: illustrative case
Hanyu Takashi MD 1
Ishibashi Ryota MD 1
Kitamura Kazushi MD 1
Nishida Namiko MD, PhD 1
Yuba Yoshiaki MD 2
Honjo Gen MD, PhD 2
Satomi Kaishi MD, PhD 3
Ichimura Koichi MD, PhD 3
Shibahara Junji MD, PhD 3
Sawada Takeshi MD, PhD 4
Ishimori Takayoshi MD, PhD 4
Takebe Noriyoshi MD 1
Hashikata Hirokuni MD, PhD 1
Toda Hiroki MD, PhD 1
1 Departments of Neurosurgery, Medical Research Institute, Kitano Hospital, Osaka, Japan
2 Departments of Pathology, Medical Research Institute, Kitano Hospital, Osaka, Japan
3 Department of Diagnostic Radiology, Medical Research Institute, Kitano Hospital, Osaka, Japan
4 Department of Pathology, Faculty of Medicine, Kyorin University, Tokyo, Japan
Correspondence Hiroki Toda: Medical Research Institute, Kitano Hospital, Osaka, Japan. hi-toda@kitano-hp.or.jp.
INCLUDE WHEN CITING Published August 26, 2024; DOI: 10.3171/CASE24247.

Disclosures Dr. Ichimura reported an endowed chair at the Department of Brain Disease Translational Research, Juntendo University Faculty of Medicine, sponsored by Idorsia Pharmaceuticals Japan Ltd., and grants from Daiichi Sankyo Co. Ltd. and from Riken Genesis Co. Ltd. outside the submitted work.

26 8 2024
26 8 2024
8 9 CASE2424712 4 2024
11 6 2024
© 2024 the authors
2024
the authors
https://creativecommons.org/licenses/by-nc-nd/4.0/ CC BY-NC-ND 4.0 (http://creativecommons.org/licenses/by-nc-nd/4.0/)

BACKGROUND

Pituicytoma is a rare glial neoplasm from pituicytes of the neurohypophysis or infundibulum. It occurs in the sella and suprasellar area, and it is extremely uncommon to observe intraventricular pituicytoma without affecting the infundibulum or infundibular recess.

OBSERVATIONS

A 69-year-old man had suffered progressive dementia for 6 months. Magnetic resonance imaging revealed a solid, homogeneously enhancing mass with flow voids within the anterior third ventricle. The sella, suprasellar area, infundibulum, and infundibular recess were unaffected. The patient underwent a transcallosal transchoroidal approach, which ended in partial removal of the tumor due to significant tumoral bleeding. A second surgery resulted in its subtotal removal. The tumor had bipolar cells, and their nuclei were immunoreactive for thyroid transcription factor–1. A DNA methylation analysis corresponded to the methylation class of pituicytoma, granular cell tumor, and spindle cell oncocytoma. Pituicytoma was the diagnosis based on these results. A systematic review identified 5 intraventricular pituicytoma cases.

LESSONS

Intraventricular pituicytoma can grow without involvement of the infundibulum or infundibular recess. The current case suggests that pituicytes of the hypothalamic tuber cinereum can also give rise to pituicytoma. Because of the hypervascular nature of intraventricular pituicytomas, it is imperative to control intraoperative bleeding with attention to the adjacent hypothalamus.

https://thejns.org/doi/10.3171/CASE24247

pituicytoma
intraventricular
third ventricle
case report
ABBREVIATIONS

CT = computed tomography
MR = magnetic resonance
MRI = magnetic resonance imaging
t-SNE = t-distributed stochastic neighbor embedding
TTF-1 = thyroid transcription factor–1.
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pmcPituicytoma is a rare glial neoplasm arising from pituicytes of the neurohypophysis or infundibulum.1 It is seen in adults in the 5th and 6th decades of life,2 with a slight male predominance.3, 4 Patients with pituicytoma can present with headaches, visual field defects, and hypopituitarism, depending on the lesion location.5 The locations of pituicytoma can be classified into the sellar and suprasellar areas.4 Occasionally, suprasellar pituicytoma and a suprasellar component of sellar pituicytoma can involve the infundibulum;2, 6–9 however, it is very rare to observe an isolated intraventricular pituicytoma with no involvement of the infundibulum.6, 10–13 Preoperative prediction of pituicytoma is crucial since it is frequently hypervascular3 and can cause significant intraoperative bleeding.6, 11, 14, 15 We describe a hypervascular intraventricular pituicytoma case and review its radiological and operative findings. In addition, the present case can provide illustrative evidence of hypothetical pituicytoma origins.4

Illustrative Case

History and Examination

A 69-year-old man had suffered a progressive cognitive and motivational decline over 6 months. His family reported an episode of dangerous driving. Additionally, the patient had gait disturbance and urinary incontinence a month before the referral. His medical history included hypertension, urolithiasis, and colorectal polyps.

The patient was disoriented to time and place. He had deficits in word registration and recall, digits backward and serial 7s subtraction, speech fluency, word similarities, lexical fluency, and go and no-go instruction, indicating moderate cognitive and persecutive impairment. His visual acuity, visual field, other cranial nerve functions, motor and sensory systems, and cerebellar functions were normal. The timed up-and-go test resulted in 12.5 seconds, indicating mild walking difficulty.

Laboratory examination indicated mild hypothalamic dysfunction causing a high prolactin level and secondary hypogonadism leading to a low testosterone level (Table 1). TABLE 1. Preoperative serum endocrine tests

Hormone	Value	Normal Range	
Prolactin (ng/mL)	30.9	4.29–13.69	
Testosterone (ng/mL)	0.38	1.3–8.7	
Luteinizing hormone (mIU/mL)	3.09	2.2–8.4	
Follicle-stimulating hormone (mIU/mL)	11.8	1.8–12.0	
Somatomedine C (ng/mL)	55	42–250	
Human growth hormone (ng/ml)	0.14	≤ 2.47	
Thyroid-stimulating hormone (mIU/mL)	3.179	0.61–4.23	
Free-thyroxine (ng/dL)	0.77	0.70–1.48	
Free-triiodothyronine (pg/mL)	2.63	1.68–3.67	

Head computed tomography (CT) revealed a contrast-enhanced mass in the third ventricle (Fig. 1A and B). Brain magnetic resonance imaging (MRI) showed a T1-weighted isointense solid mass (Fig. 1C) that enhanced brightly with gadolinium (Fig. 1D). The mass occupied the anterior third ventricle (Fig. 1E and F) and did not involve the infundibulum and infundibular recess (Fig. 1F). Hypothalamic edema (Fig. 1G) and multiple feeding arteries (Fig. 1G and H) were visible on T2-weighted MRI. Cerebral digital subtraction angiography showed that the mass was fed by the perforating arteries from the anterior cerebral, anterior communicating, and posterior cerebral arteries (Fig. 1I and J). The preoperative diagnosis included intraventricular meningioma, solitary fibrous tumor, and chordoid glioma of the third ventricle; however, the pituicytoma was not included in our preoperative differential diagnosis because the mass did not involve the infundibulum and infundibular recess (Fig. 1F). FIG. 1. Preoperative images of the tumor in the third ventricle. Head CT images show an isodense (arrow, A) and enhanced mass (arrow, B) with its feeding arteries (double-headed arrow). Axial T1-weighted MR images show an isointense solid mass (arrow, C) with bright contrast enhancement by gadolinium injection (arrow, D). A T2-weighted hyperintense mass in the third ventricle associated with edema (arrowheads, E) and multiple flow void signals of the feeding arteries and intratumoral vessels (double-headed arrows). Angiography shows the feeding arteries (arrowheads, F) from the anterior communicating artery, anterior choroidal artery, and anterior perforating arteries from the proximal anterior cerebral artery (double-headed arrow). The mass occupied the anterior part of the third ventricle, and the infundibulum (arrow, G) is intact. The mass occludes the bilateral foramen of Monro (arrows, H), and the bilateral lateral ventricles are enlarged (arrowheads). The posterior perforating arteries (double-headed arrows, I and J) from the proximal posterior cerebral artery send tortuous tumor vessels (arrowheads, J).

First Operation

A transcallosal transchoroidal approach via the right lateral ventricle exposed a firm, reddish mass in the third ventricle (Fig. 2A). The hypervascularity resulted in significant bleeding (Fig. 2B), which terminated the operation as a partial removal after confirmation of the cerebral aqueduct (Fig. 2C). Intensive tissue coagulation made the pathological diagnosis difficult. FIG. 2. Operative views via the transcallosal transchoroidal approach. The view at the first surgery shows a firm and reddish mass (arrow,A) through the right foramen of Monro behind the fornix (arrowheads) and choroidal fissure (double-headed arrow). Bleeding from the tumor was difficult to control (B). The first surgery was finished in a partial removal after confirmation of the cerebral aqueduct (arrowhead,C). Postoperative gadolinium-enhanced MR image shows a residual mass (arrow,D). An axial T2-weighted MR image shows exaggerated edema in the bilateral hypothalamus (arrowheads,E). The operative view at the second surgery shows the residual tumor (arrow,F) through the right foramen of Monro behind the fornix (arrowheads) and choroidal fissure (double-headed arrow). The thalamostriate vein (TSV) and its transition to the internal cerebral vein (ICV) are exposed. The view from the left lateral ventricle via the transseptal approach shows the left choroidal plexus (double-headed arrow, G) and a firm tumor (arrow) behind the left fornix (arrowheads). The mass was removed via the right transforaminal approach by coagulating the feeding arteries (black arrowhead, H), and the left third ventricular wall was intact (white arrowhead). The mass (arrow, I) was detached from the intact third ventricular floor (arrowheads). The mass (arrows, J) was removed from the forceps (F) by disconnecting it from the right lateral wall of the third ventricle (K). The final view confirms the cerebral aqueduct (black arrowhead, L) and the third ventricular floor (white arrowheads). Postoperative sagittal (M) and axial (N) gadolinium-enhanced MR images show subtotal removal. A T2-weighted MR image (O) shows a postoperative hyperintense change in the right hypothalamus (white arrowhead).

First Postoperative Course

The patient was conscious following the surgery; however, 2 days after surgery, he developed acute hydrocephalus, which led to a coma, and he needed ventricular drainage. A postoperative magnetic resonance (MR) image showed a remaining mass (Fig. 2D) and increased edema in the bilateral hypothalamus (Fig. 2E). A week later, the patient received a ventriculoperitoneal shunt for persistent hydrocephalus. Corticosteroids were administered to manage the edema; however, the edema persisted. The patient partially regained consciousness but developed diabetes insipidus, pneumonia, and congestive cardiac failure. Following his recuperation from these postoperative conditions, the patient’s family consented to a second surgery.

Second Operation

The patient underwent a second transcallosal transchoroidal approach. The column of the right fornix was dissected, and the choroidal fissure was re-opened to observe the transition of the right thalamostriate vein to the internal cerebral vein (Fig. 2F). The tumor was also observed through the left foramen of Monro via the opened septum pellucidum (Fig. 2G). By utilizing a 4-MHz high-frequency electrosurgical generator and nonstick bipolar forceps, hemostasis was controlled in most procedures during the tumor dissection and removal (Fig. 2H and I). The mass was severely attached to the third ventricular floor and its right-sided wall. Therefore, the tumor was removed subtotally (Fig. 2J) to minimize damage to the hypothalamus (Fig. 2K). After subtotal removal of the tumor, the third ventricular floor and the cerebral aqueduct were observed (Fig. 2L).

Second Postoperative Course and Pathological Diagnosis

After the second surgery, the patient’s level of consciousness varied from obtunded to lethargy. Postoperative MRI showed subtotal removal of the third ventricular mass (Fig. 2M and N) and a postoperative change in the right hypothalamus (Fig. 2O). Bilateral hypothalamic edema subsided (Fig. 2O).

The tissue showed interlacing fascicles of bipolar fibrillary cells with elongated nuclei (Fig. 3A), and these nuclei were immunoreactive for thyroid transcription factor–1 (TTF-1; Fig. 3B). In addition, DNA methylation analysis using the Deutsches Krebsforschungszentrum brain tumor classifier (v11b4)16 corresponded to the pituicytoma, granular cell tumor, and spindle cell oncocytoma methylation class with a score of 0.97.17 Visualization of the DNA methylome in this case, along with 2801 central nervous system tumors (GSE109381), using an unsupervised, nonlinear t-distributed stochastic neighbor embedding (t-SNE) projection, as previously described, showed that the present case had fallen into the cluster of sellar region tumors (Fig. 3C). These results supported the diagnosis of pituicytoma. FIG. 3. Photomicrographs of tumor sections. A cellular neoplasm composed of spindle cells (hematoxylin and eosin, A) positive for TTF-1 (B). Bars = 100 µm. DNA methylation-based t-SNE analysis (C)indicates that the present specimen belongs to the sella group (dotted circle).

The postoperative condition was complicated by acute respiratory failure due to acute interstitial pneumonia and panhypopituitarism. The patient needed oxygen support for the pneumonitis and vasopressin for diabetes insipidus. He also needed assistance with activities of daily living. He was transferred to a rehabilitation facility; however, he died of unknown causes 14 months after the second surgery.

Literature Review

We searched the MEDLINE and Web of Science databases for literature published before December 31, 2023, according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses protocol.18 The string used for this search was (pituicytoma[all]). Non–English-language articles were excluded. The following article types were included: case reports, case series, and observational studies of patients with pituicytoma. We excluded review articles without illustrative cases, articles without MRI findings, and articles missing clinical information on the location of the lesion. We examined 293 articles on pituicytoma (Fig. 4) and confirmed 5 intraventricular pituicytomas6, 10–13 (Table 2). The patients in these cases were men of various ages, according to available data. All cases involved the infundibular recess, except for the present case, which depressed the third ventricular floor downward and indicated flow voids on MRI. Gross-total removal has not been reported in any intraventricular pituicytoma cases. FIG. 4. Flow diagram of studies for the systematic review according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) 2020 statement.18

TABLE 2. Reported cases of intraventricular pituicytoma

Authors & Year	Age (yrs), Sex	Presentation Symptoms	MRI Characteristics	Treatment	Complications	FU (mos)	
Infund Rec Involvement	3V Floor Depression	Flow Void	Surgical Approach	Extent of Resection	Other Treatment	
Tian et al., 201312	13, M	Growth disorder, decreased vision	Yes	Yes	Yes	Transcranial	Partial	NA	Hemorrhage	Persistence (18)	
Teti et al., 201511	36, M	Polydipsia, polyuria, polyphagia, decreased libido, sleep pattern disruption	Yes	Yes	Yes	Transcranial	Subtotal	Radiation	HypoPi, DI, hemorrhage, diencephalic syndrome	Persistence (36)	
Guerrero-Pérez et al., 20192	NA	NA	Yes	Yes	Yes	NA	NA	NA	NA	NA	
Borg et al., 20206	56, M	Headache, confusion	Yes	Yes	Yes	Transcranial	Biopsy	VPS	NA	NA	
Wei et al., 202113	NA	NA	Yes	Yes	Yes	NA	NA	NA	NA	NA	
Present case	69, M	Dementia	No	Yes	Yes	Transcranial	Subtotal	VPS	Hydrocephalus, hypoPi, DI	Persistence (14)	
3V = third ventricle; DI = diabetes insipidus; FU = follow-up; hypoPi = hypopituitarism; Infund Rec = infundibular recess; NA = not available; VPS = ventriculoperitoneal shunt.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Observations

We observed an intraventricular pituicytoma originating from the third ventricular floor. The present case illustrates a unique intraventricular pituicytoma, as it did not involve the infundibulum or infundibular recess (Fig. 1F). This rare presentation has made an accurate preoperative diagnosis difficult. The reported intraventricular pituicytomas are characterized by bright gadolinium enhancement and flow voids,6, 10–13 suggesting a rich tumor vascular network.

Excessive hypervascularity is another hallmark of intraventricular pituicytoma.6, 10–13 In the present case, cerebral angiography showed that the tumor was fed extensively by the perforating arteries for the anterior and posterior hypothalamic nuclei (Fig. 1J and I), which also suggests that hypothalamic pituicytes could be the origin of the present intraventricular tumor. During operations, hypervascularity, intraventricular location, and hypothalamus adherence have prevented surgeons from totally removing the intraventricular pituicytoma, including the present and reported cases6, 11, 12 (Table 2).

Lessons

Lessons from the present case include the need to list pituicytoma in the differential diagnosis of the anterior third ventricular tumor, even if the tumor has no connection with the neurohypophysis or infundibular stalk. For pituicytomas in other locations, the loss of a T1-hyperintense posterior pituitary signal can be an indication to suspect a sellar pituicytoma.3 In addition, a lesion involving the infundibular stalk can be an indication to suspect a suprasellar pituicytoma arising from the infundibular pituicyte.2, 6–9 Preinfundibular tumor occupation can be diagnostic for suprasellar pituicytoma; however, a similar finding is often observed in craniopharyngioma.19, 20 Hypervascularity can be an indication to suspect pituicytoma.11, 21, 22 Considering that all reported intraventricular pituicytomas show multiple signs of peri- and intratumoral flow voiding,6, 10–13 the presence of a flow void sign can be a characteristic of intraventricular pituicytoma.

Another lesson from this particular case is related to tumor hypervascularity.6, 11, 15, 23–25 In the first postoperative period, exaggerated hypothalamic edema worsened the patient’s condition. This may be related to the partial removal of the tumor, as it is suspected that the postoperative vascular drainage changes from the hypervascular tumor might cause peritumoral blood flow congestion and deteriorate the peritumoral edema. Based on this hypothesis, a significant amount of tumor needs to be removed in intraventricular pituicytoma cases, although several case reports have documented significant intraoperative bleeding as a surgical difficulty preventing surgeons from totally removing the tumor safely.6, 11, 15, 23–25

Additionally, the present intraventricular tumor location and its adherence to the third ventricular floor provided some evidence for pituicytoma origins.4 The radiological and intraoperative findings in the present case suggest that pituicytes of the tuber cinereum can be the origin of some pituicytomas.4

In conclusion, intraventricular pituicytoma is an uncommon but important anterior third ventricular tumor. Its hypervascularity can be a sign of intraventricular pituicytoma, but it can pose a difficulty in surgical treatment.

Disclosures

Dr. Ichimura reported an endowed chair at the Department of Brain Disease Translational Research, Juntendo University Faculty of Medicine, sponsored by Idorsia Pharmaceuticals Japan Ltd., and grants from Daiichi Sankyo Co. Ltd. and from Riken Genesis Co. Ltd. outside the submitted work.

Author Contributions

Conception and design: Toda, Hanyu, Ichimura. Acquisition of data: Toda, Hanyu, Ishibashi, Nishida, Yuba, Honjo, Sawada, Ishimori, Takebe. Analysis and interpretation of data: Toda, Hanyu, Nishida, Yuba, Honjo, Satomi, Ichimura, Shibahara, Sawada, Ishimori. Drafting the article: Toda, Hanyu, Honjo, Satomi. Critically revising the article: Toda, Hanyu, Ishibashi, Honjo, Ichimura, Shibahara, Sawada, Ishimori. Reviewed submitted version of manuscript: Toda, Hanyu, Nishida, Honjo, Sawada, Ishimori, Hashikata. Approved the final version of the manuscript on behalf of all authors: Toda. Administrative/technical/material support: Kitamura, Yuba, Hashikata. Study supervision: Toda.

Correspondence

Hiroki Toda: Medical Research Institute, Kitano Hospital, Osaka, Japan. hi-toda@kitano-hp.or.jp.
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