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Abstract

Objective

Dermoid cysts (DCs) are congenital, slowly growing, and may cause nervous system symptoms. Related literature is limited and mainly includes case reports. We report a case series of DCs originating from the middle cranial fossa floor (MCFF) and investigate their demographic information, clinical characteristics, imaging findings, surgical procedures, and prognostic outcomes.

Methods

We reviewed the patients with DCs arising from the MCFF undergoing endoscopic endonasal surgery (EES) in our center between 2012 and 2022.

Results

A total of 5 patients with DCs were enrolled (2 males and 3 females), with a mean age of 46.2 years at the onset. All DCs originated from the MCFF with 1 case involving the middle cranial fossa bone and another 1 case affecting the dura mater. One (20.0%) patient had neurological involvement. After admission, all patients received EES with a total resection rate of 100.0% (5 of 5). After a median follow-up of 73.2 months, all patients achieved complete clinical and radiological improvements. No surgical-related complications or relapses were observed during the long-term follow-up.

Conclusion

Endoscopic endonasal surgery is considered a safe and effective approach for the treatment of DCs in the MCFF. A larger sample size and longer follow-up time are needed.

Keywords

dermoid cysts middle cranial fossa case series surgical options quality of life

Introduction

Dermoid cysts (DCs) are rare congenital tumors that account for only 0.04–0.7% of all intracranial tumors and are most common in patients aged 20–36 years.^1

-4 They are believed to arise due to ectopic inclusion of epithelial cells in the course of neural tube closure during embryologic development³ and contain dermal and epidermal elements including sebum, hair, and keratin.⁵ These tumors have rarely been reported to arise from the middle cranial fossa (MCF) but usually originate from the petrous apex or cavernous sinus and may involve the MCF.^5,6 Common clinical presentations often depend on the location of the tumor^7
-9 and whether it is ruptured,¹⁰ and can present with headaches, seizures, chemical meningitis, and visual disturbances, and olfactory hallucinations. Computerized tomography (CT) and magnetic resonance imaging (MRI) features are sufficient for accurate diagnosis,¹¹ yet this confirmation remains dependent on intraoperative histological examination. DCs are basically surgically treated diseases, and traditional approaches for DCs are multiple open approaches. Instead of such invasive techniques, some surgeons convert to resect such lesions via endoscopic endonasal surgery (EES).^12,13 If possible, all lesions should be excised to avoid tumor recurrence. However, subtotal resection is considered acceptable because of the strong adhesion of the capsule to the surrounding nerve and vascular structures.¹⁴ Growing evidence supports the advantage of multiple approaches in treating intracranial DCs, but only case studies have been performed. Thus, more information on clinical characteristics, radiographic findings, surgical procedures, and prognostic outcomes should be identified and summarized. In this study, we collected five case series of DCs arising from the middle cranial fossa floor (MCFF) treated with EES in our institution and shared our preliminary experiences and evaluated the feasibility of endoscopic endonasal surgery treatment.

Patients and methods

Study design

We retrospectively analyzed the clinical data of DCs originating from the middle cranial fossa floor (MCFF) treated with endoscopic endonasal surgery (EES) in our center from 2012 to 2022. The diagnosis of the patient population was made by an otolaryngologist and a pathologist on the basis of neurological symptoms, endoscopic examinations, radiological findings, and pathological outcomes. Clinical data including demographic information, clinical features, imaging and pathological findings, treatment, and prognostic outcomes were retrospectively collected by an independent resident doctor.

The clinical response and safety of EES treatment were evaluated. Complete response (CR) was defined by the complete disappearance of neurological symptoms and other disease-related symptoms, and by the complete improvement of radiological abnormalities related to DCs three months after EES treatment. Partial response (PR) was defined by the partial disappearance of neurological symptoms and other disease-related symptoms and by the partial improvement of radiological abonormalities three months after EES treatment. The main outcome measures were resection degree, postoperative complications, and recurrence rate. This study was approved by the Institutional Review Board of Eye & ENT Hospital of Fudan University. All participants signed informed consent forms.

Case presentations

Case 1

A 63-year-old female presented with numbness in the left face and tongue without an obvious trigger six months ago. After drug treatment in another hospital, there was no improvement. The patient was admitted and underwent CT, which revealed a cartilaginous soft tissue mass on the left MCFF (Figures 1A and 1B). The patient’s MRI demonstrated an intracranial and extracranial neurogenic tumor with cystic degeneration in the large wing of the left sphenoid bone (Figures 1C and 1D). Thus, the patient subsequently underwent endoscopic resection of the mass in the left MCF after the completion of preoperative examination and anesthetic evaluation. During the operation, the first step was to remove the left two-thirds of the middle turbinate and ipsilateral uncinate processes. The next step was to successively enlarge the ostium of the left maxillary sinus, ethmoid sinus, and sphenoid sinus. Then, the anterior wall of the sphenoid sinus was resected, and the bone of the bottom wall of the sphenoid sinus and the pterygoid root was removed. Specifically, the bone of the anterior wall of the maxillary sinus was removed via the left labigingival sulcus incision for better exposure. The lesion was located in the left skull base and posterolateral pterygoid process and extended to the MCF. The lumen of the lesion was fully exposed and enlarged, and a substantial amount of caseous outflow was observed. A bone defect in the middle cranial fossa was found, while the dura mater was intact, and the internal carotid artery was well protected. There were no significant surgery-related complications. Intravenous antibiotics were given postoperatively. On the fourth day of admission, the patient was discharged without any discomfort. The patient recovered well and the disease-related symptoms completely disappeared at the 88-month follow-up.

Figure 1.

Preoperative computed tomography and magnetic resonance imaging. (A and B) Computed tomography scanning revealed a soft tissue mass at the bottom of the left middle cranial fossa with uneven density. The mass was dilated and partially smooth, involving the left cavernous sinus, the bottom of the temporal lobe, the lateral wall of the sphenoid sinus, the parapharyngeal wall, the infraorbital fissure, the pterygopalatine fossa, and the infratemporal fossa. (C and D) Magnetic resonance imaging scanning showed a slightly irregular oval mass in the left pterygopalatine fossa, protruding outward into the infratemporal fossa and inward into the sphenoid sinus. T2WI showed uniform hyperintensity without enhancement.

Case 2

A 46-year-old male reported persistent occlusion of the right ear with low frequency hearing loss. He claimed to have received medical treatment (details are unknown) in another hospital and achieved slight improvement. Preoperative CT examination showed an enlarged soft tissue mass in the right pterygopalatine fossa, involving the ipsilateral pterygoid muscle, sphenoid wing, and infraorbital fissure. Neurogenic tumors were considered first (Figure 2A and 2B). Briefly, surgical intervention exposing the posterior wall of the maxillary sinus revealed the lesion in the pterygopalatine fossa and infratemporal fossa. The cyst cavity compressed the eustachian tube, pterygoid nerve, and paraclival internal carotid artery. Eventually, the cyst cavity was enlarged and internal fluid was removed. The patient reported that the sense of ear occlusion disappeared, their hearing was significantly improved after the operation, and there was no recurrence during the 82-month follow-up.

Figure 2.

Preoperative computed tomography. Computed tomography revealed a round dilated soft tissue mass in the right sphenoid sinus floor and pterygopalatine fossa with clear boundaries, slightly involving the infratemporal fossa, infraorbital fissure, and anterior segment of the eustachopharyngeal tube with low density and no enhancement.

Case 3

A 24-year-old female was taken to the local hospital for CT examination because of unknown nose bleeding. Imaging results revealed an abnormal signal focus adjacent to the right pterygopalatine fossa. Several months ago, the patient had experienced a twitch sensation on the lateral face of the right inferior orbital margin without facial numbness. For further diagnosis and treatment, plain CT and enhanced MRI scanning were performed in our hospital and revealed a large circular mass in the middle of the right skull base (Figures 3A-E). Consequently, infratemporal fossa lesion resection was performed. First, part of the anterior middle turbinate and the uncinate process was removed, and then the ostium of the maxillary sinus, ethmoid sinus, and sphenoid sinus were routinely enlarged. The sphenopalatine foramen was exposed to block the sphenopalatine artery and its branches, and the pterygoid nerve was also removed. Then, the anterior and posterior walls of the maxillary sinus were removed through the labigingival sulcus approach and the maxillary nerve was cut off. The cyst-like lesion was located in the infratemporal fossa, and the bean curd residue secretions were observed to spill out after exposing the pterygopalatine fossa and infratemporal fossa. The anterior skull base was well protected and no leakage of cerebrospinal fluid was observed. The patient continued to do well 62 months after surgery.

Figure 3.

Preoperative computed tomography and magnetic resonance imaging. (A, B and C) Sagittal, coronal, and horizontal enhanced computed tomography showed an expanded soft tissue mass in the right middle skull base and infratemporal fossa with low density and smooth edges. (D and E) Magnetic resonance imaging showed oval abnormal signal foci in the right middle skull base foramen ovale and infratemporal fossa with obvious hyperintensity on T2 sequences.

Case 4

A 37-year-old patient developed a right middle skull base mass with pharyngeal discomfort during physical examination three weeks ago. CT examination showed a 2 cm * 3.5 cm oval mass in the right middle skull base (not shown). Intraoperative surgery found that pterygopalatine fossa was exposed after removing the posterior wall of the maxillary sinus and then the mass was seen in the middle skull base after removing the process of the sphenoid bone. The capsule of the mass was dissected and was filled with yellow granules. Multiple imaging examinations after the operation did not find any signs of tumor recurrence (Figure 4A-D).

Figure 4.

Postoperative magnetic resonance imaging scan. Coronal (A and C) and axial (B and D) magnetic resonance imaging T2 sequences show complete dermoid cyst removal in the right middle skull base without any recurrence at the four-month and two-years follow-ups, respectively.

Case 5

A 61-year-old male developed left nasal congestion and occasional mucopurulent excretion with ear tightness and hearing loss eight years ago. He was treated in another hospital and achieved partial improvement after medical treatment (details unknown). For further diagnosis and treatment, he received horizontal and coronal MRI scanning examinations and imaging results showed an intracranial and extracranial communicating expandable mass occupying the left middle skull base (Figures 5A and 5B). For the main steps of the operations, part of the middle turbinate was routinely excised, and the ostium of the maxillary sinus, ethmoid sinus, and sphenoid sinus were enlarged in preparation for exposure of the lesion. The left anterior wall of the maxillary sinus was exposed through a labial-sulcus incision and enlarged with forceps. A circular incision was made on the posterior wall of the maxillary sinus under endoscopy to expose the pterygopalatine fossa (Figure 6A). Then, the inner and lateral plates of the pterygopalatine process were removed with yellow discharge overflow (Figure 6B). Next, the boundary was further expanded along the edge of the cyst until it was completely exposed and removed (Figure 6C-E). The remaining wound showed the dura of the middle skull base and internal carotid artery (Figure 6F). No obvious cerebrospinal fluid leakage was observed. The middle turbinate mucosa was used to cover the middle skull base and internal carotid artery. Postoperative imaging examination showed the process of lesion regression without recurrence (Figure 5C-F).

Figure 5.

Preoperative and postoperative magnetic resonance imaging scan. (A and B) T2 sequences demonstrated an intracranial and extracranial communicating mass in the left middle skull base containing fat components and compressing the left temporal lobe and the internal carotid artery. (C and D) On the first postoperative day, T2 sequences showed perioperative hemorrhage and edema around the temporal lobe. (E and F) T2 sequences showed complete dermoid cyst removal without any recurrence at the four-month follow-up.

Figure 6.

Endoscopic endonasal resection of dermoid cyst in left middle cranial fossa floor. Endoscopic appearance of dermoid cyst (A-D) and gross total resection (E and F).

Results

A total of 5 patients with DCs originating from MCF were enrolled (3 females and 2 males). The mean age of patients at diagnosis was 46.2 years. All patients underwent diagnosis, endoscopic surgery and long-term follow-up in our center. In this patient population, one patient presented with numbness of the left face and tongue, which was considered nervous system involvement. The other three patients reported hearing loss, nose bleeding, and nasal obstruction. Another patient had no symptoms and the DC was found on physical examination. In addition, all DCs originated from the MCFF (5 of 5, 100.0%). Specifically, 2 cases were from the pterygoid process, and the other 3 cases originated from the pterygopalatine fossa, the infratemporal fossa, and the great wing of the sphenoid bone. In regard to the extent of the DCs, 1 case involved the middle skull base bone, and another case affected the MCF. In almost all cases, patients underwent preoperative computed tomography (CT) or magnetic resonance imaging (MRI) examination and showed consistent features with rounded, well-delineated nonenhancd unilocular cystic hypodense masses of variable sizes containing fat and capsular calcium. However, the imaging result of one case demonstrated T1-weighted hypo- to iso- intense areas in the brain with contrast enhancement, and T2-weighted hyperintense areas. We initially thought this lesion was a schwannoma, but final pathological examination confirmed a DC. All patients underwent EES treatment and achieved total resection. The internal carotid artery was well protected during the operation, and no postoperative complications such as cerebrospinal fluid leakage or intracranial infection were found after skull base reconstruction. All patients were routinely given infection prevention, secretion drainage, and nasal irrigation postoperatively. The mean duration of follow-up was 73.2 months (range: 29–120). All patients achieved CR both clinically and radiologically within three months after EES treatment. Moreover, no relapses were observed in any of the patients during the follow-up. The detailed information of the five patients is shown in Table 1.

Table 1.

Clinical Data of Enrolled Patients.

	Age	Sex	Manifestation	Diagnosis	Location	Surgical approach	Total resection	Complications	Recurrence	Follow-up period
Case 1	63	Female	Left face and tongue numbness	Dermoid cyst	Left greater wing of sphenoid bone	Caldwell-Luc approach	Yes	No	No	120 months
Case 2	46	Male	Right hearing loss	Dermoid cyst	Right pterygopalatine fossa	Caldwell-Luc approach	Yes	No	No	82 months
Case 3	24	Female	Bilateral nose bleeding	Dermoid cyst	Right infratemporal fossa	Caldwell-Luc approach	Yes	No	No	62 months
Case 4	37	Female	No	Dermoid cyst	Right pterygoid process	Caldwell-Luc approach	Yes	No	No	57 months
Case 5	61	Male	Left nasal obstruction	Dermoid cyst	Left pterygoid process	Caldwell-Luc approach	Yes	No	No	29 months

Discussion

Skull base DCs as rare congenital diseases mostly occur in the midline region of the brain involving the MCF. The lesions mostly originate from the middle cranial fossa floor, the infratemporal fossa, the cavernous sinus, and the petrous apex.^5,6 To date, there have been no conclusive reports on the clinical features, therapeutic options, and prognostic outcomes of DCs arising from the MCFF. In this article, we summarized 5 patients’ clinical course with DCs originating from the MCFF at our center.

To our knowledge, the clinical manifestations of DCs are varied and lack of specificity, and neurological involvement has certain clinical significance. In our study, only one case was found to have neurological symptoms at onset. These cysts can enlarge before any symptoms or signs appear due to their slow growth, which depends on the location of the lesion and compression of adjacent structures. The common symptoms include focal neurological deficits, headaches, or meningitis,^7,15 and other less common manifestations, including seizures, hydrocephalus, visual field defects, exophthalmos, and oculomotor palsy. These common clinical manifestations are also seen in our data. Asymptomatic intracranial DCs are usually diagnosed by chance, and we also described one case in which the lesion was discovered by physical examination. Cysts can become spontaneous ruptures, traumatic ruptures, or rupture during surgery,^16,17 but these situations were not observed in our cases. Once ruptured, cyst components spread into the subarachnoid space and may present with a variety of symptoms and signs, including headache, nausea, vomiting, visual impairment, dizziness, epilepsy, aseptic chemical meningitis, hemiplegia, and psychiatric changes.^7,15

The use of CT and MRI not only facilitates the diagnosis, but also helps in planning the surgical approach.^3,4 Our imaging findings conform to the general features of DCs being rounded, well-delineated nonenhanced unilocular cystic hypodense masses of variable sizes containing fat and capsular calcium. Notably, in one special case, T1-weighted MRI scans showed hypo- to iso- intense areas in the brain with contrast enhancement, while T2-weighted images showed hyperintense areas. We initially thought it was a schwannoma, but it was eventually confirmed as DCs by postoperative histopathological examination. Lipoma, epidermoid cyst, arachnoid cyst, craniopharyngioma, and teratoma should be considered in the differential diagnosis. Suppression of lipoma in fat-suppressed sequences and uniform visualization are important features for differentiating these lesions from DCs. Thus, histopathological examinations remain the gold standard for diagnosis.

Surgery is the only effective treatment with the goal of a complete resection of the lesion to prevent recurrence. Radiation and chemotherapy have not been shown to be sensitive in treating DCs and thus are not recommended.¹⁸ By reviewing the literature, we concluded that multiple surgical treatment options were used for the treatment of DCs originating from the MCFF (Table 2), but the reported total resection rate needs to be further improved. Our study is the first large case series reporting the EES treatment of the DCs arising from the MCFF. Many studies have reported that the resection approach and extension are still debatable. With the development of microneurosurgical techniques, it is reported in most literature that total resection can be accomplished.^{4,7,15,19

-25} However, it has been suggested that aggressive surgical procedures may increase complications and mortality,^5,26,27 especially if the lesions adhere to the neurovascular structures.^2,28 Thus, subtotal resection is advocated for a low recurrence rate, which has been reported in only a few studies.^5,13,29 Compared with open surgery, EES can also be used in the separation and exposure of the internal carotid artery and affected nerve, when sufficiently guided by experts, and the lesions can be removed completely. The advantage of EES is that it allows reliable access to the cyst via the Caldwell-Luc approach and provides a large drainage window for endoscopic follow-up. In our study, this approach improved clinical manifestations and radiological abnormalities within three months after EES treatment. Moreover, there were no postoperative complications or relapses found during the long-term follow-up. Thus, endonasal endoscopic removal of MCF lesions is considered as a less-invasive and appropriate procedure with lower morbidity for treating these patient populations.

Table 2.

Clinical features and therapeutic results of reviewed literature.

Publishtion year	Patient no./age at diagnosis, years/sex	Tumor size in imaging	Surgical option	Extent of resection	Complications	Outcome
2019³⁰	1/60/F	Meckel’s cave	Endoscopic transorbital approach	Total resection	No	Recovery
2018²⁰	1/43/F	Infratemporal fossa	Fronto-temporo-sphenoidal craniotomy	Total resection	No	Recovery
2015¹³	2/37/M	Cavernous sinus	Endonasal endoscopic excision	Subtotal resection	No	Recovery
2015²¹	3/55/F	Infratemporal fossa	Extradural anterior infratemporal fossa approach	Total resection	No	Recovery
2014²²	4/21/F	Infratemporal fossa	Frontotemporal craniotomy	Total resection	No	Recovery
2011²³	5/3/F	Temporal fossa	Pterional carniotomy and elliptical excision	Total resection	No	Recovery
2009²⁹	6/37/F	Petrous apex	Temporal craniotomy	Subtotal resection	No	Recovery
2007¹⁵	7/49/M	Petrous apex	Extradural subtemporal approach	Total resection	No	Recovery
2006²⁴	8/41/F	Cavernous sinus	Single-flap zygomatic Osteotomy and frontotemporal approach	Total resection	No	Recovery
2004⁵	9/18/M	Parasellar region	Single-flap fronto-orbitozygomatic craniotomy	Subtotal resection	No	Recovery
2002²⁵	10/17/F	Infratemporal fossa	Lateral approach	Total resection	No	Recovery

Conclusion

Dermoid cysts are basically benign and generally become symptomatic secondary to their mass effect on neural structures. Radiological imaging is still essential for diagnosis, but ultimately depends on pathological examinations. EES is considered safe and effective in the treatment of DCs in MCFF. A larger sample size and longer follow-up time are needed.

Footnotes

Acknowledgments

The authors thank the researchers of the original studies included in this paper.

Author Contributions

HX drafted the manuscript. WL performed the statistical analysis. DW conceived and designed the study manuscript. HW and HZ acquired the data. LH supervised the study and corrected the manuscript. All authors contributed to the article and approved the submitted version.

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was financially supported by the National Natural Science Foundation of China (No. 81870703), Shanghai Shen Kang Hospital Development Center (SHDC12018118), Science and Technology Commission of Shanghai Municipality (20Y11902000, 21ZR1411700), and Shanghai Municipal Health Commission (201940143).

ORCID iD

Dehui Wang

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Endoscopic Endonasal Surgery for Dermoid Cysts Arising From the Middle Cranial Fossa Floor: A Rare Case Series

Abstract

Objective

Methods

Results

Conclusion

Keywords

Introduction

Patients and methods

Study design

Case presentations

Case 1

Case 2

Case 3

Case 4

Case 5

Results

Discussion

Conclusion

Footnotes

Acknowledgments

Author Contributions

Declaration of Conflicting Interests

Funding

ORCID iD

References