66 JOURNAL OF KOREAN SKULL BASE SOCIETY SEPTEMBER | Vol. 12 | No. 2
가톨릭대학교 의과대학 성바오로병원 이비인후과학교실
김지선, 김수빈, 오정훈, 장기홍
Department of Otolaryngology, St. Paul’s Hospital, The Catholic University of Korea, College of Medicine, Seoul, Korea
Ji-sun Kim, Su-Bin Kim, Jeong-Hoon Oh, Ki-Hong Chang
측두외로 확장된 거대 콜레스테롤 육아종 1례
J Korean Skull Base Society 12권 2호 : 66~69, 2017
종설1 종설2 원저1
증례1 원저2
증례2 증례3 증례4 증례5 증례6 증례7 증례8 증례9
Cholesterol granulomas and cysts may arise from any portions of the pneumatized temporal bone of human, resulting from foreign body reactions to the accumulation of cholesterol crystals. Most of them arise from middle ear cleft, mastoid process and petrous apex but extra-temporal extension, especially cervical extension, is rare. In general, the size is not so large before clinical detection.
The treatment principle for cholesterol cyst is surgical drainage. The authors experienced and treated a 53-years-old female patient with a huge cholesterol granulomatous cyst extending from the mastoid cavity to the posterior cranial fossa and the neck. Consistent drainage of intracranial cyst lesion was achieved after removal of cyst in mastoid cavity and the surgical defect in the neck was obliterated by utilizing a rotational temporalis myofascial flap. We report this case with relevant review of literature.
A case of huge cholesterol granulomatous cyst with massive extratemporal extension
논문 접수일 : 2017년 8월 5일 논문 완료일 : 2017년 8월 25일
주소 : Department of Otolaryngology, St. Paul’s Hospital, The Catholic University of Korea, College of Medicine, 180, Wangsan-ro, Dongdaemun-gu, Seoul, Korea Tel : +82-2-961-4501
Fax : +82-2-968-7250 E-mail : [email protected]
Ki-Hong Chang
교신저자
Cholesterol Granuloma, Posterior cranial fossa, Neck, Temporal bone Key Words
67
측두외로 확장된 거대 콜레스테롤 육아종 1례
▒ INTRODUCTION
Cholesterol granuloma and cyst are known to be formed by repeated courses of foreign body reaction to hemosiderin and cholesterol crystals, which occur from transudate of blood from mucosal edema of gas resorption, hypoxia, and negative pressure from chronic state of inefficient gas exchange within the pneumatized system.[1]
Cholesterol granuloma typically involves middle ear cavity, mastoid cavity and petrous apex, and does not usually extend extra-temporally.[2, 3] Giant cholesterol granulomas with intracranial extensions have been seldom reported in the literature,[4, 5] and the case with cervical extension is even more rare. The treatment principle for cholesterol cyst is surgical drainage. The authors recently experienced huge mastoid cholesterol granulomatous cysts with massive extra-temporal extension.
▒ CASE REPORT
A 53-years-old female patient admitted with complaints for 3 months of dizziness and right aural fullness. She had also noted hard of hearing on the same side for about a year and pulsatile tinnitus on the contralateral side. Otoscopic examination revealed normal tympanic membrane but sagging on posterior portion of the external auditory canal in the right ear. There was also painless swelling over the right mastoid tip. There was horizontal gazing nystagmus with fast component toward the left side when she gazed to the left. On pure tone audiogram, air and bone conduction threshold on the right side was 45 dB and 10dB, respectively and within normal limits on the left side. On caloric test, there was 51% canal paresis in the right horizontal semicircular canal.
Computed tomography of the temporal bone showed a huge lobulated expansile mass in the right mastoid cavity.
Fig. 2
A B Magnetic resonance imaging shows
huge expansile lobulated mass in the right mastoid cavity and posterior cranial fossa expanding into the neck at about level of the first cervical vertebra with high signal intensity in both T1 (A) and T2-weighted images (B).
Fig. 1
A B Computed tomography of the temporal
bone shows a huge lobulated mass (arrow) on the right mastoid cavity with bone defect in the tegmen tympani and the sigmoid sinus area (A), which extended into the upper neck to first cervical vertebra level (B).
68 JOURNAL OF KOREAN SKULL BASE SOCIETY SEPTEMBER | Vol. 12 | No. 2
It extended posteriorly to the posterior cranial fossa causing a bony defect at the tegmen and near the sigmoid sinus, and inferiorly to the neck to about the level of the first cervical vertebra (Fig. 1A, B). On magnetic resonance imaging (MRI), the mass showed high signal intensity in both T1 and T2-weighted images (Fig. 2A, B) without enhancement after Gadolinium infusion.
When performing the mastoidectomy, a cystic mass with brownish serous fluid found filling the antrum and aditus ad antrum. The mass was extending to the posterior cranial fossa, while another cystic mass of the same consistency found posterior to the jugular bulb. The latter mass further extended down to the level of first cervical vertebra passing through the retrofacial cell tract. The posterior fossa dura was uninjured and the middle ear was unremarkable except for edema of the middle ear mucosa.
Granulomatous tissue and cyst wall in mastoid cavity was removed through intact bridge mastoidectomy and
granulomatous tissue and cyst wall surrounding jugular bulb and first cervical vertebra was removed completely approaching through retrofacial cell track. The surgical defect in the mastoid and the neck was obliterated with abdominal fat, which was later covered by autogenous temporalis muscle fascia. Light microscopic examination of the removed specimen revealed cholesterol crystals surrounded by inflammatory cells within connective tissue with abundant red blood cells.
The patient was discharged on the sixteenth postoperative day. But on the follow up examination, there was continuous ear discharge and signs of inflammation in the grafted fascia and fat, despite frequent ear dressing and antibiotic treatment. Due to the resorption of the grafted material and persistent otorrhea, the inflamed tissue was removed and muscle flap was applied to prevent further infection. Under the general anesthesia, large curvilinear skin incision was done postauricularly to expose enough temporalis muscle and superficial temporal artery.
Seven cm sized temporalis muscle in ovoid shape, pedicled by superficial temporal artery, was designed. It was rotated posteriorly and inferiorly to obliterate the surgical defect in the mastoid area and the neck. The wound was well healed and further infection was not developed. MRI checked 5years after the operation revealed that the huge cystic mass in mastoid cavity, posterior cranial fossa and neck disappeared, and the surgical defect of the upper neck was well obliterated(Fig. 3). The patient had no specific complaints during the follow up period. So we plan to check up continuously based on outpatient clinic.
▒ DISCUSSION
Clinically, cholesterol granuloma of the temporal bone may be presented as chronic otitis media, idiopathic hemotympanium, or as localized mass.[6] Cholesterol granuloma of the middle ear are readily detected because symptom like hearing impairment appears early in the disease and are easily noted on physical examination.
Fig. 3
The follow-up magnetic resonance imaging (MRI). The shifted cerebellum was repositioned well with a few postoperative fluid collections in posterior cranial fossa on MRI at 5 years after operation.
69
측두외로 확장된 거대 콜레스테롤 육아종 1례
When occurring in the mastoid cavity, swelling on the posterior wall of the external auditory canal and postauricular area are often found. Lesions on petrous apex can cause eustachian tube dysfunction, carotid artery involvement, brain stem dysfunction, otic capsule infiltration, or cranial nerve signs such as headache, diplopia, aural fullness, dizziness, paresthesia of V2 area, hearing loss, or facial weakness.[7, 8] The patient in this case had complained of dizziness, hearing loss, aural fullness on the affected side, and pulsatile tinnitus on the contralateral side. Interestingly, pulsatile tinnitus in the unaffected side disappeared after surgery in our case. This phenomenon may be attributed to the release of vascular compression postoperatively in the affected side, which consequently relieved the dominant venous flow causing tinnitus in the unaffected side.
When managing cholesterol granulomatous cysts, surgical procedure is not required if there are no specific signs or symptoms associated with the lesion. For surgical intervention, if drainage and permanent ventilation system can be acquired, no further manipulation is needed.[9] In our case, intratemporal lesion was able to be managed with natural drainage through a middle ear cavity.
However, permanent drainage route for neck lesion was not able to be made because gravity dependent drainage was not possible. At first, we thought the surgical defect in the mastoid cavity and the neck could be managed with abdominal fat obliteration covered by fascia. But the wound infection was developed shortly after operation and as a result, pedicled temporalis muscle rotational flap was applied after wound debridement. Therefore, if surgical defect is somewhat large and postoperative infection is concerned, the vascularized muscle flap should be considered rather than abdominal fat graft.[10]
Cholesterol granulomas rarely appear in size over several centimeters,[11-13] and even more rarely have extradural extensions involving the intracranial structures and the neck.[4, 5] Therefore, we report a unique case of mastoid cholesterol granulomatous cysts with massive extra-
temporal extension, which was successfully managed with surgical drainage and coverage with pedicled temporalis muscle flap.
References
1. Roland PS, Meyerhof f WL, Judge LO, Mickey BE. Asymmetric pneumatization of the petrous apex. Otolaryngol Head Neck Surg 1990;103:80-8.
2. Graham MD, Kemink JL, Latack JT, Kartush JM. The giant cholesterol cyst of the petrous apex: a distinct clinical entit y. Lar yngoscope 1985;95:1401-6.
3. Polo R, Medina M, Labatut T, Alonso A, Vaca M. Mastoid cholesterol granuloma with posterior cranial fossa compression. Otol Neurotol 2013;34:e103-4.
4. Gamache FW, Jr., McLure T, Deck M, Linstrom C. Bilateral cholesterol granuloma of the skull base: case report and review of the literature.
Neurosurgery 1988;22:1098-101.
5. Morioka T, Fujii K, Nishio S, Miyagi Y, Nagata S, Hasuo K, et al. Cholesterol granuloma in the middle cranial fossa: report of two cases. Neuroradiology 1995;37:564-7.
6. Rinaldo A, Ferlito A, Cureoglu S, Devaney KO, Schachern PA, Paparella MM. Cholesterol granuloma of the temporal bone: a pathologic designation or a clinical diagnosis? Acta Otolaryngol 2005;125:86-90.
7. Sweeney AD, Osetinsky LM, Carlson ML, Valenzuela CV, Frisch CD, Netterville JL, et al. The natural history and management of petrous apex cholesterol granulomas. Otol Neurotol 2015;36:1714-9.
8. Isaacson B. Cholesterol granuloma and other petrous apex lesions.
Otolaryngol Clin North Am 2015;48:361-73.
9. Fong BP, Brackmann DE, Telischi FF. The long-term follow-up of drainage procedures for petrous apex cholesterol granulomas. Arch Otolaryngol Head Neck Surg 1995;121:426-30.
10. Bajpai H, Saikrishna D. The versatility of temporalis myofascial flap in maxillo-facial reconstruction: a clinical study. J Maxillofac Oral Surg 2011;10:25-31.
11. Palva T, Lehto VP, Johnsson LG, Virtanen I, M kinen J. Large cholesterol granuloma cysts in the mastoid. Clinical and histopathologic findings. Arch Otolaryngol 1985;111:786-91.
12. Martin TP, Tzifa KT, Chavda S, Irving RM. A large and uncharacteristically aggressive cholesterol granuloma of the middle ear. J Laryngol Otol 2005;119:1001-3.
13. Jaramillo M, Windle-Taylor PC. Large cholesterol granuloma of the petrous apex treated via subcochlear drainage. J Laryngol Otol 2001;115:1005-9.
