6. Crispin JC, Diaz-Jouanen E. Systemic lupus erythematosus inducedby therapy with interferon-beta in a patient with multiple sclerosis.Lupus 2005;14:495–6.

7. Polman CH, Kappos L, Dahlke F, et al. Interferon beta-1btreatment does not induce autoantibodies. Neurology 2005;64:996–1000.

8. Kivisakk P, Lundahl J, von Heigl Z, et al. No evidence for increasedfrequency of autoantibodies during interferon-beta 1b treatment ofmultiple sclerosis. Acta Neurol Scand 1998;97:320–3.

9. Colosimo C, Pozzilli C, Frontoni M, et al. No increase of serumautoantibodies during therapy with recombinant human interferon-beta 1a in relapsing-remitting multiple sclerosis. Acta Neurol Scand

1997;96:372–4.10. Hirohata S, Kosaka M. Association of anti-Sm antibodies with

organic brain syndrome secondary to systemic lupus erythematosus.Lancet 1994;343:796.

11. Swaak AJ, Huysen V, Nossent JC, et al. Antinuclear antibodyprofiles in relation to specific disease manifestations of systemic lupuserythematosus. Clin Rheumatol 1990;9:82–94.

12. Silva LM, Garcia AB, Donadi EA. Increased lymphocyte death byneglect-apoptosis is associated with lymphopenia and autoantibodiesin lupus patients presenting with neuropsychiatric manifestations. J

Neurol 2002;249:1048–54.13. McLean BN, Miller D, Thompson EJ. Oligoclonal banding of IgG in

CSF, blood-brain barrier function, and MRI findings in patients withsarcoidosis, systemic lupus erythematosus, and Behcet’s diseaseinvolving the nervous system. J Neurol Neurosurg Psychiatry

1995;58:548–54.14. Kurtzke JF. Rating neurological impairment in multiple sclerosis: an

expanded disability status scale (EDSS). Neurology (Cleveland)

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doi:10.1016/j.jocn.2006.07.011

Intracranial mucocele in pregnancy

Murat Cosar a, Mustafa Aziz Hatiboglu b,*, Emine Cosar c, A. Celal Iplıkcıoglu b

a Department of Neurosurgery, Afyon Kocatepe University Medicine Faculty, Afyon, Turkeyb Department of Neurosurgery, Okmeydani Training Hospital, Istanbul, Turkey

c Department of Obstetrics and Gynecology, Afyon Kocatepe University Medicine Faculty, Afyon, Turkey

Received 25 January 2006; accepted 2 August 2006

Abstract

Intracranial mucocele is rare. They are slow-growing lesions and usually form as a result of an obstruction of the paranasal sinuses,particularly by osteoma, fibrosis, trauma, previous surgery or inflammation. The effect of pregnancy in the development of intracranialmucocele is unclear. We report a pregnant patient who was admitted to our clinic with severe headache and diplopia. A giant mucocele inthe right frontal lobe was detected on magnetic resonance imaging. The patient underwent surgery with satisfactory outcome.� 2006 Elsevier Ltd. All rights reserved.

Keywords: Frontal lobe; Intracranial extension; Mucocele; Surgery

1. Introduction

Mucoceles are slow-growing and locally aggressive le-sions which form due to the accumulation of mucous secre-tions in a paranasal sinus secondary to obstruction causedby inflammation, fibrosis, trauma, previous surgery, ana-tomical abnormality or by a mass lesion such as an oste-oma. Most mucoceles arise in the frontal and ethmoidalsinuses, but they rarely extend into the intracranial cavity.We report a pregnant patient with a giant mucocele in the

frontal region. The possible role of pregnancy in the devel-opment of intracranial mucocele is also discussed.

2. Case report

A 35-year-old woman in the 7th month of pregnancywas admitted to our clinic with a history of severe head-ache and diplopia of 1-month duration. Also, the patienthad complained of nasal obstruction and rhinitis overthe previous few months. Neurological examination re-vealed a sixth nerve palsy of the right eye and bilateralpapilledema. The patient had no history of previous sur-gery or trauma.

MRI showed a mass lesion measuring 4 · 4 · 7 cmwhich was hypointense on T1-weighted and hyperintense

* Corresponding author. Present address: Turkali Mah. Sehit Asim Cad.Sakarya Apt. 104/2, 80690, Besiktas, Istanbul, Turkey. Tel.: +90 532 76122 90; fax: +90 212 2217800.

E-mail address: azizhatiboglu@yahoo.com (M.A. Hatiboglu).

1000 Case reports / Journal of Clinical Neuroscience 14 (2007) 1000–1003

on T2-weighted images in the right frontal lobe. The lesioninvolved the ethmoid sinuses and was compressing theright lateral ventricle (Fig. 1 A,B).

A right frontal craniotomy was performed. There was acapsule and mild edema around the lesion. The thickencapsulated mass was seen and opened. White-gray vis-cous liquid, including mucinous tissue was evacuated. Noanatomical structures were recognized between the lesionand ventricle. Postoperative culture of pus was negativeand no bacteria were seen in direct smear tests. The histo-pathological diagnosis was mucocele with ciliated respira-tory epithelium (Fig. 2).

Postoperative neurological examination revealed no def-icit. Otorhinological examination and investigation re-

vealed no CSF leak after the operation. Follow-up MRIwas obtained 1 week after the operation and showed post-operative changes and no residual mass (Fig. 3 A,B).The patient was treated medically for rhinitis by theOtolaryngology Clinic and discharged on the 10th postop-erative day. She delivered her baby 2 months after theoperation.

3. Discussion

The majority of mucoceles arise in the frontal andethmoid sinuses.1–6 Sinus inflammation, altered mucus pro-duction, and obstruction of the sinus ostium are consideredcauses of mucoceles.7 A sinus mucocele may enlarge suffi-ciently to compress intracranial structures and erode adja-cent bone.3

Although the pathogenesis of primary intracranial muco-celes is uncertain, there are two hypotheses about their for-mation. One hypothesis is the obliteration of pneumatizingair cell tracts. The other involves the inclusion of ectopicseromucinous tissue within developing bone.3 Lund et al.8

showed that cytokines, especially interleukin (IL)-1 and IL-6 are responsible for the osteolytic properties of mucoceles.

Our patient had no history of sinus obstruction, oste-oma, inflammation, fibrosis, trauma, previous surgery oranatomical abnormality. How pregnancy may precipitatesymptoms of an intracranial mucocele is unclear. However,the incidence of rhinitis is high in pregnancy, is usually re-lated to physiological changes during pregnancy and mayresult in nasal congestion and obstruction. This is calledrhinitis of pregnancy. Measurements of the nasal airwayincluding anterior rhinoscopy, peak inspiratory and expira-tory nasal flow and saccarin tests show decreased nasal pa-tency in pregnancy. The rise in the serum concentration offemale sex hormones with gestational age affects the nasalmucosa and may cause nasal obstruction and rhinitis.9–11

Fig. 1. (A) Preoperative axial T1-weighted MRI shows a mass in the rightfrontal lobe compressing the right lateral ventricle and falx. (B) Preop-erative sagittal T1-weighted MRI shows a mass in the frontal lobecompressing the lateral ventricle.

Fig. 2. Photomicrograph of a histopathological section, revealing a cystlined with ciliated respiratory epithelium, with a chronic inflammatoryreaction (H&E, ·100).

Case reports / Journal of Clinical Neuroscience 14 (2007) 1000–1003 1001

In addition, pregnancy may lead to a slight increase in thesize of the intracranial mass lesion. Several possibilitieshave been advanced to account for the manner in whichpregnancy might influence the lesion size. Suggested mech-anisms include accelerated growth rate, vascular engorge-ment and increased fluid content.12 To our knowledge,this is the first report of an intracranial mucocele in preg-nancy. We have hypothesized that the increased level ofprogesterone in pregnancy may increase the volume ofintracellular and extracellular fluid in the patient, resultingin an increase in the volume of the mucocele and therebycompressing the surrounding neural structures.

Most patients are diagnosed on clinical symptomsassociated with intracranial complications such asCSF rhinorrhea, pneumocephalus, neuro-ophthalmologi-cal disturbances, convulsion and intracranial infection.6,13

In our patient, a giant mucocele in the right frontal lobewas found and was associated with headache and diplopia.There was no evidence of CSF leak and pneumocephaluswas not seen on MRI.

Radiodiagnostic features of mucocele are related tothe duration of obstruction. On CT scans, mucocelesusually appear isodense to brain, depending on the watercontent of the mucus.5,14 The appearance of mucoceleson MRI is variable. The protein concentration and vis-cosity of the mucocele affects the signal intensities onT1-weighted images, whereas dehydration of mucocele af-fects the signal intensities on T2-weighted images. Gener-ally, mucoceles are hypointense on T1-weighted imagesand hyperintense on T2-weighted images.3,5 Differentialdiagnoses include epidermoid tumour, calcified meningi-oma, glioma, parasite infections (cystocercosis), andposttraumatic porencephaly.6

The treatment of intracranial mucocele is total extirpa-tion of the lesion. The complications of operation areCSF rhinorrea, pneumocephalus and infection. However,there was no CSF leak or pneumocephalus on MRI forour patient.

In conclusion, intracranial mucocele without known eti-ological factors in a pregnant woman is presented. Thepregnancy related physiological changes, pregnancy rhini-tis and female sex hormones during pregnancy mayaggravate the formation, volume and symptoms of intra-cranial mucocele. Delivery may decrease the symptomsbut total excision or evacuation of the mass is necessaryfor definitive treatment.

References

1. Akay KM, Onguru O, Sirin S, et al. Association of paranasal sinusosteoma and intracranial mucocele-two case report. Neurol Med Chir

(Tokyo) 2004;44:201–4.2. Alsarraf R, Goldman ND, Kuntz C, et al. Isolated intracranial

mucocele. Arch Otolaryngol Head Neck Surg 1999;125:1023–4.3. Chung DS, Park YS, Lee JH, et al. Mucocele of anterior clinoid

process: case report. Neurosurgery 1999;45:376–8.4. Kawaguchi S, Sakaki T, Okuno S, et al. Giant frontal

mucocele extending into the anterior cranial fossa. J Clin Neurosci

2002;9:86–9.5. Manen SR, Bosch DA, Peeters FLM, et al. Giant intracranial

mucocele:case report. Clin Neurol Neurosurg 1995;97:156–60.

6. Nakajima Y, Yoshimine T, Ogawa M, et al. A giant intracranialmucocele associated with an orbitoethmoidal osteoma. J Neurosurg

2000;92:697–701.7. Diaz F, Latchow R, Duvall AJ, et al. Mucoceles with intracranial and

extracranial extensions. J Neurosurg 1978;48:284–8.8. Lund VJ, Henderson B, Song Y. Involvement of cytokines and

vascular adhesion receptors in the pathology of fronto-ethmoidalmucoceles. Acta Otolaryngol 1993;113:540–6.

9. Ellegard EK, Karlsson G. Nasal congestion during pregnancy. Clin

Otolaryngol 1999;24:307–11.10. Ellegard EK, Karlsson Ng. Nasal mucociliary transport in pregnancy.

Am J Rhinol 2000;14:375–8.11. Phillpott CM, Conboy P, Al-Azzawi F, et al. Nasal

physiological changes during pregnancy. Clin Otolaryngol 2004;29:343–51.

12. Aminoff MJ. Neurologic disorders. In: Creasy RK, Resnik R, editors.Maternal-fetal Medicine. 4th ed. Philadelphia: W.B. Saunders; 1999.p. 1091–119.

Fig. 3. (A) Postoperative axial T1-weighted MRI showing no residual mass. (B) Postoperative sagittal T1-weighted MRI showing no residual mass.

1002 Case reports / Journal of Clinical Neuroscience 14 (2007) 1000–1003

13. Wang L, Kim J, Heilman CB. Intracranial mucocele as a complicationof endoscopic repair of cerebrospinal fluid rhinorrhea: case report.Neurosurgery 1999;45:1244–6.

14. Chiarini L, Nocini PF, Bedogni A, et al. Intracranial spread of a giantfrontal mucocele: case report. Br J Oral Maxillofac Surg

2000;38:637–40.

doi:10.1016/j.jocn.2006.08.003

Lipomatous meningioma

Shigeo Ohba a,*, Kazunari Yoshida a, Takekazu Akiyama a, Eiji Ikeda b, Takeshi Kawase a

a Department of Neurosurgery, Keio University School of Medicine, 35 Shinano-machi, Shinjuku-ku, Tokyo 160-8582, Japanb Department of Pathology, Keio University School of Medicine, Tokyo, Japan

Received 25 December 2005; accepted 25 March 2006

Abstract

We present a case of intracranial lipomatous meningioma in the parietal convexity in a 64-year-old woman. The mass showed lowdensity on computed tomography, was hyper-intense on T1-weighted magnetic resonance images, and had decreased intensity uponimaging with fat-suppressed sequences. Gross total removal of the tumor was performed. Histopathologically, the tumor was a menin-gioma with mixed transitional and lipomatous patterns. Immunohistochemically, the meningothelial foci were positive for epithelialmembrane antigen (EMA) and vimentin, and negative for S-100 protein. The lipomatous foci were positive for EMA, vimentin, andS-100 protein. The Ki-67 index values of the meningothelial and lipomatous foci were 1.0% and 1.8%, respectively. We review previousreports of lipomatous meningioma and discuss its clinical presentations and pathology.� 2006 Elsevier Ltd. All rights reserved.

Keywords: Immunohistochemistry; Lipomatous; Meningioma; Ki-67

1. Introduction

Meningioma is a common tumor that constitutes approx-imately 20% of all primary brain tumors.1 Lipomatousmeningioma was first reported by Bailey and Bucy in1931.2 This histological type is well described but uncommon.Roncaroli reported that lipomatous meningioma represents0.3% of meningiomas.3 In the 2000 World Health Organiza-tion Classification of Tumors of the Nervous System, menin-giomas with lipomatous changes are classified as metaplasticmeningiomas, as are meningiomas with osseous, cartilagi-nous, myxoid, and xanthomatous changes.4 Previous studieshave reported a variety of different immunoperoxidase stain-ing characteristics in lipomatous meningioma, and these arereviewed.3,5–12 In addition, we compared the Ki-67 indicesin the lipomatous and non-lipomatous areas of the currentcase, a feature not described in previous reports.

2. Case report

A 64-year-old woman was referred to our hospital be-cause incidental magnetic resonance imaging (MRI) re-vealed an abnormal mass on the right parietal convexity.

On examination, the patient had no neurological deficit,nor did she have relevant personal or family history.MRI revealed an extra-axial mass, 2.0 cm in diameter, thatwas hyper-intense on T1- and T2-weighted images(Fig. 1A). T1-weighted gadolinium-enhanced MRI re-vealed a homogenously enhanced lesion with a dural tail.The mass showed decreased intensity on MRI with fat-sup-pressed sequences (Fig. 1B). A plain computed tomogra-phy (CT) scan revealed a low-density mass (Fig. 1C).Based on the MRI, CT scan findings, and tumor location,the tumor was diagnosed preoperatively as meningiomawith a fatty component. The patient wanted the tumor re-moved, and this was carried out by parietal craniotomy.During surgery, it was discovered that the tumor had in-vaded the superior sagittal sinus (SSS). Therefore, we per-formed gross total removal of the tumor, except for theportion invading the SSS.

Macroscopically, the tumor was yellow, soft, andindistinguishable from adipose tissue. Histopathologicalexamination revealed the characteristic appearance of ameningioma composed of mature adipose tissue. Themeningothelial area included whorl formations and a fi-brous pattern. The proportion of lipid content wasabout 50%. All of the lipomatous areas took the formof mature adipocyte-like cells. There was no lipoblast-like or xanthomatous area, no atypical features and no

* Corresponding author. Tel.: +81 3 5363 3808; fax: +81 3 3354 8053.E-mail address: di045009@sc.itc.keio.ac.jp (S. Ohba).

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