신생아에서 심한 안와골 결손을 동반한 교세포 분리종...and maxillofacial region:...
TRANSCRIPT
- 307 -
: 2014 10 6, : 2014 10 16 : 2014 10 17 : , 156-755 102 : 02)6299-3181, : 02)6264-2167 E-mail : [email protected]
Copyright By The Korean Society of Perinatology
25 4, 2014 Korean J Perinatol Vol.25, No.4, Dec., 2014 http://dx.doi.org/10.14734/kjp.2014.25.4.307
: 40 1, 2,850
g,
(Fig. 1)
5 .
: 29 ,
.
.
.
4
(glial heterotopia)
(glial choristoma)
.1-3 (extracranial midline
structures) , , (nasopharynx)
(oropharynx) ,
(extracranical non-mildline structures)
.5, 6
, ,
1
1, 2, 3
1^1^1^2^3
Glial Choristoma Accompanied with Severe Bony Defect in Orbit in a Newborn
Suk Hwan Han, M.D.1, Tae Hee Kim1, Na Mi Lee, M.D.1, Jae Kyun Kim, M.D., Ph.D.2, and She Young Lee, M.D., Ph.D.3
1Departments of Pediatrics, 2Radiology, 3Otorhinolaryngology, Chung-Ang University Hospital, Chung-Ang University College of Medicine, Seoul, Korea
Glial choristoma is a mass-like lesion composed of otherwise normal, mature brain tissue that is isolated from the spinal cord and cranial cavity. In generally, this choristoma involves extracranial midline structures, especi- ally the nose, as well as the nasopharynx, oropharynx and cleft. Glial choristomas rarely involve extracranial non-midline structures such as the scalp, orbit, middle ear or mastoid and account for approximately 10% of all glial choristomas. Moreover, such glial choristoma presentations are usually seen in adults, most of whom have predisposing factors such as trauma, surgery, or chronic inflammation. Here, we present a case of a glial choristoma that invaded a neonate’s the nasopharynx, oropharynx, oral cavity and orbit.
Key Words : Brain, Choristoma, Heterotopia, Orbit, Newborn
- 308 -
×4 cm
.
.
(stridor) .
.
: (magnetic reso-
nance imaging, MRI) 3.1×2.4 cm
(Fig. 2A). 2.5×2.2 cm
(multiloculated cystic mass)
(masticator space) (buccal space)
(foramen lacerum)
(Fig. 2B).
(computed tomography, CT)
(Fig. 2C).
.
(debulking surgery) (excisional biopsy)
(glial tissue)
(choroid plexus) (Fig. 3A).
(glial fibrillary acidic protein,
GFAP) (Fig. 3B).
(cystic fluid)
. (normal cellu-
larity) (atypia) (pleo-
morphism) ,
A B Fig. 1. Gross photographs taken prior to surgery. A mass at the right buccal area (A) and a mass in the oral cavity with mouth was opened (B).
A B B C Fig. 2. The patient’s radiologic findings. Magnetic resonance imagine (MRI) showing a mixed cystic and solid lesion approximately 3.1x 2.4 cm with a bony defect in the right inferior orbital wall (A). MRI showing a multiloculated cystic mass approximately 2.5x2.2 cm with peripheral enhancement in the right masticator and buccal space as well as connected with cystic lesion in right foramen lacerum on MRI (B) and a computed tomography scan showing a bony defect in the right orbit floor (C).
: - -
- 309 -
.
(marsupialization)
.
, 2-3 , ,
.
(teratoma), (glioma),
(neurofibroma), (myofibroma), (en-
.
1907 Wolbach ,
, (spinal cord)
- .8
6
(intraparenchymal lesions, dural and leptomeningeal
lesions, intracranial extracerebral lesions, midline
lesions, distal lesions of the lung and uterus, extra-
cranial non-midline lesions) .3, 4, 9
, .3, 6
,
.4, 6, 9, 10.
.5 , (middle ear),
(maxillofacial), (temporal bone)
,1, 3, 11
.
, ,
.
.
, .6, 9
,
.
(hypercellularity), (cellular pleo-
Fig. 3. Patient’s pathologic findings. The mass is composed of mature glial tissue and choroid plexus-like area with papillary formation (A, Hematoxylin-and-eosin stain, x100). Both the glial and choroid plexus-like area showing a strong positive reaction for glial fibrillary acidic protein (B, x100).
Suk Hwan Han, et al. : - Glial Choristoma Accompanied with Severe Bony Defect in Orbit in a Newborn -
- 310 -
in the middle ear and mastoid bone: a case report. J Korean Med Sci 2004;19:155-8.
4) Gyure KA, Thompson LD, Morrison AL. A clinicopatholo- gical study of 15 patients with neuroglial heterotopias and encephaloceles of the middle ear and mastoid region. Laryn- goscope 2000;110:1731-5.
5) Dunham E, Armeni M. Glial choristoma of the temporal bone in a 7-month-old infant. JAMA Otolaryngol Head Neck Surg 2013;139:944-6.
6) McGregor DH, Cherian R, Kepes JJ, Kepes M. Case reports: heterotopic brain tissue of middle ear associated with chole- steatoma. Am J Med Sci 1994;308:180-3.
7) Mohanty S, Das K, Correa MA, D'Cruz AJ. Extranasal glial heterotopia: case report. Neurol India 2003;51:248-9.
8) Wolbach SB. Congenital Rhabdomyoma of the Heart. J Med Res 1907;16:495-520.
9) Shemanski KA, Voth SE, Patitucci LB, Ma Y, Popnikolov N, Katsetos CD, et al. Glial choristoma of the middle ear. Ear Nose Throat J 2013;92:555-7.
10) Wu L, Sun J, Zhang F. Glial heterotopia of the middle ear and Eustachian tube in children. Otolaryngol Head Neck Surg 2013;148:884-5.
11) Sun LS, Sun ZP, Ma XC, Li TJ. Glial choristoma in the oral and maxillofacial region: a clinicopathologic study of 6 cases. Arch Pathol Lab Med 2008;132:984-8.
12) Kallman JE, Loevner LA, Yousem DM, Chalian AA, Lanza DC, Jin L, et al. Heterotopic brain in the pterygopalatine fossa. AJNR Am J Neuroradiol 1997;18:176-9.
morphism)
.
, .2, 12 CT
, MRI , ,
CNS .
,
. ,
, ,
.
References
1) Heffner DK. Brain in the middle ear or nasal cavity: hetero- topia or encephalocele? Ann Diagn Pathol 2004;8:252-7.
2) Klein MV, Schwaighofer BW, Sobel DF, Fantozzi RD, Hes- selink JR. Heterotopic brain in the middle ear: CT findings. J Comput Assist Tomogr 1989;13:1058-60.
3) Lee JI, Kim KK, Park YK, Eah KY, Kim JR. Glial choristoma
= =
.
, .
, , .
10% .
, .
, , .
: , , , ,
: 2014 10 6, : 2014 10 16 : 2014 10 17 : , 156-755 102 : 02)6299-3181, : 02)6264-2167 E-mail : [email protected]
Copyright By The Korean Society of Perinatology
25 4, 2014 Korean J Perinatol Vol.25, No.4, Dec., 2014 http://dx.doi.org/10.14734/kjp.2014.25.4.307
: 40 1, 2,850
g,
(Fig. 1)
5 .
: 29 ,
.
.
.
4
(glial heterotopia)
(glial choristoma)
.1-3 (extracranial midline
structures) , , (nasopharynx)
(oropharynx) ,
(extracranical non-mildline structures)
.5, 6
, ,
1
1, 2, 3
1^1^1^2^3
Glial Choristoma Accompanied with Severe Bony Defect in Orbit in a Newborn
Suk Hwan Han, M.D.1, Tae Hee Kim1, Na Mi Lee, M.D.1, Jae Kyun Kim, M.D., Ph.D.2, and She Young Lee, M.D., Ph.D.3
1Departments of Pediatrics, 2Radiology, 3Otorhinolaryngology, Chung-Ang University Hospital, Chung-Ang University College of Medicine, Seoul, Korea
Glial choristoma is a mass-like lesion composed of otherwise normal, mature brain tissue that is isolated from the spinal cord and cranial cavity. In generally, this choristoma involves extracranial midline structures, especi- ally the nose, as well as the nasopharynx, oropharynx and cleft. Glial choristomas rarely involve extracranial non-midline structures such as the scalp, orbit, middle ear or mastoid and account for approximately 10% of all glial choristomas. Moreover, such glial choristoma presentations are usually seen in adults, most of whom have predisposing factors such as trauma, surgery, or chronic inflammation. Here, we present a case of a glial choristoma that invaded a neonate’s the nasopharynx, oropharynx, oral cavity and orbit.
Key Words : Brain, Choristoma, Heterotopia, Orbit, Newborn
- 308 -
×4 cm
.
.
(stridor) .
.
: (magnetic reso-
nance imaging, MRI) 3.1×2.4 cm
(Fig. 2A). 2.5×2.2 cm
(multiloculated cystic mass)
(masticator space) (buccal space)
(foramen lacerum)
(Fig. 2B).
(computed tomography, CT)
(Fig. 2C).
.
(debulking surgery) (excisional biopsy)
(glial tissue)
(choroid plexus) (Fig. 3A).
(glial fibrillary acidic protein,
GFAP) (Fig. 3B).
(cystic fluid)
. (normal cellu-
larity) (atypia) (pleo-
morphism) ,
A B Fig. 1. Gross photographs taken prior to surgery. A mass at the right buccal area (A) and a mass in the oral cavity with mouth was opened (B).
A B B C Fig. 2. The patient’s radiologic findings. Magnetic resonance imagine (MRI) showing a mixed cystic and solid lesion approximately 3.1x 2.4 cm with a bony defect in the right inferior orbital wall (A). MRI showing a multiloculated cystic mass approximately 2.5x2.2 cm with peripheral enhancement in the right masticator and buccal space as well as connected with cystic lesion in right foramen lacerum on MRI (B) and a computed tomography scan showing a bony defect in the right orbit floor (C).
: - -
- 309 -
.
(marsupialization)
.
, 2-3 , ,
.
(teratoma), (glioma),
(neurofibroma), (myofibroma), (en-
.
1907 Wolbach ,
, (spinal cord)
- .8
6
(intraparenchymal lesions, dural and leptomeningeal
lesions, intracranial extracerebral lesions, midline
lesions, distal lesions of the lung and uterus, extra-
cranial non-midline lesions) .3, 4, 9
, .3, 6
,
.4, 6, 9, 10.
.5 , (middle ear),
(maxillofacial), (temporal bone)
,1, 3, 11
.
, ,
.
.
, .6, 9
,
.
(hypercellularity), (cellular pleo-
Fig. 3. Patient’s pathologic findings. The mass is composed of mature glial tissue and choroid plexus-like area with papillary formation (A, Hematoxylin-and-eosin stain, x100). Both the glial and choroid plexus-like area showing a strong positive reaction for glial fibrillary acidic protein (B, x100).
Suk Hwan Han, et al. : - Glial Choristoma Accompanied with Severe Bony Defect in Orbit in a Newborn -
- 310 -
in the middle ear and mastoid bone: a case report. J Korean Med Sci 2004;19:155-8.
4) Gyure KA, Thompson LD, Morrison AL. A clinicopatholo- gical study of 15 patients with neuroglial heterotopias and encephaloceles of the middle ear and mastoid region. Laryn- goscope 2000;110:1731-5.
5) Dunham E, Armeni M. Glial choristoma of the temporal bone in a 7-month-old infant. JAMA Otolaryngol Head Neck Surg 2013;139:944-6.
6) McGregor DH, Cherian R, Kepes JJ, Kepes M. Case reports: heterotopic brain tissue of middle ear associated with chole- steatoma. Am J Med Sci 1994;308:180-3.
7) Mohanty S, Das K, Correa MA, D'Cruz AJ. Extranasal glial heterotopia: case report. Neurol India 2003;51:248-9.
8) Wolbach SB. Congenital Rhabdomyoma of the Heart. J Med Res 1907;16:495-520.
9) Shemanski KA, Voth SE, Patitucci LB, Ma Y, Popnikolov N, Katsetos CD, et al. Glial choristoma of the middle ear. Ear Nose Throat J 2013;92:555-7.
10) Wu L, Sun J, Zhang F. Glial heterotopia of the middle ear and Eustachian tube in children. Otolaryngol Head Neck Surg 2013;148:884-5.
11) Sun LS, Sun ZP, Ma XC, Li TJ. Glial choristoma in the oral and maxillofacial region: a clinicopathologic study of 6 cases. Arch Pathol Lab Med 2008;132:984-8.
12) Kallman JE, Loevner LA, Yousem DM, Chalian AA, Lanza DC, Jin L, et al. Heterotopic brain in the pterygopalatine fossa. AJNR Am J Neuroradiol 1997;18:176-9.
morphism)
.
, .2, 12 CT
, MRI , ,
CNS .
,
. ,
, ,
.
References
1) Heffner DK. Brain in the middle ear or nasal cavity: hetero- topia or encephalocele? Ann Diagn Pathol 2004;8:252-7.
2) Klein MV, Schwaighofer BW, Sobel DF, Fantozzi RD, Hes- selink JR. Heterotopic brain in the middle ear: CT findings. J Comput Assist Tomogr 1989;13:1058-60.
3) Lee JI, Kim KK, Park YK, Eah KY, Kim JR. Glial choristoma
= =
.
, .
, , .
10% .
, .
, , .
: , , , ,