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��ࡱ�>��	KM����HIJ�����������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������_����bjbj��bb��������VV�����4���������j$�5.����cT�,4444444$56��8�44����"���44��i�4�%�%�%�����,t�%�4�%�%(T(����`x茡������"� (�,�405((,�9]#<�9T(T(��9��(����%�����4444�$<���5�������������������������������������������������������������������������9���������V"x:LANGERHANS CELL HISTIOCYSTOSIS OF TEMPORAL BONE: CASE REPORTS

Mehmet Akif Aksoy1 , Armaan Incesulu 1, Ercan Kaya1 , M. Kezban G�rb�z1 , Hamdi Cakli 1 
1Eskisehir Osmangazi University Ear, Nose and Throat Department

INTRODUCTION
Langerhans cell histiocytosis (LCH) is a disease which is characterized by uncontrolled clonal proliferation of immature Langerhans cells and can cause multiple organ failure. ADDIN EN.CITE <EndNote><Cite><Author>Neilan</Author><Year>2012</Year><RecNum>22</RecNum><DisplayText>(1)</DisplayText><record><rec-number>22</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">22</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Neilan, Ryan E.</author><author>Kutz, Joe Walter Jr.</author></authors></contributors><titles><title>Langerhans Cell Histiocytosis of the Temporal Bone</title><secondary-title>Otology &amp; Neurotology</secondary-title></titles><periodical><full-title>Otology &amp; Neurotology</full-title></periodical><pages>e31-e32 10.1097/MAO.0b013e31823386d8</pages><volume>33</volume><number>4</number><keywords><keyword>Temporal bone mass-Langerhans cell histiocytosis</keyword><keyword>00129492-201206000-00042</keyword></keywords><dates><year>2012</year></dates><isbn>1531-7129</isbn><urls><related-urls><url>http://journals.lww.com/otology-neurotology/Fulltext/2012/06000/Langerhans_Cell_Histiocytosis_of_the_Temporal_Bone.42.aspx</url></related-urls></urls></record></Cite></EndNote>( HYPERLINK \l "_ENREF_1" \o "Neilan, 2012 #22" 1) Etiology of this disease still remains unclear and it can presents with various types. A comprehensive literature review reveals that the disease is first described by Liechtenstein in 1953 as Histiocytosis X. In the meeting of Histiocytosis Society in 1987, the term of  �Langerhans cell histiocytosis� had been used and replaced all previously used terms. ADDIN EN.CITE <EndNote><Cite><Author>Kleinjung</Author><Year>2003</Year><RecNum>19</RecNum><DisplayText>(2)</DisplayText><record><rec-number>19</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">19</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Kleinjung, Tobias</author><author>Woenckhaus, Matthias</author><author>Bachthaler, Maike</author><author>Wolff, Johannes E. A.</author><author>Wolf, Stephan R.</author></authors></contributors><titles><title>Langerhans� cell histiocytosis with bilateral temporal bone involvement</title><secondary-title>American Journal of Otolaryngology</secondary-title></titles><periodical><full-title>American Journal of Otolaryngology</full-title></periodical><pages>265-270</pages><volume>24</volume><number>4</number><dates><year>2003</year></dates><isbn>01960709</isbn><urls></urls><electronic-resource-num>10.1016/s0196-0709(03)00049-8</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_2" \o "Kleinjung, 2003 #19" 2)

The disease has 3 different forms including eosinophilic granuloma, Letterer-Siwe disease and Hand-Schuller-Christian disease. ADDIN EN.CITE <EndNote><Cite><Author>Yavas</Author><Year>2010</Year><RecNum>16</RecNum><DisplayText>(3)</DisplayText><record><rec-number>16</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">16</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Yavas, Ulas Savas</author><author>Incesulu, Armagan</author><author>Acikalin, Mustafa</author><author>Calisir, Cuneyt</author><author>Adapinar, Baki</author></authors></contributors><titles><title>Eosinophilic granuloma of the temporal bone with extensive bilateral otic capsule involvements: Incomplete reossification despite theraphy</title><secondary-title>International Journal of Pediatric Otorhinolaryngology Extra</secondary-title></titles><periodical><full-title>International Journal of Pediatric Otorhinolaryngology Extra</full-title></periodical><pages>74-78</pages><volume>5</volume><number>2</number><dates><year>2010</year></dates><isbn>18714048</isbn><urls></urls><electronic-resource-num>10.1016/j.pedex.2009.03.003</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_3" \o "Yavas, 2010 #16" 3) Although eosinophilic granuloma is characterized by regional bone involvement and has a better prognosis, Hand-Schuller-Christian disease is commonly seen under the age of 5 years and is characterized by multifocal osteolytic bone lesions, diabetes insipidus, exophthalmos and mucocutaneous skin lesions. On the other hand, Letterer-Siwe disease is associated with multiple organ involvement and is the form of the disease with a poor prognosis. ADDIN EN.CITE <EndNote><Cite><Author>Neilan</Author><Year>2012</Year><RecNum>22</RecNum><DisplayText>(1)</DisplayText><record><rec-number>22</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">22</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Neilan, Ryan E.</author><author>Kutz, Joe Walter Jr.</author></authors></contributors><titles><title>Langerhans Cell Histiocytosis of the Temporal Bone</title><secondary-title>Otology &amp; Neurotology</secondary-title></titles><periodical><full-title>Otology &amp; Neurotology</full-title></periodical><pages>e31-e32 10.1097/MAO.0b013e31823386d8</pages><volume>33</volume><number>4</number><keywords><keyword>Temporal bone mass-Langerhans cell histiocytosis</keyword><keyword>00129492-201206000-00042</keyword></keywords><dates><year>2012</year></dates><isbn>1531-7129</isbn><urls><related-urls><url>http://journals.lww.com/otology-neurotology/Fulltext/2012/06000/Langerhans_Cell_Histiocytosis_of_the_Temporal_Bone.42.aspx</url></related-urls></urls></record></Cite></EndNote>( HYPERLINK \l "_ENREF_1" \o "Neilan, 2012 #22" 1)

Because LCH can affect multipl diffuse sites, there is no classic clinical presentation. 55-80% of the patients show the symptom in the head and neck region. ADDIN EN.CITE <EndNote><Cite><Author>Neilan</Author><Year>2012</Year><RecNum>22</RecNum><DisplayText>(1)</DisplayText><record><rec-number>22</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">22</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Neilan, Ryan E.</author><author>Kutz, Joe Walter Jr.</author></authors></contributors><titles><title>Langerhans Cell Histiocytosis of the Temporal Bone</title><secondary-title>Otology &amp; Neurotology</secondary-title></titles><periodical><full-title>Otology &amp; Neurotology</full-title></periodical><pages>e31-e32 10.1097/MAO.0b013e31823386d8</pages><volume>33</volume><number>4</number><keywords><keyword>Temporal bone mass-Langerhans cell histiocytosis</keyword><keyword>00129492-201206000-00042</keyword></keywords><dates><year>2012</year></dates><isbn>1531-7129</isbn><urls><related-urls><url>http://journals.lww.com/otology-neurotology/Fulltext/2012/06000/Langerhans_Cell_Histiocytosis_of_the_Temporal_Bone.42.aspx</url></related-urls></urls></record></Cite></EndNote>( HYPERLINK \l "_ENREF_1" \o "Neilan, 2012 #22" 1) Involvement of the head and neck may be associated with skin lesions and lymphadenopathy, and more commonly, with   involvement of the cranial bones. Most commonly involved cranial bone is the temporal bone. ADDIN EN.CITE <EndNote><Cite><Author>de Brito Macedo Ferreira</Author><RecNum>23</RecNum><DisplayText>(4)</DisplayText><record><rec-number>23</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">23</key></foreign-keys><ref-type name="Book">6</ref-type><contributors><authors><author>de Brito Macedo Ferreira, Lidiane Maria</author><author>de Carvalho, Jo</author><author>atilde</author><author>o Deodato Di</author><author>oacute</author><author>genes</author><author>Pereira, S</author><author>eacute</author><author>rgio Tadeu Almeida</author><author>Tavares, Marylane Galv</author><author>atilde</author></authors></contributors><titles><title>Histiocytosis X of the temporal bone</title></titles><pages>575</pages><volume>72</volume><number>4</number><dates></dates><isbn>1808-8694</isbn><urls><related-urls><url>http://www.biomedsearch.com/nih/Histiocytosis-X-temporal-bone/17143443.html</url></related-urls></urls></record></Cite></EndNote>( HYPERLINK \l "_ENREF_4" \o "de Brito Macedo Ferreira,  #23" 4) 

Temporal bone involvement of LCH could be mistaken with otitis externa, otitis media, acute mastoiditis, chronic mastoiditis and other similar otological diseases, therefore differential diagnosis must be done carefully. ADDIN EN.CITE <EndNote><Cite><Author>Skoulakis</Author><Year>2008</Year><RecNum>24</RecNum><DisplayText>(5)</DisplayText><record><rec-number>24</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">24</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Skoulakis, C. E.</author><author>Drivas, E. I.</author><author>Papadakis, C. E.</author><author>Bizaki, A. J.</author><author>Stavroulaki, P.</author><author>Helidonis, E. S.</author></authors></contributors><auth-address>Departments of Otolaryngology, General Hospital of Volos, Volos, Greece.</auth-address><titles><title>Langerhans cell histiocytosis presented as bilateral otitis media and mastoiditis</title><secondary-title>Turk J Pediatr</secondary-title><alt-title>The Turkish journal of pediatrics</alt-title></titles><periodical><full-title>Turk J Pediatr</full-title><abbr-1>The Turkish journal of pediatrics</abbr-1></periodical><alt-periodical><full-title>Turk J Pediatr</full-title><abbr-1>The Turkish journal of pediatrics</abbr-1></alt-periodical><pages>70-3</pages><volume>50</volume><number>1</number><edition>2008/03/28</edition><keywords><keyword>Diagnosis, Differential</keyword><keyword>Fatal Outcome</keyword><keyword>Histiocytosis, Langerhans-Cell/*diagnosis/physiopathology/therapy</keyword><keyword>Humans</keyword><keyword>Infant</keyword><keyword>Male</keyword><keyword>Mastoiditis/*diagnosis</keyword><keyword>Rare Diseases</keyword><keyword>Tomography, X-Ray Computed</keyword></keywords><dates><year>2008</year><pub-dates><date>Jan-Feb</date></pub-dates></dates><isbn>0041-4301 (Print)&#xD;0041-4301</isbn><accession-num>18365596</accession-num><urls></urls><remote-database-provider>Nlm</remote-database-provider><language>eng</language></record></Cite></EndNote>( HYPERLINK \l "_ENREF_5" \o "Skoulakis, 2008 #24" 5) Temporal CT scan generally shows a mass that causes osteolitic lesions at temporal bone and biopsy reveals the diagnosis precisely. Treatment of LCH includes surgical excision, radiotherapy, chemotherapy, and systemic corticosteroids either alone or in different combinations.

In this article, we would like to present three patients with LCH. 


CASE REPORTS
The first patient was a 2-years old male who had admitted to the emergency service with a lump behind the left ear. The patient had a history of having that lump for about 1 month which had grown within a few days. Parents have reported no history of hearing loss or external ear discharge; however, the medical file of the patient had a bilaterally recorded otoacoustic emission test for the problem of articulating in our department 6 months ago. Physical examination revealed a reddish mass of about 1 cm in the left retroauricular region and the otological examination revealed a normal left external ear canal and intact-hyperemic tympanic membrane. Computerized tomography (CT) scan showed  a soft-tissue mass in the mastoid eroding sigmoid plate and extending into middle ear. The intracranial structures were in normal. Other physical examinations and blood tests revealed no abnormality.


FIG.  SEQ ^ekil \* ARABIC 1. Axial computed tomographic scan shows a soft-tissue mass in the mastoid eroding sigmoid plate and extending into middle ear

The patient underwent surgery with a preoperative diagnosis of a malignancy or a complication of chronic otitis media. Because a necrotic mass was found in the retroauricular region, intraoperative frozen biopsy was performed. The surgery was terminated after obtaining the biopsy result revealing histiocytic infiltration. Pathological result of the patient was recorded as Langerhans cell histiocytosis and he was referred to the Hematology department. Whole-body bone scan showed no other abnormality and the patient was diagnosed as having isolated temporal bone involvement. Chemotherapy was commenced with vinblastine and prednisone which were in use at the time of writing this paper. 

The second patient was a 5-years old male with a complaint of bilateral hearing loss and discharge for 1 year. On otologic examination, polyps were found in both external ear canals which were confirmed previously by biopsy and pathological examination. Other physical examinations and blood tests revealed no abnormality. The radiologist has interpreted the CT results of the patient that there may be a well-differentiated tumor extending into the inner ear, granulomatous infection or a congenital cholesteatoma. 

FIG.  SEQ ^ekil \* ARABIC 2. Axial computed tomographic scan reveals bilateral osteolitic defects without sclerotic margins filled with soft tissue masses involving the middle ear, mastoid, squama and petrous part of the temporal bone

The patient underwent to the surgery and right radical mastoidectomy was performed. Intraoperative macroscopic appearance and frozen biopsy did not suggest the cholesteatoma. Thus, the biopsy specimens were obtained from temporal bone and from the polyps in left external ear canal. The pathological results was reported as Langerhans cell histiocytosis and the patient found to have no multisystemic involvement. The patient who had bilateral temporal involvement was referred to the pediatric oncology and the chemotherapy was started with vinblastine and prednisolone. The patient cured with no complications during follow-ups. Hearing aid was recommended to the patient for bilateral hearing loss. 

The third patient was a 45-years old male with a complaint of hearing loss and discharge in the left ear for a six months. On otologic examination, purulent discharge was found in the left external ear canal. The timpanic membran was intakt but viewed irregular. Audiometric tests resulted mild conductive hearing loss in the left ear. Computerized tomography (CT) scan showed a soft tissue causing erosive changes the internal and external cortex and placed at back of the mastoid area and neighborhood of sigmoid sinus. The radiologist had interpreted as a possible granulomatous infection. 

FIG.  SEQ ^ekil \* ARABIC 3. Axial computed tomographic scan showed a soft tissue causing erosive changes in the internal and external cortex and placed at back of the mastoid area and neighborhood of sigmoid sinus

Whereupon we decided to surgery. During the operation, was encountered white-gray colored granulomatous lesion and lesion was totally excised. Pathological result of the patient was recorded as Langerhans cell histiocytosis and he was referred to the Hematology department. Whole-body bone scan showed no other abnormality and the patient was diagnosed as having local temporal bone involvement and follow-up treatment was recommended. After 1.5 years follow-up, control PET/BT scan showed increased FDG uptake at nasopharynx and bilateral tonsillar logy. Then tonsillectomy and nasopharynx biopsy was performed. Biopsy results reported as a chronic inflamation and the patient going to follow up.

FIG.  SEQ ^ekil \* ARABIC 4. No mastoid defect after treatment in case 3 on axial computed temporal bone tomographic scan is seen


DISCUSSION
LCH diagnosis is made with  langerhans cells which are normally found in the dermis when present outside in the dermis and clonal proliferation. Although multipl theories of origin exist including neoplastic, inflammatory, viral and genetic causes; etiology of this disease still remains unclear. ADDIN EN.CITE <EndNote><Cite><Author>Neilan</Author><Year>2012</Year><RecNum>22</RecNum><DisplayText>(1)</DisplayText><record><rec-number>22</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">22</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Neilan, Ryan E.</author><author>Kutz, Joe Walter Jr.</author></authors></contributors><titles><title>Langerhans Cell Histiocytosis of the Temporal Bone</title><secondary-title>Otology &amp; Neurotology</secondary-title></titles><periodical><full-title>Otology &amp; Neurotology</full-title></periodical><pages>e31-e32 10.1097/MAO.0b013e31823386d8</pages><volume>33</volume><number>4</number><keywords><keyword>Temporal bone mass-Langerhans cell histiocytosis</keyword><keyword>00129492-201206000-00042</keyword></keywords><dates><year>2012</year></dates><isbn>1531-7129</isbn><urls><related-urls><url>http://journals.lww.com/otology-neurotology/Fulltext/2012/06000/Langerhans_Cell_Histiocytosis_of_the_Temporal_Bone.42.aspx</url></related-urls></urls></record></Cite></EndNote>( HYPERLINK \l "_ENREF_1" \o "Neilan, 2012 #22" 1) In histopathologic examination characteristic Birbeck granules can be find and is supported by immunohistochemical detection of CD1a and S-100 protein positive. ADDIN EN.CITE <EndNote><Cite><Author>Kleinjung</Author><Year>2003</Year><RecNum>19</RecNum><DisplayText>(2)</DisplayText><record><rec-number>19</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">19</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Kleinjung, Tobias</author><author>Woenckhaus, Matthias</author><author>Bachthaler, Maike</author><author>Wolff, Johannes E. A.</author><author>Wolf, Stephan R.</author></authors></contributors><titles><title>Langerhans� cell histiocytosis with bilateral temporal bone involvement</title><secondary-title>American Journal of Otolaryngology</secondary-title></titles><periodical><full-title>American Journal of Otolaryngology</full-title></periodical><pages>265-270</pages><volume>24</volume><number>4</number><dates><year>2003</year></dates><isbn>01960709</isbn><urls></urls><electronic-resource-num>10.1016/s0196-0709(03)00049-8</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_2" \o "Kleinjung, 2003 #19" 2)

Langerhans cell histiocytosis is common in the male children at 1-4 years of age. ADDIN EN.CITE <EndNote><Cite><Author>Neilan</Author><Year>2012</Year><RecNum>22</RecNum><DisplayText>(1)</DisplayText><record><rec-number>22</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">22</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Neilan, Ryan E.</author><author>Kutz, Joe Walter Jr.</author></authors></contributors><titles><title>Langerhans Cell Histiocytosis of the Temporal Bone</title><secondary-title>Otology &amp; Neurotology</secondary-title></titles><periodical><full-title>Otology &amp; Neurotology</full-title></periodical><pages>e31-e32 10.1097/MAO.0b013e31823386d8</pages><volume>33</volume><number>4</number><keywords><keyword>Temporal bone mass-Langerhans cell histiocytosis</keyword><keyword>00129492-201206000-00042</keyword></keywords><dates><year>2012</year></dates><isbn>1531-7129</isbn><urls><related-urls><url>http://journals.lww.com/otology-neurotology/Fulltext/2012/06000/Langerhans_Cell_Histiocytosis_of_the_Temporal_Bone.42.aspx</url></related-urls></urls></record></Cite></EndNote>( HYPERLINK \l "_ENREF_1" \o "Neilan, 2012 #22" 1) Accordingly, the two patients presented here are both male children. When reviewing the literature this disease incidence is 0.5 to 5 cases per million per year but the occurrence of disease in adult individuals becomes more frequent. ADDIN EN.CITE  ADDIN EN.CITE.DATA ( HYPERLINK \l "_ENREF_6" \o "Arsovic, 2013 #21" 6)  Starting from this information our Case 3 can be considered extremely rare encountered case. 

Greater than two thirds of patients with LCH  have nondisseminated disease at presentation like our cases. ADDIN EN.CITE <EndNote><Cite><Author>Boston</Author><Year>2002</Year><RecNum>13</RecNum><DisplayText>(7)</DisplayText><record><rec-number>13</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">13</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Boston, Mark</author><author>Derkay, Craig S.</author></authors></contributors><titles><title>Langerhans&apos; cell histiocytosis of the temporal bone and skull base</title><secondary-title>American Journal of Otolaryngology</secondary-title></titles><periodical><full-title>American Journal of Otolaryngology</full-title></periodical><pages>246-248</pages><volume>23</volume><number>4</number><dates><year>2002</year></dates><isbn>01960709</isbn><urls></urls><electronic-resource-num>10.1053/ajot.2002.123452</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_7" \o "Boston, 2002 #13" 7) Howarth et al., in their series of 314 patients published in 1999, reported cranial bone involvement in 30% of the patients. The other most commonly involved bones include the femur, vertebrae and pelvis. Frontal, parietal and temporal bones are the most common site of involvement in the cranium. ADDIN EN.CITE <EndNote><Cite><Author>Bozdemir</Author><Year>2013</Year><RecNum>11</RecNum><DisplayText>(8)</DisplayText><record><rec-number>11</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">11</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Bozdemir, K.</author><author>Tarlak, B.</author><author>Cakar, H.</author><author>Doblan, A.</author><author>Kutluhan, A.</author><author>Dilek, I.</author><author>Adiyaman Sungu, N.</author></authors></contributors><auth-address>Ankara Ataturk Training and Research Hospital ENT, Department Bilkent, 06800 Ankara, Turkey.</auth-address><titles><title>Langerhans cell histiocytosis in bilateral mastoid cavity</title><secondary-title>Case Rep Otolaryngol</secondary-title><alt-title>Case reports in otolaryngology</alt-title></titles><periodical><full-title>Case Rep Otolaryngol</full-title><abbr-1>Case reports in otolaryngology</abbr-1></periodical><alt-periodical><full-title>Case Rep Otolaryngol</full-title><abbr-1>Case reports in otolaryngology</abbr-1></alt-periodical><pages>957926</pages><volume>2013</volume><dates><year>2013</year></dates><isbn>2090-6765 (Print)&#xD;2090-6773 (Linking)</isbn><accession-num>23841005</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/23841005</url></related-urls></urls><custom2>3691895</custom2><electronic-resource-num>10.1155/2013/957926</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_8" \o "Bozdemir, 2013 #11" 8)

Temporal bone is the most commonly involved site in the skull. In the literature, temporal bone involvement has been reported to be present in 14-61% of the patients. ADDIN EN.CITE <EndNote><Cite><Author>Bozdemir</Author><Year>2013</Year><RecNum>11</RecNum><DisplayText>(8)</DisplayText><record><rec-number>11</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">11</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Bozdemir, K.</author><author>Tarlak, B.</author><author>Cakar, H.</author><author>Doblan, A.</author><author>Kutluhan, A.</author><author>Dilek, I.</author><author>Adiyaman Sungu, N.</author></authors></contributors><auth-address>Ankara Ataturk Training and Research Hospital ENT, Department Bilkent, 06800 Ankara, Turkey.</auth-address><titles><title>Langerhans cell histiocytosis in bilateral mastoid cavity</title><secondary-title>Case Rep Otolaryngol</secondary-title><alt-title>Case reports in otolaryngology</alt-title></titles><periodical><full-title>Case Rep Otolaryngol</full-title><abbr-1>Case reports in otolaryngology</abbr-1></periodical><alt-periodical><full-title>Case Rep Otolaryngol</full-title><abbr-1>Case reports in otolaryngology</abbr-1></alt-periodical><pages>957926</pages><volume>2013</volume><dates><year>2013</year></dates><isbn>2090-6765 (Print)&#xD;2090-6773 (Linking)</isbn><accession-num>23841005</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/23841005</url></related-urls></urls><custom2>3691895</custom2><electronic-resource-num>10.1155/2013/957926</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_8" \o "Bozdemir, 2013 #11" 8) Temporal bone involvement is commonly associated with multisystem involvement. Moreover, the disease has been reported to manifest with otologic symptoms in only a few patients. ADDIN EN.CITE <EndNote><Cite><Author>Kleinjung</Author><Year>2003</Year><RecNum>19</RecNum><DisplayText>(2)</DisplayText><record><rec-number>19</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">19</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Kleinjung, Tobias</author><author>Woenckhaus, Matthias</author><author>Bachthaler, Maike</author><author>Wolff, Johannes E. A.</author><author>Wolf, Stephan R.</author></authors></contributors><titles><title>Langerhans� cell histiocytosis with bilateral temporal bone involvement</title><secondary-title>American Journal of Otolaryngology</secondary-title></titles><periodical><full-title>American Journal of Otolaryngology</full-title></periodical><pages>265-270</pages><volume>24</volume><number>4</number><dates><year>2003</year></dates><isbn>01960709</isbn><urls></urls><electronic-resource-num>10.1016/s0196-0709(03)00049-8</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_2" \o "Kleinjung, 2003 #19" 2)Therefore, the three patients presented here are extremely rare cases presenting with isolated temporal bone involvement and manifesting with otologic symptoms. In addition, case 2 had bilateral temporal bone involvement which has been reported to have an incidence of 30% in the literature. ADDIN EN.CITE <EndNote><Cite><Author>Skoulakis</Author><Year>2008</Year><RecNum>24</RecNum><DisplayText>(5)</DisplayText><record><rec-number>24</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">24</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Skoulakis, C. E.</author><author>Drivas, E. I.</author><author>Papadakis, C. E.</author><author>Bizaki, A. J.</author><author>Stavroulaki, P.</author><author>Helidonis, E. S.</author></authors></contributors><auth-address>Departments of Otolaryngology, General Hospital of Volos, Volos, Greece.</auth-address><titles><title>Langerhans cell histiocytosis presented as bilateral otitis media and mastoiditis</title><secondary-title>Turk J Pediatr</secondary-title><alt-title>The Turkish journal of pediatrics</alt-title></titles><periodical><full-title>Turk J Pediatr</full-title><abbr-1>The Turkish journal of pediatrics</abbr-1></periodical><alt-periodical><full-title>Turk J Pediatr</full-title><abbr-1>The Turkish journal of pediatrics</abbr-1></alt-periodical><pages>70-3</pages><volume>50</volume><number>1</number><edition>2008/03/28</edition><keywords><keyword>Diagnosis, Differential</keyword><keyword>Fatal Outcome</keyword><keyword>Histiocytosis, Langerhans-Cell/*diagnosis/physiopathology/therapy</keyword><keyword>Humans</keyword><keyword>Infant</keyword><keyword>Male</keyword><keyword>Mastoiditis/*diagnosis</keyword><keyword>Rare Diseases</keyword><keyword>Tomography, X-Ray Computed</keyword></keywords><dates><year>2008</year><pub-dates><date>Jan-Feb</date></pub-dates></dates><isbn>0041-4301 (Print)&#xD;0041-4301</isbn><accession-num>18365596</accession-num><urls></urls><remote-database-provider>Nlm</remote-database-provider><language>eng</language></record></Cite></EndNote>( HYPERLINK \l "_ENREF_5" \o "Skoulakis, 2008 #24" 5)

Otologic presentation of LCH includes a postauricular lump in 10-30% of the patients. ADDIN EN.CITE <EndNote><Cite><Author>Skoulakis</Author><Year>2008</Year><RecNum>24</RecNum><DisplayText>(5)</DisplayText><record><rec-number>24</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">24</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Skoulakis, C. E.</author><author>Drivas, E. I.</author><author>Papadakis, C. E.</author><author>Bizaki, A. J.</author><author>Stavroulaki, P.</author><author>Helidonis, E. S.</author></authors></contributors><auth-address>Departments of Otolaryngology, General Hospital of Volos, Volos, Greece.</auth-address><titles><title>Langerhans cell histiocytosis presented as bilateral otitis media and mastoiditis</title><secondary-title>Turk J Pediatr</secondary-title><alt-title>The Turkish journal of pediatrics</alt-title></titles><periodical><full-title>Turk J Pediatr</full-title><abbr-1>The Turkish journal of pediatrics</abbr-1></periodical><alt-periodical><full-title>Turk J Pediatr</full-title><abbr-1>The Turkish journal of pediatrics</abbr-1></alt-periodical><pages>70-3</pages><volume>50</volume><number>1</number><edition>2008/03/28</edition><keywords><keyword>Diagnosis, Differential</keyword><keyword>Fatal Outcome</keyword><keyword>Histiocytosis, Langerhans-Cell/*diagnosis/physiopathology/therapy</keyword><keyword>Humans</keyword><keyword>Infant</keyword><keyword>Male</keyword><keyword>Mastoiditis/*diagnosis</keyword><keyword>Rare Diseases</keyword><keyword>Tomography, X-Ray Computed</keyword></keywords><dates><year>2008</year><pub-dates><date>Jan-Feb</date></pub-dates></dates><isbn>0041-4301 (Print)&#xD;0041-4301</isbn><accession-num>18365596</accession-num><urls></urls><remote-database-provider>Nlm</remote-database-provider><language>eng</language></record></Cite></EndNote>( HYPERLINK \l "_ENREF_5" \o "Skoulakis, 2008 #24" 5) The case 1 had presented with a postauricular lump and an intact tympanic membrane which may be explained by the preferential involvement of the middle ear. ADDIN EN.CITE <EndNote><Cite><Author>Skoulakis</Author><Year>2008</Year><RecNum>24</RecNum><DisplayText>(5)</DisplayText><record><rec-number>24</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">24</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Skoulakis, C. E.</author><author>Drivas, E. I.</author><author>Papadakis, C. E.</author><author>Bizaki, A. J.</author><author>Stavroulaki, P.</author><author>Helidonis, E. S.</author></authors></contributors><auth-address>Departments of Otolaryngology, General Hospital of Volos, Volos, Greece.</auth-address><titles><title>Langerhans cell histiocytosis presented as bilateral otitis media and mastoiditis</title><secondary-title>Turk J Pediatr</secondary-title><alt-title>The Turkish journal of pediatrics</alt-title></titles><periodical><full-title>Turk J Pediatr</full-title><abbr-1>The Turkish journal of pediatrics</abbr-1></periodical><alt-periodical><full-title>Turk J Pediatr</full-title><abbr-1>The Turkish journal of pediatrics</abbr-1></alt-periodical><pages>70-3</pages><volume>50</volume><number>1</number><edition>2008/03/28</edition><keywords><keyword>Diagnosis, Differential</keyword><keyword>Fatal Outcome</keyword><keyword>Histiocytosis, Langerhans-Cell/*diagnosis/physiopathology/therapy</keyword><keyword>Humans</keyword><keyword>Infant</keyword><keyword>Male</keyword><keyword>Mastoiditis/*diagnosis</keyword><keyword>Rare Diseases</keyword><keyword>Tomography, X-Ray Computed</keyword></keywords><dates><year>2008</year><pub-dates><date>Jan-Feb</date></pub-dates></dates><isbn>0041-4301 (Print)&#xD;0041-4301</isbn><accession-num>18365596</accession-num><urls></urls><remote-database-provider>Nlm</remote-database-provider><language>eng</language></record></Cite></EndNote>( HYPERLINK \l "_ENREF_5" \o "Skoulakis, 2008 #24" 5)

The management of patients with LCH, especially in those with regional head and neck involvement, is controversial. Many clinicians address the need for invasive procedures.  ADDIN EN.CITE <EndNote><Cite><Author>de Brito Macedo Ferreira</Author><RecNum>23</RecNum><DisplayText>(4)</DisplayText><record><rec-number>23</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">23</key></foreign-keys><ref-type name="Book">6</ref-type><contributors><authors><author>de Brito Macedo Ferreira, Lidiane Maria</author><author>de Carvalho, Jo</author><author>atilde</author><author>o Deodato Di</author><author>oacute</author><author>genes</author><author>Pereira, S</author><author>eacute</author><author>rgio Tadeu Almeida</author><author>Tavares, Marylane Galv</author><author>atilde</author></authors></contributors><titles><title>Histiocytosis X of the temporal bone</title></titles><pages>575</pages><volume>72</volume><number>4</number><dates></dates><isbn>1808-8694</isbn><urls><related-urls><url>http://www.biomedsearch.com/nih/Histiocytosis-X-temporal-bone/17143443.html</url></related-urls></urls></record></Cite></EndNote>( HYPERLINK \l "_ENREF_4" \o "de Brito Macedo Ferreira,  #23" 4)Treatment options include surgical intervention, chemotherapy, radiotherapy and steroid administration. Our  Case1 was treated chemotherapy and steroids; and the disease was under control at the time of writing this paper. Case 2 was also treated with a similar regimen and has cured. Case 3 wasn�t receive additional treatment after surgical resection and has not recurrence after 2 years follow-up. 

In the literature, after complete resection of the lesion with clear margins, local recurrence rate is nearly %6 and new lesion presents in nearly %22 of patients. ADDIN EN.CITE <EndNote><Cite><Author>Binning</Author><Year>2008</Year><RecNum>15</RecNum><DisplayText>(9)</DisplayText><record><rec-number>15</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">15</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Binning, M. J.</author><author>Brockmeyer, D. L.</author></authors></contributors><auth-address>Department of Neurosurgery, University of Utah, Salt Lake City, Utah.</auth-address><titles><title>Novel multidisciplinary approach for treatment of langerhans cell histiocytosis of the skull base</title><secondary-title>Skull Base</secondary-title><alt-title>Skull base : official journal of North American Skull Base Society ... [et al.]</alt-title></titles><periodical><full-title>Skull Base</full-title><abbr-1>Skull base : official journal of North American Skull Base Society ... [et al.]</abbr-1></periodical><alt-periodical><full-title>Skull Base</full-title><abbr-1>Skull base : official journal of North American Skull Base Society ... [et al.]</abbr-1></alt-periodical><pages>53-8</pages><volume>18</volume><number>1</number><dates><year>2008</year><pub-dates><date>Jan</date></pub-dates></dates><isbn>1531-5010 (Print)&#xD;1531-5010 (Linking)</isbn><accession-num>18592019</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/18592019</url></related-urls></urls><custom2>2435467</custom2><electronic-resource-num>10.1055/s-2007-993048</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_9" \o "Binning, 2008 #15" 9) Thus  we performed nasopharynx biopsy and tonsillectomy to case 3 and no recurrence was detected from biopsy specimen. 

Although temporal bone involvement may mimic many otologic diseases, as seen in our cases, clinician should consider LCH in patients if the discharge from ear do not respond to medical treatment. In suspected cases, the computerized tomographic scan is the gold standard for imaging the temporal bone lesions. ADDIN EN.CITE  ADDIN EN.CITE.DATA ( HYPERLINK \l "_ENREF_10" \o "Yildirim-Baylan, 2012 #14" 10) Temporal CT scan can reveal an osteolytic lesion or a mass. However, it should be kept in mind that LCH has no specific radiological finding.  ADDIN EN.CITE <EndNote><Cite><Author>Bozdemir</Author><Year>2013</Year><RecNum>11</RecNum><DisplayText>(8)</DisplayText><record><rec-number>11</rec-number><foreign-keys><key app="EN" db-id="zvzxeasaz5vz97ewdawpeweywasre5wtrpsd">11</key><key app="ENWeb" db-id="">0</key></foreign-keys><ref-type name="Journal Article">17</ref-type><contributors><authors><author>Bozdemir, K.</author><author>Tarlak, B.</author><author>Cakar, H.</author><author>Doblan, A.</author><author>Kutluhan, A.</author><author>Dilek, I.</author><author>Adiyaman Sungu, N.</author></authors></contributors><auth-address>Ankara Ataturk Training and Research Hospital ENT, Department Bilkent, 06800 Ankara, Turkey.</auth-address><titles><title>Langerhans cell histiocytosis in bilateral mastoid cavity</title><secondary-title>Case Rep Otolaryngol</secondary-title><alt-title>Case reports in otolaryngology</alt-title></titles><periodical><full-title>Case Rep Otolaryngol</full-title><abbr-1>Case reports in otolaryngology</abbr-1></periodical><alt-periodical><full-title>Case Rep Otolaryngol</full-title><abbr-1>Case reports in otolaryngology</abbr-1></alt-periodical><pages>957926</pages><volume>2013</volume><dates><year>2013</year></dates><isbn>2090-6765 (Print)&#xD;2090-6773 (Linking)</isbn><accession-num>23841005</accession-num><urls><related-urls><url>http://www.ncbi.nlm.nih.gov/pubmed/23841005</url></related-urls></urls><custom2>3691895</custom2><electronic-resource-num>10.1155/2013/957926</electronic-resource-num></record></Cite></EndNote>( HYPERLINK \l "_ENREF_8" \o "Bozdemir, 2013 #11" 8)
 


REFERENCES	
 ADDIN EN.REFLIST 1.	Neilan RE, Kutz JWJ. Langerhans Cell Histiocytosis of the Temporal Bone. Otology & Neurotology 2012;33:e31-e2 10.1097/MAO.0b013e31823386d8.
2.	Kleinjung T, Woenckhaus M, Bachthaler Met al. Langerhans� cell histiocytosis with bilateral temporal bone involvement. American Journal of Otolaryngology 2003;24:265-70.
3.	Yavas US, Incesulu A, Acikalin Met al. Eosinophilic granuloma of the temporal bone with extensive bilateral otic capsule involvements: Incomplete reossification despite theraphy. International Journal of Pediatric Otorhinolaryngology Extra 2010;5:74-8.
4.	de Brito Macedo Ferreira LM, de Carvalho J, atildeet al. Histiocytosis X of the temporal bone:575.
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5.	Skoulakis CE, Drivas EI, Papadakis CEet al. Langerhans cell histiocytosis presented as bilateral otitis media and mastoiditis. The Turkish journal of pediatrics 2008;50:70-3.
6.	Arsovic N, Tomanovic N, Bukurov B. Sudden sensorineural hearing loss as first presenting symptom of unifocal Langerhans cell histiocytosis in the temporal bone. Otology & neurotology : official publication of the American Otological Society, American Neurotology Society [and] European Academy of Otology and Neurotology 2013;34:e24-5.
7.	Boston M, Derkay CS. Langerhans' cell histiocytosis of the temporal bone and skull base. American Journal of Otolaryngology 2002;23:246-8.
8.	Bozdemir K, Tarlak B, Cakar Het al. Langerhans cell histiocytosis in bilateral mastoid cavity. Case reports in otolaryngology 2013;2013:957926.
9.	Binning MJ, Brockmeyer DL. Novel multidisciplinary approach for treatment of langerhans cell histiocytosis of the skull base. Skull base : official journal of North American Skull Base Society ... [et al.] 2008;18:53-8.
10.	Yildirim-Baylan M, Cureoglu S, Paparella MM. Langerhans' cell histiocytosis of the temporal bone. Otology & neurotology : official publication of the American Otological Society, American Neurotology Society [and] European Academy of Otology and Neurotology 2012;33:e15-6.

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