성인에서 발생한 조롱박오목루를 동반한 방선균 갑상선염 1예
최현주·김보원·신민지·최보광·강지현·전윤경·김상수·김보현·김인주·김용기1
부산대학교 의학전문대학원 내과학교실, 김용기내과1
Received: 14 August 2011, Accepted: 25 October 2011 Corresponding author: Bo Hyun Kim
Division of Endocrinology and Metabolism, Department of Internal Medicine, Pusan National University Hospital, Pusan National University School of Medicine, 305 Gudeok-ro, Seo-gu, Busan 602-739, Korea
Tel: +82-51-240-7678, Fax: +82-51-254-3217, E-mail: pons71@hanmail.net
A Case of Actinomycotic Thyroiditis in an Adult with Piriform Sinus Fistula
Hyun Ju Choi, Bo Won Kim, Min Ji Shin, Bo Kwang Choi, Ji Hyun Kang, Yun Kyung Jeon, Sang Soo Kim, Bo Hyun Kim, In Ju Kim, Yong Ki Kim1
Department of Internal Medicine, Pusan National University Hospital, Pusan National University School of Medicine, Busan; Kim Yong Ki Internal Medicine Clinic1, Busan, Korea
Acute suppurative thyroiditis is an uncommon infectious thyroid disease affecting mainly children and young adults. The route of infection is frequently a pyriform sinus fistula. The major pathogens responsible for acute bacterial suppurative thyroiditis are the Streptococcus and Staphylococcus species. In contrast, Actinomyces species are a very rare cause of acute suppurative thyroiditis.
We experienced a case of a 23-year-old man who has presented general weakness and neck pain. Thyroid ultrasonography showed an ill-defined area of heterogeneous hypoechogenicity in the left lobe of the thyroid gland. Histologic examination by fine needle aspiration demonstrated gram-positive, filamentous-like organisms with branching hyphae and characteristic sulfur gran- ules. Barium esophagogram showed a linear barium-filled track at the left pyriform sinus. We report a case of actinomycotic thy- roiditis in a young adult with pyriform sinus fistula along with a brief review of related literature. (Endocrinol Metab 27:222-226, 2012) Key Words: Actinomycosis, Pyriform sinus fistula, Thyroiditis
서 론
세균성갑상선염은그빈도가낮은질환이며재발하는경우에서 조롱박오목루(pyriform siuns fistula)와관계있는것으로알려져있
다[1,2]. 조롱박오목루를동반하는경우에갑상선염은대부분좌엽
을침범하고임상진단으로는급성화농성갑상선염과재발성경부 농양으로나타난다. 조롱박오목루가동반된경우는대부분선천성 기형과관련되어있어그증상은대부분소아기에나타난다. 원인균 은주로 Staphylococcus aureus, Streptococcus pyogenes이며 Pepto- streptococcus species, Haemophilus influenzae, Salmonella species
등도 보고되어있으나방선균(actinomyces)에의한갑상선염은그
빈도가낮은것으로알려져있다[3,4].
방선균에의한감염은점막의손상이있을때주로일어나고외
상, 자궁내장치나천공, 수술등과관련이있는것으로알려져있 다. 세균배양이어렵고, 유황과립(sulfur granule)의발견도어렵기 때문에종양으로오인하여수술후절제된조직소견에서방선균증 (actinomycosis)로진단되는경우도적지않다[5,6].
저자들은경부통증및전신위약감을주소로내원한 23세남자 환자에서방선균갑상선염을경부갑상선초음파를통한세포흡인 검사를통해진단하였고수술적배농없이항생제정주로치료하여 문헌고찰과함께보고하는바이다.
증 례
환자: 23세, 남자 주소:경부통, 전신위약감
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Copyright © 2012 Korean Endocrine Society
발적을동반한부종이보였으며촉진시통증을호소하였다. 방사선학적 검사 소견:경부전산화단층촬영을시행하였고갑상선
우엽에석회화를동반한 8 mm의갑상선결절이관찰되었고갑상선
좌엽에 2 cm 크기의경계가불명확한종괴가관찰되었다. 왼쪽갑상
선종괴병변은외곽의조영이증가하는소견이며주위의연부조직 에서도조영증강과부종이관찰되었다(Fig. 1). 갑상선스캔에는불
균질한섭취소견을보였으나냉결절은관찰되지않았다(Fig. 2). 경
Fig. 1. Contrast-enhanced computed tomography scan of the neck demonstrat- ed a 2 cm sized rim enhancing lesion with thick wall and necrotic portion at left lobe of thyroid gland (arrow) and 10 mm sized nodule at right lobe of thyroid
gland (arrowhead). Fig. 2. 99mTc04 thyroid scan showed inhomogenous uptake, but there was no
definite cold nodule.
Anterior UPTAKE (%): 2.6
Fig. 3. (A) Initial thyroid ultrasonography shows about 2 × 2 cm sized ill-defined heterogenous low echoic mass in left thyroid gland. (B) Thyroid ultrasonography after administration of antibiotics for 4 weeks shows decreased mass size (0.6 × 0.8 cm).
A B
부갑상선초음파에서좌하엽에 2 × 2 cm 크기의내부에일부낭성
변성을동반하는불균등한저에코의농양소견을보여(Fig. 3A) 갑
상선세침흡인을시행하였고흡인된물질은탁한분홍색을띠고있 었다(Fig. 4). 흡인된농양은배양과함께시행한 hematoxylin and
eosin 염색에서 유황과립을보여 방선균 갑상선염(actinomycotic
thyroiditis)으로진단되었다(Fig. 5).
치료 및 경과: Penicillin G 정맥투여를시작하였고내원 10일째시 행한바륨식도조영술에서좌측조롱박오목루가확인되었다(Fig. 6).
이후항생제치료로증상호전되어다른침습적치료없이경구 amox-
icillin으로전환할수있었다. 퇴원 2주후외래에서추적갑상선초
음파를시행하였고추적관찰초음파에서병변의크기는감소하였
다(Fig. 3B). 환자는향후 6개월간항생제치료를유지하고 동반된
조롱박오목루에대해서는화학소작술또는수술적치료예정이다.
고 찰
급성갑상선염과갑상선농양은드문질환이다. 이는풍부한혈류 량, 임파선배액, 높은요오드농도등이세균의성장을억제하고갑 상선피막이감염에대한저항성을높이기때문이다. 주된원인균은 Staphylococcus aureus, Streptococcus pyogenes이며 Peptostrepto- coccus species, H. influenzae, Salmonella species도보고되고있 다. 갑상선은구조적으로세균감염에대한내성이큰기관으로충 분한내과적, 외과적인치료에도불구하고재발하는좌측경부농 양또는급성화농성갑상선염, 후인두부농양의경우조롱박오목루 Fig. 5. Histopathology demonstrated gram-positive, branching, thin, filamen-
tous-like organisms suggestive of actinomyces, and the characteristic sulfur granules (H&E stain, × 400).
Fig. 6. Barium esophagogram shows linear barium filled track at left pyriform sinus fistula (arrow).
Fig. 4. Fine needle aspiration shows yellowish pink color pus like material.
이힘들수있어적절한항생제투여후시행을고려해야하고이경 우바륨식도조영술이후에전산화촬영을시행하거나변형된발살 바조작(modified Valsalva maneuver) 후경부전산화촬영을시행하 면누관을확인할수있다[10,11].
방선균은구강, 대장, 질등에상재하는정상균무리로점막손상 이발생하면통증이없고만성으로진행하는감염을일으키며유황
과립과동굴길배농(sinus tract)이특징적이다. 방선균증은드문감
염증으로그빈도는인구 30만명중 1명꼴로보고되고있다. 남성 의경우여성보다유병률이 3배정도높고감염증의발생위치를보 면두경부, 흉부, 복부, 골반, 중추신경계를침범하며이중두경부
형(50%)의빈도가가장높게보고되어있다. 초기급성감염시통
증과급성염증을동반할수있으며이는두경부질환이나연부조 직의감염증에서흔히관찰된다[12]. 하지만세균배양이어렵고, 유 황과립의발견도어렵기때문에종양으로오인하여수술후절제된 조직소견에서방선균증으로진단되는경우도적지않다. 이전보고 된갑상선방선균증례에있어서도종양으로오인되어갑상선절제
술등의외과적절제가시행된바있다[13]. 두경부형방선균증에있
어세포흡인검사를통한균주배양의양성률은낮은것으로보고되
어있는데이는다른세균의증식, 이전항생제치료에기인한다[14].
치료는페니실린을 2주에서 6주가량정주하고이후경구약제로 전환하여총치료기간을 6개월에서 12개월까지유지하여야한다. Penicillin allergy가있는경우에는 Tetracycline, Macrolide 등으로 대체할수있다. 그리고다른동반감염이있는경우에는다른약제 를추가하여야한다. 하지만대부분의경우에있어서다른동반감 염균에대한치료없이방선균증을성공적으로치료할수있다. 외 과적절제와배농을내과적치료와함께시행하면치료기간을 3개 월에서 6개월까지단축시킬수있다는보고가있지만항생제단독 치료만으로도완치된경우의보고가늘면서초치료는페니실린정 주를시행하고이후항생제에반응이없는경우외과적치료를고려 해볼수있다[15].
조롱박오목루는외과적절제가이상적인치료이나반복된감염이 나이전수술로인해누관전체의절제가힘든경우가많다. 수술후
에서조롱박오목루를확인할수있었다. 재발하는경부감염또는 원인불명의경부감염이있을때특히갑상선내에감염이동반될 경우에는조롱박오목루가능성을고려해야한다. 그리고일반적으 로알려져있는균이외에도급성화농성갑상선염에서방선균이원 인일수있음을고려해야한다.
요 약
세균성갑상선염은빈도가 낮은질환으로 조롱박오목루와같은 동반기형확인이필요하다. 방선균갑상선염의경우이전보고에서 종양으로오인되어수술적치료후진단되는경우가있었으나본증 례는세포흡인검사를통해 방선균갑상선염을 진단하였고항생제 치료만으로도성공적인치료를하였기에보고하는바이다.
참고문헌
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Rosello M: Fine-needle aspiration cytology in the diagnosis of cervicofacial actinomycosis: report of 15 cases. Med Oral Patol Oral Cir Bucal 9:467- 470; 464-467, 2004
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