DOI: 10.4046/trd.2009.67.3.229 ISSN: 1738-3536(Print)/2005-6184(Online) Tuberc Respir Dis 2009;67:229-233 CopyrightC2009. The Korean Academy of Tuberculosis and Respiratory Diseases. All rights reserved. 수차례재발한스파르가눔증으로치료를받았던환자에서발생한폐스파르가눔증 1 예 분당제생병원 1 내과, 2 흉부외과, 3 병리과오윤정 1, 김미진 1, 조준형 1, 차치운 1, 김도훈 1, 오미정 1, 진재용 1, 최성실 2, 권계원 3 Case Report A Case of Pulmonary Sparganosis in a Patient with a History of Recurrent Sparganum Infections Yun-Jung Oh, M.D. 1, Mi-Jin Kim, M.D. 1, Jun-Hyung Cho, M.D. 1, Chi-Woon Cha, M.D. 1, Do-Hoon Kim, M.D. 1, Mi-Jung Oh, M.D. 1, Jae-Yong Chin, M.D. 1, Sung-Sil Choi, M.D. 2, Kye-Won Kwon, M.D. 3 Departments of 1 Internal Medicine, 2 Thoracic Surgery, 3 Pathology, Bundang Jesaeng General Hospital, Daejin Medical Center, Seongnam, Korea Sparganosis is a zoonosis caused by the migrating larvae of cestode genus Spirometra. We report a rare form of sparganosis that invades the lung. A 44-year-old man presented with newly appeared pulmonary nodules that were found accidentally on regular medical checkup, and on sequential chest CT, which we checked at an interval of every 2 months, revealed that the pulmonary lesion had migrated. The patient had a medical history of having undergone surgical excisions for sparganosis in muscles and in subcutaneous tissues of the lower abdomen, perianus, thigh, right axilla, and scapula area, several times over 7 years. A right middle lobectomy was performed and the lesion was diagnosed as sparganosis based on the characteristic histological findings. Key Words: Lung, Sparganosis, Recurrence 서 스파르가눔 (sparganum) 은의엽조충류 (pseudophyllidean tapewarm) 의일종인열두조충류 (Spirometra spp.) 의충미충 (plerocercoid) 을말한다 1. 스파르가눔증 (sparganosis) 은이유충이인체에침입하여발생하는질환으로, 전세계적으로광범위하게분포하고있으나, 그중상당수의인체감염의예가한국, 일본, 중국을비롯한동남아시아에서보고되었다 2. 기생부위는피하조직이가장많고, 주로복벽, 대퇴부, 음낭, 하지등을침범하며드물게는안와, 뇌, 척수등을침범하는경우도있다 3-6. 흉강 Address for correspondence: Do-Hoon Kim, M.D. Department of Internal Medicine, Bundang Jesaeng General Hospital, 255-2, Seohyun-dong, Bundang-gu, Seongnam 463-774, Korea Phone: 82-31-779-0387, Fax: 82-31-779-0897 E-mail: dhkim@dmc.or.kr Received: Jul. 2, 2009 Accepted: Aug. 11, 2009 론 도드문발생부위중하나로, 폐스파르가눔증의예는문헌상태국에서 1예, 일본에서 1예, 국내에서도 1예만이보고되었을뿐이다 7-9. 저자들은임상증상없이흉부방사선사진상보이는이동성종괴로검사중이던환자에서폐스파르가눔증을진단한 1예를경험하였기에문헌고찰과함께보고하는바이다. 증례환자 : 이, 남자, 44세주소 : 흉부 X-선사진상보이는 1 cm 크기의결절현병력 : 환자는내원 1개월전직장에서건강검진의목적으로시행한흉부 X-선사진에서 1 cm 크기의결절이발견되어본원호흡기내과외래를방문하였다. 과거력 : 환자는내원 7년전우측가슴근에발생한스파르가눔증으로수술적절제술을시행받은후, 복부와항문 229
YJ Oh et al: Pulmonary sparganosis 주위, 좌 우대퇴부, 우측견갑부의근육과피하조직에지속적으로스파르가눔증이재발하여총 6차례본원외과에서수술적절제술을시행받았다 (Table 1). 그외호흡기질환의기왕력은없었다. 가족력 : 특이사항없었다. 사회력 : 20년전군복무시특수전요원으로근무하면서뱀, 개구리등을생식한기왕력이있는환자로서, 자영업자이며 35갑년의흡연자였다. 신체검사소견 : 입원당시활력징후는혈압이 130/80 mmhg, 맥박수는 72회 / 분, 호흡수는 20회 / 분, 체온은 36.1 o C였으며, 의식은명료하였고급성병색소견은없었다. 흉부청진및심음은정상이었고, 항문주위에작은종괴와누공이관찰되었으며, 이외촉지되는종괴나임파절은없었다. 검사실소견 : 말초혈액검사상백혈구 10,000/mm 3 ( 호중구 46.3%, 림프구 45.2%, 호산구 5.6%) 로호산구증가소견은없었고, 혈색소 15.7 g/dl, 혈소판 240,000/mm 3 였으며, 혈청총면역글로불린 E (IgE) 는 732.80 IU/mL로증가되어있었다. 생화학검사상 aspartate aminotransferase (AST) 29 IU/L, alanine aminotransferase (ALT) 27 IU/L, 알칼리인산분해효소 (alkaline phosphatase) 184 IU/L, 총빌리루빈 (total biliruin) 0.54 mg/dl였으며, 기생충특이항체에대한효소면역측정법 (enzyme-linked immunosorbent assay, ELISA) 상항폐흡충, 간흡충 IgG 항체에대해서는모두음성이었고, 항스파르가눔 IgG 항체는양성이었다. 대변검사상기생충이나충란은발견되지 않았다. 방사선학적소견 : 호흡기내과외래내원당시시행한흉부 CT에서는우중엽에각각, 1 cm 크기내외의 2개의소엽화된경화성병변들이종격동에연하여관찰되었고병변주위로는비교적넓은젖빛유리음영 (ground glass opacity) 의훈륜 (halo) 을동반하고있었다. 2개월후추적검사한 CT에서는이전에보였던병변들은호전되어보이지않았고, 위치가이동하여새로운부위에가늘고긴모양의경화성병변이새로이발견되었다 (Figure 1). 조직병리학적소견 : 우중엽절제술로얻어진조직은육안적으로검체의중앙에는 2.5 1.5 1 cm 크기의경계가명확하지않은노랗고하얀빛을띤괴사가관찰되었다. 현미경적소견으로는중심부의괴사와소수의흩뿌려진석회소구 (calcospherules) 를동반한만성육아종성염증이관찰되었으며, 이는기생충감염, 특히스파르가눔증에합당한소견이었다 (Figure 2). 경과 : 환자는본원에서 1차흉부 CT 시행후타병원진료를원하여전원하였으나, 이후타병원진료를받지않고지내다가 2개월후본원호흡기내과외래를재차방문하였고추적흉부 CT를촬영하였다. 저자들은방사선학적으로보이는이동성병변과스파르가눔증이수차례재발하였던환자의과거력을바탕으로폐스파르가눔증을의심하였으며, 이에대한치료의목적으로시험적개흉술을시행하여우중엽절제술을시행하였고, 항문주위의작은종괴와누공에대해서외과와협진하여누공절제술을함께시행하였다. 수술적절제로얻은폐조직에서스 Table 1. Summary of past medical history Clinical symptoms Sites involved Diagnostic findings Treatment First attack Palpable mass Right pectoralis muscle and One dead sparganum Surgical excision (July 2002) adjacent subcutaneous tissue Second attack Multiple painful and Subcutaneous tissues of Pathological findings, Surgical excision (August 2003) palpable masses right lower abdomen, consistent with perianus, left thigh sparganosis Third attack Painful palpable mass Subcutaneous tissue of Necrotized sparganum Surgical excision (January 2005) perianus area encapsulated with fibrous tissue Forth attack Painful swelling and Subcutaneous tissue of One alive sparganum Surgical excision (January 2008) palpable mass right thigh, anus Fifth attack Painful palpable mass Muscle and subcutaneous Pathological findings, Surgical excision (November 2008) tissue of left thigh consistent with sparganosis Sixth attack Palpable mass Subcutaneous tissue of One alive sparganum Surgical excision (January 2009) right scapular area 230
Tuberculosis and Respiratory Diseases Vol. 67. No. 3, Sep. 2009 Figure 1. Initial chest CT scan shows about 1 cm sized two lobulated consolidative lesions with peripheral ground glass opacity and halo on right middle lobe that is adjacent to mediastium (A, B). A follow-up chest CT scan reveals newly appeared elongated consolidations on a different site of right middle lobe with disappearance of previous lesions compared with initial scan, suggesting the lesions have a fleeting nature (C, D). Figure 2. (A) An elongated necrotic material surrounded by palisading granulomatous reaction is noted (H&E stain, 40). (B) A few lamellated calcospherules are found in the necrotic material (H&E stain, 400). 231
YJ Oh et al: Pulmonary sparganosis 파르가눔증의특징적인조직병리학적소견을확인함으로써폐스파르가눔증을진단하였으며, 환자는수술후별다른합병증없이퇴원하였다. 이후환자는 4개월째재발의징후없이본원외과외래에서추적관찰중이다. 고찰열두조충류의충미충인스파르가눔은리본형으로가늘고길며, 유백색의납작한형태와위결절을가진기생충으로길이는 3 4 cm에서 60 80 cm까지다양하며개나고양이를종숙주로한다. 스파르가눔의일반적인생활사를보면, 먼저종숙주에서대변과함께충란이배출되면수중에서부화하여육구유충 (coracidium) 이되고이는제 1 중간숙주인물벼룩 (cyclops) 에섭취된후원미충유충 (procercoid larva) 으로성장하게된다. 원미충유충에감염된물벼룩이제 2 중간숙주인개구리, 뱀등에섭취되면충미충유충 ( 스파르가눔 ) 으로성장하게되며종숙주인개, 고양이등에침입하여성충으로성장한다. 사람은주로중간숙주의역할을하는데, 충미충유충이섭취되면장내에서성충으로발육하지못하고장벽을뚫고조직으로이행함으로써인체스파르가눔증이발생한다 10. 인체로의감염경로는첫째, 원미충에감염된물벼룩으로오염된물을마시거나, 둘째, 충미충유충 ( 스파르가눔 ) 에감염된제 2 중간숙주인개구리나뱀등을생식하였을때이며, 그밖에조리사의손혹은식기등에부착된충미충에의해우연히감염될수도있다 2,3. 본증례의경우는개구리, 뱀등을생식함으로써충미충유충에감염된것으로생각된다. 기생부위는피하조직이가장많고그중복부, 하지, 음낭등이흔하며, 안와, 뇌, 척수, 폐등의장기에드물게발생하기도한다 3-9,11. 임상적으로대부분서서히자라고, 압통이있고이행하는피하결절을보이며, 감염부위에이물감, 가려움, 염증소견을보이고, 장천공을일으켜복막염을일으키기도하고, 뇌및신경계를침범하는경우간질, 발작, 하반신마비등을일으키기도한다 2,3. 스파르가눔증은비증식형 (nonproliferative) 과증식형 (proliferative) 으로나누는데, 비증식형은천천히자라고번식을하지못하는반면증식형은분지 (branching) 와분아 (budding) 에의해성장할수있고증식이가능하여한피막내에여러개의충체가있으며골조직을제외한전장기를침범할수있어치사율이높지만매우드문것이특징이다 12. 국내및국외에서보고되는증례의대부분이 비증식형으로, 비증식형스파르가눔증에서폐를침범한경우는매우드물다. Hur 등 9 이보고한국내폐스파르가눔증의증례에서는환자가호흡기적임상증상을동반하였던것에비해, 본증례의환자는무증상인상태에서흉부방사선사진의이동성종괴로인해진단된경우이다. 또한대부분의국내및국외증례의경우각장기에서단발성으로발생하였던스파르가눔증에대한보고는많았으나, 질환발생자의장기추적결과에대한보고는없고, 국내에서드물게동일환자에서세차례재발한다수의스파르가눔충체가발견되었던비전형적인스파르가눔증에대한증례보고가있었을뿐이다 11. 본증례의환자는 7년에걸쳐총 6차례의스파르가눔증이하복부, 대퇴부, 겨드랑이와견갑부등의피하조직및근육에서발생하여수차례의외과적절제술을받은후폐에서질환이재발하였다. 스파르가눔증의진단은환자의임상증상과개구리나뱀등을생식한경험이있으면의심해볼수있고, 확실한진단은충체를외과적으로적출한후에야가능하다. 충체를외과적으로적출하지못하더라도적출된조직내에서변형된충체를발견하거나특이한조직반응이관찰되는병리소견이있으면진단이가능하다. 스파르가눔증은병리조직학적으로충체가빠져나간후에생긴불규칙한모양의공동과괴사성및육아종성염증반응이특징적이다 3,13. 그리고일반적인염증성병변과다르게이러한공동들은대개비어있고공동의내면은유충의이동에따른톱니모양의내연을형성하여특징적인 zonal phenomenon을나타낸다 3,12. 또한기생기간이오래경과할수록호산구, 상피세포및림프구등의세포침윤을유발하고만성감염으로충체가죽으면병소는석회화된다 3,13. 본증례의경우스파르가눔증을의심할수있었던병리조직학적증거로중심부의괴사와석회소구 (calcospherules) 를동반한만성육아종성염증소견이있었다. 그외병리소견만으로감별이어려운경우에는혈청학적진단법이사용될수있는데, 그중에서 ELISA 가특이 IgG 항체가의상승을나타내므로진단및수술후추적검사로사용될수있다 14. 치료는외과적절제술이가장확실한방법이고, praziquantel이나 mebendazole 의경구투여및환부에 procaine과 40% ethylalcohol 을주입하고충체의사멸과흡수를기다리는방법이있다 3. 하지만다수감염시에는외과적치료가어려울수있으며, praziquantel 투여의경우두절및경부일부가약제에저항하여후에반드시재발한다는것이알려져있다 15. 232
Tuberculosis and Respiratory Diseases Vol. 67. No. 3, Sep. 2009 본저자들은수차례외과적절제술을받았음에도지속적으로스파르가눔증이재발한환자에서발생한폐스파르가눔증을경험하였기에이를보고한다. 참고문헌 1. Mueller JF. The biology of Spirometra. J Parasitol 1974; 60:3-14. 2. Cho SY, Bae JH, Seo BS. Some aspects of human sparganosis in Korea. Korean J Parasitol 1975;13:60-77. 3. Lee BJ, Ahn SK, Kim SC, Lee SH. Clinical and histopathologic study of Sparganosis. Korean J Dermatol 1992;30:168-174. 4. Mougeot G, Cambon M, Menerath JM, Dimeglio V, Houin R. Human eye anterior chamber sparganosis. Parasite 1999;6:365-7. 5. Jeong SC, Bae JC, Hwang SH, Kim HC, Lee BC. Cerebral sparganosis with intracerebral hemorrhage: a case report. Neurology 1998;50:503-6. 6. Lo YK, Chao D, Yan SH, Liu HC, Chu FL, Huang CI, et al. Spinal cord proliferative sparganosis in Taiwan: a case report. Neurosurgery 1987;21:235-8. 7. Phunmanee A, Boonsawat W, Indharapoka B, Tuntisirin C, Kularbkeaw J. Pulmonary sparganosis: a case report with five years follow-up. J Med Assoc Thai 2001; 84:130-5. 8. Iwatani K, Kubota I, Hirotsu Y, Wakimoto J, Yoshioka M, Mori T, et al. Sparganum mansoni parasitic infection in the lung showing a nodule. Pathol Int 2006;56:674-7. 9. Hur J, Lee SC, Lee CY. Pulmonary sparganosis: a case report. Korean J Thorac Cardiovasc Surg 2003;36:43-6. 10. Beaver DC, Jung RC, Cupp EW. Clinical parasitology. 9th ed. Philadephia: Lea & Febriger; 1984. 11. Cho BK, Chun CS, Choi WY, Cho SY. A case of unusual sparganosis. Korean J Dermatol 1985;23:672-7. 12. Brown HW, Neva FA. Basic clinical parasitology. 5th ed. Norwalk: Appleton-Century-Crofts; 1983. 13. Chi JG, Chi HS, Lee SH. Histopathologic study on human sparganosis. Korean J Parasitol 1980;18:15-23. 14. Wang KC, Huh S, Hong ST, Chai JY, Choi KS, Lee SH. The fate of spargana inoculated into the cat brain and sequential changes of anti-sparganum IgG antibody levels in the cerebrospinal fluid. Korean J Parasitol 1990;28:1-10. 15. Chai JY, Yu JR, Lee SH, Kim SI, Cho SY. Ineffectiveness of praziquantel treatment for human sparganosis (a case report). Seoul J Med 1988;29:397-9. 233