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1 The Korean Journal of Gastrointestinal Endoscopy Case Report 림프종으로오인된회장말단부의살모넬라감염증 채명훈ㆍ김문영ㆍ이진형ㆍ권용환ㆍ박소연ㆍ조미연 * ㆍ정요셉 * ㆍ차승환 연세대학교원주의과대학내과학교실, * 병리학교실, 영상의학교실 A Case of Salmonella Infection in the Terminal Ileum That Was Initially Misdiagnosed as Lymphoma Myeong Hun Chae, M.D., Moon Young Kim, M.D., Jin Hyung Lee, M.D., Yong Hwan Kwon, M.D., So Yeon Park, M.D., Mee Yon Cho, M.D.*, Yo Sep Chong, M.D.* and Seung Whan Cha, M.D. Departments of Internal Medicine, *Pathology and Radiology, Yonsei University Wonju College of Medicine, Wonju, Korea Salmonella usually invades the Peyer s patch of the terminal ileum or ascending colon. A 55-year old female was referred to our hospital for general weakness and sustained fever. On the abdominal contrast computed tomography (CT) scan, we found symmetric circular hypertrophy of the intestinal wall and multiple hypertrophied lymph nodes in the terminal ileum. The positron emission tomography computed tomography (PET-CT) scan showed skipped areas of wall thickening and intense fluorodeoxyglucose (FDG) uptake in the terminal ileum and the ileocecal valve with adjacent lymphadenopathies. On the colonoscopy, multiple mass forming variable-sized ulcers on the terminal ileum were found, so a biopsy specimen and the colonic luminal fluid were obtained, and we made a diagnosis of lymphoma. However, the blood and colonic luminal fluid culture for Salmonella paratyphi-a was reported as positive, and therefore we corrected the diagnosis to Salmonella infection. We report here on a case of Salmonella infection in the terminal ileum, which looked like malignant lymphoma on the baseline radiologic image studies, including the CT and PET-CT. (Korean J Gastrointest Endosc 2009;39: ) Key Words: Salmonellosis, Lymphoma, Computed tomography, Positron emission tomography computed tomography, Colonoscopy 교신저자. 김문영연세대학교원주의과대학내과학교실 ( ), 강원원주시일산동 162 전화 : 팩스 : 이메일 : drkimmy@yonsei.ac.kr 접수 년 8 월 27 일승인 년 10 월 27 일 서론살모넬라감염증은사람과동물에서다양한임상양상으로나타나는데발열및설사를주증상으로하는장염증세가가장흔하다. 1,2 살모넬라 (Salmonella) 의 2,000개가넘는혈청형가운데오직 Salmonella typhi, Salmonella paratyphi 만이사람에게감염을일으킨다. 살모넬라균은감염된물이나음식의섭취를통해체내에들어오기때문에위장관어디에도침범할수있는데주로회장, 소장, 대장점막의 Peyer s patches를통해 1차감염후균혈증을통해폐, 간, 비장, 골수, 림프절등에서증식하면서 2차감염을일으킨다. 3,4 Peyer s patches의비대나괴사가현저해지면임상적으로복통과함께영상의학적검사에서회장말단부의부종이관찰되어급성충수염으로오인되기 도한다. 그러나다발성림프절비대를동반한종양양상의소견을보이는경우는매우드물다. 저자들은복부전산화단층촬영술및양전자방출단층촬영술에서회장말단부및회-결장장간막림프종으로의심되는소견을보였으나, 대장내시경검사를통한장관액배양검사및혈액배양검사를통해살모넬라감염증으로확인된 1예를경험하여문헌고찰과함께보고한다. 증례 55세여자가 1주일전부터전신쇠약과함께식욕부진이있어개인병원에서 5일간입원치료하였으나증세의호전이없고 3일전부터 39 o C의발열이있어항생제치료를하였으나발열이지속되어본원으로전원되었다. 과거력및가족력에서 384 The Korean Journal of Gastrointestinal Endoscopy

2 특이사항이없었고, 흡연력이나음주력도없었다. 내원당시활력징후는혈압 100/60 mmhg, 맥박수 90회 / 분, 호흡수 18회 / 분, 체온 36 o C이었으며의식은명료하였고급성병색은보이지않았다. 결막과공막은정상이었으며, 경부진찰에서만져지는림프절종대는없었다. 그외복부및다른신체검사의특이소견은없었다. 혈액검사에서백혈구 8,650/mm 3 ( 림프구 1,340/ mm 3 ), 혈색소 10.7 g/dl, 혈소판 141,000/mm 3 이었으며생화학검사는총단백질 5.8 g/dl, 알부민 2.7 g/dl, ALT 395 IU/L, AST 1134 IU/L, alkaline phosphatase 345 IU/L, 총빌리루빈 0.6 mg/dl, 직접빌리루빈 0.2 mg/dl, C-reactive protein mg/dl이었고, A, B, C, E형간염항원 / 항체검사는모두음성이었다. 심전도는정상이었고대변의잠혈반응은양성이었다. 내원당시시행한혈액배양검사에서분리된균은없었으며, Widal 응집반응검사는음성이었다. 내원직후 39 o C에이르는고열이지속되어우선응급으로복부전산화단층촬영술을시행하였다. 복부전산화단층촬영술에서비장의종대와함께회장말단부와회맹판의부종, 회-결장장간막을따라다발성의림프절종대가관찰되어회장말단부에서발생한림프종을의심하였다 (Fig. 1A, B). 내원사흘째선혈변을호소하여시행한식도위십이지장경검사및대장내시경검사에서위전정부에 1.5 cm 크기의치유기위궤양이발견되었고, 회장말단부와회맹판및상행결장의근위부에국한된다발성궤양에서활동성출혈이확인되어, 내시경적지혈치료를시행하였다 (Fig. 2). 또한림프종확진을위한조직검사를시행하였고, 지속적인고열이있는점과대장내시경적으로감염성장염을완전히배제할수없어장관액을채취하여균배양검사를추가로시행하였다. 이어림프종에대한검사로양전자방출단층촬영술을시행하였고, 그결과역시회장말단부와회맹판, 그주변의림프절에강력한 fluorodeoxyglucose (FDG) 섭취가관찰되어이역시복강내의림프종을시사하였다 (Fig. 1C, D). 그러나장관조직검사에서궤양과염증소견이외에는림프종을시사할만한특이소견이관찰되지않아, 확진을위해커져있는회-결장장간막의림프절에대한초음파유도흡인조직검사를시행하였다. 추적검사로시행한 Widal test는처음결과와마찬가지로모두음성이었다. 그러나이후보고된대장내시경검사중얻은장관액배양검사결과에서 Salmonella paratyphi-a가검출되었으며, 반복시행한혈액배양검사에서도 Salmonella paratyphi-a가검출된반면, 림프절에서시행한조직검사에서는림프종을의심할만한조직학적소견은관찰되지않았다 (Fig. 3). 이와같은결과를종합하여결국환자는 Salmonella paratyphi-a에의한살모넬라감염증으로확진되었고, 동정된 Salmonella paratyphi-a에대한감수성검사에따라 3세대 cephalosporin 계열의항생제 (Ceftriaxone 2 g/day) 치료를유지하면서격리조치하였다. 입원 10일째시행한혈액배양검사에서동 정되는균이없고생화학검사는 ALT 11 IU/L, AST 28 IU/L, alkaline phosphatase 96 IU/L, 총빌리루빈 0.5 mg/dl, 직접빌리루빈 0.1 mg/dl, C-reactive protein 0.17 mg/dl로감소하였으며발열및전신상태등이호전되어퇴원하였다. 고찰 살모넬라균은그람음성간균으로 2,000개가넘는혈청형이있다. 이가운데 typhi와 paratyphi 만이사람의몸에존재하고다른혈청형은동물들에정상적으로존재하기때문에주로이들동물을재료로만든음식을통하여또는조리자의오염된손에의해사람에게감염된다. 5 이처럼살모넬라감염증은오염된식품을통해서전파되는대표적인전신적감염증으로 Salmonella typhi나 Salmonella paratyphi에의해야기되는장티푸스 (typhoid fever), 파라티푸스 (paratypoid fever) 와다른살모넬라균에의한비장티푸스성살모넬라증 (non-typhoidal salmonellosis) 으로구분된다. 6 살모넬라감염증의임상적발현증상은위장관염, 장티프스, 균혈증, 국소감염증, 만성보균상태의다섯가지로나타나며, 7 균혈증상태에서는신체어느부위에나전이되어국소감염증을일으키거나농양을형성하여심내막염, 폐렴, 농흉, 중추신경계감염, 골수염, 관절염, 비뇨생식기감염등을유발할수있다. 이러한여러임상증상중장염은 1 2일의잠복기를거쳐갑작스런복통으로임상증세가시작된다. 설사는물과같은설사로양은일정치않으며, 구역및구토를유발할수있다. 이러한증상들은수일이내로회복되어설사는 2 3일내, 발열은 5 7일내로호전되며 14일이상지속되는경우는거의없다. 8 살모넬라감염의진단은대변, 혈액, 골수및국소감염부위에서살모넬라균을분리함으로써가능하다. 하지만대변, 혈액, 소변의배양검사의진단예민도가높지않고임상시기에따라양성도가다르며, 비교적예민도가높은골수배양검사는침습적이라는한계를갖고있다. 이에반해대장내시경검사를통한조직배양검사는상대적으로비침습적이고대변배양검사에비해많은병원체를포함하고있어, 다른진단방법보다유용하다는보고도있다. 2,9,10 본환자의경우에도복통이나설사와같은특징적인위장관감염증세는없었으나혈변과복부전산화단층촬영술에서보인회장말단부와회맹판의부종의확인을위해시행한대장내시경을통한감염된부위장관액의배양검사를통해서살모넬라감염증을진단할수있었다. 살모넬라감염증의대장내시경소견은점막출혈이나혈관의변화없이단지점막부종이나발적만을보이는경한경우와다량의점막출혈반을동반한부종및발적소견을보이는중등도의변화, 자연출혈, 접촉성출혈, 점막탈락등점막의취약성소견을보이는심한경우까지다양하며, 궤양성대장염의소견과유사할수도있다. 2,11-13 일반적으로살모넬라감염증을 Vol. 39, No. 6 December, 2009 ( ) 385

(arrow head) and multiple lymph nodes enlargement of the ileocolic mesentery

(B) It shows multiple lymph nodes enlargement of the aortocaval lymph node

fluorodeoxyglucose (FDG) uptake in the terminal ileum and ileocecal valve

enlargement (D) It shows intense FDG uptake in the the aortocaval lymph nodes

3 Figure 1. CT findings. (A) It shows heterogeneous enhanced wall thickening on the terminal ileum (arrow head) and multiple lymph nodes enlargement of the ileocolic mesentery (arrow). (B) It shows multiple lymph nodes enlargement of the aortocaval lymph node (arrow). (C) PET-CT finding also shows skipped wall thickening and intense fluorodeoxyglucose (FDG) uptake in the terminal ileum and ileocecal valve (arrow head) with adjacent mesentery and the aortocaval lymph nodes enlargement (arrow). (D) It shows intense FDG uptake in the the aortocaval lymph nodes enlargement (arrow). 진단하기위해대장내시경검사를시행하는것이추천되지는않으나, 급성대량출혈을동반한경우에는전기소작술같은치료목적으로시행할수있다. 2 본환자에서도선혈변이있어 대장내시경을시행한결과회장말단부와회맹판및상행결장근위부에국한된다발성궤양과회장말단부궤양에서활동성출혈이확인되어내시경적지혈치료를시행하였다. 386 The Korean Journal of Gastrointestinal Endoscopy

Figure 2. Colonoscopic findings. (A) It shows several edematous hyperemic mucosal patch on the ileocecal valve. (B) It shows a large deep ulcer with spontaneous bleeding in the terminal ileum.

The Ziehl-Neelsen stain failed to demonstrate acid-fast bacilli.

주름비후및궤양소견을보이고간혹소장천공에의한복막염소견이관찰되기도한다. 14 그러나본환자는복부전산화단층촬영술에서비장의종대와함께회장말단부와상행결장근위부의저음영의다소불균질한부종및비후와회-결장장간막을따라크기가최대 2.5 2.0 cm의장간막의다발성림프절종대가함께관찰되었다.

그러나본증례에서와같이복부전산화단층촬영술에서는림프절의종대나양전자방출단층촬영에서 FDG 섭취는림프종의해부학적위치와질환의진행정도를인지하는데도움이되나, 골반또는후복막의림프절상태는드물게는비뇨기계나장의염증상태등에따라영향을받을수도있기때문에이러한질환의감별진단에대해서도고려해야한다.

4 Figure 2. Colonoscopic findings. (A) It shows several edematous hyperemic mucosal patch on the ileocecal valve. (B) It shows a large deep ulcer with spontaneous bleeding in the terminal ileum. (C) It shows multiple variable sized ulcers with blood clots in the terminal ileum. Figure 3. Microscopic finding. There are several ill-defined small granulomas without necrosis (H&E, 400). The Ziehl-Neelsen stain failed to demonstrate acid-fast bacilli. 회장의 Peyer s patches에서증식한살모넬라균주는 Peyer s patches 혹은이외의림프조직안의대식세포나림프구와작용을하여더많은중성구와림프구를모으게되며이로인해 Peyer s patches와림프조직의부종과궤양을야기하며, 위장관중에는림프계가제일발달되어있는회장말단부에주로침범하여복부전산화단층촬영술등의영상학적검사에서회장말단의환형의균질한비후, 주름비후및궤양소견을보이고간혹소장천공에의한복막염소견이관찰되기도한다. 14 그러나본환자는복부전산화단층촬영술에서비장의종대와함께회장말단부와상행결장근위부의저음영의다소불균질한부종및비후와회-결장장간막을따라크기가최대 cm의장간막의다발성림프절종대가함께관찰되었다. 또한양전자방출단층촬영술에서도회장말단부및회-결장장간막의비연속적인부종, 주변부저음영의비후와함께인접한장간막 림프절과대동정맥주위림프절에강력한 FDG의섭취가관찰되어영상학적으로림프종을의심하게하였다. 그러나본증례에서와같이복부전산화단층촬영술에서는림프절의종대나양전자방출단층촬영에서 FDG 섭취는림프종의해부학적위치와질환의진행정도를인지하는데도움이되나, 골반또는후복막의림프절상태는드물게는비뇨기계나장의염증상태등에따라영향을받을수도있기때문에이러한질환의감별진단에대해서도고려해야한다. 15 본환자에서도영상학적검사에서는회장말단부에서발생한림프종과이로인한출혈을우선의심하여이의진단및지혈을위해시행한대장내시경검사에서추가적으로시행한장관액배양검사를통해살모넬라감염증으로확진할수있었다. 본증례에서보는바와같이살모넬라감염증에서림프종을의심할정도의크기로증가된다발성림프절이복부전산화단층촬영술과양전자방출단층촬영술에서나타나는경우는매우드물며이제까지국내외적으로보고된바가없다. 복강내의종양양상의병소에대한검사에있어서양전자방출단층촬영술을포함한영상학적검사는주변장기의염증상태등에의한영향을받아위양성을보일수있으므로, 가능한반드시조직학적, 혈청학적및배양검사등에의한추가적검사에의한감별진단이이루어져야한다. 요약저자들은최근발열과선혈변을주소로내원한환자의영상학적검사 ( 복부 CT 및 PET-CT) 에서회장말단부및회-결장장간막림프절종대를보여림프종으로판단하였으나, 회장점막조직검사및장간막림프절에대한조직검사에서단순한궤양과염증소견만을보였고, 대장내시경검사및혈액배양검사, 장관액배양검사를통해살모넬라감염증으로확인된 1예를경험하였다. 일반적으로살모넬라감염증은회장말단부에염증을일으켜주위림프절의부종과궤양을일으킬수있으나 Vol. 39, No. 6 December, 2009 ( ) 387

5 이와같이림프종으로오인될정도의다발성림프절종대와 PET-CT에서 fluorodeoxyglucose (FDG) 의섭취를보인이전의증례가없기에문헌고찰과함께보고한다. 색인단어 : 살모넬라감염증, 림프종, 복부전산화단층촬영술, 양전자방출단층촬영술, 대장내시경 참고문헌 1. Saphra I, Winter JW. Clinical manifestation of salmonellosis in man: an evluation of 7779 human infections identified at the New York Salmonella Center. N Engl J Med 1957;256: Ham JS, Ryu CB, Cheon GJ, et al. Clinical presentations of salmonella colitis on total colonoscopy. Korean J Gastrointest Endosc 2001;22: Kim TH, Myung SJ, Chang HS, et al. A case of salmonella paratyphi infection manifested as duodenal and terminal ileal ulcers. Korean J Gastrointest Endosc 2002;25: Darby J, Sheorey H. Searching for salmonella. Aust Fam Physician 2008;37: Oh MD. Food borne illness-salomonellosis, Virbriosis, Listeriosis. Korean J Intern Med 1998;55: Goldman L, Ausiello D. Cecil testbook of medicine. In: Keith SK, Donald K, eds. Salmonella infections (including typhoid fever). 23th ed. Philadelphia: Saunders, 2006: Cohen JI, Bartlett JA, Corey GR. Extra-intestinal manifestations of salmonella infections. Medicine (Baltimore) 1987;66: Saffouri B, Bartolomew RS, Fuchs B. Colonic involvement in salmonellosis. Dig Dis Sci 1979;24: Woo KE, Jung SA, Kang IS, et al. Value of colonoscopy and Salmonella-Shigella culture using biopsy specimens and intaluminal fluid obtained during colonoscopy in patients with acute diarrhea. Korea J Gastrointest Endosc 2002;24: Matsumoto T, Iida M, Kimura Y, Fujishima M. Culture of colonoscopically obtained biopsy specimens in acute infectious colitis. Gastrointest Endosc 1994;40: Kang JY. Endoscopic diagnosis of ileal ulceration in typhoid fever. Gastrointest Endosc 1988;34: Lee JH, Kim JJ, Jung JH, et al. Colonoscopic manifestations of typhoid fever with lower gastrointestinal bleeding. Dig Liver Dis 2004;36: Kume K, Kubo K, Murata I, et al. Endoscopic diagnosis in typhoid fever. Endoscopy 2000;32:31S. 14.Balthazar EJ, Charles HW, Megibow AJ. Salmonella- and Shigella-induced ileitis: CT findings in four patients. J Comput Assist Tomogr 1996;20: Tateishi U, Terauchi T, Inoue T, Tobinai K. Nodal status of malignant lymphoma in pelvic and retroperitoneal lymphatic pathways: PET/CT. Abdom Imaging 2009 Apr 16 [Epub ahead of print]. 388 The Korean Journal of Gastrointestinal Endoscopy

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Jkss hwp 급성복증을보이는감염성요막관기형 Infected Urachal Remnants with Symtoms of the Acute Abdomen: The Differential Diagnosis & Proper Management Jan Dy Lee, M.D., Chee Young Lim, M.D. 1, Hyoung Il Kim, M.D. 1, Chul Woon Chung,