Disseminated Septic Lesions Caused by Mimicking Cysticercosis Detected on Ultrasonography in a Pediatric Patient with Acute Myeloid Leukemia

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ABSTRACT

Staphylococcus epidermidis is a part of the normal skin flora of humans. However, it can cause serious infections in people exposed to foreign bodies or in immunocompromised patients.

A 13-year-old boy was hospitalized with fever and myalgia. Painful nodular lesions were detected on the scalp, arms, and legs. Pancytopenia and blasts were present in the peripheral blood. He was diagnosed with acute myeloid leukemia. Magnetic resonance imaging of the whole body showed multiple peripheral rim-enhancing, cyst-like lesions. Ultrasonography showed echogenic nodules inside the cystic lesions in the intramuscular space of the arms and legs. Therefore, cysticercosis was strongly suggested initially. However, an abscess was confirmed on sono-guided biopsy and S. epidermidis was isolated from a microbial culture of the tissue. We report a case of multiple disseminated lesions caused by S. epidermidis in a leukemia patient, initially mistaken for cysticercosis.

Keywords: Leukemia, myeloid, acute; Staphylococcus epidermidis; Cysticercosis

Case Report

Received: Oct 4, 2019 Revised: Dec 3, 2019 Accepted: Dec 5, 2019 Correspondence to Sun-Ja Kim

Department of Pediatrics, Cheju Halla General Hospital, 65 Doryeong-ro, Jeju 63127, the Republic of Korea.

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (https://

creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

ORCID iDs Jae Min Lee

https://orcid.org/0000-0002-1081-2403 Joon-Sik Choi

https://orcid.org/0000-0002-5587-2960 Keon Hee Yoo

https://orcid.org/0000-0002-5980-7912 Yae-Jean Kim

https://orcid.org/0000-0002-8367-3424 Sun-Ja Kim

https://orcid.org/0000-0002-1584-3970 Conflict of Interest

No potential conflict of interest relevant to this article was reported.

Disseminated Septic Lesions Caused by Staphylococcus epidermidis

Mimicking Cysticercosis Detected on Ultrasonography in a Pediatric Patient with Acute Myeloid Leukemia

Jae Min Lee ,¹ Joon-Sik Choi ,² Keon Hee Yoo ,² Yae-Jean Kim ,² Sun-Ja Kim ¹

1Department of Pediatrics, Cheju Halla General Hospital, Jeju, the Republic of Korea

2 Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, the Republic of Korea

급성 골수성 백혈병 소아환자에서 초음파 검사에서 낭미충증으로 오인된 표피

포도알균에 의한 파종 감염 병변이 발생한 증례

이재민 ,¹최준식 ,²유건희 ,²김예진 ,²김선자¹

1제주한라병원 소아청소년과

2성균관대학교 의과대학 삼성서울병원 소아청소년과

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Author Contributions

Conceptualization: Lee JM, Kim YJ; Data curation: Choi JS; Supervision: Lee JM, Yoo KH, Kim YJ, Kim SJ; Writing - original draft: Lee JM; Writing - review & editing: Lee JM.

서론

표피포도알균⁽Staphylococcus epidermidis)은그람양성균으로¹⁾사람피부에흔하게있는균이 다^.²⁾몸안의카테터나외과적이물질로인해감염된경우를제외하고는병독성이낮다^.그러 나^,백혈병등면역이저하된환자에서는심각한감염을일으킬수있다^.³⁾

낭미충증(cysticercosis)은유구조충⁽Taenia solium, pork tapeworm)의충란으로오염된음식이

나물을섭취하여발생한다^.⁴⁾인체로들어온유충은장벽의혈류를타고온몸의여러장기로 이동한다^.다른조직에비해혈액공급이풍부한뇌^,눈및근육에서유충이잘성장할수있 으며^,⁵⁾중추신경계감염이없이연부조직이나근육에만감염된경우는드물다^.⁶⁾기생부위 에따라증상이발생하기도하나^,보통수년동안무증상이거나피부에결절이만져지는등 의비특이적증상을보인다^.⁵⁾조직검사나혈청학적검사를통해진단하며^,자기공명영상이

나초음파검사에서낭종내부에두절^(scolex)이보이면의심할수있다^.^7,8)

저자들은급성골수성백혈병환자에서낭미충증과감별이필요했던표피포도알균에의한 두피^,폐^,어깨^,팔과다리에파종감염병변이발생한예를경험하였기에보고하는바이다^.

증례

제주도에거주하던¹³세남자가내원³일전부터시작된발열^,근육통으로걷기힘들어^A병 원응급실을통해입원하였다^.환자는내원²주전독감으로항바이러스제를²일간복용하 다자의중단하였다^.이외다른과거력은없었다^.여행력은없었고^,가족중비슷한증상을보 인사람도없었다^.

A 병원입원시활력징후는혈압 117/54 mmHg, 심박수¹⁰⁸회^/분^,호흡수²⁰회^/분^,체온^38.9°C, 산소포화도^99%이었다^.신체진찰에서우측폐음이감소하였고수포음이들렸다^.좌상복부

압통이있었으나^,간과비장은만져지지않았다^.혈액검사에서백혈구^14,480/mm³⁽호중구

4.4%, 림프구^17.1%,단핵구^78.4%,절대호중구수^650/mm³^,절대림프구수^2,476/mm³^),혈

색소 9.6 g/dL, 적혈구용적률^26.7%,혈소판^69,000/mm³이었다^{. C-}반응단백 21.09 mg/dL (정

상치 0–0.3 mg/dL), 크레아틴인산활성효소 366 IU/L (정상치 50–200 IU/L), 젖산탈수소효소

1,260 IU/L (정상치 119–229 IU/L), 페리틴 891.67 ng/mL (정상치 22–322 ng/mL) 이었다^.요잠혈 은양성이었다^.가래배양검사와혈액배양검사에서동정된균은없었다^.흉부방사선검사

에서가슴막삼출을동반한우하엽폐렴소견으로 ceftriaxone, roxithromycin 치료를시작하

였다^.근육통에대해서는근염이나횡문근융해증을의심하여수액치료를시작하였다^.환자 는입원³일째백혈구^13,510/mm³⁽절대호중구수^500/mm³^),혈색소 7.9 g/dL, 혈소판^49,000/

mm³으로범혈구감소증이진행하여시행한말초혈액도말검사에서모세포^80%로급성백

혈병을진단받았다^.같은날추가검사및치료를위해^B병원으로전원하였다^.

B 병원입원당시신체검진에서압통이없는여러개의경부림프절이만져졌고^,압통을동

반하는결절이오른쪽측두부두피및팔과다리에서여러개만져졌다^{. B}병원입원²일째시 행한골수생검에서급성골수성백혈병으로진단되었다^.입원³일째시행한전신자기공명

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영상검사에서오른쪽측두부두피에조영증강되는결절과양쪽어깨^,팔과다리에서가장 자리조영증강되는병변이다수보였다^.대부분의병변은근육내에위치하고^,일부는피부 와피하지방에위치하였다^{(Fig. 1).}흉부고해상도단층검사에서가장자리조영증강을보이 는여러개의결절이폐에서발견되었다^{(Fig. 2).}두검사에서비정형과립구육종^,파종감염 결절^,결핵^,진균감염^,낭미충증등이의심되었다^.원인감별을위해혈액배양검사^,객담및

기관지폐포세척액의세균배양^,결핵균도말과배양및 polymerase chain reaction, 진균배양

검사및 aspergillus antigen 검사^,경기관지폐생검^,혈청기생충특이항체^,분변기생충충란

검사등을시행하였으나모두음성소견이었다^.

Fig. 1. Whole-body magnetic resonance imaging shows multiple peripheral rim-enhancing lesions in the shoulder, upper limbs, and lower extremities (red circle). The lesions are mostly in the intramuscular space and look like cystic lesions. Peripheral rim-enhancing lesions are also seen in the lung (blue circle).

B A

Fig. 2. (A) coronal view and (B) axial view of chest computed tomography show multiple nodular low-density lesions in the bilateral subpleural area with ground-glass opacification.

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환자는호중구감소증이지속되는상태에서발열과압통을동반한종아리부종이호전되지

않아서^,호중구감소성발열및폐렴에대해경험적항생제로^cefepime과 doxycycline 치료를

유지하면서항암치료를보류하였다^.입원¹⁰일째팔과다리의연부조직초음파를시행하였 다^.초음파검사에서팔과다리근육내여러개의낭성병변이보였다^.낭성병변내부에는에 코성결절이있었다^.일부병변에서는부스러기를동반하여농양가능성이고려되었으나^,낭 성병변내부에보이는결절은낭미충증에서보이는두절이강력히의심되었다^{(Fig. 3).}입원 11일째오른쪽종아리근육내병변에서초음파유도하조직생검및배양검사를무균적으로

시행후 teicoplanin을추가하였다^.낭미충증가능성에대해뇌자기공명영상검사를시행하

였으나^,특이소견은없었다^.조직생검결과가입원¹⁶일째에농양으로확인되었고^,조직배 양검사에서 methicillin 내성표피포도알균이동정되었다^.심내막염으로인한색전또는다 발성농양이발생했을가능성을고려하여심초음파를시행하였으나정상소견이었다^.당시

사용중인 teicoplanin에대한표피포도알균의최소억제농도가^{8 μg/mL}으로감수성기준최

대치에해당하였고^{, 5}일사용후에도임상적으로호전을보이지않아항생제를 vancomycin 으로변경하였다^.환자가면역저하상태인점을감안하여낭미충증가능성에대해서도항원

충제(albendazole, praziquantel) 치료를병행하였다. Vancomycin과항원충제치료⁵일후인입

원²⁰일째발열과압통은호전되었다. Praziquantel 6일, albendazole 7일투여후^,입원¹¹일째 에재시행했던혈청기생충항체검사가음성인것을확인하여항원충제치료는중단하였다^.

Vancomycin 치료를지속하면서입원²¹일째급성골수성백혈병에대해항암치료를시작하

였다. Vancomycin은총²⁵일사용후중단하였고^,입원⁴⁴일째퇴원하였다^.퇴원⁶일후항암

치료를위해재입원하였고^,당시시행한흉부방사선검사에서폐결절은없어졌다^. 본증례보고는제주한라병원임상연구심의위원회의심사후승인을받았다 (2019-L07).

B A

Fig. 3. Ultrasonography of the (A) right elbow and (B) right lower leg show echogenic nodular lesions (red arrow) inside echogenic cystic lesions in the intramuscular space. (A) Debris (blue arrow) inside the cystic lesion may be the formation of an abscess. (A), (B) The echogenic nodular lesion (red arrow) inside the cystic lesion was thought to be strongly suggestive of a scolex in cysticercosis.

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고찰

본증례는급성골수성백혈병을진단받은환자에서전신다발낭성병변이발생하여^,이에 대해낭미충증과세균감염의감별이필요하였다^.

유구낭미충⁽cysticercus cellulosae)은유구조충⁽T. solium, pork tapeworm)의유충으로감염된돼 지근육내분포한다^.⁴⁾덜익힌돼지고기를먹어감염되며^,사람의소장에서성충으로성장 해알을낳아대변으로배출된다^.낭미충증은유구조충의충란으로오염된음식이나물을먹

어감염되어발생한다^.^4,5,7)사람의장에서충란속에있던유충이나와장벽혈관으로들어가

신체각부분으로이동하게된다^.^4,5,8)낭미충증은중남미^,아프리카^,인도^,중국등이유행지

역이나^,사람들의이동이많은현재는전세계적으로분포한다^.뇌를침범하는가장흔한기 생충감염이며^,뇌에발생한경우를뇌낭미충증(neurocysticercosis)이라한다^.대부분뇌감 염을동반하며^,근육감염만있는경우는드물다^.⁸⁾감염후유충이기생하는장소에따라다 양한증상이발생하기도하나^,특별한증상없이피부에결절이만져지거나수년동안무증 상으로지내다가유충이죽으면서나오는항원에증상이발생하는경우가많다^.^5,8)과거제주 도에서는돼지에게사람대변을먹여유구조충감염과낭미충증이유행했지만^{, 1980}년대부

터이를금지하여감염률이급격히감소했다^.⁶⁾¹⁹⁹⁰년이후우리나라돼지에서유구낭미충

이검출되지않고^{, 2006}년제주도주민의대변검사에서유규조충감염률은^0%였다^.^8,9)낭미

충증확진은조직검사이나혈청학적검사가진단에유용하며^,뇌낭미충증은컴퓨터단층촬 영이나자기공명영상검사가도움이된다^.⁸⁾피하조직이나근육에기생하는경우에는영상 학적진단이쉽지않으나^,자기공명영상이나초음파검사에서낭종내부에두절이보이면 진단에도움이된다^.⁷⁾

본증례의환자는팔과다리초음파검사에서근육내낭성병변내부에결절이두절로보여 낭미충증이의심되어항원충제 praziquantel 및 albendazole 치료를약¹주동안시행하였다^.

그러나효과적인낭미충증치료기간은 praziquantel 3주, albendazole 2주이다^.⁵⁾환자는과거

낭미충증이유행하였던제주도에거주하였지만^,최근낭미충증감염률이낮고^,^6,8-10)환자의 연령이어리며^,시내빌라에거주하였고^,오염원을접촉한적이없으며해외여행력이없어 낭미충증가능성은낮았다^.또한낭미충증가능성에대해부적절한치료를하였음에도증상 과검사소견이호전되었다^.

표피포도알균은그람양성, coagulase 음성균으로서보통사람피부에존재하는비병원성균 이지만^,입원환자나체내이물질을가진환자에서감염을일으킬수있다^1-3)^.특히^,면역저하 자에서심각한감염을잘일으킨다^.한편표피포도알균에의해건강한소아환자에서간농양 이발생한사례^,¹¹⁾약물중독자가아닌대동맥판막협착증과만성간질환이있는성인에서심 내막염과심장농양이발생한사례^,¹²⁾신생아에서유선염과유방농양이발생한사례¹³⁾를보 면병독성이약한표피포도알균도심각한감염을일으킬수있다^.따라서^,본증례환자에서 발생한전신다발병변은표피포도알균에의한파종감염병변으로보인다^.이처럼^,백혈병 환자에서는다양한균주에대한감염에취약하고^,특히일반적으로병독성이덜하다고여겨 지는표피포도알균과같은균주조차도전신의다발성감염을일으킬수있기에환자의감염 관리에각별한주의가필요할것으로생각된다^.

(6)

저자들은급성골수성백혈병환자에서영상학적으로낭미충증과감별이필요했던표피포 도알균에의한전신다발병변이발생한예를경험하였기에보고하는바이다^.

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요약

표피 포도알균은 사람 피부에 있는 정상균이나, 체내 이물질을 가진 사람이나 면역 저하자에게는 심각한 감염을 일으킬 수 있다. 13세 남자가 발열, 근육통으로 입원하였고 두피, 팔과 다리에 통증이 있는 결절성 병변이 만져졌다. 혈액검사에서 범 혈구 감소증과 모세포 80% 소견을 보였고 급성 골수성 백혈병으로 진단되었다. 전신 자기공명영상 검사에서 가장자리 조 영 증강을 보이는 다발 낭성 병변이, 초음파 검사에서 에코성 낭성 병변과 그 내부의 에코성 결절이 팔과 다리의 근육 내부 에서 관찰되어, 낭미충증이 강력히 의심되었다. 그러나 초음파 유도하 조직 검사에서 농양이 확인되었고, 조직 배양검사에 서 표피 포도알균이 동정되었다. 저자들은 백혈병 환자에서 낭미충증으로 오인되었던 표피 포도알균에 의한 전신 다발 병 변이 발생한 예를 경험하였기에 보고하는 바이다.