DOI QR코드

DOI QR Code

Immunoglobulin G4-Related Disease Involving the Pterygopalatine Fossa, Mimicking Invasive Aspergillosis: A Case Report and Literature Review

침습아스페르길루스증으로 오인된 날개입천장오목에 발생한 면역글로불린 G4 관련 질환: 증례 보고 및 문헌 고찰

  • Jin Young Son (Department of Radiology, Yeouido St. Mary's Hospital, College of Medicine, The Catholic University of Korea) ;
  • Jee Young Kim (Department of Radiology, Eunpyeong St. Mary's Hospital, College of Medicine, The Catholic University of Korea) ;
  • Jin Hee Cho (Department of Otorhinolaryngology-Head and Neck Surgery, Yeouido St. Mary's Hospital, College of Medicine, The Catholic University of Korea) ;
  • Eun Jung Lee (Department of Hospital Pathology, Yeouido St. Mary's Hospital, College of Medicine, The Catholic University of Korea)
  • 손진영 (가톨릭대학교 의과대학 여의도성모병원 영상의학과) ;
  • 김지영 (가톨릭대학교 의과대학 은평성모병원 영상의학과) ;
  • 조진희 (가톨릭대학교 의과대학 여의도성모병원 이비인후과) ;
  • 이은정 (가톨릭대학교 의과대학 여의도성모병원 병원병리과)
  • Received : 2020.04.27
  • Accepted : 2020.10.02
  • Published : 2021.07.01

Abstract

We report a case of Immunoglobulin G4 (IgG4) related disease involving the pterygoplataine fossa. A 83-year-old male presented with left ocular pain and visual disturbance. CT showed an isodense soft tissue lesion in the left pterygopalatine fossa with bony sclerotic changes and erosion. MRI revealed an infiltrative soft tissue mass in the left pterygopalatine fossa as a T2 slightly low signal intensity and heterogeneous enhancement. The patient underwent left ethmoidectomy, and biopsy of the mass was conducted. The histopathological diagnosis was IgG4-related disease. In this case, it was difficult to differentiate invasive aspergillosis, which is common in immunocompromised patients, considering the patient's clinical history of diabetes mellitus. This report describes the imaging findings of IgG4-related disease mimicking invasive sinusitis such as invasive aspergillosis.

본 증례는 좌안의 통증과 시력저하를 호소한 83세 남자 환자에게서 진단된 면역글로불린 G4 (immunoglobulin G4; 이하 IgG4) 관련 질환의 CT와 MRI 소견에 대한 보고이다. 이는 CT에서 주변의 골미란을 동반한 날개입천장 오목의 등밀도 연부조직 종괴로 관찰되었으며 이병변은 자기공명영상 T2 강조영상에서 등신호에서 저신호강도를 보이며 비균질한 조영증강을 보였다. 환자는 벌집굴 절제술과 조직검사를 시행하였고 조직학적 검사상 IgG4 관련 질환으로 진단되었다. 환자는 기저질환으로 당뇨병이 있었고, 이런 면역저하 환자군에서 흔하게 보이는 침습적 진균성 부비동염과의 영상학적 감별이 어려운 경우였다. 저자들은 침습적 진균성 부비동염과 유사한 영상 소견을 보였던 IgG4 관련 질환의 증례를 문헌 고찰과 함께 보고하고자 한다.

Keywords

References

  1. Wallace ZS, Naden RP, Chari S, Choi HK, Della-Torre E, Dicaire JF, et al. The 2019 American College of Rheumatology/European League Against Rheumatism classification criteria for IgG4-related disease. Ann Rheum Dis 2020;79:77-87
  2. Fujita A, Sakai O, Chapman MN, Sugimoto H. IgG4-related disease of the head and neck: CT and MR imaging manifestations. Radiographics 2012;32:1945-1958
  3. Horger M, Lamprecht HG, Bares R, Spira D, Schmalzing M, Claussen CD, et al. Systemic IgG4-related sclerosing disease: spectrum of imaging findings and differential diagnosis. AJR Am J Roentgenol 2012;199:W276-W282
  4. Ryu G, Cho HJ, Lee KE, Lee JJ, Hong SD, Kim HY, et al. Clinical significance of IgG4 in sinonasal and skull base inflammatory pseudotumor. Eur Arch Otorhinolaryngol 2019;276:2465-2473
  5. Koeller KK. Radiologic features of sinonasal tumors. Head Neck Pathol 2016;10:1-12
  6. Kim SH, Mun SJ, Kim HJ, Kim SL, Kim SD, Cho KS. Differential diagnosis of sinonasal lymphoma and squamous cell carcinoma on CT, MRI, and PET/CT. Otolaryngol Head Neck Surg 2018;159:494-500
  7. Kim E, Kim JY, Cho JH, Kang C. Image features of aspergillosis arising from the pterygopalatine fossa: a case report and literature review. Oral Radiology 2014;30:192-195
  8. Katsura M, Mori H, Kunimatsu A, Sasaki H, Abe O, Machida T, et al. Radiological features of IgG4-related disease in the head, neck, and brain. Neuroradiology 2012;54:873-882
  9. Wang Z, Feng R, Chen Y, Duan M, Wang M, Jin Z, et al. CT features and pathologic characteristics of IgG4-related systemic disease of submandibular gland. Int J Clin Exp Pathol 2015;8:16111-16116