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양안 언가지모양혈관염을 동반한 기구치-후지모토병 1예

Bilateral Frosted Branch Angiitis in Kikuchi-Fujimoto Disease

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초록

목적: 양안에 언가지모양혈관염을 동반한 기구치-후지모토병 1예를 경험하였기에 보고하고자 한다. 증례요약: 33세 남자 환자가 양안의 시력 저하로 내원하였다. 1주일 전부터 두통, 오한, 근육통, 옆구리 통증이 있었다. 초진 당시 최대교정시력은 우안 0.1, 좌안 0.2였고, 안저검사상 양안 후극부 및 주변부 망막에 전체적으로 퍼져 있는 언가지모양의 혈관초가 망막혈관에서 관찰되었으며, 형광안저혈관조영술에서 혈관초가 형성된 망막혈관에서 형광색소의 염색과 누출이 있었다. 말초혈액검사, 혈액배양검사, 복부 컴퓨터단층촬영, 뇌 컴퓨터단층촬영, 뇌척수액 검사상 특이 소견은 없었다. 경부 초음파 검사에서 림프절의 비대가 관찰되었고, 세침흡입조직검사에서 기구치-후지모토병으로 진단되었다. 전신 스테로이드 치료를 시작하였고, 1달 뒤 양안 교정시력은 1.0으로 호전되었다. 결론: 언가지모양혈관염 소견을 보이는 환자에서 기구치-후지모토병 등의 전신질환 동반 가능성에 대한 검사가 필요할 것으로 생각된다.

Abstract

Purpose: A case of frosted branch angiitis in Kikuchi-Fujimoto disease is reported. Case summary: A 33-year-old male complained of a sudden decrease in visual acuity that developed in both eyes 5 days prior. He suffered from a headache, chills, myalgia, and flank pain 1 week before. The initial best-corrected visual acuity (BCVA) was 0.1 in the right eye and 0.2 in the left eye. On slit lamp examination, no inflammatory finding was observed in the anterior chamber and vitreous body of both eyes. On fundus examination, a diffuse vascular sheathing-like frosted branch was found in the retinal vessels, and retinal hemorrhage was observed. Fluorescein angiography showed staining and leakage of dye along the vascular sheathing. Serological findings were negative, showing no evidence of an autoimmune disease or viral infection. Neck ultrasonography revealed non-tender left cervical lymph node enlargement >1 cm in diameter. Ultrasound-guided fine needle aspiration cytology showed findings compatible with Kikuchi-Fujimoto disease, including necrotic changes and pronounced karyorrhexis, plus histiocyte and lymphocyte infiltration without neutrophils. We started systemic steroid therapy. One month after treatment, the BCVA of both eyes improved to 1.0. Conclusions: In patients with frosted branch angiitis, systemic disease such as Kikuchi-Fujimoto disease should be considered.

참고문헌 (15)

  1. Norris AH, "Kikuchi-Fujimoto disease: a benign cause of fever and lymphadenopathy", Am J Med, 101, pp.401~405 (1996)[DOI]
  2. Rocher F, "Kikuchi's disease and ocular manifestation", J Fr Ophtalmal, 29, pp.932~936 (2006)[DOI]
  3. Ito Y, "Frosted branch angiitis in a child", Jpn J Clin Ophthalmol, 30, pp.797~803 (1976)
  4. Bosch X, "Enigmatic Kikuchi-Fujimoto disease: a comprehensive review", Am J Clin Pathol, 122, pp.141~152 (2004)[DOI]
  5. Famularo G, "Kikuchi Fujimoto lymphadenitis: case report and literature review", AM J Hematol, 74, pp.60~63 (2003)[DOI]
  6. Cheng CY, "Clinical presentations, laboratory results and outcomes of patients with Kikuchi's disease: emphasis on the association between recurrent Kikuchi's disease and autoimmune diseases", J Microbiol Immunol Infect, 43, pp.366~371 (2010)[DOI]
  7. Kim SH, "Bilateral anterior uveitis as an unusual manifestation of Kikuchi-Fujimoto disease", Rheumatology (Oxford), 43, pp.1056~1057 (2004)[DOI]
  8. Taguri AH, "Bilateral panuveitis: a possible association with Kikuchi-Fujimoto disease", Am J Ophthalmol, 132, pp.419~421 (2001)[DOI]
  9. Zou W, "Bilateral occlusive retinal vasculitis in Kikuchi-Fujumoto disease", Clin Exp Ophthalmol, 35, pp.875~877 (2007)[DOI]
  10. Galor A, "Papillary conjunctivitis associated with Kikuchi disease", Cornea, 27, pp.944~946 (2008)[DOI]
  11. Biswas J, "Bilateral frosted branchangiitis in an 8-year-old indian girl", Retina, 16, pp.444~445 (1996)[DOI]
  12. Masuda K, "A case of frosted branch angiitis with yellowish-white placoid lesions: fluorescein and indocyanine green angiography findings", Jpn J Ophthalmol, 42, pp.484~489 (1998)
  13. Duker JS, "Retinal vasculitis in Crohn's disease", Am J Ophthalmol, 103, pp.664~668 (1987)[DOI]
  14. Dorfman RF, "Kikuchi's histiocytic necrotizing lymphadenitis: an analysis of 108 cases with emphasis of differential diagnosis", Semin Diagn Pathol, 5, pp.329~345 (1988)
  15. Imamura M, "An ultrastructural study of subacute necrotizing lymphadenitis", Am J Pathol, 107, pp.292~299 (1982)

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