Skip to main content
Log in

Kikuchi–Fujimoto's Necrotizing Lymphadenitis in Association with Discoid Lupus Erthematosus: A Case Report

  • Case Report
  • Published:
Journal of Cutaneous Medicine and Surgery

Abstract

Background

Kikuchi–Fujimoto's necrotizing lymphadenitis (KFNL) is a rare, benign, self-limited condition characterized by constitutional symptoms, lymphadenopathy, and skin lesions.

Objective

We report a case of KFNL in a 43-year-old East Indian woman with a ten-year history of discoid lupus erythematosus (DLE) of the scalp and a three-month history of a erythematous plaque on the left nasal bridge, cervical lymphadenopathy, and fever. Skin biopsy samples were taken from the face and lymph node.

Results

Histopathological examination of the skin revealed a mixed infiltrate of inflammatory cells, nuclear dust, and histiocytes phagocytosing nuclear debris in the reticular dermis. The lymph node showed interfollicular liquefactive necrosis, immunoblasts, and a similar cellular infiltrate as the skin. The non-necrotic areas demonstrated follicular hyperplasia. These pathological changes are associated with a diagnosis of KFNL.

Conclusions

KFNL is reported in association with systemic lupus erythematosus, but only two other cases of systemic KFNL in association with DLE exist in the literature. This case is unique in that the patient presented with cutaneous and systemic KFNL in the setting of longstanding DLE.

Sommaire

Antécédents

La maladie de Kikuchi–Fujimoto, ou lymphadénite histiocytaire nécrosante, est une maladie rare, bénigne et résolutive, qui se caractérise par des symptômes constitutionnels, une adénopathie et des lésions cutanées.

Objectifs

Nous rapportons un cas de lymphadénite histiocytaire nécrosante chez une femme de 43 ans, originaire des Indes orientales, ayant un lupus érythémateux discoïde du cuir chevelu depuis dix ans et présentant, depuis trois mois, une plaque érythémateuse sur la voûte nasale gauche, une adénopathie cervicale et de la fièvre. Des échantillons de biopsie ont été prélevés du visage et des ganglions lymphatiques.

Résultats

L'examen histopathologique de la peau a révélé un infiltrat mixte de cellules inflammatoires, de poussière nucléaire et de phagocytes de débris nucléaires dans le derme réticulaire. Le ganglion lymphatique a montré une nécrose liquéfiante interfolliculaire, des immunoblastes et un infiltrat cellulaire similaire à la peau. Les zones non nécrosées ont présenté une hyperplasie folliculaire. Ces changements pathologiques sont associés à un diagnostic de la maladie de Kikuchi-Fujimoto.

Conclusion

La maladie de Kikuchi–Fujimoto est rapportée en présence d'un lupus érythémateux discoïde. Deux autres cas seulement ont été décrits dans les publications spécialisées. Le présent cas est unique du fait que la lymphadénite histiocytaire nécrosante s'est déclarée en présence d'un lupus érythémateux discoïde de longue date.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

FIGURE 1
FIGURE 2
FIGURE 3
FIGURE 4
FIGURE 5

Similar content being viewed by others

References

  1. M. Kikuchi (1972) ArticleTitleLymphadenitis showing focal reticulum cervical hyperplasia with nuclear debris and phagocytes Acta Hematol Jpn 35 379–380

    Google Scholar 

  2. Y Fujimoto Y Kojima K. Yamaguchi (1972) ArticleTitleCervical subacute necrotizing lymphadenitis Naika 30 920–927

    Google Scholar 

  3. AH Norris AM Krasinskas KE Salhany et al. (1996) ArticleTitleKikuchi–Fujimoto disease: a benign cause of fever and lymphadenopathy Am J Med 101 401–405 Occurrence Handle10.1016/S0002-9343(96)00231-8 Occurrence Handle1:STN:280:DyaK2s%2FisleksA%3D%3D

    Article  CAS  Google Scholar 

  4. C Lopez M Oliver R Olavarria et al. (2000) ArticleTitleKikuchi–Fujimoto necrotizing lymphadenitis associated with cutaneous lupus erythematosus: a case report Am J Dermatopathol 22 328–333 Occurrence Handle10.1097/00000372-200008000-00006 Occurrence Handle1:STN:280:DC%2BD3M%2FjsF2jtQ%3D%3D

    Article  CAS  Google Scholar 

  5. S Lecoules M Michel V Zarrouk et al. (2003) ArticleTitle[Recurrent Kikuchi's disease in a patient with discoid lupus] Rev Med Interne 24 613–616 Occurrence Handle10.1016/S0248-8663(03)00222-4 Occurrence Handle1:STN:280:DC%2BD3svht1Wqtg%3D%3D

    Article  CAS  Google Scholar 

  6. D Biasi P Caramaschi A Carletto et al. (1996) ArticleTitleThree clinical reports of Kikuchi's lymphadenitis combined with systemic lupus erythematosus Clin Rheumatol 15 81–83 Occurrence Handle10.1007/BF02231692 Occurrence Handle1:STN:280:DyaK2s%2Fotlajuw%3D%3D

    Article  CAS  Google Scholar 

  7. MD Eisner J Amory B Mullaney et al. (1996) ArticleTitleNecrotizing lymphadenitis associated with systemic lupus erythematosus Semin Arthritis Rheum 26 477–482 Occurrence Handle10.1016/S0049-0172(96)80028-X Occurrence Handle1:STN:280:DyaK2s%2FitlOrug%3D%3D

    Article  CAS  Google Scholar 

  8. HC Lin CY Su CC Huang et al. (2003) ArticleTitleKikuchi's disease: a review and analysis of 61 cases Otolaryngol Head Neck Surg 128 650–653 Occurrence Handle10.1016/S0194-5998(02)23291-X

    Article  Google Scholar 

  9. A Quintas–Cardama M Fraga SN Cozzi et al. (2003) ArticleTitleFatal Kikuchi–Fujimoto disease: the lupus connection Ann Hematol 82 186–188 Occurrence Handle10.1007/s00277-003-0611-7

    Article  Google Scholar 

  10. A Yen P Fearneyhough SS Raimer et al. (1997) ArticleTitleEBV-associated Kikuchi's histiocytic necrotizing lymphadenitis with cutaneous manifestations J Am Acad Dermatol 36 342–346 Occurrence Handle10.1016/S0190-9622(97)80413-6 Occurrence Handle1:STN:280:DyaK2s7ptFamtQ%3D%3D

    Article  CAS  Google Scholar 

  11. YJ Jang KH Park HJ. Seok (2000) ArticleTitleManagement of Kikuchi's disease using glucocorticoid J Laryngol Otol 114 709–711 Occurrence Handle10.1258/0022215001906561 Occurrence Handle1:STN:280:DC%2BD3M%2FltlCjsg%3D%3D

    Article  CAS  Google Scholar 

  12. R Blewitt S Kumar J. Abraham (2000) ArticleTitleRecurrence of Kikuchi's lymphadenitis after 12 years J Clin Pathol 53 157–158 Occurrence Handle10.1136/jcp.53.2.157 Occurrence Handle1:STN:280:DC%2BD3c3jtVeluw%3D%3D

    Article  CAS  Google Scholar 

  13. RF Dorfman GJ. Berry (1988) ArticleTitleKikuchi's histiocytic necrotizing lymphadenitis: an analysis of 108 cases with emphasis on differential diagnosis Semin Diagn Pathol 5 329–345 Occurrence Handle3217625 Occurrence Handle1:STN:280:DyaL1M7hsVSjsg%3D%3D

    PubMed  CAS  Google Scholar 

  14. J Spies K Foucar CT Thompson et al. (1999) ArticleTitleThe histopathology of cutaneous lesions of Kikuchi's disease (necrotizing lymphadenitis): a report of five cases Am J Surg Pathol 23 1040–1047 Occurrence Handle10.1097/00000478-199909000-00006 Occurrence Handle1:STN:280:DyaK1MvgsFOlug%3D%3D

    Article  CAS  Google Scholar 

  15. SA Pileri F Facchetti S Ascani et al. (2001) ArticleTitleMyeloperoxidase expression by histiocytes in Kikuchi's and Kikuchi-like lymphadenopathy Am J Pathol 159 915–924 Occurrence Handle10.1016/S0002-9440(10)61767-1 Occurrence Handle1:STN:280:DC%2BD3Mrht1Cqsg%3D%3D

    Article  CAS  Google Scholar 

  16. RE Felgar EE Furth MA Wasik et al. (1997) ArticleTitleHistiocytic necrotizing lymphadenitis (Kikuchi's disease): in situ end-labeling, immunohistochemical, and serologic evidence supporting cytotoxic lymphocyte-mediated apoptotic cell death Mod Pathol 10 231–241 Occurrence Handle9071731 Occurrence Handle1:STN:280:DyaK2s3isFemug%3D%3D

    PubMed  CAS  Google Scholar 

  17. KJ Cho SS Lee SK. Khang (1996) ArticleTitleHistiocytic necrotizing lymphadenitis. A clinico-pathologic study of 45 cases with in situ hybridization for Epstein–Barr virus and hepatitis B virus J Korean Med Sci 11 409–414 Occurrence Handle10.3346/jkms.1996.11.5.409 Occurrence Handle1:STN:280:DyaK2s%2FovVCmug%3D%3D

    Article  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Shane G. Silver.

About this article

Cite this article

Silver, S.G., Hong, H.Ch., Ting, P.T. et al. Kikuchi–Fujimoto's Necrotizing Lymphadenitis in Association with Discoid Lupus Erthematosus: A Case Report. J Cutan Med Surg 8, 442–445 (2004). https://doi.org/10.1007/s10227-004-0119-3

Download citation

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10227-004-0119-3

Keywords

Navigation