Interstitial Pneumonitis: A Rare Manifestation of Kikuchi-Fujimoto Disease
Keywords:
Kikuchi Fujimoto Disease, Lymphadenopathy, Interstitial pneumonitisAbstract
Background: Kikuchi-Fujimoto Disease (KFD) or subacute histiocytic necrotizing lymphadenitis is an uncommon cause of lymphadenopathy. The etiology of KFD remains unknown but it is postulated to be associated with post-viral infection and autoimmune processes. Most patients present with relapsing and remitting course of fever and lymphadenopathy, mostly cervical area. However, KFD patients could present with other uncommon manifestations including aseptic meningitis, hepatitis, uveitis, etc. Pulmonary involvement is rare in KFD and was reported by only a few groups. Herein, we report a case of KFD presenting with interstitial pneumonitis. Case Presentation: A 32-year-old Thai female initially presented with prolonged fever and cervical lymphadenopathy. She was diagnosed with KFD proved by histopathology of cervical lymph node biopsy. Her KFD course was complicated with aseptic meningitis, recurrent fever and lymphadenopathy responded to systemic corticosteroid. She recently presented with dyspnea on exertion, cough and hypoxia. The chest computed tomography revealed diffuse ground glass and interstitial nodules. Extensive investigations for infectious etiologies were normal. Our patient was given therapeutic trial of systemic corticosteroid and her symptoms were completely resolved. Follow-up chest radiographic imaging became normal. Conclusion: Although pulmonary involvement in KFD is rare, physician should be aware of this complication in order to promptly diagnose and provide appropriate treatments
Downloads
References
2. Fujimoto Y, Kozima Y, Yamaguchi K. Cervical subacute necrotizing lymphadenitis: a new clinicopathologic entity. Naika. 1972;20:920-7.
3. Sopena B, Rivera A, Vazquez-Trinanes C et al. Autoimmune manifestations of Kikuchi disease. Semin Arthritis Rheum. 2012;41:900-6.
4. Garcia-Zamalloa A, Taboada-Gomez J, Bernardo-Galan P et al.Bilateral pleural effusion and interstitial lung disease as unusual manifestations of Kikuchi-Fujimoto disease: case report and literature review. BMC Pulm Med. 2010;10:54.
5. Hua F, Zhu L. Kikuchi Fujimoto disease associated with cryptogenic organizing pneumonia: case report and literature review. BMC Infect Dis. 2010;10:64.
6. Wong CY, Law GT, Shum TT et al. Pulmonary haemorrhage in a patient with Kikuchi disease. Monaldi Arch Chest Dis. 2001;56:118-20.
7. Bosch X, Guilabert A, Miquel R, Campo E. Enigmatic Kikuchi-Fujimoto disease: a comprehensive review. Am J Clin Pathol.2004;122:141-52.
8. Bogusz AM, Bhargava P. Recurrent histiocytic necrotizing lymphadenitis with a long latency in a patient with autoimmunity: a case report and review of literature. Int J Surg Pathol. 2013;21:
287-96.
9. Sah SK, Pant R, Piper K, et al. Recurrent Kikuchi-Fujimoto disease: case report. Br J Oral Maxillofac Surg. 2007;45:231-3.
10. Song JY, Lee J, Park DW, et al. Clinical outcome and predictive factors of recurrence among patients with Kikuchi’s disease. Int J Infect Dis. 2009;13:322-6.
11. Kumar TS, Scott JX, Agarwal I, Danda D. Management of Kikuchi disease using glucocorticoids. J Clin Rheumatol. 2006;12:47.