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<p>Kikuchi-Fujimoto Disease (KFD) is a rare, self-limiting necrotizing lymphadenitis, seldom observed in children. Its atypical clinical presentation often mimics infectious or malignant conditions. Secondary hemophagocytic lymphohistiocytosis (HLH), a severe hyperinflammatory syndrome, is an exceptionally rare but critical complication of KFD. We report a case of a 12-year-old male presenting with persistent fever and bilateral parotid swelling unresponsive to empirical antibiotics and antivirals. Imaging revealed bilateral parotid gland enlargement without features suggestive of neoplasia. Histopathological examination of an incisional biopsy confirmed necrotizing lymphadenitis consistent with KFD. Further investigations revealed hyperferritinemia, hypertriglyceridemia, and parvovirus B19 positivity. Bone marrow aspiration demonstrated hemophagocytosis, fulfilling HLH-2004 diagnostic criteria. The patient was treated with high-dose intravenous methylprednisolone, broad-spectrum antibiotics (Meropenem and Vancomycin), liposomal Amphotericin B, and prophylactic Trimethoprim-Sulfamethoxazole. Marked clinical improvement and resolution of systemic inflammatory features were observed following initiation of immunosuppressive therapy. This case underscores a rare pediatric presentation of KFD mimicking bilateral parotitis and complicated by secondary HLH. It highlights the importance of early histopathological evaluation and comprehensive hematologic workup in children with persistent lymphadenopathy and systemic symptoms. Prompt recognition and multidisciplinary management are essential to improve outcomes in such complex presentations.</p>

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Parotid Glands as Pandora’s Box: A Case of Mistaken Swelling—Pediatric Kikuchi-Fujimoto Disease with Secondary HLH Masquerading as Infective Parotitis

  • 期刊论文

  • 发表日期：

    2025年8月4日

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  • Kiran Kumar,
  • Sandeep Dabhekar,
  • Rohit Bhondekar,
  • K. Khadeeja,
  • Nittika Garg

摘要

Kikuchi-Fujimoto Disease (KFD) is a rare, self-limiting necrotizing lymphadenitis, seldom observed in children. Its atypical clinical presentation often mimics infectious or malignant conditions. Secondary hemophagocytic lymphohistiocytosis (HLH), a severe hyperinflammatory syndrome, is an exceptionally rare but critical complication of KFD. We report a case of a 12-year-old male presenting with persistent fever and bilateral parotid swelling unresponsive to empirical antibiotics and antivirals. Imaging revealed bilateral parotid gland enlargement without features suggestive of neoplasia. Histopathological examination of an incisional biopsy confirmed necrotizing lymphadenitis consistent with KFD. Further investigations revealed hyperferritinemia, hypertriglyceridemia, and parvovirus B19 positivity. Bone marrow aspiration demonstrated hemophagocytosis, fulfilling HLH-2004 diagnostic criteria. The patient was treated with high-dose intravenous methylprednisolone, broad-spectrum antibiotics (Meropenem and Vancomycin), liposomal Amphotericin B, and prophylactic Trimethoprim-Sulfamethoxazole. Marked clinical improvement and resolution of systemic inflammatory features were observed following initiation of immunosuppressive therapy. This case underscores a rare pediatric presentation of KFD mimicking bilateral parotitis and complicated by secondary HLH. It highlights the importance of early histopathological evaluation and comprehensive hematologic workup in children with persistent lymphadenopathy and systemic symptoms. Prompt recognition and multidisciplinary management are essential to improve outcomes in such complex presentations.