Steroid-dependent Kimura Disease in a Child Treated with Cetirizine and Montelukast
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Steroid-dependent Kimura Disease in a Child Treated with Cetirizine and Montelukast
cervical, axillary, and inguinal lymphadenopathy and fever at the age of 3 years. Cervical lymph node biopsy had reported an illdefined granulomatous lesion and with a positive Mantoux test, but no microbial confirmation, he had received anti-tuberculous therapy for 18 months. As the lymphadeno-pathy and fever persisted, his lymph node biopsy was repeated and he was diagnosed as Kimura disease at 5 years and 3 months of age. He was started on oral steroids to which he initially responded but the lymphadenopathy and fever would recur as the steroids were tapered to 10 mg (0.6 mg/kg/day). Methotrexate and azathioprine were tried sequentially as steroid-sparing agents, but were discontinued due to transaminitis. On steroids for about 20 months with significant growth retardation, he was referred to us for further management.
Kimura disease or eosinophilic lymphogranuloma, is a rare disease of unknown etiology, affecting middle-aged Asian men and occasionally children [1]. It is characterized by a triad of painless unilateral cervical adenopathy or subcutaneous masses predominantly in head or neck, blood and tissue eosinophilia, and elevated serum IgE levels [1]. We share our experience with using cetirizine and montelukast in a long-standing, steroiddependent, severely growth retarded child with the disease, and propose this combination as an alternative to steroids, immunosuppressive drugs, radiation or surgery.
On examination, he was febrile with mild cushingoid facies and had non-tender left axillary and inguinal lymph nodes. The systemic examination was normal. His clinical features, anthropometry, investigations and course are summarized in Fig. 1. There were no features of renal involvement. His lymph node biopsy slides from both earlier biopsies were reviewed and
An 8-year-9-month-old male child, firstborn to third-degree consanguineous parents presented to his pediatrician with
Abbreviations: C: Centirizine; M: Montelukast; P: Prednisolone; LN: Lymphadenopathy; wt: weight; ht: height; w: weeks; I: Investigations; CBC: Complete blood picture; ESR: Erythrocytesedimentation rate; Hb: Hemoglobin; WBC: white blood cell count; Plt: Platelets; E: eosinophils; Rx: treatment.
Fig. 1 Disease course in the index patient with Kimura disease.
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CORRESPONDENCE were consistent with the diagnosis of Kimura disease.
affordable, easily available, orally administered and not needing any specific monitoring for side effects. After remission, we omitted montelukast first because we believed that cetirizine has a better safety profile for long t
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