Kimura disease with Allergic Bronchopulmonary Aspergillosis: a case report

Fan, Ruiyun; Xu, Guo; Chen, Ying; Lv, Jinghuan; Zhang, Zhongwei

doi:10.1186/s13223-022-00683-1

Cited by 4 publications

(2 citation statements)

References 21 publications

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“…KD is characterized by painless, rm, diffuse, single, or multiple subcutaneous masses 1 to 7 cm in diameter, principally in the head and/or neck (76%), particularly around the parotid gland and in the submandibular region. Although extremely rare, lesions have also been reported in the axillary and inguinal regions, the trunk, abdomen, chest wall, peripheral extremities, epiglottis, long bones, breasts, genitals, orbits, and ocular appendages [7,8]. The overlying skin is usually not signi cantly affected.…”

Section: Discussionmentioning

confidence: 99%

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Kimura Disease: A Rare Case in Vietnamese Woman

Le,

Tran,

Pham

2023

Preprint

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Background: Kimura disease (KD) is a rare benign chronic inflammatory condition that predominantly affects Asian males. It is characterized by subcutaneous tissue masses in the head and neck region, enlarged lymph nodes, increased blood eosinophilia, and elevated serum total IgE levels. In this report, we describe a rare case of KD in a young Vietnamese female. Case summary: A 31-year-old Vietnamese woman presented to the hospital with 2 masses in the bilateral cheeks and 1 mass behind the left ear that persisted for 15 years, recurrent skin itching, elevated serum total IgE levels and increased blood eosinophilia. No medical history of the individual or family was recorded. We performed an excision biopsy of the postauricular mass which revealed follicular hyperplasia with small vessel hyperplasia, diffuse infiltration of eosinophils in lymphoid follicles, and several eosinophilic microabscesses. After a comprehensive review, the final diagnosis for this patient was KD and atopic dermatitis comorbidity. Conclusion: KD is not limited to males, as this report demonstrated. The histopathological examination plays an important role in the diagnosis of KD. This case illustrated the characteristic description of KD and highlights the need for awareness of this rare disease in Asian women.

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Section: Discussionmentioning

confidence: 99%

“…Systemic associations include nephrotic syndrome, eczema, asthma, sinusitis, tuberculosis, and Loe er syndrome [10][11][12]. Typically, the kidneys and skin are affected; the incidence of renal pathology ranges from 10-60% [7,13,14].…”

Section: Discussionmentioning

confidence: 99%

Kimura Disease: A Rare Case in Vietnamese Woman

Le,

Tran,

Pham

2023

Preprint

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Kimura's disease

Matchin¹,

Matz²

2023

Kazan Med J

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The analysis of published clinical observations on the diagnosis and treatment of patients with Kimura's disease, which is a rather rare chronic inflammatory disease of human soft tissues of unknown etiology, is presented. The onset of Kimura's illness is hardly noticeable. The disease proceeds sluggishly, with a varied clinical picture, most often with the formation of single or multiple subcutaneous nodules or tubercles and swelling in the head and neck on one or both sides. Other localizations of lesions are also described: limbs, orbit, eyelids, palate, pharynx, armpit, chest, inguinal region. The affected areas gradually increase in size over many months or years. Other clinical manifestations of Kimura's disease are presented in the form of edema of the extraocular muscle, juvenile temporal arteritis, sleep apnea in cases of damage to the larynx, and life-threatening coronary artery spasm in children. There are reports of other rare systemic clinical signs of Kimura's disease in the form of pruritus, eczema and rash. The disease is often associated with regional lymphadenopathy and/or salivary gland involvement, eosinophilia, and an increase in serum immunoglobulin E from 800 to 35,000 IU/mL. The disease may be accompanied by kidney damage in the form of glomerulonephritis and nephrotic syndrome. Most reported cases of Kimura's disease occur in young Asian men in their 20s and 30s, sporadically the disease is diagnosed in Europeans, North Americans, Caucasians, and Africans. Diagnosis of Kimura's disease is often clinically difficult. It is primarily based on excisional biopsy of the lesion or regional lymph nodes, and the histopathological findings are consistent regardless of the location of the lesion. Morphologically, Kimura's disease is characterized by the presence in biopsy specimens of such histological features as reactive follicular hyperplasia with prominent follicles surrounded by fibrous tissue. Interfollicular infiltrates are rich in plasma cells and eosinophils, which form diffuse eosinophilic microabscesses associated with granulomatous inflammation. The vessels are numerous, with an increase in postcapillary venules. At the same time, the blood vessels remain thin-walled, with cuboidal endothelial cells. Treatment issues are highlighted, and differences between Kimura's disease and angiolymphoid hyperplasia with eosinophilia are presented.

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Dupilumab combined with corticosteroid therapy for Kimura disease with multiple systemic masses: a case report and literature review

Lyu,

Cui,

et al. 2024

Front. Immunol.

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To date, the pathogenesis of Kimura’s disease remains unclear, there is no unified diagnostic criterion, the clinical phenotype shows considerable heterogeneity, and there is a lack of optimal treatment strategies. Due to its rarity, treatment strategies for KD are still under exploration. This paper reports a case of a 37-year-old Chinese female presenting with generalized erythematous papules and pruritic eruptions for 12 years, followed by the onset of limb swellings 3 years later, ultimately diagnosed as Kimura’s disease. Considering the patient’s multiple lymphadenopathies and limb swellings with concurrent atopic dermatitis, the treatment regimen included initial dupilumab dosage of 600 mg (300 mg administered in two injections), followed by subcutaneous injections of 300 mg every two weeks for four months. Concurrent oral corticosteroid therapy (methylprednisolone, initial dose 16 mg/kg/day, gradually tapered with tumor regression) was also administered. Following treatment, the patient did not experience severe adverse effects, and the multiple nodules markedly decreased in size. Additionally, serum IgE levels, eosinophil, and basophil counts showed significant reductions. These results demonstrate the significant efficacy of dupilumab combined with oral corticosteroids in treating Kimura’s disease with concurrent atopic dermatitis.

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Kimura disease with Allergic Bronchopulmonary Aspergillosis: a case report

Cited by 4 publications

References 21 publications

Kimura Disease: A Rare Case in Vietnamese Woman

Kimura Disease: A Rare Case in Vietnamese Woman

Kimura's disease

Dupilumab combined with corticosteroid therapy for Kimura disease with multiple systemic masses: a case report and literature review

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