Bilateral orbital Kimura’s disease: A case report and brief review of literature

Agrawal, Sahil; Sen, Seema; Sabu, Shilpa; Modaboyina, Sujeeth; Bajaj, Mandeep S; Das, Deepsekhar

doi:10.4103/ijo.ijo_2648_21

Cited by 6 publications

(7 citation statements)

References 11 publications

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“…Infectious etiologies have been postulated, but it is now thought that KD is related to an autoimmune or delayed-type hypersensitivity reaction. The association with asthma, allergic rhinitis and conjunctivitis, atopic dermatitis, peripheral hypereosinophilia, and elevated serum IgE levels supports the idea of an aberrant allergic response [4]. Some have suggested a role for interleukins (IL-4 and IL-5) and mast cells in regulating immunoglobulin (IgE) synthesis and eosinophil infiltration [2].…”

Section: Introductionmentioning

confidence: 81%

Kimura's Disease: A Literature Review Based on a Clinical Case

Brito,

Baptista,

Pereira

et al. 2023

Cureus

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Kimura's disease (KD) was first described in 1937. It is a rare, benign, and chronic immune-mediated inflammatory disorder affecting the subcutaneous tissue, salivary glands, and lymph nodes. The disease is more common in the second to third decades of life in middle-aged Southeast Asian countries. The cause of Kimura's disease remains unknown; some authors believe it is related to an autoimmune or delayed-type hypersensitivity reaction. It commonly presents as a solitary painless lymph node in the head and neck or generalized lymphadenopathy (67%-100%) associated with peripheral eosinophilia and elevated IgE levels. Renal involvement may occur in some patients. Diagnosis is made by histology.A 21-year-old Caucasian man with no relevant medical history presented with a non-tender swelling of the left hemiface without other associated symptoms. Laboratory investigations revealed a leukocyte count with eosinophilia (2.29×10^9/L-26.5%) and elevated total IgE and IgG4. He had no renal dysfunction. He underwent surgical resection of the lesion in the jugal, infraorbital, and left nasolabial regions, and the anatomopathological examination revealed the characteristics of Kimura's disease. Three months after surgery, an MRI showed an increase in the size of the mass, and he was started on corticosteroids. Six months after surgery, he presented with a slight increase in the size of the mass and was started on Ciclosporin, which allowed a progressive reduction in the dose of corticosteroid with evidence of a progressive reduction in swelling. Ciclosporin was stopped due to toxic serum levels, and he was started on mycophenolate mofetil. The dose was increased because of the increase in facial mass; on mycophenolate mofetil 2500mg/day, the patient remains stable.KD is a chronic disorder of unknown etiology that mainly affects young people in Southeast Asia and is rare in Western countries, as in the case of this young man. Studies have shown no significant differences in region or race, complications, multiplicity, laterality, anatomical distribution, maximum size, eosinophil count, or IgE levels between age groups. There is no consensus on the optimal treatment for KD; several treatments have been used, including surgery, systemic corticosteroids, immunosuppressants, and radiation. Due to the tendency to relapse and the rarity of the disease, there is no consensus on treatment options for relapse.

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Section: Introductionmentioning

confidence: 81%

Kimura's Disease: A Literature Review Based on a Clinical Case

Brito,

Baptista,

Pereira

et al. 2023

Cureus

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“…[12] They claimed the two diseases were the same, with subcutaneous ALHE signifying any earlier stage that eventually developed into Kimura's disease. [12] Later research [13] , however, based on histopathological results, separated the disorders into two distinct categories. Kimura's disease is currently assumed to be an uncommon chronic inflammatory or allergic condition, and ALHE is considered a benign vascular neoplasm.…”

Section: Kimuramentioning

confidence: 99%

“…Kimura's disease is currently assumed to be an uncommon chronic inflammatory or allergic condition, and ALHE is considered a benign vascular neoplasm. [13] Although several studies have been done, the etiology and pathophysiology of Kimura's response are all possible etiologies. [14] In this case, the patient was a 40-year-old female with the chief complaint of a mass on her left nasal orbit without lymphadenopathy and systemic symptoms.…”

Section: Kimuramentioning

confidence: 99%

“…[7] Findings from CT with contrast enhancement usually show homogeneously enhancing non-specific lesions. [13] Based on literature studies [14] ,…”

Section: Kimuramentioning

confidence: 99%

“…[14] Kimura's disease is currently assumed to be an uncommon chronic inflammatory or allergic condition, and ALHE is considered a benign vascular neoplasm. [13] Several ranging from cuboidal to dome-shaped. [1], [7], [14], [16] In this case, the patient underwent surgical excision under general anesthetic, and the mass was obliterated.…”

Section: Kimuramentioning

confidence: 99%

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Kimura's Disease Finding on Ocular Adnexal Mass

Tiendie,

Fatmariyanti,

Ridholia

2024

VSEHJ

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Introduction: Kimura's disease is an unknown chronic lymphoproliferative inflammatory disease affecting the skin, soft tissues, and lymph nodes. Until September 2020, only 200 cases of Kimura's disease were reported worldwide, however, their exact incidence is unknown. Here, we are interested in reporting a patient with Kimura's disease of the ocular adnexa due to its rarity and to enhance the knowledge of ophthalmologists about confirming the diagnosis of Kimura's disease. Case Presentation: A 40-year-old female presented with a chief complaint of a mass on her left nasal orbit for the last year. The mass gradually increased for six months, and it was painless. The systemic laboratory workup revealed eosinophilia and increased serum IgE. Contrast CT-Scan revealed a benign tumor, suspect dermoid cyst. She underwent surgery, and the histopathologic showed Kimura's disease. The patient was followed up on a scheduled basis, and there was no recurrence during four months of monitoring. Conclusions: A clinical, systemic laboratory, and histopathological examination is required to confirm the diagnosis of Kimura's ocular adnexa disease and determine the best therapy for the patient due to the high recurrence rate. Combining surgical excision with postoperative radiation is recommended as the most effective treatment in terms of controlling the residual lesion and minimizing the recurrence rate while causing the fewest side effects. Eosinophil screening regularly is advised to evaluate the recurrence rate.

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