Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A

Sugimoto, Kunihisa; Enya, Takuji; Morimoto, Yuichi; Oshima, Rina; Miyazaki, Katsumi; Okada, Mitsuru

doi:10.1186/s13223-021-00549-y

Cited by 4 publications

(2 citation statements)

References 28 publications

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“…11,12 This autoimmune mechanism can impacts the development of KD’s clinical manifestations and possibly accounts for the observed efficacy of immunosuppression therapy. Bilateral lacrimal gland enlargement is an infrequent orbital manifestation of pediatric KD, 13 and its association with a lateral ventricular lesion of the brain, to the best of our knowledge, has never been reported. In fact, the only published cases of central nervous system involvement in KD are represented by brain embolis.…”

Section: Discussionmentioning

confidence: 79%

Bilateral Dacryoadenitis and Central Nervous System Involvement in a Child With Kimura Disease

Camacho¹,

Chahud²,

Boasquevisques³

et al. 2022

Ophthalmic Plastic &Amp; Reconstructive Surgery

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Section: Discussionmentioning

confidence: 79%

Bilateral Dacryoadenitis and Central Nervous System Involvement in a Child With Kimura Disease

Camacho¹,

Chahud²,

Boasquevisques³

et al. 2022

Ophthalmic Plastic &Amp; Reconstructive Surgery

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“…Table 1 summarizes the clinical features of patients with periorbital KD. 3–38 The average age of patients was 31.9 years (standard deviation: 17.8 years). A total of 81.1% (30/37) of identified patients were male.…”

Section: Resultsmentioning

confidence: 99%

Kimura Disease of the Periorbita and Earlobe in a 15-Year-Old Male: A Case Report and Review of the Literature

Han

Dermarkarian

Estopinal³

et al. 2022

Ophthalmic Plastic &Amp; Reconstructive Surgery

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Purpose: The authors describe a case of Kimura disease (KD) affecting the lacrimal gland, the ipsilateral periorbita, and the contralateral earlobe. The authors also review the literature for characteristics and outcomes of KD affecting the periorbita or earlobe. Methods: The authors review retrospectively a single chart and abstract data from the English language literature. Results: A 15-year-old Samoan male presented with a left upper eyelid mass and later the contralateral earlobe and ipsilateral periorbita. Surgical resection was partially effective and normal vision and eye motility were maintained. Histopathology was consistent with KD. Thirty-seven cases of periorbital KD were identified in the literature. The average age of patients with periorbital KD was 31.9 years (standard deviation: 17.8 years), with 81.1% (30/37) of patients being male and 69.4% (25/36) Asian. Over half of periorbital KD patients had lacrimal gland involvement (51.3%; 19/37). Fifteen patients were initially treated with surgery; 3 (20%) had recurrence of the disease. Another 15 patients were initially treated with corticosteroids; 12 (80%) had recurrence of the disease. Ophthalmologic data, when reported, demonstrated that, most patients had no visual (77.8%; 14/18 patients) or motility disturbances (82.4%; 14/17 patients) but most had proptosis 71.4% (10/14). Only 4 cases of earlobe involvement in KD were identified, all in women. Conclusions: In the literature, orbital KD was rare but often affected the lacrimal gland and caused proptosis, commonly in young adult Asian males. Vision and extraocular motility were usually normal. Earlobe involvement was very rare. This unique case of KD that affected the orbit and contralateral earlobe corroborates the prior literature that KD is prone to recurrence but may not adversely affect vision or extraocular motility.

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Orbital Kimura disease: maintenance therapy using mycophenolate mofetil

Fenech,

Ajanaku,

McCormick

et al. 2024

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Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A

Cited by 4 publications

References 28 publications

Bilateral Dacryoadenitis and Central Nervous System Involvement in a Child With Kimura Disease

Bilateral Dacryoadenitis and Central Nervous System Involvement in a Child With Kimura Disease

Kimura Disease of the Periorbita and Earlobe in a 15-Year-Old Male: A Case Report and Review of the Literature

Orbital Kimura disease: maintenance therapy using mycophenolate mofetil

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