Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A
Abstract Background Kimura’s disease (KD) is a rare chronic inflammatory disease of unknown etiology. Clinically, KD is characterized by nodular subcutaneous masses, that are typically localized to the neck and head. Involvement of the lacrimal glands and limbs is uncommon and seldom reported. Case...
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| Format: | Article |
| Language: | English |
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BMC
2021-05-01
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| Series: | Allergy, Asthma & Clinical Immunology |
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| Online Access: | https://doi.org/10.1186/s13223-021-00549-y |
| _version_ | 1818832568440586240 |
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| author | Keisuke Sugimoto Takuji Enya Yuichi Morimoto Rina Oshima Kohei Miyazaki Mitsuru Okada |
| author_facet | Keisuke Sugimoto Takuji Enya Yuichi Morimoto Rina Oshima Kohei Miyazaki Mitsuru Okada |
| author_sort | Keisuke Sugimoto |
| collection | DOAJ |
| description | Abstract Background Kimura’s disease (KD) is a rare chronic inflammatory disease of unknown etiology. Clinically, KD is characterized by nodular subcutaneous masses, that are typically localized to the neck and head. Involvement of the lacrimal glands and limbs is uncommon and seldom reported. Case presentation We report a case of a 4-year-old Japanese boy presenting with bilateral upper eyelid swelling with nodular subcutaneous lesions and peripheral eosinophilia. Based on clinical, histopathological, and laboratory findings, the patient was diagnosed with KD. An itchy subcutaneous mass on the left arm developed at the age of 14 years. Treatment with steroids was effective. However, as the steroids were tapered after the patient developed side effects, the masses relapsed within a few months. Treatment with cyclosporine A was then initiated, which led to an improvement of clinical features and serial levels of cytokines. Conclusions We report a rare case of KD with a peculiar clinical presentation. The patient responded well to treatment with cyclosporine A. |
| first_indexed | 2024-12-19T02:05:06Z |
| format | Article |
| id | doaj.art-e6234eafe5ed41e89e93d005c273f577 |
| institution | Directory Open Access Journal |
| issn | 1710-1492 |
| language | English |
| last_indexed | 2024-12-19T02:05:06Z |
| publishDate | 2021-05-01 |
| publisher | BMC |
| record_format | Article |
| series | Allergy, Asthma & Clinical Immunology |
| spelling | doaj.art-e6234eafe5ed41e89e93d005c273f5772022-12-21T20:40:57ZengBMCAllergy, Asthma & Clinical Immunology1710-14922021-05-011711610.1186/s13223-021-00549-yKimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine AKeisuke Sugimoto0Takuji Enya1Yuichi Morimoto2Rina Oshima3Kohei Miyazaki4Mitsuru Okada5Department of Pediatrics, Kindai University Faculty of MedicineDepartment of Pediatrics, Kindai University Faculty of MedicineDepartment of Pediatrics, Kindai University Faculty of MedicineDepartment of Pediatrics, Kindai University Faculty of MedicineDepartment of Pediatrics, Kindai University Faculty of MedicineDepartment of Pediatrics, Kindai University Faculty of MedicineAbstract Background Kimura’s disease (KD) is a rare chronic inflammatory disease of unknown etiology. Clinically, KD is characterized by nodular subcutaneous masses, that are typically localized to the neck and head. Involvement of the lacrimal glands and limbs is uncommon and seldom reported. Case presentation We report a case of a 4-year-old Japanese boy presenting with bilateral upper eyelid swelling with nodular subcutaneous lesions and peripheral eosinophilia. Based on clinical, histopathological, and laboratory findings, the patient was diagnosed with KD. An itchy subcutaneous mass on the left arm developed at the age of 14 years. Treatment with steroids was effective. However, as the steroids were tapered after the patient developed side effects, the masses relapsed within a few months. Treatment with cyclosporine A was then initiated, which led to an improvement of clinical features and serial levels of cytokines. Conclusions We report a rare case of KD with a peculiar clinical presentation. The patient responded well to treatment with cyclosporine A.https://doi.org/10.1186/s13223-021-00549-yKimura’s diseaseCyclosporinePediatricsLacrimal gland |
| spellingShingle | Keisuke Sugimoto Takuji Enya Yuichi Morimoto Rina Oshima Kohei Miyazaki Mitsuru Okada Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A Allergy, Asthma & Clinical Immunology Kimura’s disease Cyclosporine Pediatrics Lacrimal gland |
| title | Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A |
| title_full | Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A |
| title_fullStr | Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A |
| title_full_unstemmed | Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A |
| title_short | Kimura’s disease with recurrent bilateral lacrimal gland involvement in a male Japanese child successfully treated with cyclosporine A |
| title_sort | kimura s disease with recurrent bilateral lacrimal gland involvement in a male japanese child successfully treated with cyclosporine a |
| topic | Kimura’s disease Cyclosporine Pediatrics Lacrimal gland |
| url | https://doi.org/10.1186/s13223-021-00549-y |
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