Kimura's disease involving bilateral lacrimal glands and extraocular muscles along with ipsilateral face: A unique case report

Chakraborti, Chandana; Saha, Ajoy K.; Bhattacharjee, Ankita; Lakra, Rupanjili

doi:10.4103/ijo.ijo_810_19

Cited by 13 publications

(4 citation statements)

References 10 publications

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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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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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“…The prominent eosinophilic component of the infiltrate in the current case prompted the consideration of Kimura's disease, which may involve the extraocular muscles and orbit, [7][8][9][10] and was suggested by the high total serum IgE levels. Elevated IgE is, however, nonspecific and may be associated with allergic disease, primary immunodeficiencies, infections, malignancies, and other inflammatory disorders.…”

Section: Discussionmentioning

confidence: 99%

Myositis of an Extraocular Muscle, a Possible Drug Reaction: Histopathologic and Immunopathologic Analysis

Charles

Rebhun²,

Lidder³

et al. 2021

Ophthalmic Plastic &Amp; Reconstructive Surgery

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A 58-year-old man presented with left-sided orbital inflammation, including chemosis and a lateral rectus abduction defect. Initially presumed to represent cellulitis, the condition responded poorly to oral and intravenous antibiotics. CT showed the epicenter of an infiltrate to involve the lateral rectus. The patient improved dramatically when oral prednisone was added. Lateral rectus biopsy displayed intramuscular polyclonal lymphoid infiltrates, rich with eosinophils. Complete resolution of the inflammatory process was confirmed by a follow-up CT. The presumptive diagnosis was idiopathic orbital myositis, an uncommon condition of unknown etiology. However, the patient had taken rosuvastatin, which has been rarely associated with diplopia and ophthalmoplegia, raising the question of whether this case was truly idiopathic.

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“…[ 39 ] 1 2014 Chinese Male 53 1.7 × 1.1 × 1.1 neck surgical excision no 1.01 × 10 9 / L 537.2 recurrent / 68 Hsu, S. N. [ 40 ] 1 2015 Chinese Male 33 / head, lower extremity (edema) surgical excision, PTA, Cilostazol 50 mg, Pentoxifylline 400 mg, prednisolone 1 mg/kg/day, Cetirizine 5 mg twice a day no 34 to 51% 12,400-17,200 recurrent / not clear Ye, X. [ 41 ] 1 2015 Chinese Male 47 5 in diameter cervical, subaxillary and inguinal region prednisone 0.5 mg/kg/day, thalidomide 50 mg/day no 26.11% 1081.34 recurrent tapering of prednisolone 36 Wang, H. [ 42 ] 1 2016 Chinese Male 72 / head surgical excision, Chinese herbal remedies, cetirizine hydrochloride and olmesartan 20 mg/day, gamma immunoglobulin 10 g/day for 5 days, intravenous pulse methylprednisolone therapy 500 mg for 3 days, hydroxychloroquine 0.4 g/day, prednisone 50 mg/day, a single dose of intravenous cyclophosphamide 500 mg yes 35% 149,000 recurrent / 43 Matsuo, T. [ 11 ] 1 2017 Japanese Male 42 / head surgical excision, prednisone 40 mg/day, cyclosporine 75 mg/day yes 9.40% 735 recurrent tapering of prednisolone 82 Chakraborti, C. [ ...…”

Section: Discussionmentioning

confidence: 99%

Kimura’s disease mimicking thoracic spine dumbbell neurogenic tumor: a case report and literature review

2020

BMC Surg

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Background Kimura’s disease is a rare, benign chronic inflammatory disease of unknown etiology that mostly affects Asians. The disease typically presents as subcutaneous masses in the head or neck region that are predominantly found in the preauricular and submandibular areas. Case presentation A 7-year-old boy presenting with paralysis of both lower extremities and a thoracic spine dumbbell mass was initially diagnosed with a neurogenic tumor, but the pathological and laboratory examinations confirmed the diagnosis of Kimura’s disease. The paralysis symptom disappeared rapidly, but the patient had developed a recurrent mass in the cervical vertebral canal at the 9-month follow-up. Conclusion To our knowledge, no prior published literature has revealed Kimura’s disease cases that mimic dumbbell neurogenic tumors. Here, we report such a case of Kimura’s disease for the first time and provide a brief review of the literature.

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Kimura's disease involving bilateral lacrimal glands and extraocular muscles along with ipsilateral face: A unique case report

Cited by 13 publications

References 10 publications

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

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

Myositis of an Extraocular Muscle, a Possible Drug Reaction: Histopathologic and Immunopathologic Analysis

Kimura’s disease mimicking thoracic spine dumbbell neurogenic tumor: a case report and literature review

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