PURPOSE: To report a case of multiple solitary plasmacytomas that presented with painful erythematous swelling of the upper eyelid. The patient was diagnosed with extramedullary plasmacytoma, and was later found to have multiple metastases of the bone and soft tissue during follow up. CASE SUMMARY: A 55-year-old female patient presented with painful erythematous swelling of the left upper eyelid that persisted for 1 month prior to examination. Under suspicion of lacrimal gland inflammation, anti-inflammatory medication was started but the symptoms worsened. Orbital computed tomography showed that a mass infiltrated the left lacrimal gland. We performed incisional biopsy of the mass via eyelid crease incision. Based on histopathological examination, the mass was diagnosed as extramedullary plasmacytoma and the patient was treated with radiation. After a 10-month follow-up period, multiple metastases on the left parotid gland, thoracic spine, lumbar spine and pelvic bone were observed. Finally, we diagnosed the patient with multiple solitary plasmacytomas. CONCLUSIONS: We report the first case of multiple solitary plasmacytomas presenting with upper eyelid painful erythematous swelling. This condition should be considered for patients presenting with eyelid inflammation. In addition, long term follow up should be conducted to detect metastasis or recurrence.
Biopsy ; Eyelids* ; Female ; Follow-Up Studies ; Humans ; Inflammation ; Lacrimal Apparatus ; Middle Aged ; Neoplasm Metastasis ; Orbit ; Parotid Gland ; Pelvic Bones ; Plasmacytoma* ; Recurrence ; Spine

PURPOSE: Eruptive vellus hair cysts (EVHC) are benign lesions that affect the pediatric population and are rarely seen congenitally or in young adults. EVHCs are small, cystic papules that usually occur on the chest and proximal extremities. EVHCs of the eyelids have been reported infrequently. We experienced a case of solitary EVHC that developed on the eyelid in a middle-aged male. Herein, we present our case with a brief review of the literature. CASE SUMMARY: A 44-year-old male presented with a history of an asymptomatic, palpable mass in the right upper eyelid that had been slowly growing for 2 years. Physical examination revealed non-tender, firm and round mass in right upper eyelid. Computed tomography scan of the orbit showed a 7.0 x 9.0 x 9.5 mm-sized focal bulging contour in the right upper eyelid. The patient underwent sub-brow incision and excisional biopsy of the eyelid mass. Histopathological examination revealed a 7.0 x 6.0 x 4.0 mm-sized cystic structure lined by squamous epithelium containing laminated keratinous materials and multiple vellus hair shafts. There was no local recurrence during the postoperative follow-up. CONCLUSIONS: We experienced a rare case of EVHC that developed on the eyelid. Based on our experience, EVHC should be considered when determining an eyelid mass, especially around hair follicles.
Adult ; Biopsy ; Epithelium ; Extremities ; Eyelids* ; Follow-Up Studies ; Hair Follicle ; Hair* ; Humans ; Male ; Orbit ; Physical Examination ; Recurrence ; Thorax ; Young Adult

PURPOSE: The authors of the present study describe a rare case of angiolymphoid hyperplasia with eosinophilia (ALHE) of the eyelid. CASE SUMMARY: A 63-year-old male who was diagnosed with ALHE based on biopsy of an inguinal mass presented with an eyelid mass of 1 month duration. A light brown, solitary, 1.0 x 0.5 cm-sized mass involved the right upper eyelid. There was no lymphadenopathy, but eosinophilia was present. An excisional biopsy of the mass was performed for diagnosis and management. Macroscopic examination of the excised mass revealed a well-defined, smooth, firm, yellowish-red colored lesion measuring 1.0 x 0.6 x 0.5 cm. Histopathology showed the proliferation of small blood vessels, many of which were lined by enlarged endothelial cells with uniform ovoid nuclei and intracytoplasmic vacuoles. The distinctive endothelial cells were described as having a cobblestone appearance. In addition, a perivascular and interstitial infiltrate composed primarily of lymphocytes and eosinophils was present. ALHE was finally confirmed with clinical and microscopic examination. CONCLUSIONS: The authors of the present study report a rare case of ALHE of the eyelid and suggest that a differential diagnosis should be considered.
Angiolymphoid Hyperplasia with Eosinophilia ; Biopsy ; Blood Vessels ; Diagnosis, Differential ; Endothelial Cells ; Eosinophilia ; Eosinophils ; Eyelids ; Humans ; Light ; Lymphatic Diseases ; Lymphocytes ; Male ; Middle Aged ; Vacuoles

PURPOSE: To report a case of isolated squamous cell carcinoma of the orbit. CASE SUMMARY: A 75-year-old man with over a 50 pack-year history of smoking presented discomfort and visual disturbance of the left eye for several months. His best-corrected visual acuity was 0.3, intraocular pressure was 9 mm Hg, and extraocular movements were normal. Slit-lamp examinations revealed no specific findings in the anterior segment in the left eye. However, retinal exams showed an oval-shaped, juxtapapillary mass-like lesion associated with retinal folding in the left eye. A huge, distorted echoic mass with an initial prominent spike and low-to-medium internal reflectivity with diminishing amplitude was observed on ocular ultrasonography. Enhanced CT revealed a highly-intense, irregular-circumscribed heterogeneous mass (2.0 x 2.0 x 1.5) in the superomedial quadrant of the left eye. Metastatic workups, including bone scan and CT of the head, neck, chest, and abdomen, were unremarkable. One week after the initial visit, the patient experienced pain and reduced visual acuity (light perception) in the left eye. Following the diagnosis, enucleation with tumor resection and hydroxyapatite implantation was performed. Histopathologic examination revealed a moderated-differentiated squamous cell carcinoma invading the sclera. The patient subsequently underwent radiation treatment and no evidence of recurrence was reported 6 months after surgery.
Abdomen ; Aged ; Carcinoma, Squamous Cell ; Durapatite ; Eye ; Head ; Humans ; Intraocular Pressure ; Neck ; Orbit ; Recurrence ; Retinaldehyde ; Sclera ; Smoke ; Smoking ; Thorax ; Visual Acuity

Purpose: To report a case of apocrine hidrocystoma in the lacrimal gland.Case summary: A 51-year-old woman presented with a left upper eyelid mass that occurred two years before presenting tohospital. Upon physical examination, a painless palpable mass on the left upper eyelid was observed. Based on slit-lamp examination,a pinkish mass with a well-circumscribed border on the left upper temporal conjunctiva was observed. Orbital computedtomography showed a well-defined contrast enhanced mass of 8 × 9 × 9 mm in the lacrimal lesion. The mass was removed completelyunder local anesthesia. Biopsy confirmed that the mass was an apocrine hidrocystoma. There was no recurrence at oneyear postoperatively. Conclusions This is the first report of an apocrine hidrocystoma in a lacrimal lesion in Korea. Apocrine hidrocystoma is rarelyfound on lacrimal gland lesions, but it should be considered in the differential diagnosis of a lacrimal gland mass.

PURPOSE: We report a case of hemorrhagic lymphangiectasia of the conjunctiva with a 360° connected circumference, which recovered spontaneously.CASE SUMMARY: A 44-year-old female patient presented with congestion of the right eye 1 day prior to her visit. There was no history of any systemic disease or trauma, but she had experienced relapses of the same episode three times before the visit. There were no accompanying symptoms such as decreased vision or pain. Blood analysis, orbital computed tomography, and angiographic findings showed no remarkable finding. Slit lamp examination showed circumferential lymphatic dilatation extending 360° under the conjunctiva of the eye at a distance of about 6 mm behind the limbus of the right eye, which was accompanied by intralymphatic bleeding. Irregular local lymphatic dilatations were observed on the bulbar conjunctiva at 4 and 8 o'clock of the left eye. The bleeding spontaneously resolved in about 2 weeks, but the translucent enlarged lymphatic vessels were still observed on slit lamp examination and anterior segment optical coherence tomography.CONCLUSIONS: This is the first reported case of hemorrhagic lymphangiectasia involving 360° of the bulbar conjunctiva in Republic of Korea.

PURPOSE: Desmoplastic trichilemmoma is a rare variant of trichilemmoma first described in 1990. Desmoplastic trichilemmoma on the eyelid has not been previously reported in Korea. We report our experience with a case of desmoplastic trichilemmoma of the eyelid. CASE SUMMARY: A 72-year-old male patient presented with a round mass on the upper eyelid, which was noticed 1 year prior and caused irritation and itching. Excisional biopsy of the mass was performed. Histopathological examination revealed findings suggestive of a desmoplastic trichilemmoma. There was no local recurrence during the 2-year follow-up. CONCLUSIONS: Desmoplastic trichilemmoma should be considered when determining the type of eyelid mass.
Aged ; Biopsy ; Eyelids ; Follow-Up Studies ; Humans ; Korea ; Male ; Pruritus ; Recurrence

PURPOSE: To present a rare case of idiopathic orbital myositis involving levator palpebrae superioris. CASE SUMMARY: A 27-year-old male presented with a 1-week history of redness, discomfort, swelling, and drooping of his left upper eyelid. A computed tomography scan showed isolated enlargement of the right superior rectus/levator muscle complex. On examination, there was a left blepharoptosis, although eye movements were normal. The authors treated the patient with 3rd-generation cephalosporin; however, after 3 days, the symptoms did not improve. Subsequently, the patient was diagnosed with idiopathic orbital myositis and treated with oral corticosteroids for 1 month; the symptoms gradually resolved. CONCLUSIONS: Idiopathic orbital myositis is a subtype of nonspecific orbital inflammation primarily involving the extraocular muscles. Although the exact cause of orbital myositis is unknown, an immune-mediated pathophysiologic mechanism appears to be one of the causes. Medial rectus myositis is the most common, and isolated levator muscle myositis is very rare. The authors of the present study reported a case of orbital myositis involving the levator palpebrae superioris which should be considered a differential diagnosis of blepharoptosis with eyelid swelling.
Adrenal Cortex Hormones ; Adult ; Blepharoptosis ; Diagnosis, Differential ; Eye Movements ; Eyelids ; Humans ; Inflammation ; Male ; Muscles ; Myositis ; Orbit ; Orbital Myositis

PURPOSE: To evaluate the association of apolipoprotein E (APOE) polymorphism and cataracts in the Korean population. METHODS: The present research included participants from a population-based study in Incheon, Korea. A sample of 126 adults genotyped for polymorphisms of APOE underwent a medical interview, an eye examination which included visual acuity testing, slitlamp cataract evaluation and fundus examination. The APOE polymorphism was determined using a polymerase chain reaction method. RESULTS: Eighty-eight participants (69.8%) were diagnosed with cataracts or had undergone cataract surgery in 1 or both eyes, and 38 participants (30.2%) demonstrated no signs of cataract. The frequencies of the APOE genotypes and alleles were not significantly different from the cataract and the control group. APOE epsilon2 carriers were less likely to have cataracts than non-epsilon2 carriers with an odds ratio of 0.367 which was almost statistically significant with the multiple logistic regression analysis (p = 0.052). CONCLUSIONS: There was no significant correlation of APOE genotype and cataracts. However, a slight negative association of APOE epsilon2 and cataracts were found in the Korean population.
Adult ; Alleles ; Apolipoprotein E2 ; Apolipoproteins ; Apolipoproteins E ; Cataract ; Eye ; Genotype ; Humans ; Korea ; Logistic Models ; Odds Ratio ; Polymerase Chain Reaction ; Visual Acuity

Purpose: To report a case of actinomycosis infection originating in the orbitonasal cavity that quickly invaded the cerebrum.Case summary: A-57-year-old male with a history of right tooth extraction 7 days before the first visit presented with right eyelid swelling and pain that had developed 4 days prior to the visit and had become increasingly worse. The best-corrected visual acuity was light perception. The intraocular pressure of the patient was 37 mmHg in the right eye. Eyelid abscess, subconjunctival hemorrhage, and severe chemosis was observed. Orbital computed tomography showed multiple air pockets and enhancing soft tissue along the periorbital, maxillary, and ethmoid sinus. Emergent endoscopic sinus surgery, lateral canthotomy, and intravenous antifungal treatment were performed due to suspicion of mucormycosis infection. Two days after treatment, exenteration combined with otolaryngology and neurosurgery were performed, as brain magnetic resonance imaging scans indicated that intraorbital lesions had invaded the dura and frontal sinus. On Day 8 of treatment, Actinomyces odontolyticus was identified and intravenous Ampicillin was administered. Despite systemic antibiotic treatment, the infection could not be controlled. The patient died 28 days after treatment due to persistent intracranial hemorrhage and brain edema. Conclusions For patients with severe eyelid swelling with a history of tooth extraction, actinomycosis infection should be considered. Delays in diagnosis and treatment of this infection could lead to serious consequences.