Tubulointerstitial nephritis and uveitis (TINU) syndrome: a case report and review of the literature

Purpose: We aim to describe the first case of tubulointerstitial nephritis and uveitis (TINU) syndrome reported in Scandinavia and to underline the importance of the syndrome, which should be better known among ophthalmologists. Methods: We report an 11‐year‐old boy who was admitted to hospital because of general fatigue and weight loss. Blood tests showed renal insufficiency and a renal biopsy revealed acute tubulointerstitial nephritis. One week after admission the patient developed transitory blurred vision and flickering shadows in the left eye. Slit‐lamp examination revealed bilateral anterior non‐granulomatous uveitis and TINU syndrome was diagnosed. Results: Because of renal insufficiency the patient was treated with systemic prednisone 50 mg/day for 3 weeks. Evolution was favourable, and prednisone was tapered over 10 weeks. Uveitis was treated with topical steroid with good effect. The only complication after 18 months of follow‐up was transitory cushingoid aspect. Conclusions: Tubulointerstitial nephritis and uveitis syndrome is a rare syndrome that is probably underdiagnosed in clinical practice. Co‐operation between ophthalmologists and nephrologists/paediatricians is crucial for early diagnosis and instigation of treatment. Uveitis may occur before tubulointerstitial nephritis or the renal symptoms may be so mild that the patient is unaware of them. Therefore, ophthalmologists play an important role in the initial discovery of patients with TINU syndrome. Tubulointerstitial nephritis tends to be self‐limiting, whereas uveitis tends to relapse and recurrences tend to be more severe than the initial uveitis. Therefore, the patient must be ophthalmologically monitored very carefully.     

The intermediate uveitis with systemic symptoms: a case report]

The case of 26-year old male patient with typical clinical intermediate uveitis (vitritis, periphlebitis) with accompanying leucopenia, bradycardia and demyelination focal areas in brain of unknown etiology is presented. The asymptomatic periphlebitis was also found in the eyes of his twin brothers (30 years old) and sister (20 years old).     

Syphilitic uveitis in HIV-positive patients: report of a case series, treatment outcomes, and comprehensive review of the literature

AIM: To report the clinical characteristics, treatment and outcomes of active syphilitic uveitis in human immunodeficiency virus (HIV) positive patients and compare them with the previously published data. METHODS: Retrospective analysis of the case series from an infectious disease center in southern China was conducted. Comprehensive review of previously published cases of HIV positive syphilitic uveitis was conducted using the PubMed and Web of Science databases and the references listed in the identified articles. RESULTS: Twelve HIV positive patients with active syphilitic uveitis were collected. All were male, with age of 36.3y (range 27 to 53y). Five (41.7%) had a history of syphilis, and three of them had received anti-syphilis treatment. Ocular manifestations included corneal epithelial defect (13%), complicated cataract (17.4%), vitreous opacity (82.6%), optic disc edema (26.1%), macular edema (30.4%), neuro-retinitis (43.5%), and retinal hemorrhage (26.1%). After standardized syphilitic treatment, intraocular inflammation was reduced and vision improved in all cases. The literature review summarizes 105 previously reported cases of HIV positive syphilitic uveitis. High serum rapid plasma regain (RPR) titers may be associated with severe uveitis and poor vision. Treatment with penicillin, ceftriaxone sodium, or penicillin plus benzylpenicillin instead of using benzylpenicillin alone can significantly improve best-corrected visual acuity (BCVA) in HIV positive ocular syphilis patients. CONCLUSION: For HIV positive syphilitic uveitis patients, prompt diagnosis and appropriate treatment and follow-up are paramount. In our series, the clinical manifestations are diverse. Syphilis patients treated by penicillin G or long-acting penicillin before may still develop syphilitic uveitis. Patients who relapse after long-term penicillin treatment can still benefit from penicillin G.     

Solar retinopathy: a review of the literature and case report

Accounts of solar retinopathy have existed for centuries, but only recently have researchers begun to investigate the mechanisms responsible for producing solar retinal injury. The vast majority of solar retinal injuries occur as a result of viewing a solar eclipse without adequate protection. The extent of structural retinal damage and associated visual impairment is dependent upon the intensity and duration of solar exposure. The exact mechanisms which operate to produce solar retinal compromise are not completely known, but is believed to involve a thermally enhanced photochemical process. Despite the lack of a standardized treatment protocol, most cases of solar retinopathy will improve significantly over time without treatment. Prevention remains the mainstay of therapy. Solar retinopathy must be differentiated from other subtle macular diseases in the absence of a confirmed history of solar exposure.     

Eyelid lipomas: a case report and review of the literature

Simple lipomas of the eyelid are rare. We present a case of a 61-year-old man, who presented with 6 months of a slowly worsening blepharoptosis. On examination, that patient was noted to have a palpable, soft mass in the medial left upper eyelid. Histopathological examination of the mass revealed mature adipose tissue most consistent with lipoma. Simple lipomas of the eyelid are very unusual but should be considered in the differential diagnosis of patients presenting with mechanical ptosis.     

Xylene keratopathy: a case report and review of the literature

PURPOSE: To report a case of direct contact xylene-related vacuolar keratopathy. METHODS: Interventional case report and review of the literature. RESULTS: Xylene keratopathy is a distinct clinical entity characterized by significantly decreased visual acuity, increased corneal thickness, and the presence of corneal epithelial and stromal vacuoles on a diffuse gray stromal background. The vacuoles are diffusely scattered, subtle, and clear on slit-lamp examination. Occasionally, white, round, denser areas can also be observed. Xylene keratopathy disappeared gradually over 4 weeks with topical steroid treatment and did not cause permanent sequelae. CONCLUSIONS: Xylene exposure should be considered as possible etiologic agent of vacuolar epithelial and stromal keratopathy. Exposure may occur directly as a splash of liquid or through its vapors.     

Optic neuropathy following orbital irradiation for Graves' ophthalmopathy: a case report and literature review

Radiation optic neuropathy (RON) is a devastating complication of radiotherapy to the anterior visual pathway resulting in acute, profound and sometimes irreversible visual loss. Cumulative doses of radiation that exceed 50 Gy or radiation fractions of greater than 2 Gy are usually required for RON to develop. Several factors, such as diabetes and pre-existing compression of the optic nerve, are associated with a higher risk for developing RON at lower doses of radiation. We report a case of presumed subacute RON following orbital irradiation for Graves' ophthalmopathy, successfully treated with megadoses of intravenous corticosteroids.     

Orbital leiomyoma: report of a case and review of the literature

A 10-year-old female presented with slowly progressive proptosis of 1 year's duration. Orbital MRI revealed an extraconal oval mass occupying the lateral half of the mid and posterior orbit. The tumor was isointense to the extraocular muscle and cerebral gray matter on T1-weighted images and hyperintense on T2-weighted images. The patient underwent superolateral orbitotomy, and it was felt that the tumor was totally excised in a piecemeal fashion. Histopathologic examination showed that the tumor was composed of spindle cells arranged in a fibrous stroma rich in dilated sinusoidal capillaries. The nuclei of tumor cells were oval with blunted ends. There were no mitotic figures. Immunohistochemically, the tumor stained positive with smooth muscle actin, desmin, and vimentin. Based on the histopathologic and immunohistochemical staining results, a diagnosis of orbital leiomyoma was made. At 34 months follow-up, the patient continues to have 20/20 vision and is free of tumor recurrence clinically. Orbital leiomyoma is an uncommon tumor. Search of the English literature showed only 15 previous cases of orbital leiomyoma that have been published since 1963.     

Dorzolamide-induced immune thrombocytopenia: a case report and literature review

PURPOSE: To report a severe case of dorzolamide-induced immune thrombocytopenia, to review the literature on this topic, and to draw attention to the serious potential side effects of this topical sulfonamide. CASE REPORT: An 83-year-old man with primary open-angle glaucoma in both eyes who was taking topical dorzolamide therapy for 3.5 years developed a severe thrombocytopenia (10,000 platelets/microL). The amount of platelets was not influenced by prednisone therapy but increased rapidly to 100,000/microL after the withdrawal of dorzolamide. LITERATURE REVIEW: Although the potential capacity of sulfonamides to induce thrombocytopenia is well known, no case of dorzolamide-induced immune thrombocytopenia was published in the medical literature until July 2000. CONCLUSION: Considering the possible severe side effects that can be induced by topical dorzolamide 2%, ophthalmologists should carefully evaluate during the medical history of their patients the risk of sensitivity to sulfonamides.     

Gaze-evoked nystagmus: a case report and literature review.

BACKGROUND: A sustained gaze-evoked nystagmus (GEN) is an important ocular finding that may indicate serious neurologic pathology. It is also a finding that can be missed easily during routine extraocular muscle (EOM) testing. This report presents a case that should familiarize the reader with GEN and presents a novel approach to testing EOM function. CASE REPORT: The mother of an otherwise healthy 4-year-old girl noted that her daughter's eyes crossed occasionally, the right lid drooped on one occasion, and she had been having strange headaches. An asymmetric, sustained, gaze-evoked nystagmus was detected using a different approach to EOM testing. Magnetic resonance imaging found a large, brainstem astrocytoma in the cerebellar-pontine angle. CONCLUSION: EOM function often is overlooked or underperformed but is an important part of the battery of clinical tests to rule out neurologic problems. Most forms of EOM testing will check for muscle palsies but little else. If the time is taken to extend the patient's gaze to the extreme ends, to attempt to hold the gaze in all 9 positions, and to maintain an accurate speed, the clinician can stand to gain much more information regarding the neurologic system.     

Aarskog syndrome: a case report and literature review.

BACKGROUND: Aarskog syndrome (facial-digital-genital syndrome) is an X-linked inherited disorder that causes multiple limb and genital abnormalities. Although ophthalmic manifestations are noted rarely, findings may include optic nerve hypoplasia, retinal vessel tortuosity, deficient ocular elevation, hyperopia, and anisometropia. CASE REPORT: An 8-year-old boy with Aarskog syndrome presented with complaints of letter reversals, letter additions, and an occasional "crooked" eye when he became frustrated or tired. He was currently enrolled in a special education program because of poor academics. The examination found anisometropic amblyopia, superior ophthalmoplegia, and high hyperopic astigmatism. Glasses were prescribed, and further binocular, accommodative and perceptual testing will be performed once adaptation has occurred. CONCLUSION: Knowledge of this rare condition can benefit the practitioner as well as the patient. An understanding of the associated conditions will aid and simplify the examination process. A search of the English-language literature is reported.     

Electric cataract: a case report and review of the literature.

A case of electrically induced cataract in both eyes in a 12-year-old boy, after a high-voltage electric shock, is reported. He sustained skin burns on the neck, chest, abdomen, and inner left arm. The cataract developed first in the left eye and later on in the right eye. The child regained normal vision in both eyes after cataract extraction and aphakic correction with spectacles. The need for awareness of the possibility of this complication and screening of all cases of electrical injuries is stressed. The majority of cases respond well to surgery, but final visual acuity will depend on the other ocular damage due to electrical current. The clinical features and pathogenesis of this condition are briefly reviewed.     

Intracranial inflammatory pseudotumor: a case report and review of the literature

Purpose: To describe a rare case of intracranial inflammatory pseudotumor. Case Report: We describe a case of primary intracranial pseudotumor presenting with ophthalmoplegia. Seventeen well-documented cases reported in the literature are summarized. Conclusion: Inflammatory pseudotumor may present to the neuro-ophthalmologist as a visual field defect, monocular visual loss, or diplopia and may be associated with a monoclonal or polyclonal gammopathy.     

Bacterial subretinal abscess: a case report and review of the literature

PURPOSE: To report a case of Klebsiella subretinal abscess with a successful visual outcome with treatment and to review the literature pertaining to focal intraocular infection in bacterial endophthalmitis. METHODS: Clinical data including medical history, findings on physical examination, blood cultures, and an abdominal computed tomographic scan were collected in a 32-year-old man with Klebsiella sepsis, liver abscesses, and a focal subretinal abscess. Ocular data including visual acuity, fundus photographs, fluorescein angiography, and ultrasound were evaluated, as were results of culture and histopathologic studies. RESULTS: Despite immediate intervention, including vitreous tap and intravitreal antibiotics, the eye deteriorated, with enlargement of the abscess. A pars plana vitrectomy was performed in which the subretinal abscess material was removed after an extensive retinectomy of the involved area in association with an endophotocoagulative barrier and intravitreal amikacin without gas or oil tamponade. Culture confirmed Klebsiella subretinal infection. A retinal detachment occurred 1 month postoperatively and was successfully repaired. Visual acuity was 20/30 and has remained stable for 14 months. CONCLUSION: Klebsiella endophthalmitis with subretinal abscess formation is a rare but devastating ocular condition. In the present case, prompt intervention with extensive retinectomy, complete abscess excision, and intravitreal antibiotic therapy resulted in unprecedented visual recovery.     

Sarcoid granuloma simulating amelanotic melanoma of the iris: a case report

We report the case of a unilateral vascular iris tumor in a 24-year-old patient. Clinically, the iris lesion appeared similar to amelanotic melanoma. A systemic work-up (initial check-up), including an angiotensin-converting enzyme determination, provided the diagnosis of sarcoid granuloma. A biopsy confirmation was not needed. We concluded that iris granuloma can be the only ocular manifestation of sarcoidosis and this context is likely to be confused with amelanotic melanoma and metastatic carcinoma.