Refractory rhino-orbito-cerebral mucormycosis treated with intraconal amphotericin B / Mucormicose rino-orbito-cerebral refratária tratada com anfotericina B intraconal

ABSTRACT We report the case of a 46-year-old diabetic man receiving treatment for rhino-orbital-cerebral mucormycosis with liposomal amphotericin B and surgical debridement. The patient's condition worsened clinically, accompanied by the loss of ocular motility and a visual acuity of absence of light perception. Radiological extension of the infection was evidenced, with invasion of the cavernous sinus. Based on ophthalmological findings, exenteration (a potentially disfiguring procedure) was indicated, but we opted for wide surgical debridement and administration of amphotericin B via intraconal catheter. Clinical improvement and resolution of inflammation occurred after 2 weeks of treatment. Thus, rhino-orbital-cerebral mucormycosis was effectively controlled through intraconal administration of amphotericin B, while avoiding exenteration. The intervention should be considered as an adjuvant treatment in selected rhino-orbital-cerebral mucormycosis cases before attempting exenteration.

RESUMO Os autores relatam o caso de um homem diabético de 46 anos de idade em tratamento para mucormicose rino-orbital-cerebral com anfotericina B lipossomal e desbridamento cirúrgico. Piora clínica foi observada, com acometimento de motilidade ocular e acuidade visual de ausência de percepção luminosa, sendo evidenciada extensão radiológica da infecção, com invasão do seio cavernoso. Com base nos achados oftalmológicos, uma exenteração, procedimento potencialmente desfigurante, foi indicada, mas optamos por realizar amplo desbridamento cirúrgico e administração intraconal de anfotericina B por cateter. Melhora clínica e resolução da inflamação ocorreram após duas semanas de tratamento. Dessa forma, a mucormicose rino-orbital-cerebral foi efetivamente controlada com a administração intraconal de anfotericina B, evitando a exenteração. A intervenção deve ser considerada como um tratamento adjuvante em casos selecionados de mucormicose rino-orbital-cerebral antes que a exenteração seja realizada.

Refractory rhino-orbito-cerebral mucormycosis treated with intraconal amphotericin B.

We report the case of a 46-year-old diabetic man receiving treatment for rhino-orbital-cerebral mucormycosis with liposomal amphotericin B and surgical debridement. The patient's condition worsened clinically, accompanied by the loss of ocular motility and a visual acuity of absence of light perception. Radiological extension of the infection was evidenced, with invasion of the cavernous sinus. Based on ophthalmological findings, exenteration (a potentially disfiguring procedure) was indicated, but we opted for wide surgical debridement and administration of amphotericin B via intraconal catheter. Clinical improvement and resolution of inflammation occurred after 2 weeks of treatment. Thus, rhino-orbital-cerebral mucormycosis was effectively controlled through intraconal administration of amphotericin B, while avoiding exenteration. The intervention should be considered as an adjuvant treatment in selected rhino-orbital-cerebral mucormycosis cases before attempting exenteration.

Optical coherence tomography evaluation of retinal nerve fiber layer in longitudinally extensive transverse myelitis.

OBJECTIVE: To compare optical coherence tomography (OCT) measurements on the retinal nerve fiber layer (RNFL) of healthy controls and patients with longitudinally extensive transverse myelitis (LETM) without previous optic neuritis. METHOD: Twenty-six eyes from 26 patients with LETM and 26 control eyes were subjected to automated perimetry and OCT for comparison of RNFL measurements. RESULTS: The mean deviation values from perimetry were significantly lower in patients with LETM than in controls (p<0.0001). RNFL measurements in the nasal quadrant and in the 3-o'clock segment were significantly smaller in LETM eyes than in controls. (p=0.04 and p=0.006, respectively). No significantly differences in other RNFL measurements were found. CONCLUSION: Patients with LETM may present localized RNFL loss, particularly on the nasal side of the optic disc, associated with slight visual field defects, even in the absence of previous episodes of optic neuritis. These findings emphasize the fact that patients with LETM may experience attacks of subclinical optic nerve damage.

Optical coherence tomography evaluation of retinal nerve fiber layer in longitudinally extensive transverse myelitis / Avaliação da camada de fibras nervosas da retina na mielite transversal longitudinalmente extensa usando tomografia de coerência óptica

OBJECTIVE: To compare optical coherence tomography (OCT) measurements on the retinal nerve fiber layer (RNFL) of healthy controls and patients with longitudinally extensive transverse myelitis (LETM) without previous optic neuritis. METHOD: Twenty-six eyes from 26 patients with LETM and 26 control eyes were subjected to automated perimetry and OCT for comparison of RNFL measurements. RESULTS: The mean deviation values from perimetry were significantly lower in patients with LETM than in controls (p<0.0001). RNFL measurements in the nasal quadrant and in the 3-o'clock segment were significantly smaller in LETM eyes than in controls. (p=0.04 and p=0.006, respectively). No significantly differences in other RNFL measurements were found. CONCLUSION: Patients with LETM may present localized RNFL loss, particularly on the nasal side of the optic disc, associated with slight visual field defects, even in the absence of previous episodes of optic neuritis. These findings emphasize the fact that patients with LETM may experience attacks of subclinical optic nerve damage.

OBJETIVO: Comparar as medidas da camada de fibras nervosas da retina (CFNR) usando a tomografia de coerência óptica (TCO) em indivíduos normais e pacientes com mielite transversal longitudinalmente extensa (MTLE) sem episódio prévio de neurite óptica. MÉTODO: Vinte e seis olhos de 26 pacientes com MTLE e 26 olhos normais foram submetidos à campimetria computadorizada e TCO para comparação das medidas da CFNR. RESULTADOS: Valores do parâmetro desvio médio da campimetria computadorizada foram significativamente menores nos pacientes com MTLE do que nos controles (p<0,001). Medidas da CFNR no quadrante nasal e no segmento 3 horas foram significativamente menores nos olhos dos pacientes com MTLE do que nos olhos normais (p=0,04 e p=0,006, respectivamente). Não foi encontrada diferença significante nas outras medidas da CFNR avaliadas. CONCLUSÃO: Pacientes com MTLE podem apresentar perda localizada da CFNR, particularmente na região nasal do disco óptico, associada a defeitos discretos de campo visual, mesmo na ausência de episódio prévio de neurite óptica. Estes achados sugerem que pacientes com MTLE podem apresentar acometimento subclínico do nervo óptico.

Evaluation of retinal nerve fiber layer thickness measurements using optical coherence tomography in patients with tobacco-alcohol-induced toxic optic neuropathy.

Three patients with progressive visual loss, chronic alcoholism and tabagism were submitted to a complete neuro-ophthalmic examination and to retinal nerve fiber layer (RNFL) measurements using optical coherence tomography (OCT) scanning. Two patients showed marked RNFL loss in the temporal sector of the optic disc. However, a third patient presented RNFL measurements within or above normal limits, based on the Stratus-OCT normative database. Such findings may be due to possible RNFL edema similar to the one that may occur in the acute phase of toxic optic neuropathies. Stratus-OCT was able to detect RNFL loss in the papillomacular bundle of patients with tobacco-alcohol-induced toxic optic neuropathy. However, interpretation must be careful when OCT does not show abnormality in order to prevent diagnostic confusion, since overestimation of RNFL thickness measurements is possible in such cases.

Using frequency-doubling perimetry to detect optic neuropathy in patients with Graves' orbitopathy.

PURPOSE: to test the ability of frequency-doubling technology (FDT) perimetry to detect dysthyroid optic neuropathy (DON). METHODS: Fifteen eyes of 15 patients with DON and 15 healthy control eyes were studied. Eligible eyes had a diagnosis of DON based on visual field abnormalities on standard automated perimetry and had visual acuity better than 20/30. FDT testing was performed using both the C-20-5 screening test and the C-20 full-threshold test. Normal and DON eyes were compared with regard to FDT mean sensitivity. RESULTS: Sensitivity ranges were 40.0%-86.7% for the screening test, and 53.3%-100.0% (total deviation) and 20.0-93.3 (pattern deviation) for the C-20 threshold test. The corresponding specificity ranges were 86.7-100.0, 33.3-93.3, and 26.7-100.0, respectively. The best sensitivity/specificity ratios were for one abnormal point depressed <5% in the screening test (86.7%/86.7%), one point depressed <1% in the total deviation analysis (80.0%/86.7%), and one point depressed <2% in the pattern deviation analysis (80.0%/86.7%). DON eyes presented significantly lower than normal average sensitivity in the central, pericentral, and peripheral areas. CONCLUSIONS: FDT perimetry is a useful screening tool for DON in eyes with normal or only slightly reduced visual acuity.

Scanning laser polarimetry with enhanced corneal compensation for detection of axonal loss in band atrophy of the optic nerve.

PURPOSE: To compare the abilities of scanning laser polarimetry (SLP) with enhanced corneal compensation (ECC) and variable corneal compensation (VCC) modes for detection of retinal nerve fiber layer (RNFL) loss in eyes with band atrophy (BA) of the optic nerve. DESIGN: Cross-sectional study. METHODS: Thirty-seven eyes from 37 patients with BA and temporal visual field defect from chiasmal compression and 40 eyes from 40 healthy subjects were studied. Subjects underwent standard automated perimetry and RNFL measurements using an SLP device equipped with VCC and ECC. Receiver operating characteristic (ROC) curves were calculated for each parameter. Pearson correlation coefficients were obtained to evaluate the relationship between RNFL thickness parameters and severity of visual field loss, as assessed by the temporal mean defect. RESULTS: All RNFL thickness parameters were significantly lower in eyes with BA compared with normal eyes with both compensation modes. However, no statistically significant differences were observed in the areas under the ROC curves for the different parameters between GDx VCC and ECC (Carl Zeiss Meditec, Inc, Dublin, California, USA). Structure-function relationships also were similar for both compensation modes. CONCLUSIONS: No significant differences were found between the diagnostic accuracy of GDx ECC and that of VCC for detection of BA of the optic nerve. The use of GDx ECC does not seem to provide a better evaluation of RNFL loss on the temporal and nasal sectors of the peripapillary retina in subjects with BA of the optic nerve.

Diagnostic ability of optical coherence tomography with a normative database to detect band atrophy of the optic nerve.

PURPOSE: To evaluate the diagnostic ability of stratus optical coherence tomography (OCT) with a normative database to detect band atrophy (BA) of the optic nerve. DESIGN: Cross-sectional study. METHODS: StratusOCT retinal nerve fiber layer thickness scans were obtained in 37 eyes with BA and permanent temporal visual field defect and 37 healthy eyes. The categorization of eyes according to the normative database of the instrument and sensitivity and specificity values were reported. RESULTS: The average thickness parameter demonstrated the highest sensitivity for detection of abnormalities in eyes with BA, followed by the parameters related to the temporal and nasal quadrants. Values of sensitivity were relatively lower for the 30 degree segments. CONCLUSION: StratusOCT with a normative database performed well in detecting retinal nerve fiber layer (RNFL) loss in eyes with BA. Clinicians should be aware of possible detection failure in the 30 degree segmental analysis, particularly at the 3 o'clock meridian.

Evaluation of macular thickness measurements for detection of band atrophy of the optic nerve using optical coherence tomography.

PURPOSE: To evaluate the ability of optical coherence tomography (OCT) macular thickness parameters to differentiate between eyes with band atrophy (BA) of the optic nerve and healthy eyes. DESIGN: Cross-sectional study. PARTICIPANTS: The study included 1 eye of each of 40 consecutive patients with BA of the optic nerve and permanent temporal hemianopic visual field (VF) defects owing to chiasmal compression and 31 age- and gender-matched healthy subjects. METHODS: All patients underwent VF assessment with kinetic Goldmann perimetry and Humphrey 24-2 full-threshold standard automated perimetry (SAP). Macular and retinal nerve fiber layer (RNFL) thickness scans were obtained using the commercially available Stratus OCT. The severity of VF defect in patients with BA was evaluated by the temporal mean defect (TMD), calculated as the average of the 22 values of the temporal total deviation plot of the SAP 24-2 test, excluding the 2 points immediately above and below the blind spot. MAIN OUTCOME MEASURES: Receiver operating characteristic (ROC) curves and sensitivities at fixed specificities were calculated for each parameter. Spearman's rank correlation coefficients were used to evaluate the relationship between RNFL and macular thickness parameters and severity of VF loss as measured by the TMD. RESULTS: The macular thickness parameters related to the nasal hemiretina had the best performance to detect damage in BA eyes. No statistically significant difference (P = 0.19) was found between the ROC curve areas (AUCs) for the best macular thickness parameter (temporal/nasal macular thickness, AUC = 0.96) and the best RNFL parameter (average thickness, AUC = 0.99). Lower values of TMD, indicating more severe VF loss, were associated with lower macular thickness measurements. The highest correlation was observed for the parameter nasal average macular thickness (rho = 0.693, R2 = 48%, P<0.001). CONCLUSION: Eyes with BA of the optic nerve show significant thinning of the retinal thickness on the nasal macular area, which is associated with the severity of VF damage in these eyes. Macular thickness measurements could potentially be used to evaluate the amount of ganglion cell loss in patients with BA of the optic nerve and could prove clinically useful for detection of damage and for monitoring these patients.

Bilateral progressive enophthalmos as the presenting sign of metastatic breast carcinoma.

A 51-year-old woman, without systemic symptoms, complained of slowly progressive receding eyes. She had severe bilateral enophthalmos and markedly restricted extraocular movements in all fields of gaze. Computed tomography showed an infiltrative homogenous mass filling both orbits. Gynecologic and systemic investigation revealed bilateral lobular breast carcinoma, without metastasis to other organs. She was started on chemotherapy and 2 years after the diagnosis remains in good general health, despite persistence of severe enophthalmos. This case serves to emphasize that bilateral progressive enophthalmos may be the presenting sign of metastatic breast carcinoma even when local symptoms in the breast are absent. Clinicians should be aware of this sign to establish an early diagnosis and treatment.

Axonal loss after traumatic optic neuropathy documented by optical coherence tomography.

PURPOSE: To report longitudinal retinal nerve fiber layer (RNFL) thickness measurements using optical coherence tomography (OCT) in a patient with traumatic optic neuropathy. DESIGN: Observational case report. METHODS: A 14-year-old boy with severe optic nerve trauma had repeated OCT scans of the peripapillary retinal nerve fiber layer at 3 days, 20 days, 40 days, and 70 days after injury. RESULTS: There was gradual loss of nerve fibers as shown by the OCT color-coded map, RNFL thickness profile, and RNFL thickness measurements around the optic disk. At 70 days of follow-up, severe thinning of the RNFL was observable. CONCLUSIONS: These findings suggest that OCT is able to assess and monitor axonal loss after traumatic optic neuropathy.

Intraocular pressure fluctuations in medical versus surgically treated glaucomatous patients.

The purpose of this study was to compare the IOP fluctuations in the daytime tension curves of glaucoma patients under ocular hypotensive therapy with those of patients previously submitted to trabeculectomy. We also evaluated the IOP peaks and fluctuations for the same patients in response to the water-drinking test (WDT). The study included 30 primary open-angle glaucoma (POAG) patients using ocular hypotensive medications and with no history of previous intraocular surgery (medical group), and 30 POAG patients previously submitted to one or more trabeculectomies though taking no medication at the time of the study (surgical group). All patients were submitted to a diurnal tension curve--DTC (8:30-17:00/3-hour intervals) followed by the WDT. The IOP peak and IOP fluctuation during the diurnal tension curve were significantly greater in the medical group than in the surgical group. The same was observed following the WDT. From an overall baseline IOP of 10.6 mmHg, the mean IOP change following the WDT was 13% in the surgical group and 40% in the medical group. Patients submitted to trabeculectomy have less TOP fluctuations during the diurnal tension curve and following a water-drinking provocative test. This effect could represent an additional benefit of surgery in controlling the intraocular pressure of glaucomatous patients.