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Anatomic and Visual Function Outcomes in Paediatric Idiopathic Intracranial Hypertension Sidney M Gospe III,1 M Tariq Bhatti,1,2 Mays a El-Dairi1

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Anatomic and Visual Function Outcomes in Paediatric Idiopathic Intracranial Hypertension Sidney M Gospe III,1 M Tariq Bhatti,1,2 Mays a El-Dairi1 Downloaded from http://bjo.bmj.com/ on April 14, 2016 - Published by group.bmj.com Clinical science Anatomic and visual function outcomes in paediatric idiopathic intracranial hypertension Sidney M Gospe III,1 M Tariq Bhatti,1,2 Mays A El-Dairi1 1Department of ABSTRACT Monitoring of papilledema severity and early Ophthalmology, Duke Background There is a paucity of literature describing detection of optic atrophy in patients with IIH University Medical Center, Durham, North Carolina USA risk factors for vision loss in paediatric idiopathic have been greatly aided by advances in spectral 2Department of Neurology, intracranial hypertension (IIH). We investigate the final domain optical coherence tomography (SD-OCT). Duke University Medical visual function, spectral domain optical coherence This non-invasive imaging modality is now com- Center, Durham, North tomography (SD-OCT) and enhanced depth imaging monly used to measure the thickness of the peripa- Carolina, USA (EDI)-OCT findings in children with papilledema caused pillary RNFL in IIH, glaucoma and other optic 5–7 Correspondence to by IIH. neuropathies. Moreover, the high resolution of Dr Mays A El-Dairi, Methods Medical records of 31 patients with SD-OCT has allowed pathology of specific layers of Department of Ophthalmology, paediatric IIH (age ≤17 years) were retrospectively the retina to be analysed qualitatively and quantita- Duke Eye Center, Duke reviewed. Optic disc photographs on presentation and tively, and we have recently described examples of university, P.O. Box 3802, Durham, NC 27710, USA; automated perimetry, SD-OCT and EDI-OCT imaging on focal atrophy of retinal layers in the macula in [email protected] final follow-up visit were statistically analysed to identify adult patients with IIH that is distinct from the patient characteristics and anatomic findings associated already-well-described RGC and RNFL changes.8 Received 17 April 2015 with irreversible vision loss. Though less common than in adults, IIH may Revised 13 July 2015 Results Permanent visual acuity or visual field loss affect the paediatric population.9 The longstanding Accepted 29 July 2015 Published Online First developed in 19% of study eyes. Papilledema of belief that paediatric patients are less prone to poor 12 August 2015 modified Frisén grade ≥3 on presentation was highly visual outcomes has been challenged in recent – predictive of permanent vision loss (p<0.001), while years.10 12 The demographics of paediatric IIH are associations between pubertal status and visual function dictated by patients’ pubertal status: pubertal outcome failed to reach statistical significance. SD-OCT patients tend to be obese females (similar to the revealed optic atrophy in 13% and photoreceptor loss in adult population), while pre-pubertal children show 19% of eyes, with both findings highly associated with no tendency towards a particular sex or body vision loss (p<0.0001). Optic disc drusen was noted in habitus.13 14 48% of study eyes by EDI-OCT but was not found to be In this retrospective longitudinal study, we aimed predictive of visual outcome. to address the relative paucity of information Conclusions Clinical observation of high papilledema regarding visual outcomes in paediatric IIH and to grade on presentation is predictive of poor visual examine potential predictors of visual morbidity. outcomes. Vision loss is associated not only with optic We applied SD-OCT and enhanced depth imaging atrophy but also with photoreceptor damage. (EDI)-OCT to assess anatomic abnormalities of the Interestingly, a high proportion of study eyes had optic retina and optic nerve that may correlate with disc drusen, which was not associated with vision loss, visual disability following treatment of IIH. but can be a diagnostic challenge in distinguishing true papilledema from pseudopapilledema. METHODS All patients with IIH age 17 and under, presenting to the paediatric neuro-ophthalmology clinic between 1 January 2010 and 31 August 2013, were INTRODUCTION included. Patients were required to meet the Patients with idiopathic intracranial hypertension revised diagnostic criteria for IIH proposed by (IIH) often present with headache and visual Friedman, Liu and Digre: (1) observation of papil- symptoms due to elevated intracranial pressure ledema or sixth nerve palsy, (2) otherwise normal (ICP) not caused by an intracranial mass, venous neurological examination, (3) contrast-enhanced sinus thrombosis or central nervous system inflam- neuroimaging excluding secondary aetiologies of mation.1 A hallmark finding of IIH is papilledema. elevated ICP, (4) normal CSF composition and (5) When profound or chronic, papilledema may lead lumbar puncture opening pressure exceeding 28 cm 1 to irreversible visual field defects, decreased visual H2O (or 25 cm H2O if not sedated). Inclusion acuity and the development of optic atrophy from additionally required optic disc photography per- death of retinal ganglion cells (RGCs) and their formed on presentation and Spectralis (Heidelberg, axons that comprise the retinal nerve fibre layer Carlsbad, California, USA) SD-OCT imaging of the (RNFL).2 To alleviate symptoms and prevent per- macula obtained following treatment. manent visual disability, IIH is typically treated Data collected from the initial patient visit with dietary interventions combined with pharma- included age, gender, pubertal status (as determined fl To cite: Gospe SM, cotherapy to reduce cerebrospinal uid (CSF) pro- by menarche in females and growth of facial hair in Bhatti MT, El-Dairi MA. Br J duction3 or surgical intervention via optic nerve boys), serum haemoglobin (if available), visual Ophthalmol 2016;100: sheath fenestration (ONSF) or CSF diversion acuity and optic disc photos. Type of therapy admi- – 505 509. procedures.4 nistered (eg, diuretics, ONSF, CSF diversion Gospe SM, et al. Br J Ophthalmol 2016;100:505–509. doi:10.1136/bjophthalmol-2015-307043 505 Downloaded from http://bjo.bmj.com/ on April 14, 2016 - Published by group.bmj.com Clinical science procedures) and assessments of visual acuity and visual field (using the automated Humphrey Field Analyzer/HFA II-i; Carl Zeiss Meditec, Dublin, California, USA) were documented. Using optic disc photographs, the severity of papilledema on presentation for both eyes of each patient was graded by the modified Frisén scale15 by a masked reader (MAE-D), and the eye with more severe disc swelling (or the right eye if the papil- ledema was symmetric) was selected for further analysis. Humphrey visual fields were categorised as normal or demon- strating an enlarged blindspot, single or double arcuate scotoma, central scotoma or paracentral scotoma; the latter two classifica- tions required depressions within 10° of fixation, with a foveal threshold <30 dB being denoted a central scotoma and a foveal Figure 1 Visual acuity pre-treatment and post-treatment. Scatterplot threshold of ≥30 dB denoted a paracentral scotoma. Finally, raw of visual acuity measurements by the logarithm of the minimum angle of Spectralis SD-OCT and (when available) EDI-OCT data were resolution (LogMAR) in the study eye for each subject. The visual acuity inspected in a masked fashion for each patient, and the presence by Snellen equivalent is marked on the right axis. Pre-pubertal subjects of retinal or optic nerve pathology was documented. are displayed on the left and pubertal subjects on the right. Visual acuity Data analysis was performed through the construction of con- on presentation is plotted as blue diamonds, and visual acuity at most 2 recent follow-up visit is plotted as red squares. Patients undergoing optic tingency tables and χ testing in order to test the predictive nerve sheath fenestration in the study eye are marked with orange value for adverse visual outcomes of various patient character- arrows, and those undergoing cerebrospinal fluid diversion procedures fi istics on presentation. Poor visual outcomes were classi ed as are marked with green arrows. HM, hand-motion; LP, light perception. permanent central vision loss (visual acuity of 20/40 or worse or a central/paracentral scotoma on visual field testing) or per- manent peripheral vision loss (single or double arcuate scotoma with normal visual acuity maintained normal acuity, while all on visual field testing). Correlation between anatomic abnormal- eight presenting with abnormal visual acuity demonstrated some ities on final SD-OCT imaging and adverse visual outcomes degree of improvement over the course of treatment (figure 1). were similarly tested. Ultimately, 28 study eyes had normal visual acuity, 2 had moder- ately diminished visual acuity (one pubertal, one pre-pubertal) RESULTS and 1 had profound loss of visual acuity (pubertal). Automated Thirty-one patients met criteria for inclusion in the study perimetry data were available for 23 patients (11 of 12 pubertal (table 1), with an average follow-up time of 30.7±16.2 months and 12 of 19 pre-pubertal). Five patients (four pubertal, one (mean±SD). There were 12 pubertal (all female) and 19 pre- pre-pubertal) demonstrated permanent peripheral visual field pubertal (12 male, 7 female) children. The mean age on presen- loss in the study eye, and four patients (three pubertal, one pre- tation was 15.4±1.4 years for pubertal children and 7.8 pubertal) demonstrated permanent central or paracentral scot- ±3.4 years for pre-pubertal children. The mean grade of papil- omas. A total of seven patients had at least one form of perman- ledema on presentation was 2.6±1.4 in the study eyes overall, ent visual field loss in the study eye. with no significant difference between pubertal and pre-pubertal Final SD-OCT images of the optic nerve head and macula group (p=0.26). On presentation, 23 patients had normal were reviewed for the study eye of each patient. RNFL OCT visual acuity (20/20–20/30), 5 had moderately diminished visual demonstrated a final average RNFL thickness of 106.9 acuity (20/40–20/80) and 3 had profound visual acuity loss (20/ ±29.1 mm. Four eyes (all pubertal) demonstrated optic atrophy, 100—light perception) in the study eyes.
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