de Boer, Joke H

Kalmann, Rachel

Laban, Kamil G

Leguit, Roos J

Radstake, Timothy R D J

Uijttewaal, Peter H

van der Veen, Rob L P

PubMed

Utrecht University Repository

findings are in contrast to previouspopulation-based studies in childrenand adults. Gopinath et al. (2010)observed in 1700 children with an ageof 12-years that former low birthweightnewborns had narrower retinal arteri-oles which is in accordance to otherinvestigations in children (Mitchellet al. 2008). For adult individuals, anassociation between birthweight andretinal arteriolar calibre but not withretinal venular calibre was reported.The authors found an arteriolar calibrenarrowing of 2.4 µm for each kg whichwas still significant after adjusting formultiple covariates including averagearterial blood pressure measurementover 6 years (Liew et al., 2008). Theseresults are in contrast to our data whichmay be caused by our younger cohortand potentially stricter blood pressuretreatment regimes in the last years.Our results are limited because 46%of all participants did not report theirbirthweight at study entry (Fieß et al.2019). In addition, birthweight wasself-reported and not validated byreviewing individual charts. Further-more, missing data about gestationalage and postnatal occurrence ofretinopathy of prematurity may beconfounders which have to be consid-ered when interpreting our data.In summary, this large population-based study shows no associationbetween lower birthweight and centralretinal venular equivalents, while theassociation for central retinal arterialequivalent was weakened by arterialblood pressure.AcknowledgementOpen access funding enabled andorganized by Projekt DEAL.[Correction added on 01 October 2020,after first online publication: ProjektDeal funding statement has beenadded.]ReferencesFieß A, Schuster AK, Nickels S, Elflein HM,Schulz A, Beutel ME, Blettner M & PfeifferN (2019): Association of low birth weightwith myopic refractive error and lowervisual acuity in adulthood: results from thepopulation-based Gutenberg Health Study(GHS). The British journal of ophthalmol-ogy 103: 99–105.Gopinath B, Baur LA, Wang JJ, Teber E,Liew G, Cheung N, Wong TY & Mitchell P.(2010): Smaller birth size is associated withnarrower retinal arterioles in early adoles-cence. Microcirculation (New York, N.Y.:1994) 17: 660–668.Hellstrom A, Dahlgren J, Marsal K & Ley D(2004): Abnormal retinal vascular morphol-ogy in young adults following intrauterinegrowth restriction. Pediatrics 113: e77–80.Liew G, Wang JJ, Duncan BB et al. (2008):Low birthweight is associated with narrowerarterioles in adults. Hypertension (Dallas,Tex: 1979) 51: 933–938.Mitchell P, Liew G, Rochtchina E, Wang JJ,Robaei D, Cheung N & Wong TY (2008):Evidence of arteriolar narrowing in low-birth-weight children. Circulation 118: 518–524.Received on February 5th, 2020.Accepted on September 2nd, 2020.CorrespondenceAchim Fieß, MDDepartment of OphthalmologyUniversity Medical Center of the JohannesGutenberg University MainzLangenbeckstr. 155131 MainzGermanyTel. +49 (0)6131 17 5150Fax. +49 (0)6131 17 8495E-mail: Achim.fiess@gmail.comAll authors: The Gutenberg Health Study is fundedthrough the government of Rhineland-Palatinate(‘Stiftung Rheinland-Pfalz f€ur Innovation’, contractAZ 961-386261/733), the research programmes‘Wissen schafft Zukunft’ and ‘Center for Transla-tional Vascular Biology (CTVB)’ of the JohannesGutenberg University of Mainz, and its contractwith Boehringer Ingelheim and PHILIPS MedicalSystems, including an unrestricted grant for theGutenberg Health Study. Schuster AK holds theprofessorship for ophthalmic healthcare researchendowed by ‘Stiftung Auge’ and financed by‘Deutsche Ophthalmologische Gesellschaft’ and‘Berufsverband der Augen€arzte Deutschland e.V.’Wild PS is funded by the Federal Ministry ofEducation and Research (BMBF 01EO1503) and heis PI of the German Center for CardiovascularResearch (DZHK). Pfeiffer N receives financialsupport by Novartis, Ivantis, Santen, Thea, Boeh-ringer Ingelheim Deutschland GmbH & Co. KG,Alcon, Sanoculis. Beutel ME received speakerhonorarium from Pfizer Deutschland GmbH, ShireDeutschland GmbH and currently receives researchgrants from the governments of Rhineland-Palati-nate and Nordrhein-Westfalen, the German Asso-ciation of Psychoanalysis and Psychotherapy(DGPT), the German Research Foundation, theGerman Ministry of Research, the German CancerAid and the European Union. Fieß A., Ponto KA.,Schulz A., Urschitz MS., Nickels S., M€unzel T.,Lackner KJ.: none.Financial disclosure/conflict of interest: Pfeiffer Nreceives financial support and grants from Novartis,Ivantis, Santen, Thea, Boehringer IngelheimDeutschland GmbH & Co. KG, Alcon, andSanoculis. Schuster AK received research fundingfrom Allergan, Bayer Vital, Novartis, PlusOptix andHeidelberg Engineering. Nickels S received reim-bursement of travel expenses from Heidelberg Engi-neering. Fieß A., Ponto KA., Schulz A., UrschitzMS., M€unzel T., Beutel M., Lackner KJ.: none.Boehringer Ingelheim, PHILIPS Medical Systemsand Novartis Pharma provided funding towardsthis study. There are no patents, products indevelopment or marketed products to declare. Thisdoes not alter the authors’ adherence to all thejournal policies on sharing data and materials. Thefunders had no role in study design, data collectionand analysis, decision to publish, or preparation ofthe manuscript.Severe outcome ofidiopathic inflammatorymass lesions primarilylocated in the posteriororbit and orbital apexKamil G. Laban,1,2,3Rachel Kalmann,1,2 Roos J. Leguit,4Peter H. Uijttewaal,2Timothy R.D.J. Radstake,1,3,5Joke H. deBoer1,2 andRob L.P. van derVeen21Ophthalmo-Immunology Unit, UniversityMedical Center Utrecht, University Utrecht,Utrecht, The Netherlands; 2Department ofOphthalmology, University Medical CenterUtrecht, University Utrecht, Utrecht, TheNetherlands; 3Laboratory of TranslationalImmunology, University Medical CenterUtrecht, University Utrecht, Utrecht, TheNetherlands; 4Department of Pathology,University Medical Center Utrecht,University Utrecht, Utrecht, TheNetherlands; 5Department of Rheumatology& Clinical Immunology, University MedicalCenter Utrecht, University Utrecht, Utrecht,The Netherlandsdoi: 10.1111/aos.14656ª 2020 The Authors. Acta Ophthalmologica publishedby John Wiley & Sons Ltd on behalf of ActaOphthalmologica Scandinavica FoundationThis is an open access article under the terms of theCreative Commons Attribution-NonCommercial-NoDerivs License, which permits use and distribution in anymedium, provided the original work is properly cited, theuse is non-commercial and no modifications oradaptations are made.Editor,I diopathic orbital inflammation(IOI) is a heterogeneous noninfec-tious inflammatory process that causesa mass or an enlargement of orbitale774Acta Ophthalmologica 2021structures, and is a diagnosis of exclu-sion (Mombaerts et al. 2016). Theorbital localization of IOI can influencethe disease course and the clinicalcharacteristics (Lee et al. 2020). Thediagnostic process of IOI can be chal-lenging due to the heterogeneous nat-ure, with diagnostic overlap comparedto other orbital conditions. Histo-pathological assessment of the affectedtissue is recommended for nonmyositisIOI (Mombaerts et al. 2017), althoughlesions isolated to the posterior orbitcan be difficult to approach. As sug-gested in previous work (Li et al. 2013)and in our own experience, posteriorIOIs tend to have a severe clinicalpresentation with poor outcome. Wetherefore investigated patients withsuspected posterior IOI and comparedcharacteristics between cases with afavourable or unfavourable outcome.The posterior orbit was defined asthe most posterior 1/3rd of the orbitand could include posterior extension.From a single centre cohort of 176patients with suspected IOI of a ter-tiary referral centre for orbital diseasein the Netherlands, eight patients withisolated posterior involvement wereincluded. Of these, seven patients hadhistopathologically confirmed diagno-sis of IOI, while in one patient biopsywas not deemed possible and wasdiagnosed as suspected IOI based thor-ough clinical, radiological and labora-tory examination. The medianduration between symptom onset andpresentation was 22 days (range 4–483 days). All patients received stan-dard corticosteroid treatments (i.e.intravenous pulse therapy of 500–1000 mg per day for 3 days) withadditional (immunosuppressive, i.e.Rituximab) therapy on clinical indica-tion. Although seven patients reactedon therapy positively with a reductionof pain, five patients were regarded ashaving an unfavourable outcome over-all with a visual acuity of <0.5 decimal.Characteristics of the patients areshown in Table 1. Two patients withan unfavourable visual outcome had anIOI related death; the first patient didnot respond to treatment and devel-oped a severely painful blind eye, withpersisting pain after enucleation andsuicide in combination with a concur-rent mental health condition. The sec-ond patient suffered from asubarachnoid haemorrhage because ofIOI ingrowth into the cavernous sinusand internal carotid artery as shown byautopsy.Although the outcome groups aretoo small to perform statistic testing,patients with a more severe outcomewere generally older, presented with alower visual acuity and were diabetic inthe majority of cases (Table 1). Poste-rior IOI tends to have a serious clinicalpresentation with severe orbital pain,loss of visual acuity and poor visualoutcome after treatment. Althoughinitial treatment can be effective toreduce pain, visual acuity improvementis often absent.In contrast to the typical Tolosa-Hunt syndrome, in which visual acuityis usually not affected, the majority ofpatients in our study presented with, orresulted in, a poor visual acuity.Tolosa-Hunt syndrome is most com-monly described as a granulomatousinflammatory condition that affects theorbital apex, superior orbital fissureand cavernous sinus (Lueck 2018). Thedistinction between an isolated poste-rior IOI and Tolosa-Hunt, however,can be unclear (Lueck 2018).Once IOI of the posterior orbit isdiagnosed, treatment should be ensuedrapidly in our opinion. In almost allour cases, there was initial relief of painon high-dose steroid treatment. How-ever, visual acuity did not improve, anddeteriorated further in some patients.We believe that this is due to lastingmass effect on the tissues surroundingthe optic nerve and cranial nerveswithin the tight space of the orbitalapex, even after decongestion by ster-oids. Therefore, steroid treatmentalone is not always sufficient for treat-ing posterior IOI. Further research inTable 1. Patient and disease characteristics based on visual outcome.Variable AllVisualoutcome <0.5Visualoutcome ≥0.5Total patients, n 8 5 3Age (year), mean (SD) 66.3 (14.2) 74.2 (12.1) 53.0 (3.3)Sex (female), n 3 (38%) 1 (20%) 2 (67%)Diabetes, n 3 (38%) 3 (60%) –BCVA at presentation (decimal), nNLP 3 (38%) 3 (60%) –LP–0.05 2 (25%) 1 (20%) 1 (33%)0.05–0.5 1 (13%) – 1 (33%)0.5–0.7 2 (25%) 1 (20%) 1 (33%)Total ophthalmoplegia, n 3 (38%) 1 (20%) 2 (67%)IOP ipsilateral, mean (SD) 18.2 (6.6) 13.8 (1.5) 24.3 (6.0)IOP contralateral, mean (SD) 17.3 (3.9) 14.8 (1.6) 20.6 (3.4)Motility reduction, nModerate 3 (38%) 2 (40%) 1 (33%)Severe 5 (63%) 3 (60%) 2 (67%)Modified Werner criteria, median (IQR) 23 (20–26) 24 (21–26) 23 (20–25)Onset to ophthalmologist (days), median (IQR) 22 (6–75) 15 (6–62) 22 (14–64)Treatment, nOral prednisone only 2 (25%) 1 (20%) 1 (33%)IV MP + oral prednisone 6 (75%) 4 (80%) 2 (67%)Additional treatment, n 2 (25%) 1 (20%) 1 (33%)Initial response, nVisual improvement 3 (38%) – 3 (100%)Pain improvement 7 (88%) 5 (100%) 2 (67%)Motility improvementFollow-up time (months), median (IQR) 18 (7–30) 8 (3–45) 22 (18–24)Visual (decimal) and clinical outcome, nNLP 5 (63%) 5 (100%) –LP–0.05 – – –0.05–0.5 – – –0.5–1.0 3 (38%) – 3 (100%)Residual pain (mild) 2 (25%) 1 (20%) 1 (33%)Residual pain (moderate/severe) 3 (38%) 2 (40%) –BCVA = best corrected visual acuity, IOP = intraocular pressure, IQR = interquartile range,LP = light perception, MP = methylprednisolone, n = number, NLP = no light perception.e775Acta Ophthalmologica 2021early and aggressive managementstrategies for posterior IOI is war-ranted.ReferencesLee MJ, Planck SR, Choi D et al. (2020): Non-specific orbital inflammation: current under-standing and unmet needs. Prog Retin EyeRes. 100885 [Epub ahead of print].Li Y, Lip G, Chong V, Yuan J & Ding Z(2013): Idiopathic orbital inflammationsyndrome with retro-orbital involvement: aretrospective study of eight patients. PLoSOne 8: e57126.Lueck CJ (2018): Time to retire the Tolosa-Hunt syndrome? Pract Neurol 18: 350–351.Mombaerts I, Bilyk JR, Rose GE et al. (2017):Consensus on diagnostic criteria of idio-pathic orbital inflammation using a modifieddelphi approach. JAMA Ophthalmol 135:769–776.Mombaerts I, Rose GE & Garrity JA (2016):Orbital inflammation: biopsy first. SurvOphthalmol 61: 664–669.Correspondence:Kamil G. LabanDepartment of OphthalmologyUniversity Medical Center UtrechtRoom E 03.136, P.O. Box 855003508 GA UtrechtThe NetherlandsTel: +31 88 7560818Fax: +31 88 7555417Email: k.g.laban@umcutrecht.nle776Acta Ophthalmologica 2021

Severe outcome of idiopathic inflammatory mass lesions primarily located in the posterior orbit and orbital apex

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Severe outcome of idiopathic inflammatory mass lesions primarily located in the posterior orbit and orbital apex

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