lacrimal gland abcess

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  fusiformenlargementofrightlateralrectusmusclewithout involvementofthe tendon.Their patient showed improve-ment with steroid treatment. In our case, the diplopia andocular alignment at the primary gaze improved after treat-ment for Graves disease. However, the ocular motility showed worsening ofabductioninthe righteye andadduc-tion in the left eye. These changes of ocular motility might account for the improvement of exotropia.It is well known that patients with TED have a greaterincidence of myasthenia gravis (MG); both entities repre- sentautoimmunediseases. 1,7,10  Vargasandcolleagues 10 re-ported patients with TED who showed e xotropia due toconcomitant MG. Chen and colleagues 7 suggested that  MG may develop many years after the onset of Graves dis-ease, and exotropia is rare in patient with TED. They rec-ommended that the development of an exotropia in apatient with TED should alert clinicians to the possibledevelopment of MG. In our case, the possibility of concomitant MG was ruled out, because the patient didnot have ptosis characteristic features of MG, and hissymptoms were not associated with diurnal variations orcorrelated with fatigue. Moreover, the anti-acetylcholinereceptor antibody test was negative. However, the patient  will be closely followed because of the possibility of concomitant MG. Literature Search  PubMed and Google Scholar were searched on June 1,2017, without language or data restrictions, using thefollowing terms singly and in combination:  thyroid eyedisease ,  Graves disease ,  strabismus  ,  exotropia , and  diplopia. References 1. Biousse V, Newman NJ. Neuro-Ophthalmology Illustrated. 2nd ed.New York: Thieme; 2016:321-465.2. Mizen T. Thyroid eye disease. Semin Ophthalmol 2003;18:243-7.3. Schotthoefer EO, Wallace DK. Strabismus associated with thyroideye disease. Curr Opin Ophthalmol 2007;18:361-5.4. Gruters A. Ocular manifestations in children and adolescents withthyrotoxicosis. Exp Clin Endocrinol Diabetes 1999;107:172-4.5. Krassas GE. Thyroid eye disease in children and adolescents—newtherapeutic approaches. J Pediatr Endocrinol Metab 2001;14:97-100.6. Uretsky SH, Kennerdell JS, Gutai JP. Graves’ ophthalmopathy inchildhood and adolescence. Arch Ophthalmol 1980;98:1963-4.7. Chen CS, Lee AW, Miller NR, Lee AG. Double vision in a patient  with thyroid disease: what’s the big deal? Surv Ophthalmol 2007;52:434-9.8. Erdurmus M, Celebi S, Ozmen S, Bucak YY. Isolated lateral rectusmuscle involvement as a presenting sign of euthyroid Graves disease. J AAPOS 2011;15:395-7.9. RobertsCJ,MurphyMF,AdamsGG,LundVJ.Strabismusfollowingendoscopic orbital decompression for thyroid eye disease. Strabismus2003;11:163-71.10. VargasME,WarrenFA,KupersmithMJ.Exotropiaasasignofmyas-thenia gravis in dysthyroid ophthalmopathy. Br J Ophthalmol 1993;77:822-3. Lacrimal gland abscess in achild as a raremanifestation of IgG4-related disease Edward L. Raab, MD, JD, a,b Hamideh S. Moayedpardazi, MD, a Steven M. Naids, MD, a  Alan H. Friedman, MD, a,c and Murray A. Meltzer, MD a Dacryoadenitisisunusualinchildren,andnoninfectiouscausescanbe manifestationsofserious undiagnosedsystemic conditions.Thisreport brings further attention to this rarely-encountered conditionand emphasizes its association with IgG4-related disease, a groupof inflammatory disorders of high current interest whose spectrumof manifestations continues to evolve. The pediatric ophthalmolo-gist could be in the position of identifying a patient’s serious andpreviously unappreciated illness. Case Report  A 3-year-old boy presented at the Emergency Department of the Icahn School of Medicine at Mount Sinai for care of orbital cellulitis, suspected because of worsening swellingand redness around his right eye of 2 weeks’ duration.Hismotherstatedthathewasnotinpainbutthattheuppereyelid now felt “hard” and was “drooping.” There was ahistory of recurrent episodes of swelling around the right eye since the child was 9 months of age.On examination, near visual acuity was 20/20 in botheyes. Pupil sizes were equal, with no afferent defect. Color vision was intact. There was limited abduction and eleva-tion of the right eye. The globe was displaced downwardand forward, and there was prolapse of reddish tissuefrom the lateral aspect of the upper conjunctival fornix. Anterior segment structures were normal, and the opticdisk and posterior pole were unremarkable. The left eyeand adnexae were normal in all respects. The child wasactive,afebrile,andwithnoevidenceofrespiratoryorotherinfection.Initiallaboratorystudywasremarkableonlyfora white blood cell count of 16,700/cu mm (normal range,5,300–12,500/cu mm) and an elevation of C-reactive  Author affiliations: Departments of    a Ophthalmology,  b Pediatrics, and   c  Pathology, IcahnSchool of Medicine at Mount Sinai, New York, New YorkSubmitted April 29, 2017. Revision accepted August 7, 2017.Published online December 27, 2017.Correspondence: Edward L.Raab, MD,JD, Department of Ophthalmology, Icahn School of Medicine at Mount Sinai, 1 Gustave Levy Place, Box 1183, New York, NY 10029(email: ). J AAPOS 2018;22:73-75.Copyright   2018, American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.1091-8531/$36.00  Journal of AAPOS  Volume 22 Number 1 / February 2018 Raab et al 73  protein (30.4 mg/l; normal, 0.00–0.5 mg/l), both of whichsubsided. A blood culture obtained on admission showedno growth after 4 days. Magnetic resonance imaging (MRI) with contrast re- vealed a cavitary abscess in an enlarged right lacrimalgland, without extension to the subperiosteal space orcavernous sinus; flattening and indentation of the lateralaspect of the globe were present. The brain and sinuses were not involved (Figure 1). With neither deteriorationnor improvement of his clinical condition by the thirdday of treatment with intravenous clindamycin and despitespontaneous purulent drainage from the superior fornix,surgery was performed via an anterior approach. A largeamount of purulent material was encountered within theabscess (Figure 2) and was removed. A culture showed Staphylococcus aureus  .Because one of the treating physicians (MAM) hadexperience with the group of conditions known asIgG4-related disease, and given the history of recurrent right periorbital swelling since infancy, a biopsy of thethickened abscess wall was obtained for IgG4 staining. The incision was closed in layers after copious irriga-tion of the cavity with gentamycin solution. The post-operative course was uneventful, and the patient  was discharged after 2 days to continue clindamycinorally for 2 weeks. As of the 14th postoperative day,there was substantial resolution of swelling, withpartially improved abduction and elevation of the eye. The biopsy showed an epithelial lining as well asfibrosis and chronic nongranulomatous inflammation FIG 1.  Magnetic resonance imaging with contrast showing rightlacrimal gland abscess cavity (A) and flattening of the globe (B) byenlarged right lacrimal gland. FIG 2.  Lacrimal gland abscess cavity opened showing purulent con-tents. FIG 3.  A, Biopsy specimen, showing lymphoplasmacytic infiltration,fibrosis, and vascular obliteration (hematoxylin-eosin, srcinal magni-fication  400). B, Positive staining for IgG4 (B).  Journal of AAPOS  74 Raab et al Volume 22 Number 1 / February 2018  (Figure 3 A). Lacrimal acini were present. The excisedtissue contained cells that stained positively forIgG4 (Figure 3B). Because of noncompliance withseveral attempts to carry out our plan to obtain rhe-umatology consultation and to follow the outcomeof this child’s illness, we cannot report further informa-tion. Discussion  Causes of acute and subacute dacryoadenitis across all agegroups include infections by a wide range of agents.Noninfectiouscauses,moretypicallyinadults,includethy-roid ophthalmopathy, Wegener’s granulomatosis, andsarcoidosis (see eAppendix). The diagnosis is readily suggested by upper eyelidswelling that is most prominent laterally, an  S  -shapedappearance of the eyelid margin, and visible swelling andprolapse of the palpebral lobe, as our patient demon-strated. 1  MRIis the preferred confirming study. Flatteningof the lateral aspect of the eye from pressure by theenlargedglandisahighlyreliablesign. 1  Thecharacteristicson imaging substantially eliminate from considerationrhabdomyosarcoma, vascular malformations, and metasta-tic disease. Contrast enhancement has been advocated asindispensable in order to show abscess formation(Figure 2). 2 Infectious agents reach the lacrimal gland most typically byspreadfromtheadjacentparanasalsinuses.Blood-bornespread, usually from respiratory infections or extensionfrom the skin or ocular surface via the lacrimal ductulesare additional routes. The most serious sequel of dacryoa-denitis, although unusual in children, is purulent abscessformation. which has occurred more prominently withthe emergence of methicillin-resistant staphylococcusaureus. 3  When intensive treatment with intravenous antibioticshas been ineffective, and vision remains threatened, surgi-cal drainage is required. Culture studies to dictate therapy areof limited value, because they are often negative both inblood and in the abscess contents. The history of recurrent eyelid swelling since infancy in this patient suggested the possibility of somethingother than a local infection, a suspicion confirmed by ex-amination of the excised abscess wall. Since an IgG4-related entity was not suspected at the time of the pa-tient’s presentation, blood levels of this indicator werenot obtained. However, the accumulating literature indi-cates that demonstration in tissue is a more persuasivedetermining criterion. 4,5 C-reactive protein, althoughelevated in this patient, reflects inflammation in generaland is often normal in IgG4-related disease. By consensus, the characteristic histologic features are lym-phoplasmacytic infiltration, storiform (irregularly  whorled) fibrosis, and obliterative phlebitis, as demon-strated in our case. 6  The classification, multiple clinical settings and char-acteristics, and diagnostic features of IgG4-related dis-orders continue to evolve. 6-10; Appendix Serologic criteriaand the number or percentage of IgG4-positive cells ina biopsy necessary for this diagnosis, for example, havebeen organ- or tissue type–specific and variably re-ported. 6  This entity can cause fibrosis from inflamma-tion and often irreversible damage to most body tissues. Therefore, early recognition and prompt treatment are critical. Prednisone has been effective asfirst-line therapy, with rituximab as an alternativesteroid-sparing agent. At the minimum, referral forrheumatology investigation is an important adjunctivemeasure. Occurrence in the orbit, especially bilaterally,could indicate the presence of more widespreadinvolvement, 8 placing the pediatric ophthalmologist inthe role of “first responder” to identification and treat-ment. IgG4-related disease should be considered in thedifferential diagnosis of orbital masses or inflamma-tions, especially when recurrent, in the pediatric agegroup. References 1. SayaniA,SmithDE.Clinicalrecognitionandmanagementofatypicaldacryoadenitis. Clin Pediatr (Phila) 2015;54:956-60.2. Parvizi N, Choudhury N, Singh A. Complicated periorbitalcellulitis: case report and literature review. J Laryng Otol 2012;126:94-6.3. Mathias MT, Horsley MB, Mawn LA, et al. Atypical presentations of orbital cellulitis caused by methacillin-resistant stapylococcus aureus.Ophthalmology 2012;119:1238-43.4. TeoL,SeahLL,ChooCT,CheeSP,CheeE,LoolA.A surveyofthehistopathology of lacrimal gland lesions in a tertiary referral center.Orbit 2013;32:1-7.5. Andrew NH, McNab AA, Delva D. Review of 268 lacrimal glandbiopsies in an Australian cohort. Clin Exp Ophthalmol 2015;43:5-11.6. Karim F, Loeffen J, Bramer W, et al. IgG4-related disease: a system-atic review of this unrecognized disease in pediatrics. Pediatr Rheu-matol Online J 2016;14:18.7. Deshpande V, Zen Y, Chan JK, Yi EE, Sato Y, Yoshino T, et al.Consensus statement on the pathology of IgG4-related disease. Mod Pathol 2012;25:1181-92.8. Wu A, Andrew NH, McNab AA, Selva D. Bilateral IgG4-relatedophthalmic disease: a strong indication for systemic imaging. Br JOphthalmol 2016;100:1409-11.9. CheukW,YuenHKL,ChanJKC.Chronicsclerosingdacryoadenitis:part of the spectrum of IgG4-related sclerosing disease? Am J SurgPathol 2007;31:643-5.10. Umehara H, Okazaki K, Masaki Y, et al., Research Program forIntractable Disease by Ministry of Health, Labor and Welfare(MHLW) Japan G4 team. A novel clinical entity, IgG4-related dis-ease (IgG4RD); general concept and details. Mod Rheumatol 2012;22:1-14.  Journal of AAPOS  Volume 22 Number 1 / February 2018 Raab et al 75   eAppendix. Supplemental references 1. Andrew NH, Kearney D, Sladden N, et al. Idiopathic dacryoadenitis:clinical features, histopathology, and treatment outcomes. Am JOphthalmol 2016;163:148-153.e1.2. Chakaborti C, Biswas R, Mondal M, Mukhopadhya U, Datta J. Tuberculous dacryoadenitis in a child. Nepal J Ophthalmol 2011;3:210-13.3. Cheuk W, Yuen HK, Chan AC, et al. Ocular adnexal lymphomaassociated with IgG4 chronic sclerosing dacryoadenitis: a previously undescribed complication of IgG4-related sclerosing disease. Am JSurg Pathol 2008;32:1159-67.4. GooldLA,MadgeSN,AuA,etal.Acutesuppurativebacterialdacryoa-denitis: a case series. Br J Ophthalmol 2013;97:735-8.5. Kalapesi FB, Garrott HM, Moldovan C, Williams M, Ramanan A,Herbert HM. IgG4 orbital inflammation in a 5-year-old child present-ing as an orbital mass. Orbit 2013;32:137-40.6. Krishna N, Lyda W. Acute suppurative dacryoadenitis as a sequel tomumps. Arch Ophthalmol 1958;59:350-51.7. Patel N, Khalil HM, Amirfeyz R, Kaddour HS. Lacrimal gland abscesscomplicating acute sinusitis. Int J Pediatr Otorhinolaryng 2003;67:917-19.8. Sen DK. Acute suppurative dacryoadenitis caused by cysticercuscellulosa. J Pediatr Ophthalmol Strabismus 1982;19:100-102.  Journal of AAPOS  Volume 22 Number 1 / February 2018 Raab et al 75.e1
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