Collins, ME

Marcet, MM

Petronic-Rosic, V

Sweiss, NJ

Case Reports in Ophthalmological Medicine

PubMed

Eyelid involvement in sarcoidosis is very rare. A search of the medical literature indicates one previous report of sarcoidosis with destructive eyelid lesions. We describe the case of a 50-year-old woman with severe systemic sarcoidosis, which included her eyelids. To our knowledge, the case presented herein represents the first to show the full-thickness histopathology of destructive eyelid lesions in sarcoidosis

Megan E. Collins

Vesna Petronic-Rosic

Nadera J. Sweiss

Marcus M. Marcet

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Full-Thickness Eyelid Lesions in Sarcoidosis

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Title Full-thickness eyelid lesions in sarcoidosisAuthor(s) Collins, ME; Petronic-Rosic, V; Sweiss, NJ; Marcet, MMCitation Case Reports in Ophthalmological Medicine, 2013, v. 2013, p.579121Issued Date 2013URL http://hdl.handle.net/10722/191107Rights Creative Commons: Attribution 3.0 Hong Kong LicenseHindawi Publishing CorporationCase Reports in Ophthalmological MedicineVolume 2013, Article ID 579121, 3 pageshttp://dx.doi.org/10.1155/2013/579121Case ReportFull-Thickness Eyelid Lesions in SarcoidosisMegan E. Collins,1 Vesna Petronic-Rosic,2 Nadera J. Sweiss,3 and Marcus M. Marcet41 Department of Ophthalmology, University of Wisconsin, Madison, WI, USA2 Section of Dermatology, University of Chicago, Chicago, IL, USA3 Section of Rheumatology, University of Illinois, Chicago, IL, USA4Department of Ophthalmology, University of Hong Kong, Cyberport, Hong KongCorrespondence should be addressed to Marcus M. Marcet; marcet@hku.hkReceived 10 March 2013; Accepted 17 April 2013Academic Editors: N. Fuse, M. Rosner, and S. SchwartzCopyright © 2013 Megan E. Collins et al. This is an open access article distributed under the Creative Commons AttributionLicense, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properlycited.Eyelid involvement in sarcoidosis is very rare. A search of the medical literature indicates one previous report of sarcoidosis withdestructive eyelid lesions.We describe the case of a 50-year-oldwomanwith severe systemic sarcoidosis, which included her eyelids.To our knowledge, the case presented herein represents the first to show the full-thickness histopathology of destructive eyelidlesions in sarcoidosis.1. IntroductionSarcoidosis is a multisystem inflammatory disease of un-known etiology characterized histopathologically by non-caseating granulomas. The condition has a female predilec-tion with a peak incidence in the 3rd and 5th decades of life.Pulmonary involvement is the most common manifestation.Common extrapulmonary manifestations include the skin,central nervous system, liver, kidney, and musculoskeletalsystem. Furthermore, sarcoidosis can affect the heart, periph-eral nervous system, salivary glands, eye, and ocular adnexa.Ocular involvement occurs in 25%–60% of patients withsystemic sarcoidosis [1, 2]. While the disease can involve anyocular tissue, anterior uveitis and lacrimal gland involvementare themost frequently reported findings [3]. Eyelid manifes-tations of sarcoidosis include “millet seed” nodules, ulceratednodules, plaques, and swollen eyelids [4]. Eyelid involvementin sarcoidosis is very rare. To our knowledge, there is onlyone previous report in the literature that describes full-thickness eyelid involvement. Our case is the first to show thefull-thickness histopathology of destructive eyelid lesions insarcoidosis.2. Case ReportA 50-year-old female with severe systemic sarcoidosis, in-cluding pulmonary, skin, and joint disease, presented forevaluation of ocular involvement.The patient had noted eye-lid nodules for over 5 years; however, she recently developedeyelid deformities, tearing, and eye irritation.On examination, she had notching of her upper andlower eyelids and extensive scarring of the posterior lamellaleading to entropion and focal trichiasis (Figure 1). Slit lampexamination showed complete inspissation of themeibomianglands and superficial keratitis corresponding with misdi-rected eyelashes.The remainder of the anterior and posteriorsegment examination was within normal limits. Medicaltherapy did not adequately control the patient’s symptoms.Thepatient subsequently underwent full-thicknesswedgeresections in both upper eyelids and her right lower eyelidresulting in symptomatic improvement and elimination ofthe localized areas of trichiasis. Histopathologic evaluationof all three full-thickness wedge resections showed typical“naked,” noncaseating granulomas composed of epitheliodhistiocytes and multinucleated giant cells (Figure 2). Serialsections showed extensive destruction of both the anteriorand posterior lamellar anatomies, including derangement ofthe tarsal architecture, obliteration of the meibomian glands,and infiltration of the dermis and orbicularis oculi muscle.Entropic migration of the mucocutaneous junction and eye-lashmisdirectionwere also seen. Ziehl-Neelsen andGomori’smethenamine silver stains were negative formicroorganisms.2 Case Reports in Ophthalmological Medicine(a) (b)Figure 1: (a) Preoperative photograph of the patient showing eyelid notching, entropion, and trichiasis. Multiple sarcoid nodules on the faceare present. (b) Preoperative photograph of everted right upper eyelid showing focal trichiasis, cicatricial entropion, and thickened hyperemicconjunctival scarring. Inspissatedmeibomian glands are seen in themedial third of the eyelid.There is grossly apparent loss of themeibomianglands in the lateral two thirds of the eyelid.dotp(a) ∗(b)∗(c) (d)Figure 2: (a) Photomicrograph of sagittally sectioned full-thickness wedge resection showing a thickened eyelid with noncaseatinggranulomas infiltrating dermis, orbicularis oculi muscle, and tarsus. The mucocutaneous junction is seen (arrow). There is absence of themeibomian glands (hematoxylin-eosin, ×20). The eyelid is designed as follows: tp = tarsal plate; o = orbicularis oculi muscle; d = dermis.(b) Cross-section through the meibomian gland orifice (arrow) with sarcoidal obliteration of the meibomian glands (asterisk) (hematoxylin-eosin, ×100). (c) Tarsal (asterisk) granulomas withmultinucleated giant cells and normal overlying palpebral conjunctiva (hematoxylin-eosin,×200). (d)Highermagnification view of tarsus showing noncaseating granuloma containing epitheliod histiocytes (hematoxylin-eosin ×400).3. DiscussionEarly reports have limited discussion of the incidence of eye-lid involvement in sarcoidosis [3, 5]. Eyelid manifestations ofsarcoidosis include “millet seed” nodules, ulcerated nodules,plaques, and swollen eyelids [4]. In 1986, Jabs and Johnsdescribed eyelid nodules in 5/183 patients (0.27%) with sys-temic sarcoidosis [1]; however, a 2007 review of 26 patientswith sarcoidosis and orbital disease found eyelid involve-ment in 11.5% of patients [2]. The actual incidence of eyelidCase Reports in Ophthalmological Medicine 3involvement remains uncertain. In addition, limited data hasbeen published on the correlation of eyelid involvement anddisease severity. For example, our patient had severe systemicsarcoidosis resistant to aggressive immunosuppressive andimmunomodulating therapy.A Pubmed literature search (http://www.ncbi.nlm.nih.gov/pubmed/, accessed 10 March 2013) of “eyelid and sar-coid” indicates one previous case [6] of involvement of boththe anterior and posterior lamellae of the eyelids. In 2001Moin et al. first described destructive full-thickness sar-coidosis lesions of the eyelids [6]. The authors showed ahigh magnification image of a small segment of tissue; how-ever, an oriented figure with the full-thickness microscopicanatomy of the eyelid was absent [6]. To our knowledge,a full-thickness histologic depiction of an eyelid showingdestructive lesions in sarcoidosis has not been previouslypublished. In addition, the complete obliteration of theeyelid meibomian glands by the sarcoidosis granulomatousinfiltrates seen in our case represents new findings. Dry eyesin patients with sarcoidosis are often attributed to sarcoidinfiltration of the lacrimal gland. Based on our findings,sarcoid destruction of the meibomian glands may representan additional etiology for dry eyes in sarcoidosis patients witheyelid involvement.Conflict of InterestsTheauthors have no financial or proprietary conflicts of inter-ests in the material presented.References[1] D. A. Jabs and C. J. Johns, “Ocular involvement in chronicsarcoidosis,” American Journal of Ophthalmology, vol. 102, no.3, pp. 297–301, 1986.[2] V. C. Prabhakaran, P. Saeed, B. Esmaeli et al., “Orbital andadnexal sarcoidosis,”Archives of Ophthalmology, vol. 125, no. 12,pp. 1657–1662, 2007.[3] C. D. Obenauf, H. E. Shaw, C. F. Sydnor, and G. K. Klintworth,“Sarcoidosis and its ophthalmicmanifestations,”American Jour-nal of Ophthalmology, vol. 86, no. 5, pp. 648–655, 1978.[4] S. Brownstein, A. D. Liszauer, W. D. Carey, and D. A. Nicolle,“Sarcoidosis of the eyelid skin,” Canadian Journal of Ophthal-mology, vol. 25, no. 5, pp. 256–259, 1990.[5] J. G. Hall and K. L. Cohen, “Sarcoidosis of the eyelid skin,”American Journal of Ophthalmology, vol. 119, no. 1, pp. 100–101,1995.[6] M. Moin, R. C. Kersten, F. Bernardini, and D. R. Kulwin,“Destructive eyelid lesions in sarcoidosis,” Ophthalmic Plasticand Reconstructive Surgery, vol. 17, no. 2, pp. 123–125, 2001.Submit your manuscripts athttp://www.hindawi.comStem CellsInternationalHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014MEDIATORSINFLAMMATIONofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Behavioural NeurologyEndocrinologyInternational Journal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Disease MarkersHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014BioMed Research InternationalOncologyJournal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Oxidative Medicine and Cellular LongevityHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014PPAR ResearchThe Scientific World JournalHindawi Publishing Corporation http://www.hindawi.com Volume 2014Immunology ResearchHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Journal ofObesityJournal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014 Computational and  Mathematical Methods in MedicineOphthalmologyJournal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Diabetes ResearchJournal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Research and TreatmentAIDSHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Gastroenterology Research and PracticeHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Parkinson’s DiseaseEvidence-Based Complementary and Alternative MedicineVolume 2014Hindawi Publishing Corporationhttp://www.hindawi.com

Full-thickness eyelid lesions in sarcoidosis

Eyelid involvement in sarcoidosis is very rare. A search of the medical literature indicates one previous report of sarcoidosis with destructive eyelid lesions. We describe the case of a 50-year-old woman with severe systemic sarcoidosis, which included her eyelids. To our knowledge, the case presented herein represents the first to show the full-thickness histopathology of destructive eyelid lesions in sarcoidosis.</jats:p

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Hindawi Publishing CorporationCase Reports in Ophthalmological MedicineVolume 2013, Article ID 579121, 3 pageshttp://dx.doi.org/10.1155/2013/579121Case ReportFull-Thickness Eyelid Lesions in SarcoidosisMegan E. Collins,1 Vesna Petronic-Rosic,2 Nadera J. Sweiss,3 and Marcus M. Marcet41 Department of Ophthalmology, University of Wisconsin, Madison, WI, USA2 Section of Dermatology, University of Chicago, Chicago, IL, USA3 Section of Rheumatology, University of Illinois, Chicago, IL, USA4Department of Ophthalmology, University of Hong Kong, Cyberport, Hong KongCorrespondence should be addressed to Marcus M. Marcet; marcet@hku.hkReceived 10 March 2013; Accepted 17 April 2013Academic Editors: N. Fuse, M. Rosner, and S. SchwartzCopyright © 2013 Megan E. Collins et al. This is an open access article distributed under the Creative Commons AttributionLicense, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properlycited.Eyelid involvement in sarcoidosis is very rare. A search of the medical literature indicates one previous report of sarcoidosis withdestructive eyelid lesions.We describe the case of a 50-year-oldwomanwith severe systemic sarcoidosis, which included her eyelids.To our knowledge, the case presented herein represents the first to show the full-thickness histopathology of destructive eyelidlesions in sarcoidosis.1. IntroductionSarcoidosis is a multisystem inflammatory disease of un-known etiology characterized histopathologically by non-caseating granulomas. The condition has a female predilec-tion with a peak incidence in the 3rd and 5th decades of life.Pulmonary involvement is the most common manifestation.Common extrapulmonary manifestations include the skin,central nervous system, liver, kidney, and musculoskeletalsystem. Furthermore, sarcoidosis can affect the heart, periph-eral nervous system, salivary glands, eye, and ocular adnexa.Ocular involvement occurs in 25%–60% of patients withsystemic sarcoidosis [1, 2]. While the disease can involve anyocular tissue, anterior uveitis and lacrimal gland involvementare themost frequently reported findings [3]. Eyelid manifes-tations of sarcoidosis include “millet seed” nodules, ulceratednodules, plaques, and swollen eyelids [4]. Eyelid involvementin sarcoidosis is very rare. To our knowledge, there is onlyone previous report in the literature that describes full-thickness eyelid involvement. Our case is the first to show thefull-thickness histopathology of destructive eyelid lesions insarcoidosis.2. Case ReportA 50-year-old female with severe systemic sarcoidosis, in-cluding pulmonary, skin, and joint disease, presented forevaluation of ocular involvement.The patient had noted eye-lid nodules for over 5 years; however, she recently developedeyelid deformities, tearing, and eye irritation.On examination, she had notching of her upper andlower eyelids and extensive scarring of the posterior lamellaleading to entropion and focal trichiasis (Figure 1). Slit lampexamination showed complete inspissation of themeibomianglands and superficial keratitis corresponding with misdi-rected eyelashes.The remainder of the anterior and posteriorsegment examination was within normal limits. Medicaltherapy did not adequately control the patient’s symptoms.Thepatient subsequently underwent full-thicknesswedgeresections in both upper eyelids and her right lower eyelidresulting in symptomatic improvement and elimination ofthe localized areas of trichiasis. Histopathologic evaluationof all three full-thickness wedge resections showed typical“naked,” noncaseating granulomas composed of epitheliodhistiocytes and multinucleated giant cells (Figure 2). Serialsections showed extensive destruction of both the anteriorand posterior lamellar anatomies, including derangement ofthe tarsal architecture, obliteration of the meibomian glands,and infiltration of the dermis and orbicularis oculi muscle.Entropic migration of the mucocutaneous junction and eye-lashmisdirectionwere also seen. Ziehl-Neelsen andGomori’smethenamine silver stains were negative formicroorganisms.2 Case Reports in Ophthalmological Medicine(a) (b)Figure 1: (a) Preoperative photograph of the patient showing eyelid notching, entropion, and trichiasis. Multiple sarcoid nodules on the faceare present. (b) Preoperative photograph of everted right upper eyelid showing focal trichiasis, cicatricial entropion, and thickened hyperemicconjunctival scarring. Inspissatedmeibomian glands are seen in themedial third of the eyelid.There is grossly apparent loss of themeibomianglands in the lateral two thirds of the eyelid.dotp(a) ∗(b)∗(c) (d)Figure 2: (a) Photomicrograph of sagittally sectioned full-thickness wedge resection showing a thickened eyelid with noncaseatinggranulomas infiltrating dermis, orbicularis oculi muscle, and tarsus. The mucocutaneous junction is seen (arrow). There is absence of themeibomian glands (hematoxylin-eosin, ×20). The eyelid is designed as follows: tp = tarsal plate; o = orbicularis oculi muscle; d = dermis.(b) Cross-section through the meibomian gland orifice (arrow) with sarcoidal obliteration of the meibomian glands (asterisk) (hematoxylin-eosin, ×100). (c) Tarsal (asterisk) granulomas withmultinucleated giant cells and normal overlying palpebral conjunctiva (hematoxylin-eosin,×200). (d)Highermagnification view of tarsus showing noncaseating granuloma containing epitheliod histiocytes (hematoxylin-eosin ×400).3. DiscussionEarly reports have limited discussion of the incidence of eye-lid involvement in sarcoidosis [3, 5]. Eyelid manifestations ofsarcoidosis include “millet seed” nodules, ulcerated nodules,plaques, and swollen eyelids [4]. In 1986, Jabs and Johnsdescribed eyelid nodules in 5/183 patients (0.27%) with sys-temic sarcoidosis [1]; however, a 2007 review of 26 patientswith sarcoidosis and orbital disease found eyelid involve-ment in 11.5% of patients [2]. The actual incidence of eyelidCase Reports in Ophthalmological Medicine 3involvement remains uncertain. In addition, limited data hasbeen published on the correlation of eyelid involvement anddisease severity. For example, our patient had severe systemicsarcoidosis resistant to aggressive immunosuppressive andimmunomodulating therapy.A Pubmed literature search (http://www.ncbi.nlm.nih.gov/pubmed/, accessed 10 March 2013) of “eyelid and sar-coid” indicates one previous case [6] of involvement of boththe anterior and posterior lamellae of the eyelids. In 2001Moin et al. first described destructive full-thickness sar-coidosis lesions of the eyelids [6]. The authors showed ahigh magnification image of a small segment of tissue; how-ever, an oriented figure with the full-thickness microscopicanatomy of the eyelid was absent [6]. To our knowledge,a full-thickness histologic depiction of an eyelid showingdestructive lesions in sarcoidosis has not been previouslypublished. In addition, the complete obliteration of theeyelid meibomian glands by the sarcoidosis granulomatousinfiltrates seen in our case represents new findings. Dry eyesin patients with sarcoidosis are often attributed to sarcoidinfiltration of the lacrimal gland. Based on our findings,sarcoid destruction of the meibomian glands may representan additional etiology for dry eyes in sarcoidosis patients witheyelid involvement.Conflict of InterestsTheauthors have no financial or proprietary conflicts of inter-ests in the material presented.References[1] D. A. Jabs and C. J. Johns, “Ocular involvement in chronicsarcoidosis,” American Journal of Ophthalmology, vol. 102, no.3, pp. 297–301, 1986.[2] V. C. Prabhakaran, P. Saeed, B. Esmaeli et al., “Orbital andadnexal sarcoidosis,”Archives of Ophthalmology, vol. 125, no. 12,pp. 1657–1662, 2007.[3] C. D. Obenauf, H. E. Shaw, C. F. Sydnor, and G. K. Klintworth,“Sarcoidosis and its ophthalmicmanifestations,”American Jour-nal of Ophthalmology, vol. 86, no. 5, pp. 648–655, 1978.[4] S. Brownstein, A. D. Liszauer, W. D. Carey, and D. A. Nicolle,“Sarcoidosis of the eyelid skin,” Canadian Journal of Ophthal-mology, vol. 25, no. 5, pp. 256–259, 1990.[5] J. G. Hall and K. L. Cohen, “Sarcoidosis of the eyelid skin,”American Journal of Ophthalmology, vol. 119, no. 1, pp. 100–101,1995.[6] M. Moin, R. C. Kersten, F. Bernardini, and D. R. Kulwin,“Destructive eyelid lesions in sarcoidosis,” Ophthalmic Plasticand Reconstructive Surgery, vol. 17, no. 2, pp. 123–125, 2001.Submit your manuscripts athttp://www.hindawi.comStem CellsInternationalHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014MEDIATORSINFLAMMATIONofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Behavioural NeurologyEndocrinologyInternational Journal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Disease MarkersHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014BioMed Research InternationalOncologyJournal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Oxidative Medicine and Cellular LongevityHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014PPAR ResearchThe Scientific World JournalHindawi Publishing Corporation http://www.hindawi.com Volume 2014Immunology ResearchHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Journal ofObesityJournal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014 Computational and  Mathematical Methods in MedicineOphthalmologyJournal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Diabetes ResearchJournal ofHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Hindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Research and TreatmentAIDSHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Gastroenterology Research and PracticeHindawi Publishing Corporationhttp://www.hindawi.com Volume 2014Parkinson’s DiseaseEvidence-Based Complementary and Alternative MedicineVolume 2014Hindawi Publishing Corporationhttp://www.hindawi.com

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Full-thickness eyelid lesions in sarcoidosis

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