- Email: [email protected]
Orbital myositis and psoriatic arthritis
Correspondence expressed in keratoconic corneas: ribosomal protein S6 kinase 70-kDa (RPS6KB1) and FKBP12-rapamycin complex-associated protein 1 (FRAP1). Therefore, these genes, or downstream target molecules of the proteins they encode, may represent candidates for KC. Clinically, we propose examining patients for keratoconus, including corneal topography, should form part of the ocular assessment when examining patients with TS. Douglas AM Lyall,* Edward S. Tobias,† Sathish Srinivasan,* Colin Willoughby‡ From the *Department of Ophthalmology, University Hospital
Ayr, Ayrshire and Arran NHS, Ayr, Scotland; the †Institute of Medical Genetics, College of Medical, Veterinary and Life Sciences, University of Glasgow, Glasgow, Scotland; and the ‡ Centre for Vision and Vascular Science, Queen’s University Belfast, Northern Ireland, U.K. Fig. 2—Functional and physical protein interactions of TSC1 and TCS2 identified using STRING.
Correspondence to: Douglas AM Lyall, MRCOphth: [email protected] REFERENCES
directly with the TSC1/TSC2 complex, the functional and physical protein interactions of TSC1 and TSC2 were identified using the online database and web resource STRING (Search Tool for the Retrieval of Interacting Genes/Proteins: string.embl.de/) (Fig. 2). The expression of the genes encoding these TSC1 and TSC2 interacting proteins was determined using NEIBank, a National Eye Institute database of assembled expressed sequence tag gene expression data from eye tissue libraries (neibank. nei.nih.gov/index.shtml). The NEI human keratoconus cDNA library (NbLib0073) was also searched to determine expression of these genes in KC.5 Two genes that encode proteins that interact with TSC1 and TSC2 are
Orbital myositis and psoriatic arthritis We report a case of orbital myositis (OM) associated with psoriatic arthritis (PA). This association has only been reported once before in a child, although OM has been more frequently described in conjunction with Crohn’s and other autoimmune diseases. A 36-year-old male with a 2-year history of seronegative PA presented with acute onset binocular diplopia. His PA included skin lesions, arthritis of his back, and left wrist where magnetic resonance imaging (MRI) showed extensive synovitis, bone marrow edema, and early erosive change of the joints. On first presentation, ophthalmological findings included a convergent strabismus with limitation of right eye abduction, painful oculomotility, and 3 mm of right proptosis with medial displacement. Routine blood test investigations were normal. Antineutrophil cytoplasm antibodies (ANCA), antinuclear antibodies
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CAN J OPHTHALMOL—VOL. 47, NO. 6, DECEMBER 2012
1. Rabinowitz YS. Keratoconus. Surv Ophthalmol. 1998;42:297-319. 2. Crino PB, Nathanson KL, Henske EP. The tuberous sclerosis complex. N Engl J Med. 2006;355:1345-56. 3. Ross AT, Dickerson WW. Tuberous sclerosis. Arch Neurol Psychiatry. 1943;50:233-57. 4. Wong R, Rajendram R, Poole TR. Keratoconus in tuberous sclerosis. Eye (London, England). 2007;21:287-8. 5. Rabinowitz YS, Dong L, Wistow G. Gene expression profile studies of human keratoconus cornea for NEIBank: a novel cornea-expressed gene and the absence of transcripts for aquaporin 5. Invest Ophthalmol Vis Sci. 2005;46:1239-46. Can J Ophthalmol 2012;47:e41– e42
0008-4182/11/$-see front matter © 2012 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. dx.doi.org/10.1016/j.jcjo.2012.03.018
(ANA), human leukocyte antigens (HLA) B27, immunoglobulins, complement C3 and C4, rheumatoid factor, and DNA antibodies were negative. Thyroid microsomal antibodies, acetylcholine receptor auto-antibodies, thyroid hormone level, Lyme serology, sVDRL, and angiotensin-converting enzyme (ACE) level were all normal. Chest x-ray was normal. Computed tomography scan and MRI of the orbits (Fig. 1) showed an enhancing thickened right lateral rectus involving the tendon. In view of the presence of ophthalmic symptoms despite oral NSAID, he was started on 60 mg of prednisolone daily for 1 week with progressive dose reduction over 1 month. His symptoms improved initially but relapsed and further attempt with smoother dose reduction over 6 months failed for doses ⬍ 5 mg. A biopsy of the right lateral rectus was then carried out. It showed a nonspecific, nongranulomatous, chronic inflammation but specific features could have been altered
Correspondence Only 1 report in the literature has mentioned this association before in a child.4 The 9-year-old patient presented with similar episodes of left eye pain associated with reduced eye movements and diplopia. Several muscles were involved in the inflammation. She responded well to nonsteroidal antiinflammatory medication with no relapse after 3 months but residual impairment of right ocular motility. A case of inflammation of the left trochlea tendon complex of the superior oblique muscle mimicking a Brown’s syndrome was also reported previously and authors suggested the tendon inflammation was related to the psoriasis.5 OM has been reported in the literature in association with other autoimmune diseases and this is the second case report of its occurrence in conjunction with a psoriatic arthritis. Isabelle Ssi-Yan-Kai, Andrew Pearson Royal Berkshire Hospital-NHS Foundation Trust, Ophthalmology, Reading, UK Fig. 1—CT-Scan of the orbits: in the left orbit, the right lateral rectus and tendon appears enhanced and thickened.
by the prior treatment with steroids. Repeat biological tests remained normal. Orbital inflammatory disease (OID) involves a variety of pathologic processes and clinical presentations. OID may be idiopathic or secondary to infectious disease, trauma or retained foreign body, orbital neoplastic disease, or systemic inflammatory disease.1 In this case, none of these etiologies was identified from the clinical assessment, biological investigations, or histology. The patient’s history of PA was the only relevant clinical factor. PA belongs to the group of auto-immune disorders such as Crohn’s disease, rheumatoid arthritis, systemic lupus, or ankylosing spondylitis where OM has been reported.2 PA, in common with other auto-immune disorders, may produce abnormal pro-inflammatory mediators3 that could possibly affect the orbit with similar mechanisms.
Devastating herpes zoster ophthalmicus following orbital decompression Herpes zoster ophthalmicus (HZO) occurs with reactivation of latent varicella zoster virus harbored in the trigeminal ganglion as cell-mediated immunity wanes. Age, medical immunosuppression with steroids or chemotherapy, hematologic malignancy, and HIV are recognized risk factors. We present a case of devastating HZO that occurred after orbital decompression. Surgical stress and use of oral prednisone in the postoperative period are believed to have contributed to reduced cellmediated immunity.
Correspondence to: Isabelle Ssi-Yan-Kai, MD: [email protected]
REFERENCES 1. Gordon LK. Diagnostic dilemmas in orbital inflammatory disease. Ocul Immunol Inflamm. 2003;11:3-15. 2. Squires RH Jr, Zwiener RJ, Kennedy RH. Orbital myositis and Crohn’s disease. J Pediatr Gastroenterol Nutr. 1992;15:448-51. 3. Gordon LK. Orbital inflammatory disease: a diagnostic and therapeutic challenge. Eye (Lond). 2006;20:1196-206. 4. Ajitsaria R, Dale R, Ferguson V, Mayou S, Cavanagh N. Psoriasis, psoriatic arthropathy and relapsing orbital myositis. Clin Exp Dermatol. 2001; 26:274-5. 5. Thorne JE, Volpe NJ, Liu GT. Magnetic resonance imaging of acquired Brown syndrome in a patient with psoriasis. Am J Ophthalmol. 1999;127:233-5. Can J Ophthalmol 2012;47:e42– e43
0008-4182/11/$-see front matter © 2012 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. dx.doi.org/10.1016/j.jcjo.2012.04.005
A 79-year-old female with a history of hyperthyroidism was experiencing frequent left globe luxation with increasing difficulty in repositioning the eye. An uneventful 2-wall (inferior and medial) decompression was carried out. A tapering course of oral prednisone was started at 60 mg daily. The patient had no problems until day 14 postoperatively when she developed a stabbing pain along the right superior orbital rim with tenderness to touch. Four days later, a full-blown episode of HZO was evident with vesicles in the distribution of the right ophthalmic nerve, severe panuveitis, complete ophthalmoplegia, and encephalitis (Figs. 1A, B). Magnetic resonance imaging revealed enhancement of the right optic nerve sheath. Intravenous CAN J OPHTHALMOL—VOL. 47, NO. 6, DECEMBER 2012
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Ayr, Ayrshire and Arran NHS, Ayr, Scotland; the †Institute of Medical Genetics, College of Medical, Veterinary and Life Sciences, University of Glasgow, Glasgow, Scotland; and the ‡ Centre for Vision and Vascular Science, Queen’s University Belfast, Northern Ireland, U.K. Fig. 2—Functional and physical protein interactions of TSC1 and TCS2 identified using STRING.
Correspondence to: Douglas AM Lyall, MRCOphth: [email protected] REFERENCES
directly with the TSC1/TSC2 complex, the functional and physical protein interactions of TSC1 and TSC2 were identified using the online database and web resource STRING (Search Tool for the Retrieval of Interacting Genes/Proteins: string.embl.de/) (Fig. 2). The expression of the genes encoding these TSC1 and TSC2 interacting proteins was determined using NEIBank, a National Eye Institute database of assembled expressed sequence tag gene expression data from eye tissue libraries (neibank. nei.nih.gov/index.shtml). The NEI human keratoconus cDNA library (NbLib0073) was also searched to determine expression of these genes in KC.5 Two genes that encode proteins that interact with TSC1 and TSC2 are
Orbital myositis and psoriatic arthritis We report a case of orbital myositis (OM) associated with psoriatic arthritis (PA). This association has only been reported once before in a child, although OM has been more frequently described in conjunction with Crohn’s and other autoimmune diseases. A 36-year-old male with a 2-year history of seronegative PA presented with acute onset binocular diplopia. His PA included skin lesions, arthritis of his back, and left wrist where magnetic resonance imaging (MRI) showed extensive synovitis, bone marrow edema, and early erosive change of the joints. On first presentation, ophthalmological findings included a convergent strabismus with limitation of right eye abduction, painful oculomotility, and 3 mm of right proptosis with medial displacement. Routine blood test investigations were normal. Antineutrophil cytoplasm antibodies (ANCA), antinuclear antibodies
e42
CAN J OPHTHALMOL—VOL. 47, NO. 6, DECEMBER 2012
1. Rabinowitz YS. Keratoconus. Surv Ophthalmol. 1998;42:297-319. 2. Crino PB, Nathanson KL, Henske EP. The tuberous sclerosis complex. N Engl J Med. 2006;355:1345-56. 3. Ross AT, Dickerson WW. Tuberous sclerosis. Arch Neurol Psychiatry. 1943;50:233-57. 4. Wong R, Rajendram R, Poole TR. Keratoconus in tuberous sclerosis. Eye (London, England). 2007;21:287-8. 5. Rabinowitz YS, Dong L, Wistow G. Gene expression profile studies of human keratoconus cornea for NEIBank: a novel cornea-expressed gene and the absence of transcripts for aquaporin 5. Invest Ophthalmol Vis Sci. 2005;46:1239-46. Can J Ophthalmol 2012;47:e41– e42
0008-4182/11/$-see front matter © 2012 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. dx.doi.org/10.1016/j.jcjo.2012.03.018
(ANA), human leukocyte antigens (HLA) B27, immunoglobulins, complement C3 and C4, rheumatoid factor, and DNA antibodies were negative. Thyroid microsomal antibodies, acetylcholine receptor auto-antibodies, thyroid hormone level, Lyme serology, sVDRL, and angiotensin-converting enzyme (ACE) level were all normal. Chest x-ray was normal. Computed tomography scan and MRI of the orbits (Fig. 1) showed an enhancing thickened right lateral rectus involving the tendon. In view of the presence of ophthalmic symptoms despite oral NSAID, he was started on 60 mg of prednisolone daily for 1 week with progressive dose reduction over 1 month. His symptoms improved initially but relapsed and further attempt with smoother dose reduction over 6 months failed for doses ⬍ 5 mg. A biopsy of the right lateral rectus was then carried out. It showed a nonspecific, nongranulomatous, chronic inflammation but specific features could have been altered
Correspondence Only 1 report in the literature has mentioned this association before in a child.4 The 9-year-old patient presented with similar episodes of left eye pain associated with reduced eye movements and diplopia. Several muscles were involved in the inflammation. She responded well to nonsteroidal antiinflammatory medication with no relapse after 3 months but residual impairment of right ocular motility. A case of inflammation of the left trochlea tendon complex of the superior oblique muscle mimicking a Brown’s syndrome was also reported previously and authors suggested the tendon inflammation was related to the psoriasis.5 OM has been reported in the literature in association with other autoimmune diseases and this is the second case report of its occurrence in conjunction with a psoriatic arthritis. Isabelle Ssi-Yan-Kai, Andrew Pearson Royal Berkshire Hospital-NHS Foundation Trust, Ophthalmology, Reading, UK Fig. 1—CT-Scan of the orbits: in the left orbit, the right lateral rectus and tendon appears enhanced and thickened.
by the prior treatment with steroids. Repeat biological tests remained normal. Orbital inflammatory disease (OID) involves a variety of pathologic processes and clinical presentations. OID may be idiopathic or secondary to infectious disease, trauma or retained foreign body, orbital neoplastic disease, or systemic inflammatory disease.1 In this case, none of these etiologies was identified from the clinical assessment, biological investigations, or histology. The patient’s history of PA was the only relevant clinical factor. PA belongs to the group of auto-immune disorders such as Crohn’s disease, rheumatoid arthritis, systemic lupus, or ankylosing spondylitis where OM has been reported.2 PA, in common with other auto-immune disorders, may produce abnormal pro-inflammatory mediators3 that could possibly affect the orbit with similar mechanisms.
Devastating herpes zoster ophthalmicus following orbital decompression Herpes zoster ophthalmicus (HZO) occurs with reactivation of latent varicella zoster virus harbored in the trigeminal ganglion as cell-mediated immunity wanes. Age, medical immunosuppression with steroids or chemotherapy, hematologic malignancy, and HIV are recognized risk factors. We present a case of devastating HZO that occurred after orbital decompression. Surgical stress and use of oral prednisone in the postoperative period are believed to have contributed to reduced cellmediated immunity.
Correspondence to: Isabelle Ssi-Yan-Kai, MD: [email protected]
REFERENCES 1. Gordon LK. Diagnostic dilemmas in orbital inflammatory disease. Ocul Immunol Inflamm. 2003;11:3-15. 2. Squires RH Jr, Zwiener RJ, Kennedy RH. Orbital myositis and Crohn’s disease. J Pediatr Gastroenterol Nutr. 1992;15:448-51. 3. Gordon LK. Orbital inflammatory disease: a diagnostic and therapeutic challenge. Eye (Lond). 2006;20:1196-206. 4. Ajitsaria R, Dale R, Ferguson V, Mayou S, Cavanagh N. Psoriasis, psoriatic arthropathy and relapsing orbital myositis. Clin Exp Dermatol. 2001; 26:274-5. 5. Thorne JE, Volpe NJ, Liu GT. Magnetic resonance imaging of acquired Brown syndrome in a patient with psoriasis. Am J Ophthalmol. 1999;127:233-5. Can J Ophthalmol 2012;47:e42– e43
0008-4182/11/$-see front matter © 2012 Canadian Ophthalmological Society. Published by Elsevier Inc. All rights reserved. dx.doi.org/10.1016/j.jcjo.2012.04.005
A 79-year-old female with a history of hyperthyroidism was experiencing frequent left globe luxation with increasing difficulty in repositioning the eye. An uneventful 2-wall (inferior and medial) decompression was carried out. A tapering course of oral prednisone was started at 60 mg daily. The patient had no problems until day 14 postoperatively when she developed a stabbing pain along the right superior orbital rim with tenderness to touch. Four days later, a full-blown episode of HZO was evident with vesicles in the distribution of the right ophthalmic nerve, severe panuveitis, complete ophthalmoplegia, and encephalitis (Figs. 1A, B). Magnetic resonance imaging revealed enhancement of the right optic nerve sheath. Intravenous CAN J OPHTHALMOL—VOL. 47, NO. 6, DECEMBER 2012
e43