Immune checkpoint inhibitors (ICIs)-related ocular myositis

We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal...
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Autor*in:	Garibaldi, Matteo [verfasserIn] Calabrò, Fabio [verfasserIn] Merlonghi, Gioia [verfasserIn] Pugliese, Silvia [verfasserIn] Ceccanti, Marco [verfasserIn] Cristiano, Lara [verfasserIn] Tartaglione, Tommaso [verfasserIn] Petrucci, Antonio [verfasserIn]

Format:	E-Artikel
Sprache:	Englisch

Erschienen:	2020

Schlagwörter:	Ocular myositis Immune checkpoint inhibitors-related Myositis (irMyositis) Idiopathic inflammatory myopathies Immune checkpoint inhibitors (ICIs) Anti-PD-1 Pembrolizumab

Übergeordnetes Werk:	Enthalten in: Neuromuscular disorders - Amsterdam [u.a.] : Elsevier Science, 1991, 30, Seite 420-423
Übergeordnetes Werk:	volume:30 ; pages:420-423

DOI / URN:	10.1016/j.nmd.2020.02.013

Katalog-ID:	ELV004229223

Internformat


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100	1		\|a Garibaldi, Matteo \|e verfasserin \|0 (orcid)0000-0003-1830-2886 \|4 aut
245	1	0	\|a Immune checkpoint inhibitors (ICIs)-related ocular myositis
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520			\|a We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms.
650		4	\|a Ocular myositis
650		4	\|a Immune checkpoint inhibitors-related Myositis (irMyositis)
650		4	\|a Idiopathic inflammatory myopathies
650		4	\|a Immune checkpoint inhibitors (ICIs)
650		4	\|a Anti-PD-1
650		4	\|a Pembrolizumab
700	1		\|a Calabrò, Fabio \|e verfasserin \|4 aut
700	1		\|a Merlonghi, Gioia \|e verfasserin \|0 (orcid)0000-0001-6790-8417 \|4 aut
700	1		\|a Pugliese, Silvia \|e verfasserin \|4 aut
700	1		\|a Ceccanti, Marco \|e verfasserin \|4 aut
700	1		\|a Cristiano, Lara \|e verfasserin \|4 aut
700	1		\|a Tartaglione, Tommaso \|e verfasserin \|4 aut
700	1		\|a Petrucci, Antonio \|e verfasserin \|4 aut
773	0	8	\|i Enthalten in \|t Neuromuscular disorders \|d Amsterdam [u.a.] : Elsevier Science, 1991 \|g 30, Seite 420-423 \|h Online-Ressource \|w (DE-627)320469093 \|w (DE-600)2008287-3 \|w (DE-576)094505535 \|x 1873-2364 \|7 nnns
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912			\|a GBV_ILN_2005
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912			\|a GBV_ILN_2014
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912			\|a GBV_ILN_2020
912			\|a GBV_ILN_2021
912			\|a GBV_ILN_2025
912			\|a GBV_ILN_2027
912			\|a GBV_ILN_2034
912			\|a GBV_ILN_2038
912			\|a GBV_ILN_2044
912			\|a GBV_ILN_2048
912			\|a GBV_ILN_2049
912			\|a GBV_ILN_2050
912			\|a GBV_ILN_2056
912			\|a GBV_ILN_2059
912			\|a GBV_ILN_2061
912			\|a GBV_ILN_2064
912			\|a GBV_ILN_2065
912			\|a GBV_ILN_2068
912			\|a GBV_ILN_2111
912			\|a GBV_ILN_2112
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912			\|a GBV_ILN_4126
912			\|a GBV_ILN_4242
912			\|a GBV_ILN_4251
912			\|a GBV_ILN_4305
912			\|a GBV_ILN_4313
912			\|a GBV_ILN_4323
912			\|a GBV_ILN_4324
912			\|a GBV_ILN_4325
912			\|a GBV_ILN_4326
912			\|a GBV_ILN_4333
912			\|a GBV_ILN_4334
912			\|a GBV_ILN_4335
912			\|a GBV_ILN_4338
912			\|a GBV_ILN_4393
936	b	k	\|a 44.90 \|j Neurologie
951			\|a AR
952			\|d 30 \|h 420-423

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author_variant	m g mg f c fc g m gm s p sp m c mc l c lc t t tt a p ap
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hierarchy_sort_str	2020
bklnumber	44.90
publishDate	2020
allfields	10.1016/j.nmd.2020.02.013 doi (DE-627)ELV004229223 (ELSEVIER)S0960-8966(20)30039-0 DE-627 ger DE-627 rda eng 610 DE-600 44.90 bkl Garibaldi, Matteo verfasserin (orcid)0000-0003-1830-2886 aut Immune checkpoint inhibitors (ICIs)-related ocular myositis 2020 nicht spezifiziert zzz rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms. Ocular myositis Immune checkpoint inhibitors-related Myositis (irMyositis) Idiopathic inflammatory myopathies Immune checkpoint inhibitors (ICIs) Anti-PD-1 Pembrolizumab Calabrò, Fabio verfasserin aut Merlonghi, Gioia verfasserin (orcid)0000-0001-6790-8417 aut Pugliese, Silvia verfasserin aut Ceccanti, Marco verfasserin aut Cristiano, Lara verfasserin aut Tartaglione, Tommaso verfasserin aut Petrucci, Antonio verfasserin aut Enthalten in Neuromuscular disorders Amsterdam [u.a.] : Elsevier Science, 1991 30, Seite 420-423 Online-Ressource (DE-627)320469093 (DE-600)2008287-3 (DE-576)094505535 1873-2364 nnns volume:30 pages:420-423 GBV_USEFLAG_U SYSFLAG_U GBV_ELV SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_32 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_90 GBV_ILN_95 GBV_ILN_100 GBV_ILN_101 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_224 GBV_ILN_370 GBV_ILN_602 GBV_ILN_702 GBV_ILN_2003 GBV_ILN_2004 GBV_ILN_2005 GBV_ILN_2011 GBV_ILN_2014 GBV_ILN_2015 GBV_ILN_2020 GBV_ILN_2021 GBV_ILN_2025 GBV_ILN_2027 GBV_ILN_2034 GBV_ILN_2038 GBV_ILN_2044 GBV_ILN_2048 GBV_ILN_2049 GBV_ILN_2050 GBV_ILN_2056 GBV_ILN_2059 GBV_ILN_2061 GBV_ILN_2064 GBV_ILN_2065 GBV_ILN_2068 GBV_ILN_2111 GBV_ILN_2112 GBV_ILN_2113 GBV_ILN_2118 GBV_ILN_2122 GBV_ILN_2129 GBV_ILN_2143 GBV_ILN_2147 GBV_ILN_2148 GBV_ILN_2152 GBV_ILN_2153 GBV_ILN_2190 GBV_ILN_2336 GBV_ILN_2507 GBV_ILN_2522 GBV_ILN_4035 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4242 GBV_ILN_4251 GBV_ILN_4305 GBV_ILN_4313 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4326 GBV_ILN_4333 GBV_ILN_4334 GBV_ILN_4335 GBV_ILN_4338 GBV_ILN_4393 44.90 Neurologie AR 30 420-423
spelling	10.1016/j.nmd.2020.02.013 doi (DE-627)ELV004229223 (ELSEVIER)S0960-8966(20)30039-0 DE-627 ger DE-627 rda eng 610 DE-600 44.90 bkl Garibaldi, Matteo verfasserin (orcid)0000-0003-1830-2886 aut Immune checkpoint inhibitors (ICIs)-related ocular myositis 2020 nicht spezifiziert zzz rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms. Ocular myositis Immune checkpoint inhibitors-related Myositis (irMyositis) Idiopathic inflammatory myopathies Immune checkpoint inhibitors (ICIs) Anti-PD-1 Pembrolizumab Calabrò, Fabio verfasserin aut Merlonghi, Gioia verfasserin (orcid)0000-0001-6790-8417 aut Pugliese, Silvia verfasserin aut Ceccanti, Marco verfasserin aut Cristiano, Lara verfasserin aut Tartaglione, Tommaso verfasserin aut Petrucci, Antonio verfasserin aut Enthalten in Neuromuscular disorders Amsterdam [u.a.] : Elsevier Science, 1991 30, Seite 420-423 Online-Ressource (DE-627)320469093 (DE-600)2008287-3 (DE-576)094505535 1873-2364 nnns volume:30 pages:420-423 GBV_USEFLAG_U SYSFLAG_U GBV_ELV SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_32 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_90 GBV_ILN_95 GBV_ILN_100 GBV_ILN_101 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_224 GBV_ILN_370 GBV_ILN_602 GBV_ILN_702 GBV_ILN_2003 GBV_ILN_2004 GBV_ILN_2005 GBV_ILN_2011 GBV_ILN_2014 GBV_ILN_2015 GBV_ILN_2020 GBV_ILN_2021 GBV_ILN_2025 GBV_ILN_2027 GBV_ILN_2034 GBV_ILN_2038 GBV_ILN_2044 GBV_ILN_2048 GBV_ILN_2049 GBV_ILN_2050 GBV_ILN_2056 GBV_ILN_2059 GBV_ILN_2061 GBV_ILN_2064 GBV_ILN_2065 GBV_ILN_2068 GBV_ILN_2111 GBV_ILN_2112 GBV_ILN_2113 GBV_ILN_2118 GBV_ILN_2122 GBV_ILN_2129 GBV_ILN_2143 GBV_ILN_2147 GBV_ILN_2148 GBV_ILN_2152 GBV_ILN_2153 GBV_ILN_2190 GBV_ILN_2336 GBV_ILN_2507 GBV_ILN_2522 GBV_ILN_4035 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4242 GBV_ILN_4251 GBV_ILN_4305 GBV_ILN_4313 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4326 GBV_ILN_4333 GBV_ILN_4334 GBV_ILN_4335 GBV_ILN_4338 GBV_ILN_4393 44.90 Neurologie AR 30 420-423
allfields_unstemmed	10.1016/j.nmd.2020.02.013 doi (DE-627)ELV004229223 (ELSEVIER)S0960-8966(20)30039-0 DE-627 ger DE-627 rda eng 610 DE-600 44.90 bkl Garibaldi, Matteo verfasserin (orcid)0000-0003-1830-2886 aut Immune checkpoint inhibitors (ICIs)-related ocular myositis 2020 nicht spezifiziert zzz rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms. Ocular myositis Immune checkpoint inhibitors-related Myositis (irMyositis) Idiopathic inflammatory myopathies Immune checkpoint inhibitors (ICIs) Anti-PD-1 Pembrolizumab Calabrò, Fabio verfasserin aut Merlonghi, Gioia verfasserin (orcid)0000-0001-6790-8417 aut Pugliese, Silvia verfasserin aut Ceccanti, Marco verfasserin aut Cristiano, Lara verfasserin aut Tartaglione, Tommaso verfasserin aut Petrucci, Antonio verfasserin aut Enthalten in Neuromuscular disorders Amsterdam [u.a.] : Elsevier Science, 1991 30, Seite 420-423 Online-Ressource (DE-627)320469093 (DE-600)2008287-3 (DE-576)094505535 1873-2364 nnns volume:30 pages:420-423 GBV_USEFLAG_U SYSFLAG_U GBV_ELV SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_32 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_90 GBV_ILN_95 GBV_ILN_100 GBV_ILN_101 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_224 GBV_ILN_370 GBV_ILN_602 GBV_ILN_702 GBV_ILN_2003 GBV_ILN_2004 GBV_ILN_2005 GBV_ILN_2011 GBV_ILN_2014 GBV_ILN_2015 GBV_ILN_2020 GBV_ILN_2021 GBV_ILN_2025 GBV_ILN_2027 GBV_ILN_2034 GBV_ILN_2038 GBV_ILN_2044 GBV_ILN_2048 GBV_ILN_2049 GBV_ILN_2050 GBV_ILN_2056 GBV_ILN_2059 GBV_ILN_2061 GBV_ILN_2064 GBV_ILN_2065 GBV_ILN_2068 GBV_ILN_2111 GBV_ILN_2112 GBV_ILN_2113 GBV_ILN_2118 GBV_ILN_2122 GBV_ILN_2129 GBV_ILN_2143 GBV_ILN_2147 GBV_ILN_2148 GBV_ILN_2152 GBV_ILN_2153 GBV_ILN_2190 GBV_ILN_2336 GBV_ILN_2507 GBV_ILN_2522 GBV_ILN_4035 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4242 GBV_ILN_4251 GBV_ILN_4305 GBV_ILN_4313 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4326 GBV_ILN_4333 GBV_ILN_4334 GBV_ILN_4335 GBV_ILN_4338 GBV_ILN_4393 44.90 Neurologie AR 30 420-423
allfieldsGer	10.1016/j.nmd.2020.02.013 doi (DE-627)ELV004229223 (ELSEVIER)S0960-8966(20)30039-0 DE-627 ger DE-627 rda eng 610 DE-600 44.90 bkl Garibaldi, Matteo verfasserin (orcid)0000-0003-1830-2886 aut Immune checkpoint inhibitors (ICIs)-related ocular myositis 2020 nicht spezifiziert zzz rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms. Ocular myositis Immune checkpoint inhibitors-related Myositis (irMyositis) Idiopathic inflammatory myopathies Immune checkpoint inhibitors (ICIs) Anti-PD-1 Pembrolizumab Calabrò, Fabio verfasserin aut Merlonghi, Gioia verfasserin (orcid)0000-0001-6790-8417 aut Pugliese, Silvia verfasserin aut Ceccanti, Marco verfasserin aut Cristiano, Lara verfasserin aut Tartaglione, Tommaso verfasserin aut Petrucci, Antonio verfasserin aut Enthalten in Neuromuscular disorders Amsterdam [u.a.] : Elsevier Science, 1991 30, Seite 420-423 Online-Ressource (DE-627)320469093 (DE-600)2008287-3 (DE-576)094505535 1873-2364 nnns volume:30 pages:420-423 GBV_USEFLAG_U SYSFLAG_U GBV_ELV SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_32 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_90 GBV_ILN_95 GBV_ILN_100 GBV_ILN_101 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_224 GBV_ILN_370 GBV_ILN_602 GBV_ILN_702 GBV_ILN_2003 GBV_ILN_2004 GBV_ILN_2005 GBV_ILN_2011 GBV_ILN_2014 GBV_ILN_2015 GBV_ILN_2020 GBV_ILN_2021 GBV_ILN_2025 GBV_ILN_2027 GBV_ILN_2034 GBV_ILN_2038 GBV_ILN_2044 GBV_ILN_2048 GBV_ILN_2049 GBV_ILN_2050 GBV_ILN_2056 GBV_ILN_2059 GBV_ILN_2061 GBV_ILN_2064 GBV_ILN_2065 GBV_ILN_2068 GBV_ILN_2111 GBV_ILN_2112 GBV_ILN_2113 GBV_ILN_2118 GBV_ILN_2122 GBV_ILN_2129 GBV_ILN_2143 GBV_ILN_2147 GBV_ILN_2148 GBV_ILN_2152 GBV_ILN_2153 GBV_ILN_2190 GBV_ILN_2336 GBV_ILN_2507 GBV_ILN_2522 GBV_ILN_4035 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4242 GBV_ILN_4251 GBV_ILN_4305 GBV_ILN_4313 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4326 GBV_ILN_4333 GBV_ILN_4334 GBV_ILN_4335 GBV_ILN_4338 GBV_ILN_4393 44.90 Neurologie AR 30 420-423
allfieldsSound	10.1016/j.nmd.2020.02.013 doi (DE-627)ELV004229223 (ELSEVIER)S0960-8966(20)30039-0 DE-627 ger DE-627 rda eng 610 DE-600 44.90 bkl Garibaldi, Matteo verfasserin (orcid)0000-0003-1830-2886 aut Immune checkpoint inhibitors (ICIs)-related ocular myositis 2020 nicht spezifiziert zzz rdacontent Computermedien c rdamedia Online-Ressource cr rdacarrier We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms. Ocular myositis Immune checkpoint inhibitors-related Myositis (irMyositis) Idiopathic inflammatory myopathies Immune checkpoint inhibitors (ICIs) Anti-PD-1 Pembrolizumab Calabrò, Fabio verfasserin aut Merlonghi, Gioia verfasserin (orcid)0000-0001-6790-8417 aut Pugliese, Silvia verfasserin aut Ceccanti, Marco verfasserin aut Cristiano, Lara verfasserin aut Tartaglione, Tommaso verfasserin aut Petrucci, Antonio verfasserin aut Enthalten in Neuromuscular disorders Amsterdam [u.a.] : Elsevier Science, 1991 30, Seite 420-423 Online-Ressource (DE-627)320469093 (DE-600)2008287-3 (DE-576)094505535 1873-2364 nnns volume:30 pages:420-423 GBV_USEFLAG_U SYSFLAG_U GBV_ELV SSG-OLC-PHA GBV_ILN_20 GBV_ILN_22 GBV_ILN_23 GBV_ILN_24 GBV_ILN_31 GBV_ILN_32 GBV_ILN_40 GBV_ILN_60 GBV_ILN_62 GBV_ILN_63 GBV_ILN_65 GBV_ILN_69 GBV_ILN_70 GBV_ILN_73 GBV_ILN_74 GBV_ILN_90 GBV_ILN_95 GBV_ILN_100 GBV_ILN_101 GBV_ILN_105 GBV_ILN_110 GBV_ILN_151 GBV_ILN_224 GBV_ILN_370 GBV_ILN_602 GBV_ILN_702 GBV_ILN_2003 GBV_ILN_2004 GBV_ILN_2005 GBV_ILN_2011 GBV_ILN_2014 GBV_ILN_2015 GBV_ILN_2020 GBV_ILN_2021 GBV_ILN_2025 GBV_ILN_2027 GBV_ILN_2034 GBV_ILN_2038 GBV_ILN_2044 GBV_ILN_2048 GBV_ILN_2049 GBV_ILN_2050 GBV_ILN_2056 GBV_ILN_2059 GBV_ILN_2061 GBV_ILN_2064 GBV_ILN_2065 GBV_ILN_2068 GBV_ILN_2111 GBV_ILN_2112 GBV_ILN_2113 GBV_ILN_2118 GBV_ILN_2122 GBV_ILN_2129 GBV_ILN_2143 GBV_ILN_2147 GBV_ILN_2148 GBV_ILN_2152 GBV_ILN_2153 GBV_ILN_2190 GBV_ILN_2336 GBV_ILN_2507 GBV_ILN_2522 GBV_ILN_4035 GBV_ILN_4037 GBV_ILN_4112 GBV_ILN_4125 GBV_ILN_4126 GBV_ILN_4242 GBV_ILN_4251 GBV_ILN_4305 GBV_ILN_4313 GBV_ILN_4323 GBV_ILN_4324 GBV_ILN_4325 GBV_ILN_4326 GBV_ILN_4333 GBV_ILN_4334 GBV_ILN_4335 GBV_ILN_4338 GBV_ILN_4393 44.90 Neurologie AR 30 420-423
language	English
source	Enthalten in Neuromuscular disorders 30, Seite 420-423 volume:30 pages:420-423
sourceStr	Enthalten in Neuromuscular disorders 30, Seite 420-423 volume:30 pages:420-423
format_phy_str_mv	Article
bklname	Neurologie
institution	findex.gbv.de
topic_facet	Ocular myositis Immune checkpoint inhibitors-related Myositis (irMyositis) Idiopathic inflammatory myopathies Immune checkpoint inhibitors (ICIs) Anti-PD-1 Pembrolizumab
dewey-raw	610
isfreeaccess_bool	false
container_title	Neuromuscular disorders
authorswithroles_txt_mv	Garibaldi, Matteo @@aut@@ Calabrò, Fabio @@aut@@ Merlonghi, Gioia @@aut@@ Pugliese, Silvia @@aut@@ Ceccanti, Marco @@aut@@ Cristiano, Lara @@aut@@ Tartaglione, Tommaso @@aut@@ Petrucci, Antonio @@aut@@
publishDateDaySort_date	2020-01-01T00:00:00Z
hierarchy_top_id	320469093
dewey-sort	3610
id	ELV004229223
language_de	englisch
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author	Garibaldi, Matteo
spellingShingle	Garibaldi, Matteo ddc 610 bkl 44.90 misc Ocular myositis misc Immune checkpoint inhibitors-related Myositis (irMyositis) misc Idiopathic inflammatory myopathies misc Immune checkpoint inhibitors (ICIs) misc Anti-PD-1 misc Pembrolizumab Immune checkpoint inhibitors (ICIs)-related ocular myositis
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topic_title	610 DE-600 44.90 bkl Immune checkpoint inhibitors (ICIs)-related ocular myositis Ocular myositis Immune checkpoint inhibitors-related Myositis (irMyositis) Idiopathic inflammatory myopathies Immune checkpoint inhibitors (ICIs) Anti-PD-1 Pembrolizumab
topic	ddc 610 bkl 44.90 misc Ocular myositis misc Immune checkpoint inhibitors-related Myositis (irMyositis) misc Idiopathic inflammatory myopathies misc Immune checkpoint inhibitors (ICIs) misc Anti-PD-1 misc Pembrolizumab
topic_unstemmed	ddc 610 bkl 44.90 misc Ocular myositis misc Immune checkpoint inhibitors-related Myositis (irMyositis) misc Idiopathic inflammatory myopathies misc Immune checkpoint inhibitors (ICIs) misc Anti-PD-1 misc Pembrolizumab
topic_browse	ddc 610 bkl 44.90 misc Ocular myositis misc Immune checkpoint inhibitors-related Myositis (irMyositis) misc Idiopathic inflammatory myopathies misc Immune checkpoint inhibitors (ICIs) misc Anti-PD-1 misc Pembrolizumab
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title	Immune checkpoint inhibitors (ICIs)-related ocular myositis
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title_full	Immune checkpoint inhibitors (ICIs)-related ocular myositis
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author_browse	Garibaldi, Matteo Calabrò, Fabio Merlonghi, Gioia Pugliese, Silvia Ceccanti, Marco Cristiano, Lara Tartaglione, Tommaso Petrucci, Antonio
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title_sort	immune checkpoint inhibitors (icis)-related ocular myositis
title_auth	Immune checkpoint inhibitors (ICIs)-related ocular myositis
abstract	We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms.
abstractGer	We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms.
abstract_unstemmed	We present extensive clinical, serological, morphological and muscle imaging data of a 66-year-old man with isolated bilateral ptosis and external ophthalmoplegia secondary to Immune checkpoint inhibitors (Pembrolizumab). He had elevated CK level (>5000 UI/L). No facial, bulbar, proximal, distal or axial muscular weakness was observed. Electromyography (EMG) showed myopathic pattern, with spontaneous activity. Myositis specific antibodies and anti-striational antibodies were negative. Cardiac and respiratory functions were preserved. Skeletal muscle MRI was unremarkable, whereas extraocular muscles revealed bilateral hyperintensities in inferior rectus, medial rectus and superior oblique muscles in both T1 and STIR sequences, with mild muscle atrophy. Muscle biopsy showed endomysial inflammatory infiltrates, MHC-1 expression was observed in clusters of non-necrotic cells. CD56 positive cells were observed in perifascicular regions. Patient discontinued Pembrolizumab and received corticosteroid treatment with progressive clinical improvement and CK normalization. Our findings support this clinical entity, suggesting that isolated ocular myositis represents a subgroup of generalised myositis with predominant ocular symptoms.
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title_short	Immune checkpoint inhibitors (ICIs)-related ocular myositis
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author2	Calabrò, Fabio Merlonghi, Gioia Pugliese, Silvia Ceccanti, Marco Cristiano, Lara Tartaglione, Tommaso Petrucci, Antonio
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up_date	2024-07-06T22:17:14.761Z
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Immune checkpoint inhibitors (ICIs)-related ocular myositis

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