Wall-eyed bilateral internuclear ophthalmoplegia (webino syndrome) and myelopathy in pyoderma gangrenosum

Abstract

A 35-year-old female with pyoderma gangrenosum developed paraparesis with a sensory level at L1. Three months later she complained of diplopia and was found to have bilateral internuclear ophthalmoplegia with exotropia and no ocular convergence. The term Webino syndrome has been coined to design this set of neuro-opthalmologic findings. Although it was initially attributed to lesions affecting the medial longitudinal fasciculus and the medial rectus subnuclei of the oculomotor complex in the midbrain the exact location of the lesion is still disputed. In the present case both myelopathy and Webino syndrome were probably due to vascular occlusive disease resulting from central nervous system vasculitis occurring in concomitance to pyoderma gangrenosum.

Case report: A variant of wall-eyed bilateral internuclear ophthalmoplegia from unilateral pons infarction

“…Studies have shown that the disease is often associated with brainstem ischemia and hydrocephalus, immunotherapy of tumors, and diseases such as multiple sclerosis. This study summarized 39 case reports, including 42 WEBINO patients (Inocencio and Ballecer, 1985;Lana et al, 1990;Fay and Strominger, 1999;Korkmaz et al, 2002;Ozer et al, 2005;Chen and Lin, 2007;Kim et al, 2008;Matsumoto et al, 2008Matsumoto et al, , 2019Ushio et al, 2008;Jacob et al, 2010;Sierra-Hidalgo et al, 2010;Shinoda et al, 2011;Beh and Frohman, 2012;Jadhav and Prasad, 2012;Sakamoto et al, 2012;Bar et al, 2014;Chakravarthi et al, 2014;de Mora et al, 2014;Mathis et al, 2014;Muralidhar et al, 2014;Nakajima et al, 2014;Toufeeq and Dave, 2014;Agarwal et al, 2015;Ljevak et al, 2015;Man et al, 2015;de Souza et al, 2017;Sajjadi et al, 2017;Zou and Chen, 2017;Keereman et al, 2018;Papageorgiou et al, 2018;Im et al, 2020;Vázquez-Justes et al, 2020;Yazdi et al, 2020;Ansari et al, 2021;Jo et al, 2021;Petrik et al, 2021;Sinha et al, 2021;Wako et al, 2022). The age range of the ...…”

Case report: A variant of wall-eyed bilateral internuclear ophthalmoplegia from unilateral pons infarction

“…Studies have shown that the disease is often associated with brainstem ischemia and hydrocephalus, immunotherapy of tumors, and diseases such as multiple sclerosis. This study summarized 39 case reports, including 42 WEBINO patients (Inocencio and Ballecer, 1985;Lana et al, 1990;Fay and Strominger, 1999;Korkmaz et al, 2002;Ozer et al, 2005;Chen and Lin, 2007;Kim et al, 2008;Matsumoto et al, 2008Matsumoto et al, , 2019Ushio et al, 2008;Jacob et al, 2010;Sierra-Hidalgo et al, 2010;Shinoda et al, 2011;Beh and Frohman, 2012;Jadhav and Prasad, 2012;Sakamoto et al, 2012;Bar et al, 2014;Chakravarthi et al, 2014;de Mora et al, 2014;Mathis et al, 2014;Muralidhar et al, 2014;Nakajima et al, 2014;Toufeeq and Dave, 2014;Agarwal et al, 2015;Ljevak et al, 2015;Man et al, 2015;de Souza et al, 2017;Sajjadi et al, 2017;Zou and Chen, 2017;Keereman et al, 2018;Papageorgiou et al, 2018;Im et al, 2020;Vázquez-Justes et al, 2020;Yazdi et al, 2020;Ansari et al, 2021;Jo et al, 2021;Petrik et al, 2021;Sinha et al, 2021;Wako et al, 2022). The age range of the ...…”

Fatal Orbital Pyoderma Gangrenosum with Central Nervous System Involvement

“…1,7 There are a few previous case reports of central nervous system involvement in patients with PG, although usually representing independent foci of attainment. 8,9 Malignant pyoderma is a term used to describe a rapidly evolving ulcer in the head and neck without previous history of systemic disorders, which is considered by many authors to be a rare subtype of PG. 5,10,11 This variant may show some particularities, namely the absence of undermined borders, worse response to treatment and more frequent relapses.…”

Case report: A variant of wall-eyed bilateral internuclear ophthalmoplegia from unilateral pons infarction

“…Studies have shown that the disease is often associated with brainstem ischemia and hydrocephalus, immunotherapy of tumors, and diseases such as multiple sclerosis. This study summarized 39 case reports, including 42 WEBINO patients (Inocencio and Ballecer, 1985;Lana et al, 1990;Fay and Strominger, 1999;Korkmaz et al, 2002;Ozer et al, 2005;Chen and Lin, 2007;Kim et al, 2008;Matsumoto et al, 2008Matsumoto et al, , 2019Ushio et al, 2008;Jacob et al, 2010;Sierra-Hidalgo et al, 2010;Shinoda et al, 2011;Beh and Frohman, 2012;Jadhav and Prasad, 2012;Sakamoto et al, 2012;Bar et al, 2014;Chakravarthi et al, 2014;de Mora et al, 2014;Mathis et al, 2014;Muralidhar et al, 2014;Nakajima et al, 2014;Toufeeq and Dave, 2014;Agarwal et al, 2015;Ljevak et al, 2015;Man et al, 2015;de Souza et al, 2017;Sajjadi et al, 2017;Zou and Chen, 2017;Keereman et al, 2018;Papageorgiou et al, 2018;Im et al, 2020;Vázquez-Justes et al, 2020;Yazdi et al, 2020;Ansari et al, 2021;Jo et al, 2021;Petrik et al, 2021;Sinha et al, 2021;Wako et al, 2022). The age range of the ...…”

Fatal Orbital Pyoderma Gangrenosum with Central Nervous System Involvement

“…1,7 There are a few previous case reports of central nervous system involvement in patients with PG, although usually representing independent foci of attainment. 8,9 Malignant pyoderma is a term used to describe a rapidly evolving ulcer in the head and neck without previous history of systemic disorders, which is considered by many authors to be a rare subtype of PG. 5,10,11 This variant may show some particularities, namely the absence of undermined borders, worse response to treatment and more frequent relapses.…”

Case report: A variant of wall-eyed bilateral internuclear ophthalmoplegia from unilateral pons infarction

“…Studies have shown that the disease is often associated with brainstem ischemia and hydrocephalus, immunotherapy of tumors, and diseases such as multiple sclerosis. This study summarized 39 case reports, including 42 WEBINO patients (Inocencio and Ballecer, 1985;Lana et al, 1990;Fay and Strominger, 1999;Korkmaz et al, 2002;Ozer et al, 2005;Chen and Lin, 2007;Kim et al, 2008;Matsumoto et al, 2008Matsumoto et al, , 2019Ushio et al, 2008;Jacob et al, 2010;Sierra-Hidalgo et al, 2010;Shinoda et al, 2011;Beh and Frohman, 2012;Jadhav and Prasad, 2012;Sakamoto et al, 2012;Bar et al, 2014;Chakravarthi et al, 2014;de Mora et al, 2014;Mathis et al, 2014;Muralidhar et al, 2014;Nakajima et al, 2014;Toufeeq and Dave, 2014;Agarwal et al, 2015;Ljevak et al, 2015;Man et al, 2015;de Souza et al, 2017;Sajjadi et al, 2017;Zou and Chen, 2017;Keereman et al, 2018;Papageorgiou et al, 2018;Im et al, 2020;Vázquez-Justes et al, 2020;Yazdi et al, 2020;Ansari et al, 2021;Jo et al, 2021;Petrik et al, 2021;Sinha et al, 2021;Wako et al, 2022). The age range of the ...…”

Fatal Orbital Pyoderma Gangrenosum with Central Nervous System Involvement

“…1,7 There are a few previous case reports of central nervous system involvement in patients with PG, although usually representing independent foci of attainment. 8,9 Malignant pyoderma is a term used to describe a rapidly evolving ulcer in the head and neck without previous history of systemic disorders, which is considered by many authors to be a rare subtype of PG. 5,10,11 This variant may show some particularities, namely the absence of undermined borders, worse response to treatment and more frequent relapses.…”