Idiopathic orbital inflammation (orbital inflammatory pseudotumour): an unusual cause of transient ischaemic attack.

Unilateral periorbital pain, cranial nerve palsies and a dramatic response to corticosteroid therapy are the hallmarks of clinical presentation in Tolosa-Hunt-Syndrome (THS) and Idiopathic Pseudotumor of the Orbit (IIPO). Both are unspecific chronic granulomatous diseases of unknown origin, sharing clinical as well as paraclinical characteristics. We observed two patients suffering from acute granulomatous IIPO, who also fulfilled the criteria of THS. Patient 1 developed leftsided infiltration of the medial ocular muscle with periorbital pain and cranial nerve palsy. After an initial response to corticosteroid therapy, contralateral relapse occurred with a THS-like infiltration of the sinus cavernosus and narrowing of the intracavernous internal carotid artery. Granulomatous infiltration of the right sinus cavernosus with secondary involvement of the ipsilateral nervus opticus and a slight exophthalmos was seen in Case 2. According to the literature, MRI and CT show identical signal intensity with different localisation: IIPO preferentially intra- and THS retroorbital. Apart from neuroradiological findings, almost similar histopathology and clinical presentation makes it difficult to distinguish between these two syndromes. Similarities between these two syndromes have been discussed for more than 20 years. Our two cases show their close relationship and we suggest that both diseases belong to the same pathological process.

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“…Apart from our Case 1, there is one further case report in the literature of carotid narrowing in IIPO proven by angiography [2].…”

Section: Discussionmentioning

confidence: 92%

“…Intracranial expansion as in Case 1 through the superior orbital fissure is known. To date only 19 cases of extraorbital IIPO have been reported [2]. Retro-orbital THS is characterised by inflammatory infiltration of the sinus cavernosus and/or the superior orbital fissure [7].…”

Section: Discussionmentioning

confidence: 99%

Idiopathic inflammatory pseudotumor of the orbit and Tolosa-Hunt Syndrome - are they the same disease?

Wasmeier

Pfadenhauer

Rösler

2002

Journal of Neurology

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“…Typically, there is a temporal sequence of events that demonstrates that the inflammatory process initiates within the orbit and then spills over adjacent structures. Kaye et al 7 and Borruat et al 12 adequately document in their case study that inflammation was originally confined to the orbital contents and extended with time beyond the orbit. In the cases reported by Frohman et al 2 and De Jesús et al, 13 the authors suggested that when more extensive orbital disease was seen compared with the extraorbital inflammation, its origin was probably within the orbit.…”

Section: Discussionmentioning

confidence: 95%

“…4 -7 To date, up to 15 patients with IOI have been reported with intracranial involvement (Table 1). 2,3,[5][6][7][8][9][10][11][12][13] In the vast majority of patients with idiopathic orbital and extraorbital inflammation, the cranial cavity or sinuses are usually involved (Table 1).…”

Section: Discussionmentioning

confidence: 99%

Sclerosing Inflammation in the Orbit and in the Pterygopalatine and Infratemporal Fossae

Cruz

Akaishi

Chahud

et al. 2003

Ophthalmic Plastic & Reconstructive Surgery

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“…El compromiso externo (compresivo) es raro, siendo pocas las causas reportadas que han causado un IL destacando por frecuencia las neoplasias de cabeza y cuello (meningioma, adenoma de hipófisis, histiocitoma fibrosos y neurilemoma del X nervio craneal) (9,10) , la trombosis del seno cavernoso (11) y la mucormicosis rino-orbito-cerebral (12) ; casi todas ellas comprometiendo a la ACI en su segmento intracavernoso. Existen escasos reportes de la EIOI como causa de enfermedad cerebrovascular (accidente isquémico transitorio o infarto cerebral) por compresión de la ACI (13,14,15) . Se realizó una búsqueda bibliográfica en Medline usando los términos "orbital pseudotumor", "idiopathic orbital inflammatory disease", "idiopathic orbital inflammation", "cerebral infarction", "ischemic stroke", "watershed infarction" y "borderzone infarct"; y en Lilacs y Scielo usando los términos "seudotumor orbiFigura 2.…”

Section: Discussionunclassified

Infarto cerebral limítrofe secundario a enfermedad inflamatoria orbitaria idiopática

et al. 2018

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Conflictos de interés: Los autores declaran no tener conflictos de interésResumen Los infartos limítrofes son aquellos ubicados en regiones entre dos territorios vasculares contiguos y la enfermedad inflamatoria orbitaria idiopática es un proceso inflamatorio orbitario inespecífico. Presentamos el caso de un varón de 60 años con exoftalmos, ptosis palpebral, disminución de agudeza visual derecha y afección de los nervios oculomotores derechos. Los estudios de imágenes mostraron una masa retro ocular derecha con invasión de los senos cavernosos y esfenoidal. Durante la hospitalización, el paciente sufrió un infarto cerebral limítrofe por compresión de la arteria carótida interna derecha en su segmento intracavernoso. La exéresis y la anatomía patológica de la lesión orbitaria fueron compatibles con la enfermedad inflamatoria orbitaria idiopática. Nuestro caso muestra a la enfermedad inflamatoria orbitaria idiopática como una causa inhabitual de infarto cerebral limítrofe. Palabras clave: Infarto Cerebral; Seudotumor Orbitario; Seno Cavernoso; Trastornos Cerebrovasculares (fuente: DeCS BIREME). Abstract Watershed cerebral infarctions are those located in regions between two adjacent vascular territories and Idiopathic orbital inflammatory disease is a nonspecific inflammatory process of the orbit. We report the case of a 60-year-old man who presented suddenly in the right eye: exophthalmos, ptosis, reduced visual acuity and paresis of the extraocular muscles innervated by the III, IV and VI cranial nerves. Imaging studies showed a right retro-ocular mass with invasion of the ipsilateral cavernous and sphenoid sinus. During hospitalization, suddenly he presented a watershed infarction with narrowing of the right internal carotid artery in its intracavernous portion. Surgical excision of the lesion was performed and pathology showed a characteristic infiltration of idiopathic orbital inflammatory disease. Our case shows idiopathic orbital inflammatory disease as an uncommon cause of watershed cerebral infarction.

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Idiopathic orbital inflammation (orbital inflammatory pseudotumour): an unusual cause of transient ischaemic attack.

Cited by 17 publications

References 7 publications

Idiopathic inflammatory pseudotumor of the orbit and Tolosa-Hunt Syndrome - are they the same disease?

Idiopathic inflammatory pseudotumor of the orbit and Tolosa-Hunt Syndrome - are they the same disease?

Sclerosing Inflammation in the Orbit and in the Pterygopalatine and Infratemporal Fossae

Infarto cerebral limítrofe secundario a enfermedad inflamatoria orbitaria idiopática

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