Tolosa-Hunt Syndrome: Clinical Manifestations in Children

Tsirigotaki, Maria; Ntoulios, George; Lioumpas, Michail; Voutoufianakis, S.; Vorgia, Pelagia

doi:10.1016/j.pediatrneurol.2019.02.013

Cited by 26 publications

(23 citation statements)

References 3 publications

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“…The age of our patients ranged from 4 to 17 years, with an average age of onset of symptoms at 12.0 AE 4.3 years and there was a female predominance (66.7%), according to previous study, 13,15 while in adults, THs predominate in men (64.4 vs. 35.6%), with an average age of 44.4 AE 14.7 years. 3 The granulomatous inflammation that characterizes THS affects the cavernous sinus, the superior orbital fissure, or the orbit.…”

Section: Discussionmentioning

confidence: 69%

“…3,25 The facial nerve is most commonly affected in adults, but two of our patients had peripheral facial paralysis. 8,15 Paresis of ocular motor nerves can appear without other associated symptoms, but in most cases, it is accompanied by pain or even preceded by it, within 2 weeks. 3,17 This pain is nonspecific and poorly characterized, but in our study, it was located in the orbital region, ipsilateral to nerve involvement and had mild to severe intensity.…”

Section: Discussionmentioning

confidence: 99%

“…However, MRI consists of a mandatory exam, as it evidences and locates the lesion. 8,11 It is known that THS results from a granulomatous inflammatory process, but its etiology is not yet well established, 13,15 for this reason, all patients in this review underwent several laboratory tests to exclude other diseases with similar signs and symptoms. In this investigation, vascular, autoimmune, neoplastic, and infectious diseases were excluded.…”

Section: Discussionmentioning

confidence: 99%

“…However, there are several reports of cases of childhood involvement, showing a similar clinical presentation to adult patients. [5][6][7][8][9][10][11][12][13][14][15][16] The etiology of THS has not yet been clarified and its diagnosis is one of exclusion. During the investigation of THS it is necessary to exclude other causes of recurrent multiple cranial neuropathies, such as infectious (bacterial, viral, and fungal), neoplastic, vascular diseases (aneurysms, carotidcavernous fistula, or thrombosis) and other etiologies (sarcoidosis, Wegener's granulomatosis, and eosinophilic granuloma) that may present the same signs and symptoms.…”

Section: Introductionmentioning

confidence: 99%

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Tolosa-Hunt Syndrome in Childhood and Adolescence: A Literature Review in the Last 10 Years

Gama¹,

Silva‐Néto²

2020

Neuropediatrics

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Objectives The aim of the study is to review the articles published in the last 10 years on Tolosa-Hunt syndrome (THS) in childhood and adolescence to learn about its clinical and epidemiological characteristics and its therapeutic management. Methods Based on the literature search in the major medical databases and using the descriptors “Tolosa-Hunt syndrome and children” and “Tolosa-Hunt syndrome and adolescent” we included articles published between 2009 and 2019. We found 51 articles, but only 11 met the inclusion criteria. Results There were eight women and four men with THS, whose average age was 12.0 ± 4.3 years, ranging from 4 to 17 years. Magnetic resonance imaging showed inflammation in the cavernous sinus (100%), orbit (25%), and superior orbital fissure (8.3%), unilateral in all patients. The internal carotid artery (58.3%), third (66.7%), sixth (33.3%), and fourth (25%) cranial nerves were injured. Headache was located in the orbital region, ipsilaterally to nerve involvement and had mild to severe intensity. Corticosteroid was the treatment used in 91.7% of the patients. They were followed for an average of 13.4 ± 12.4 months. Only two patients had recurrence of the disease. Conclusion THS is infrequent in childhood and adolescence and knowledge of its clinical and epidemiological characteristics and its therapeutic management is necessary for pediatricians.

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Section: Discussionmentioning

confidence: 69%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Tolosa-Hunt Syndrome in Childhood and Adolescence: A Literature Review in the Last 10 Years

Gama¹,

Silva‐Néto²

2020

Neuropediatrics

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“…However, age of onset is 11 years by median though there has been a case report of 6 years old child reported, and predominantly affect female children than males, symptomatically most frequently presenting with 3rd CN involvement, followed by 6th, 4th, occasionally 5th CN, and rarely seventh CN involvement has been published in children, while 1/3 the patients may report multiple CN palsy [16] [17] [18].…”

Section: Introductionmentioning

confidence: 99%

Tolosa Hunt Syndrome: Current Diagnostic Challenges and Treatment

Msigwa¹,

Li²,

Cheng³

2020

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Tolosa-Hunt syndrome (THS) is an uncommon diagnosis with an incidence of nearly 1 to 2 cases per million hallmarked by the presence of painful ophthalmoplegia (PO) due to a granulomatous inflammation (GI). Diagnostically, the major THS challenges encountered are owing to the exclusion of other GI presenting conditions necessitating multi-specialization consultations. This article presents uniquely advances in diagnosis and challenges encountered attempting to exclude THS mimics, details on physical examination and laboratory investigations have been incorporated. Tolosa Hunt MRI protocol (contrast-enhanced MRI), restricted diffusion and CISS MRI have lately proved to be precise investigations for THS diagnosis and follow up, on the contrary, number of false-negative/positive MRI diagnoses appears to be rising, hence proposed that MRI or biopsy shouldn't be mandatory criteria for diagnosis as opposed to IHS 2018 guidelines. Despite corticosteroids being the cornerstone therapy for THS, there are controversies concerning the better administration route, optimal dosage, and therapy longevity, through case reports, high dose IV mPSL of 500 mg BID for 3 days, followed with maintenance dose of prednisone 60 mg resulted in the earliest recovery, hereafter standardized guidelines are required. Alternatively, infliximab (300 mg infusion), azathioprine, methotrexate, and acupuncture can be used, farther studies are required to appraise the effectiveness and their safety. On the other side, microsurgery can be utilized for GI evacuation however associated risks of permanent CN VI palsy have been stated, nonetheless, GKRS can be employed when contraindication or corticosteroid intolerability exists though the hazard of late malignancy is a drawback.

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Third Nerve Palsy

Bosley

2021

Controversies in Neuro-Ophthalmic Management

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Tolosa-Hunt Syndrome: Clinical Manifestations in Children

Cited by 26 publications

References 3 publications

Tolosa-Hunt Syndrome in Childhood and Adolescence: A Literature Review in the Last 10 Years

Tolosa-Hunt Syndrome in Childhood and Adolescence: A Literature Review in the Last 10 Years

Tolosa Hunt Syndrome: Current Diagnostic Challenges and Treatment

Third Nerve Palsy

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