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Ital J Pediatr 2022 Jun 3;48(1):82. Epub 2022 Jun 3.

Department of Clinical and Experimental Medicine, Unit of Rare Diseases of the Nervous System in Childhood, Section of Paediatrics and Child Neuropsychiatry, University of Catania, Catania, Italy.

Background: Ophthalmoplegic migraine, renamed "Recurrent Painful Ophthalmoplegic Neuropathy" (RPON) in 2013 by the International Headache Society is a rare neurologic disorder characterized by recurrent attacks of ophthalmoplegia associated to ipsilateral headache. The etiology is still unknown. Typical magnetic resonance imaging findings show a focal nerve thickening and contrast enhancement. Read More

Oxf Med Case Reports 2022 May 23;2022(5):omac050. Epub 2022 May 23.

Department of Ophthalmology, Aleppo University Hospital, Aleppo, Syria.

We are presenting a rare case of an acute complete external ophthalmoplegia with positive polymerase chain reaction (PCR) for SARS-CoV-2. Our case is the first case that depicts development of Tolosa-Hunt Syndrome (THS) following infection with COVID-19, with a challenging diagnosis and spontaneous improvement. A 65-year-old diabetic female presented with a complete external ophthalmopegia in the left eye and a severe left-sided headache. Read More

J Neuroophthalmol 2022 06 25;42(2):154-162. Epub 2022 Mar 25.

Department of Neurology (IL, ML), Division of Neuroimmunology and Neuroinfectious Disease, Massachusetts General Hospital and Harvard Medical School, Boston, Massachusetts; and Treadwell Virtual Library for the Massachusetts General Hospital (ML), Boston, Massachusetts.

Background: A worldwide mass vaccination campaign against the coronavirus disease 2019 (COVID-19) pandemic is currently underway. Although the safety data of the clinical trials did not report specific concerns regarding neuro-ophthalmological adverse events, they involved a limited number of individuals and were conducted over a relatively short time. The aim of the current review is to summarize the available postmarketing data regarding the occurrence of neuro-ophthalmological and other ocular complications of the COVID-19 vaccines. Read More

Rinsho Shinkeigaku 2022 Apr 29;62(4):281-285. Epub 2022 Mar 29.

Department of Neurology, Anti-aging and Vascular medicine, Division of Internal Medicine, National Defense Medical College.

The patient was a 14-year-old boy with two previous episodes of self-remitting right ophthalmoplegia with right temporal pain at ages 9 and 12. In 2019, he developed right eyelid ptosis and diplopia 2 days after a pulsating right-sided temporoparietal headache. Recurrent headaches with ophthalmoplegia responded to high-dose steroid therapy, and the clinical features resembled recurrent painful ophthalmoplegic neuropathy (RPON). Read More

BMC Endocr Disord 2022 Mar 14;22(1):65. Epub 2022 Mar 14.

The First Department of Medicine, Wakayama Medical University, Wakayama, Japan.

Background: Early diagnosis of lymphoma involving the central nervous system is sometimes difficult but emergent to avoid the delay of therapeutic initiation. Pituitary insufficiencies are usually associated with lymphoma in the pituitary gland. There have been no cases of lymphoma originating from extra pituitary gland with hypopituitarism that simultaneously presenting unilateral upper cranial nerve palsies and ophthalmalgia. Read More

J Yeungnam Med Sci 2022 Mar 11. Epub 2022 Mar 11.

Department of Neurology, Dongsan Medical Center, Keimyung University School of Medicine, Daegu, Korea.

Invasive sphenoid sinus aspergillosis can mimic Tolosa-Hunt syndrome (THS), leading to frequent misdiagnoses and potentially fatal consequences. We report a case of invasive sphenoid sinus aspergillosis initially misdiagnosed as THS. A 79-year-old man presented with right periorbital pain, ophthalmoplegia, and loss of vision. Read More

Eur Neurol 2022 Mar 9:1-8. Epub 2022 Mar 9.

Department of Neurology, Beijing Tongren Hospital, Capital Medical University, Beijing, China.

Background: Several case series of patients with Tolosa-Hunt syndrome have been described in the literature; however, few studies have focused on the cerebrospinal fluid (CSF) characteristics. This study aimed to analyse the CSF characteristics of patients with Tolosa-Hunt syndrome.

Methods: Fifty-five patients who fulfilled the 3rd Edition of the International Classification of Headache Disorders diagnostic criteria for Tolosa-Hunt syndrome were included in this study. Read More

BMJ Case Rep 2022 Mar 1;15(3). Epub 2022 Mar 1.

Neurology, Leeds Teaching Hospitals NHS Trust, Leeds, UK.

Tolosa-Hunt syndrome is understood as a steroid-responsive, relapsing-remitting, unilateral headache disorder associated with ipsilateral cranial neuropathies, of a probable granulomatous aetiology. The diagnosis is made clinically from the history and examination, supported by appropriate imaging. Here the authors report a case of Tolosa-Hunt syndrome with a headache phenotype mimicking a trigeminal autonomic cephalalgias (hemicrania continua), and serial MRI studies showing a stable enlarged pituitary. Read More

Surg Neurol Int 2022 20;13:27. Epub 2022 Jan 20.

Department of Neurosurgery, Tokai University Hachioji Hospital, Ishikawamachi, Hachioji, Tokyo, Japan.

Background: The diagnostic criteria for Tolosa-Hunt syndrome (THS) were updated in 2013 in the 3 Edition of the International Classification of Headache Disorders. It is now possible to diagnose THS based on the presence of granulomatous inflammation demonstrated on magnetic resonance imaging (MRI) without confirmation by biopsy. No previous study has reported the use of arterial spin labeling (ASL) perfusion MRI for diagnosing THS. Read More

Cureus 2021 Dec 30;13(12):e20838. Epub 2021 Dec 30.

Department of Rheumatology, Brown University, Warren Alpert Medical School, Providence, USA.

Tolosa-Hunt syndrome (THS) is a rare neuro-immunological disorder characterized by severe periorbital headaches and ophthalmoplegia. In some patients, THS may occur in parallel with other autoimmune disorders. The underlying etiology of THS remains to be definitively established. Read More

Eur J Ophthalmol 2022 Jan 10:11206721211073447. Epub 2022 Jan 10.

Microbiology Department, "Giovanni Paolo II" Hospital, Ragusa, 159145ASP 7 Ragusa, Italy.

Purpose: to describe a rare case of orbital apex syndrome caused by aspergillosis with acute presentation.

Case Description: retrospective case report of a 70-year-old man who developed unilateral ophthalmoplegia overnight. He was initially given the diagnosis of suspect Tolosa-Hunt syndrome, but biopsy of the involved tissue showed aspergillosis. Read More

Neurol India 2021 Nov-Dec;69(6):1805-1807

Department of Neurology, Nalanda Medical College and Hospital, Patna, Bihar, India.

Ophthalmoplegic migraine (OM) also called recurrent painful ophthalmoplegic neuropathy (RPON) is not a so common disorder. It is characterized by childhood onset, ophthalmoplegia and migraine type of headache. The most common involved nerve is third cranial nerve. Read More

Arch Soc Esp Oftalmol (Engl Ed) 2021 Dec 9;96(12):677-681. Epub 2021 Nov 9.

Servicio de Neurología Clínica, Universidad Nacional de Colombia, Bogotá, Colombia; Grupo de investigación en Neurología de la Universidad Nacional de Colombia, NEUOUNAL, Bogotá, Colombia.

Painful ophthalmoplegia syndrome is characterised by the presence of peri-orbital or peri-cranial pain that is accompanied by paralysis of the oculomotor nerves. The differential diagnosis is broad, and requires a rigorous study. Three clinical cases of patients with painful ophthalmoplegia syndrome are described, in which multiple extension studies were required to obtain an aetiological diagnosis. Read More

J Neuroophthalmol 2021 12;41(4):547-552

Departments of Ophthalmology (SV, AC, KD, BK, MS, SK, NM, JW), Rheumatology (JB), Neurology (AC, KD, BK, MS, JW), and Radiology and Imaging Sciences (EQ), University of Utah Hospital, Salt Lake City, Utah.

Abstract: An 11-year-old boy presented with 2 weeks of intermittent headache, right orbital pain, and constant diplopia. Brain MRI showed dural thickening and enhancement of the right lateral cavernous sinus, right orbital apex, and tentorium. Initial cerebral spinal fluid analysis showed only mild pleocytosis, and serum diagnostics were unrevealing. Read More

BMJ Case Rep 2021 Nov 11;14(11). Epub 2021 Nov 11.

Pediatric Neurology and Neurodevelopmental disorders, Department of Neurology, Sree Chitra Tirunal Institute for Medical Sciences and Technology, Thiruvananthapuram, India

Recurrent painful ophthalmoplegic neuropathy (RPON), previously called ophthalmoplegic migraine, is a rare condition characterised by recurrent episodes of headache and ophthalmoplegia. We report a case of 11-year-old girl with recurrent painful ophthalmoplegia due to isolated right oculomotor nerve involvement. MR brain imaging showed enhancing lesion of cisternal segment of right oculomotor nerve. Read More

N Z Med J 2021 10 22;134(1544):175-178. Epub 2021 Oct 22.

University of Otago, Christchurch, New Zealand; Department of Neurology, Canterbury District Health Board, Christchurch, New Zealand.

Here we discuss a presentation of Tolosa-Hunt syndrome (THS) in a 44-year-old female with new right-eye ptosis, ophthalmoplegia and headache. Four days prior, she had almost identical ptosis and ophthalmoplegia in her left eye, which resolved. Cavernous sinus inflammation and symptom improvement with glucocorticoid treatment indicated THS with bilateral eye involvement, a presentation which may be undervalued by the current THS classification. Read More

Eur J Hybrid Imaging 2021 Oct 7;5(1):18. Epub 2021 Oct 7.

Department of Nuclear Medicine, CUB-Hôpital Erasme, Anderlecht, Belgium.

A 73-year-old man with a history of marginal zone lymphoma was admitted to the emergency room for diplopia and ipsilateral headache. The Fluorine-18-fluorodeoxyglucose positron emission tomography/computed tomography (F-FDG PET/CT) demonstrated intense and symmetrical hypermetabolism of the cavernous sinuses, and hypermetabolic lesions diffusely in the lymph nodes and bones. The diagnosis of high-grade relapse of lymphomatous disease was made. Read More

Pediatr Neurol 2021 12 9;125:16-17. Epub 2021 Sep 9.

Department of Pediatrics, National Hospital Organization Beppu Medical Center, Beppu, Oita, Japan. Electronic address:

Acta Neurol Taiwan 2021 Mar;30(1):39-42

Department of Ophthalmology, Chi Mei Medical Center, Tainan, Taiwan.

Purpose: Case presentation of newly diagnosed systemic lupus erythematosus (SLE) presenting initially as Tolosa-Hunt syndrome (THS).

Study Design: Retrospective clinical case.

Method: Case report. Read More

Cureus 2021 Jul 31;13(7):e16791. Epub 2021 Jul 31.

Neurosurgery, Thomas Jefferson University Hospital, Philadelphia, USA.

Coronavirus disease 2019 (COVID-19) infection is associated with a plethora of neurological complications. Newly developed vaccinations targeting the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) viral spike protein represent a great epidemiological promise with respect to the resolution of the pandemic. However, vaccinations are not without side effects and, in rare cases, have even been implicated in various autoimmune phenomena. Read More

JNMA J Nepal Med Assoc 2021 Jul 1;59(238):604-607. Epub 2021 Jul 1.

Department of Neurology, Kathmandu Medical College and Teaching Hospital, Sinamangal, Kathmandu, Nepal.

Tolosa-Hunt Syndrome is a rare disease characterized by painful ophthalmoplegia affecting third, fourth, and/or sixth cranial nerve caused by non-specific inflammation in the cavernous sinus or superior orbital fissure of unknown etiology. We presented a 67-year-old female with Tolosa-Hunt Syndrome. She had a right-sided headache and periorbital pain with double vision. Read More

Zh Nevrol Psikhiatr Im S S Korsakova 2021 ;121(7):14-21

Research Center of Neurology, Moscow, Russia.

Objective: To study clinical/laboratory signs of primary vasculitis (PV) of the internal carotid artery (ICA) and vertebral artery (VA).

Material And Methods: We examined 31 patients (23 men, 74%, mean age - 36.2±5. Read More

J Curr Ophthalmol 2021 Apr-Jun;33(2):104-111. Epub 2021 Jul 5.

Guru Nanak Eye Centre, Department of Ophthalmology, Maulana Azad Medical College, New Delhi, India.

Purpose: To review the diagnostic criteria for Tolosa-Hunt syndrome (THS) and utility of recent modifications.

Methods: We searched PubMed for keywords Tolosa Hunt and magnetic resonance imaging. We compared the three editions of International Classification of Headache Disorders and isolated case reports and case series with the assessment of cavernous internal carotid artery (ICA) caliber to find the prevalence of vascular anomalies. Read More

Tzu Chi Med J 2021 Jul-Sep;33(3):314-316. Epub 2020 Oct 15.

Department of Neurology, Hualien Tzu Chi Hospital, Buddhist Tzu Chi Medical Foundation, Hualien, Taiwan.

The Tolosa-Hunt syndrome (THS) is caused by granulomatous inflammation in the cavernous sinus, superior orbital fissure, or orbit characterized by painful ophthalmoplegia. Here, we report a case of recurrent THS in a 48-year-old female, who initially showed a poor response to low-dose steroids. Each episode involved different cranial nerves and painful ophthalmoplegia. Read More

eNeurologicalSci 2021 Sep 22;24:100359. Epub 2021 Jul 22.

Department of Internal Medicine, Hamad General Hospital, Hamad Medical Corporation, Doha, Qatar.

Background: Tolosa Hunt syndrome (THS) is a rare disease that manifests mainly as painful unilateral ophthalmoplegia. It is caused by an inflammatory process of unknown aetiology within the cavernous sinus with a rare intracranial extension. The International Classification of Headache Disorders (ICHD)- 3 diagnostic criteria aids in its diagnosis. Read More

Indian J Pediatr 2022 Apr 3;89(4):407-408. Epub 2021 Aug 3.

Department of Neurological Sciences, Christian Medical College, Vellore, Tamil Nadu, 632004, India.

CEN Case Rep 2022 02 17;11(1):31-35. Epub 2021 Jul 17.

Department of Surgery, Aso Iizuka Hospital, Iizuka, Japan.

Immunodeficient patients are susceptible to systemic fungal infections; however, these rarely cause secondary peritonitis. A 66-year-old man with multiple myeloma and diabetes mellitus on continuous ambulatory peritoneal dialysis (CAPD) presented with cloudy ascitic fluid. He had been treated with corticosteroids for 1 month for Tolosa-Hunt syndrome. Read More

BMC Neurol 2021 Jul 5;21(1):264. Epub 2021 Jul 5.

Division of Neurology, Department of Clinical Medicine, Universidade Federal do Ceará, Rua Capitão Francisco Pedro, 1290, 60430-370, Fortaleza, Brazil.

Background: The term "Tolosa-Hunt syndrome" (THS) has been used to refer to painful ophthalmoplegia associated with nonspecific inflammation of the cavernous sinus and many processes can result in a similar clinical picture, including infectious, inflammatory and neoplastic diseases. Rosai-Dorfman disease (RDD) is a lymphoproliferative disorder that rarely affects the central nervous system. We report a case of isolated CNS Rosai-Dorfman disease involving the cavernous sinus and presenting as "Tolosa-Hunt syndrome". Read More

Cureus 2021 May 27;13(5):e15281. Epub 2021 May 27.

Ophthalmology, Eye Center South, Dothan, USA.

Tolosa-Hunt syndrome (THS) is a fascinating condition that is ipso facto a cavernous sinus syndome. As such it is associated with inflammation of the cavernous sinus walls and contents with spread to contiguous structures such as the orbital apex and superior orbital fissure. Therefore it does not come as a surprise that there is overlap with the condition of orbital pseudotumor. Read More

Acta Neurol Belg 2021 Jun 28. Epub 2021 Jun 28.

Department of Neurology, Kyoto University Graduate School of Medicine, Kyoto, Japan.