Trigeminal Sensory Neuropathy a Study of 22 Cases

Lecky, B. R. F.; Hughes, R. A. C.; Murray, Niall

doi:10.1093/brain/110.6.1463

Cited by 120 publications

(46 citation statements)

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“…Although it demonstrated an afferent pattern of abnormal responses in two patients (Cases 1 and 3), normal results of BRS in TSN (as seen in Case 2) were reported previously. 9 TSN has been observed most frequently in CREST syndrome (calcinosis, Raynaud's phenomenon, esophageal dysmotility, sclerodactyly and telangiectasia) or SSc in overlap with other disorders, like myositis. The antinuclear and anti-RNP antibodies have frequently been found in SSc with TSN.…”

Section: Discussionmentioning

confidence: 99%

Trigeminal sensory neuropathy associated with systemic sclerosis: report of three Brazilian cases

Ribeiro

Fialho

Souza

et al. 2009

Arq. Neuro-Psiquiatr.

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Systemic sclerosis (SSc) is a connective tissue disease manifest by fibrotic tissue changes, microvascular disease, and autoimmune abnormalities. The prevalence of different neurological manifestations in SSc has ranged from 0.8% 1 to 18.5% 2 according to the adopted criteria. Trigeminal sensory neuropathy (TSN) causes numbness in the mandibular or maxillary divisions of the nerve in about 2/3 of the cases, and in the distribution of all divisions in the remaining cases. The sensory abnormalities evolve slowly and usually spread contralaterally in an asymmetric pattern. The numbness may be accompanied by burning dysesthesia that is distinct from trigeminal neuralgia 3 . TSN is an infrequent complication of SSc. Although epidemiological studies are scarce, the prevalence of TSN associated with SSc in the largest series available was 4% 4 . A fairly diligent review of the literature revealed no previous report of TSN as a complication of SSc in Brazil. We present three cases of such association. CASES Case 1A 42-year-old male had presented progressive facial hypoesthesia and dysesthesia for the last three years and was diagnosed with the diffuse form of SSc two years ago. At his first neurological evaluation, he had sclerodermic fascies, sclerodactyly, Raynaud's phenomenon, bilateral facial and lingual hypoesthesia, left hemifacial dysesthesia, and an absent left corneal reflex. Nailfold capillaroscopy was performed and showed devascularization and ectasias. ANA was positive (1/640) and anti-RNP was also positive. Nerve conduction studies (NCS) revealed sensory myelinic polyneuropathy in all limbs and face. Blink reflex studies (BRS) demonstrated absent responses on both sides to left supraorbital nerve stimulation. Electromyography (EMG) was normal. He was on methotrexate and prednisone without improvement of the TSN. Gabapentin 800 mg daily and nortryptilin 100 mg daily were introduced with only a partial response at their maximum tolerable doses. Case 2A 47-year-old female had presented Raynaud' s phenomenon and arthritis in her hands for the last three years and was feeling progressive numbness in the inferior half of the face for the last two years, without any previous diagnosis. At her first neurological evaluation, she had sclerodermic fascies, sclerodactyly, hypoesthesia in the maxillary and mandibular divisions territory bilaterally. ANA was positive (1/80), an esophagogram showed an increased esophageal caliber and a thoracic tomography confirmed this finding and revealed signs of pulmonary fibrosis. All electrophysiological studies performed (NCS, BRS, EMG) were normal. She had never been on medication for TSN or SSc yet. Case 3A 50-year-old female had presented hypoesthesia only in the maxillary division of the right trigeminal nerve about three years before her first neurological evaluation. Six months later, she started presenting hypoesthesia in the mandibullary division, effort dyspnea, Raynaud' s phenomenon and myalgia. She was diagnosed with SSc in overlap with myositis 10 months before our firs...

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Section: Discussionmentioning

confidence: 99%

Trigeminal sensory neuropathy associated with systemic sclerosis: report of three Brazilian cases

Ribeiro

Fialho

Souza

et al. 2009

Arq. Neuro-Psiquiatr.

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“…ISTN (i.e., not associated with systemic neuropathy) is a rare clinical condition [40]. Several studies described an association between chronic trigeminal sensory neuropathy and connective tissue diseases [41]. In patients with ISTN, sensory loss develops gradually; first, it is usually localised in the perioral region on one side and then extends, over a period of months or a few years, to the other side, the intraoral mucosa and the tongue, and subsequently the whole face.…”

Section: Involvement Of the Gasserian Ganglionmentioning

confidence: 99%

“…In patients with ISTN, sensory loss develops gradually; first, it is usually localised in the perioral region on one side and then extends, over a period of months or a few years, to the other side, the intraoral mucosa and the tongue, and subsequently the whole face. Pain is a relatively common symptom; it is more often constant, and usually described as burning or aching [40,41]. In the earlier stage of disease, whereas the masseter inhibitory reflex may be abnormal, the blink reflex may be normal.…”

Section: Involvement Of the Gasserian Ganglionmentioning

confidence: 99%

Neurophysiological assessment of craniofacial pain

2006

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This review deals with the diagnostic usefulness of neurophysiological testing in patients with craniofacial pain. Neurophysiological testing of trigeminal nerve function relies on trigeminal reflexes and laser-evoked potentials (LEPs). This review briefly describes the physiology of trigeminal reflexes and LEPs, reports normal values and highlights the neurophysiological abnormalities in the main clinical conditions.

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“…The interferons are associated with numerous adverse reactions including various neuropsychiatric manifestations and neurological syndromes. There are several reports of cranial nerve palsies occurring during interferon-alpha therapy [2][3][4][5] and, in all of them, immune mechanisms are suggested. The occurrence and exacerbation of autoimmune disorders (sarcoidosis, thyroiditis, hemolytic anemia, psoriasis, thrombocytopenia) in patients treated with interferon-alpha has been reported previously.…”

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confidence: 99%