Hemiatrofia facial progressiva (doença de Parry-Romberg): relato de dois casos associados a trigeminalgia e câimbras

Brito, Jussara; Holanda, Maurus Marques de Almeida; Holanda, Giovanna Siqueira De; Silva, José Alberto Gonçalves da

doi:10.1590/s0004-282x1997000300020

Cited by 6 publications

(3 citation statements)

References 9 publications

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“… 73 Severe migrainosus state, with frontoparietal white matter lesions on MRI, has likewise been described. 41 In addition, facial pain 4 , 91 and trigeminal neuralgia 23 , 77 , 88 , 91 have been commonly reported: we have found facial pain in 11 patients (8.5% of the patients).…”

Section: Discussionmentioning

confidence: 55%

“…Other neurological CNS manifestations have been more episodically described: limb (contralateral, ipsilateral) and trunk atrophy, 4 cerebral microhemorrhages, 94 cerebellar hemorrhage, 2 movement disorders (including paroxysmal kinesigenic dyskinesia, 46 torticollis, 95 mandibular cramps, 77 dystonia, 71 and facial 2 or masticatory 66 spasms), facial nerve palsy 26 (that could also be due to local atrophy), 1 dysphonia (vocal cord palsy), 52 sympathetic hyperactivity, 23 bilateral pyramidal tract involvement, 96 mirror movements, 69 brain tumor, 13 alien limb, 18 cognitive disturbances, and progressive mental retardation due to cerebral atrophy (with or without focal seizures). 1 , 28 , 79 Ocular motility disturbances may also be observed, due to restrictive myopathy (in part inflammatory) 97 and oculomotor palsies.…”

Section: Discussionmentioning

confidence: 99%

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Neurological Manifestations in Parry–Romberg Syndrome

et al. 2015

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Parry–Romberg syndrome (PRS) is a variant of morphea usually characterized by a slowly progressive course. Clinical and radiological involvement of the central nervous system may be observed in PRS.We describe 2 patients with PRS and neurological symptoms (one with trigeminal neuralgia associated with deafness, and the second with hemifacial pain associated with migraine without aura) in conjunction with abnormal cerebral MRI including white matter T2 hyperintensities and enhancement with gadolinium. Despite the absence of specific immunosuppressive treatments, both patients have presented stable imaging during follow-up without any clinical neurologic progression. We have performed a large review of the medical literature on patients with PRS and neurological involvement (total of 129 patients)Central nervous system involvement is frequent among PRS patients and is inconsistently associated with clinical abnormalities. These various neurological manifestations include seizures, headaches, movement disorders, neuropsychological symptoms, and focal symptoms. Cerebral MRI may reveal frequent abnormalities, which can be bilateral or more often homolateral to the skin lesions, localized or so widespread so as to involve the whole hemisphere: T2 hyperintensities, mostly in the subcortical white matter, gadolinium enhancement, brain atrophy, and calcifications. These radiological lesions do not usually progress over time. Steroids or immunosuppressive treatments are controversial since it remains unclear to what extent they are beneficial and there is often no neurological progression.

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Section: Discussionmentioning

confidence: 55%

Section: Discussionmentioning

confidence: 99%

Neurological Manifestations in Parry–Romberg Syndrome

et al. 2015

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“…Caracteriza-se por atrofia progressiva de uma das hemifaces, com alteração da pele, tecido subcutâneo, tecido muscular, cartilagem e até osso. Geralmente acomete um lado da face, mas pode estender-se para o hemicorpo ipsilateral, inclusive com envolvimento cerebral, podendo o portador apresentar crises convulsivas (1) , malformações vasculares intracranianas (11) , alterações nos nervos cranianos, além de envolvimento do sistema nervoso periférico e autônomo (12) . O comprometimento ocular ocorre em 10 a 15% dos portadores, incluindo enoftalmo progressivo, atrofia palpebral, ulceração corneana, heterocromia de íris, irite, iridociclite, coroidite, estrabismo restritivo, paralisia do nervo oculomotor, catarata, alterações pupilares, papilite e vasculite retiniana (1,(13)(14) .…”

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Uso de Polietigel® intra-orbitário em paciente com atrofia hemifacial progressiva: relato de caso

Sampaio¹,

Kafuri²,

Schellini³

et al. 2004

Arq. Bras. Oftalmol.

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Trigeminal Neuropathic Pain in a Patient With Progressive Facial Hemiatrophy (Parry-Romberg Syndrome)

et al. 2011

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Background: We reviewed the literature on published cases of progressive facial hemiatrophy (Parry-Romberg syndrome) to identify possible pathophysiological mechanisms of the syndrome. Objective: To describe the somatosensory phenotype of a previously unreported patient with progressive facial hemiatrophy and facial pain. Design: Case report and 4-month follow-up period. Setting: University-based tertiary referral headache center. Patient: A 37-year-old woman with progressive facial hemiatrophy and strictly left-sided facial pain over 12 years. Intervention: Greater occipital nerve blockade with lidocaine, 2% (2 mL), and methylprednisolone sodium phosphate (80 mg). Main Outcome Measures: Trigeminal sensory phenotype on quantitative sensory testing using thermal threshold and Von Frey hairs. The case report includes patient photographs, neuroimaging, and neurophysiological findings. Results: On the left side, there was continuous pain in V 1 and V 2 and intermittent sharp shooting pains in V 3. The sensory examination showed areas on the left side with pinprick hyperalgesia, cold and heat hyperalgesia, and dynamic mechanical allodynia. The pain in V 1 and V 3 and the allodynia dramatically improved after greater occipital nerve blockade. In the cases reported in the literature, a constant component of the pain was always part of the phenotype, and positive or negative trigeminal sensory signs were frequently described. Conclusions: The phenotype of our patient suggests neuropathic pain involving all 3 branches of the trigeminal nerve, and the patient fulfills newly defined stricter criteria for neuropathic pain. Similar to our case, phenotypes of the other published cases seem to agree with trigeminal neuropathic pain rather than trigeminal neuralgia specifically.

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Hemiatrofia facial progressiva (doença de Parry-Romberg): relato de dois casos associados a trigeminalgia e câimbras

Cited by 6 publications

References 9 publications

Neurological Manifestations in Parry–Romberg Syndrome

Neurological Manifestations in Parry–Romberg Syndrome

Uso de Polietigel® intra-orbitário em paciente com atrofia hemifacial progressiva: relato de caso

Trigeminal Neuropathic Pain in a Patient With Progressive Facial Hemiatrophy (Parry-Romberg Syndrome)

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