PubMed · 11462692
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R Kato. 2001. [Hemifacial atrophy, progressive].. https://pubmed.ncbi.nlm.nih.gov/11462692/
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A 48-year-old white woman presented with a 2-year history of progressive facial hemiatrophy involving the right side of the chin and tongue, associated with mastigatory spasm. Neurological examination showed no abnormalities. Computed tomography and magnetic resonance imaging of the brain and electrophysiological investigations were normal. Histopathological examination of a skin biopsy specimen from the chin revealed atrophy of the subcutaneous fat with homogenization of dermal collagen fibres. Phenytoin 100 mg t.d.s. relieved the mastigatory spasm.
The authors report the case of a 45-years-old female patient, with progressive hemiatrophy of the left face and tongue. The laboratorial and image studies were normal, including routine blood exams, cerebral-spinal fluid, hepatic function tests, renal and rheumatic; besides X ray, computerized tomography and magnetic resonance of skull. The electroneuromyography showed peripheral neurogenic alterations of motor trigeminal and hypoglossal nerves, markedly on the left side. Those findings are compatible with Romberg's facial hemiatrophy diagnosis, motivating discussion of several aspects of this rare disease according to the account and literature.
A 46-year-old woman who presented with progressive facial hemiatrophy (PFH) following localized scleroderma is described. The patient had a markedly deformed and depressed plaque surrounded by erythema on the right cheek. She also showed linear scleroderma with hair loss on the occipital area and morphea lesions on the neck and shoulder. Histological findings of the facial and other atrophic lesions were consistent with localized scleroderma. Therefore, the PFH and localized scleroderma may have had the same pathological background in this case.