A case of overlapping adult‐onset linear scleroderma and Parry‐Romberg syndrome presenting with widespread ipsilateral neurogenic involvement
| Autor: | Koji Shinoda, Takuya Matsushita, Shoko Sadashima, Saeko Inamizu, Hirofumi Ochi, Noriko Isobe, Toru Iwaki, Masutaka Furue, Yoshinao Oda, Ryo Yamasaki, Tomomi Yonekawa, Yuki Kuma, Jun Ichi Kira, Gaku Tsuji |
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| Rok vydání: | 2019 |
| Předmět: |
Muscle biopsy
integumentary system medicine.diagnostic_test business.industry Parry–Romberg syndrome General Medicine Anatomy medicine.disease Pathology and Forensic Medicine 03 medical and health sciences 0302 clinical medicine medicine.anatomical_structure Atrophy Ptosis 030220 oncology & carcinogenesis Skin biopsy medicine Linear Scleroderma Neurology (clinical) Eyelid medicine.symptom skin and connective tissue diseases business Localized Scleroderma 030217 neurology & neurosurgery |
| Zdroj: | Neuropathology. 40:109-115 |
| ISSN: | 1440-1789 0919-6544 |
| DOI: | 10.1111/neup.12614 |
| Popis: | Linear scleroderma is a variant of localized scleroderma. We report a 43-year-old woman who had developed left arm weakness and linear scleroderma on her back during pregnancy at 25 years of age, followed by left hemifacial atrophy and left leg weakness. She had multiple linear scleroderma lesions on her trunk and left limbs, left eyelid ptosis, impairment of vertical movement and abduction of the left eye, left hemifacial atrophy, and weakness and atrophy of the sternocleidomastoid, trapezius, and proximal limb muscles on the left side. On serology, antibodies to U1-ribonucleoprotein and Jo-1 were positive; anti-scleroderma-70 antibody was negative. Skin biopsy demonstrated increased hypertrophic collagen fibers without inflammatory infiltrates. Needle electromyography of left limb muscles revealed mild neurogenic patterns; left quadriceps muscle biopsy showed chronic neurogenic changes. Brain magnetic resonance imaging revealed mild left hemispheric atrophy. This is a rare case of linear scleroderma and Parry-Romberg syndrome presenting with widespread ipsilateral neurogenic manifestations. |
| Databáze: | OpenAIRE |
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