A case of overlapping adult-onset linear scleroderma and Parry-Romberg syndrome presenting with widespread ipsilateral neurogenic involvement

Ryo Yamasaki, Tomomi Yonekawa, Saeko Inamizu, Koji Shinoda, Hirofumi Ochi, Takuya Matsushita, Noriko Isobe, Gaku Tsuji, Shoko Sadashima, Yuki Kuma, Yoshinao Oda, Toru Iwaki, Masutaka Furue, Jun ichi Kira

研究成果: ジャーナルへの寄稿記事

抄録

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.

元の言語英語
ページ(範囲)109-115
ページ数7
ジャーナルNeuropathology
40
発行部数1
DOI
出版物ステータス出版済み - 2 1 2020

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Facial Hemiatrophy
Localized Scleroderma
Extremities
Atrophy
Blepharoptosis
Biopsy
Muscles
Ribonucleoproteins
Superficial Back Muscles
Antibodies
Quadriceps Muscle
Electromyography
Serology
Needles
Leg
Arm
Collagen
Magnetic Resonance Imaging
Pregnancy
Skin

All Science Journal Classification (ASJC) codes

  • Pathology and Forensic Medicine
  • Clinical Neurology

これを引用

A case of overlapping adult-onset linear scleroderma and Parry-Romberg syndrome presenting with widespread ipsilateral neurogenic involvement. / Yamasaki, Ryo; Yonekawa, Tomomi; Inamizu, Saeko; Shinoda, Koji; Ochi, Hirofumi; Matsushita, Takuya; Isobe, Noriko; Tsuji, Gaku; Sadashima, Shoko; Kuma, Yuki; Oda, Yoshinao; Iwaki, Toru; Furue, Masutaka; Kira, Jun ichi.

:: Neuropathology, 巻 40, 番号 1, 01.02.2020, p. 109-115.

研究成果: ジャーナルへの寄稿記事

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abstract = "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.",
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AU - Yonekawa, Tomomi

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AU - Shinoda, Koji

AU - Ochi, Hirofumi

AU - Matsushita, Takuya

AU - Isobe, Noriko

AU - Tsuji, Gaku

AU - Sadashima, Shoko

AU - Kuma, Yuki

AU - Oda, Yoshinao

AU - Iwaki, Toru

AU - Furue, Masutaka

AU - Kira, Jun ichi

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N2 - 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.

AB - 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.

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