{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,21]],"date-time":"2026-07-21T12:07:18Z","timestamp":1784635638172,"version":"3.55.0"},"reference-count":12,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"48","license":[{"start":{"date-parts":[[2019,11,1]],"date-time":"2019-11-01T00:00:00Z","timestamp":1572566400000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:title>Abstract<\/jats:title>\n          <jats:sec>\n            <jats:title>Rationale:<\/jats:title>\n            <jats:p>Multicentric carpotarsal osteolysis (MCTO) is a rare hereditary disease caused by mutations in <jats:italic toggle=\"yes\">MafB<\/jats:italic>, a negative regulator of osteoclastogenesis.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Patient concerns:<\/jats:title>\n            <jats:p>A 20-year-old, Japanese woman with scoliosis visited our institute for treatment. Scoliosis was apparent since she was 12 years old, but she had not sought treatment until the age of 19. Medical examination showed a typical facial appearance associated with a small forehead and hypotelorism; shortening of the fingers of both hands and both upper limbs was observed, in addition to clubfoot. No caf\u00e9 au lait spots or mental retardation were observed. On the other hand, the trunk showed evidence of an irregular waistline and a rib hump that obviously suggested scoliosis. Neurological deficit was not observed. Spirometry showed decreased forced vital capacity (FVC). Although proteinuria was observed, renal dysfunction and hypertension were not seen. The major curve of scoliosis was 82\u00b0 (MC, Th7\u2013L2; Th11 apical vertebra), and the upper curve was 77\u00b0 (UC, Th1-6; Th3 apical vertebra). In a recumbent-traction position, the major curve was 54\u00b0 and the upper curve was 56\u00b0. The pelvic incidence minus lumbar lordosis (PI\u2013LL) angle was &lt;10\u00b0 and no mismatch was observed; thoracic kyphosis was decreased to 16\u00b0.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Diagnosis:<\/jats:title>\n            <jats:p>The patient was diagnosed with symptomatic scoliosis secondary to MCTO.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Interventions:<\/jats:title>\n            <jats:p>We decided to perform a correction and fusion from Th2 to L3 using a posterior spinal instrumentation.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Outcomes:<\/jats:title>\n            <jats:p>Postoperative x-ray demonstrated scoliosis angle correction from 77\u00b0 to 38\u00b0 at Th1\u20136 and 82\u00b0 to 39\u00b0 at Th7\u2013L2. Postoperative x-ray demonstrated thoracic kyphosis angle correction from 16\u00b0 to 21\u00b0. The patient's height increased from 155 to 161\u200acm.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Lessons:<\/jats:title>\n            <jats:p>It has been 24 months since the operation, and no exacerbation has been observed. To the best of our knowledge, this is the first report of surgical treatment of scoliosis secondary to MCTO.<\/jats:p>\n          <\/jats:sec>","DOI":"10.1097\/md.0000000000017828","type":"journal-article","created":{"date-parts":[[2019,11,26]],"date-time":"2019-11-26T19:58:55Z","timestamp":1574798335000},"page":"e17828","source":"Crossref","is-referenced-by-count":6,"title":["A CARE-compliant article"],"prefix":"10.1097","volume":"98","author":[{"given":"Kunihiko","family":"Miyazaki","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery, Faculty of Medicine, Kagawa University, Kita-gun, Kagawa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Satoshi","family":"Komatsubara","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Faculty of Medicine, Kagawa University, Kita-gun, Kagawa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Koki","family":"Uno","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, National Hospital Organization Kobe Medical Center, Hyogo, Japan."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryuji","family":"Fujihara","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Faculty of Medicine, Kagawa University, Kita-gun, Kagawa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tetsuji","family":"Yamamoto","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Faculty of Medicine, Kagawa University, Kita-gun, Kagawa"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2019,11]]},"reference":[{"key":"R1-20230915","doi-asserted-by":"crossref","first-page":"2287","DOI":"10.1002\/ajmg.a.36641","article-title":"Multicentric carpotarsal osteolysis syndrome is caused by only a few domein-specific mutation in MAFB, a negative regulator of RANKL-induced osteoclastogenesis","volume":"164A","author":"Mumm","year":"2014","journal-title":"Am J Med Genet A"},{"key":"R2-20230915","doi-asserted-by":"crossref","first-page":"88","DOI":"10.1302\/0301-620X.67B1.3968152","article-title":"The syndrome of idiopathic osteolysis: classification, review, and case report","volume":"67","author":"Hardegger","year":"1985","journal-title":"J Bone Joint Surg Br"},{"key":"R3-20230915","doi-asserted-by":"crossref","first-page":"339","DOI":"10.1302\/0301-620X.47B2.339","article-title":"Osteoarticular changes in a case of essential osteolysis","volume":"47","author":"Lagier","year":"1965","journal-title":"J Bone Joint Surg Br"},{"key":"R4-20230915","first-page":"223","article-title":"Acroosteolysis of the wrist, hollow foot, scoliosis and strabismus in a young girl with renal insufficiency [Article in French]","volume":"77","author":"Derot","year":"1961","journal-title":"Bull Mem Soc Med Hop Paris"},{"key":"R5-20230915","first-page":"25","article-title":"Carpo-tarsal osteolysis: case report and review of the literature","volume":"4","author":"Urlus","year":"1993","journal-title":"Genet Couns"},{"key":"R6-20230915","doi-asserted-by":"crossref","first-page":"29","DOI":"10.1007\/BF00347095","article-title":"Acro-osteolysis occurring in a patient with idiopathic multicentric osteolysis","volume":"5","author":"De Smet","year":"1980","journal-title":"Skeletal Radiol"},{"key":"R7-20230915","doi-asserted-by":"crossref","first-page":"242","DOI":"10.4184\/asj.2013.7.3.242","article-title":"Gorham's disease of spine","volume":"7","author":"Sekharappa","year":"2013","journal-title":"Asian Spine J"},{"key":"R8-20230915","doi-asserted-by":"crossref","first-page":"99","DOI":"10.1148\/108.1.99","article-title":"Hereditary osteolysis. 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