{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,3]],"date-time":"2026-07-03T00:22:32Z","timestamp":1783038152233,"version":"3.54.6"},"reference-count":82,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"6","license":[{"start":{"date-parts":[[2025,2,10]],"date-time":"2025-02-10T00:00:00Z","timestamp":1739145600000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Cosmet Laser Ther"],"published-print":{"date-parts":[[2025,2,10]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Gorham-Stout disease (GSD) is an extremely rare osteolytic disorder characterized by resorption of skeletal bone due to aggressive, nonneoplastic lymphatic proliferation.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>The authors report the 14th case of GSD of the craniovertebral junction (CVJ), which resulted in cranial settling, basilar impression, and an acquired Chiari malformation type I (CM-I) in a pediatric patient. A 4-year-old girl initially presented with recurrent symptoms of pharyngitis, bacteremia, and neck pain. She continued experiencing waxing and waning symptoms of fever and neck pain that gradually worsened. At 12 years old, imaging revealed progressively worsening osteolytic changes with bone marrow edema involving the CVJ and subaxial cervical spine with progressive cranial settling, basilar impression, and 14 mm of tonsillar herniation, consistent with an acquired CM-I. She underwent a frontal bone biopsy, which confirmed the diagnosis of GSD. Treatment with sirolimus and bisphosphonates stabilized the osteolysis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>GSD represents a rare group of osteolytic bone disorders with a highly variable clinical course. A high index of suspicion is warranted, as diagnosis can be challenging. Early referral to a specialized center is key, as treatment is directed toward symptomatic management and a multidisciplinary team approach. GSD is another syndrome associated with CVJ bony changes and tonsillar herniation.<\/jats:p>\n                    <jats:p>\n                      <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"https:\/\/thejns.org\/doi\/10.3171\/CASE24298\">https:\/\/thejns.org\/doi\/10.3171\/CASE24298<\/jats:ext-link>\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case24298","type":"journal-article","created":{"date-parts":[[2025,2,10]],"date-time":"2025-02-10T01:50:13Z","timestamp":1739152213000},"source":"Crossref","is-referenced-by-count":2,"title":["Gorham-Stout disease of the craniovertebral junction causing basilar impression and Chiari malformation type I: illustrative case"],"prefix":"10.3171","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0009-0005-5284-9895","authenticated-orcid":true,"given":"Hashim","family":"Syed","sequence":"first","affiliation":[{"name":"Department of Neurosurgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephen","family":"Glennon","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3508-8156","authenticated-orcid":true,"given":"Nahom","family":"Teferi","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Chenoweth","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathryn","family":"Eschbacher","sequence":"additional","affiliation":[{"name":"Department of Pathology, University of Iowa Hospitals and Clinics, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Polly J.","family":"Ferguson","sequence":"additional","affiliation":[{"name":"Stead Family Department of Pediatrics, Division of Pediatric Rheumatology, Allergy and Immunology, University of Iowa Carver College of Medicine, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yutaka","family":"Sato","sequence":"additional","affiliation":[{"name":"Stead Family Department of Pediatrics, Division of Neuroradiology, University of Iowa Carver College of Medicine, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Patrick","family":"Hitchon","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brian J.","family":"Dlouhy","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"issue":"5","key":"bib1","series-title":"World J Orthop","first-page":"694","article-title":"Vanishing bone disease (Gorham-Stout syndrome): a review of a rare entity","volume":"5","author":"Nikolaou","year":"2014","unstructured":"Nikolaou VSChytas DKorres DEfstathopoulos N.Vanishing bone disease (Gorham-Stout syndrome): a review of a rare entity. World J Orthop. 2014;5(5):694-698."},{"issue":"5","key":"bib2","series-title":"Pediatr Blood Cancer","first-page":"e27614","article-title":"Efficacy of systemic sirolimus in the treatment of generalized lymphatic anomaly and Gorham-Stout disease","volume":"66","author":"Ricci","year":"2019","unstructured":"Ricci KWHammill AMMobberley-Schuman PEfficacy of systemic sirolimus in the treatment of generalized lymphatic anomaly and Gorham-Stout disease. Pediatr Blood Cancer. 2019;66(5):e27614."},{"issue":"1","key":"bib3","series-title":"J Oral Maxillofac Surg","first-page":"199","article-title":"Vanishing bone disease: a review","volume":"69","author":"Kiran","year":"2011","unstructured":"Kiran DNAnupama A.Vanishing bone disease: a review. J Oral Maxillofac Surg. 2011;69(1):199-203."},{"issue":"3","key":"bib4","series-title":"J Oral Maxillofac Pathol","first-page":"411","article-title":"Gorham's disease: a diagnostic challenge","volume":"18","author":"Saify","year":"2014","unstructured":"Saify FYGosavi SR.Gorham's disease: a diagnostic challenge. J Oral Maxillofac Pathol. 2014;18(3):411-414."},{"issue":"1","key":"bib5","series-title":"EFORT Open Rev","first-page":"35","article-title":"Current concepts from diagnosis to management in Gorham-Stout disease: a systematic narrative review of about 350 cases","volume":"7","author":"Angelini","year":"2022","unstructured":"Angelini AMosele NPagliarini ERuggieri P.Current concepts from diagnosis to management in Gorham-Stout disease: a systematic narrative review of about 350 cases. EFORT Open Rev. 2022;7(1):35-48."},{"issue":"1","key":"bib6","series-title":"BMC Neurol","article-title":"Gorham-Stout disease affecting the spine with cerebrospinal fluid leakage and Chiari-like tonsillar herniation: a rare case report and review of literature","volume":"23","author":"Xing","year":"2023","unstructured":"Xing QQMiao MZhang QWWu YHe FF.Gorham-Stout disease affecting the spine with cerebrospinal fluid leakage and Chiari-like tonsillar herniation: a rare case report and review of literature. BMC Neurol. 2023;23(1):59."},{"issue":"5","key":"bib7","series-title":"Pediatr Blood Cancer","first-page":"832","article-title":"Clinical features and prognosis of generalized lymphatic anomaly, kaposiform lymphangiomatosis, and Gorham-Stout disease","volume":"63","author":"Ozeki","year":"2016","unstructured":"Ozeki MFujino AMatsuoka KNosaka SKuroda TFukao T.Clinical features and prognosis of generalized lymphatic anomaly, kaposiform lymphangiomatosis, and Gorham-Stout disease. Pediatr Blood Cancer. 2016;63(5):832-838."},{"issue":"5","key":"bib8","series-title":"J Bone Joint Surg Am","first-page":"985","article-title":"Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis","volume":"37-A","author":"Gorham","year":"1955","unstructured":"Gorham LWStout AP.Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis. J Bone Joint Surg Am. 1955;37-A(5):985-1004."},{"key":"bib9","series-title":"Front Immunol","article-title":"The molecular mechanism of Gorham syndrome: an update","volume":"14","author":"Xiang","year":"2023","unstructured":"Xiang JZhong W.The molecular mechanism of Gorham syndrome: an update. Front Immunol. 2023;14:1165091."},{"key":"bib10","series-title":"Bone","first-page":"47","article-title":"Viewpoints on vessels and vanishing bones in Gorham-Stout disease","volume":"63","author":"Dellinger","year":"2014","unstructured":"Dellinger MTGarg NOlsen BR.Viewpoints on vessels and vanishing bones in Gorham-Stout disease. Bone. 2014;63:47-52."},{"key":"bib11","series-title":"Ann N Y Acad Sci","first-page":"203","article-title":"Gorham\u2019s disease: an osseous disease of lymphangiogenesis?","volume":"1131","author":"Radhakrishnan","year":"2008","unstructured":"Radhakrishnan KRockson SG.Gorham\u2019s disease: an osseous disease of lymphangiogenesis? Ann N Y Acad Sci. 2008;1131:203-205."},{"issue":"14","key":"bib12","series-title":"Circulation","first-page":"1413","article-title":"Podoplanin-expressing cells derived from bone marrow play a crucial role in postnatal lymphatic neovascularization","volume":"122","author":"Lee","year":"2010","unstructured":"Lee JYPark CCho YPPodoplanin-expressing cells derived from bone marrow play a crucial role in postnatal lymphatic neovascularization. Circulation. 2010;122(14):1413-1425."},{"issue":"1","key":"bib13","series-title":"Lymphology","first-page":"18","article-title":"Primary versus trauma-induced Gorham-Stout disease","volume":"51","author":"Tanoue","year":"2018","unstructured":"Tanoue NMoedano LWitte MMontague MLukefahr ABernas M.Primary versus trauma-induced Gorham-Stout disease. Lymphology. 2018;51(1):18-27."},{"issue":"6","key":"bib14","series-title":"Am J Med","first-page":"1151","article-title":"Gorham\u2019s syndrome: a case report and review of the literature","volume":"83","author":"Choma","year":"1987","unstructured":"Choma NDBiscotti CVBauer TWMehta ACLicata AA.Gorham\u2019s syndrome: a case report and review of the literature. Am J Med. 1987;83(6):1151-1156."},{"key":"bib15","series-title":"Front Surg","article-title":"Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham-Stout syndrome: a case report and literature review","volume":"9","author":"Zhang","year":"2022","unstructured":"Zhang HHan CPang DSurgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham-Stout syndrome: a case report and literature review. Front Surg. 2022;9:981025."},{"issue":"4","key":"bib16","series-title":"Childs Nerv Syst","first-page":"695","article-title":"Gorham-Stout disease of skull base leading to cranial settling and rhinorrhea: a case-based review","volume":"38","author":"Maroufi","year":"2022","unstructured":"Maroufi SFHabibi ZDabbagh Ohadi MAMohammadi ENejat F.Gorham-Stout disease of skull base leading to cranial settling and rhinorrhea: a case-based review. Childs Nerv Syst. 2022;38(4):695-703."},{"issue":"7","key":"bib17","series-title":"Medicina (Kaunas)","first-page":"681","article-title":"Gorham-Stout disease with multiple bone involvement-challenging diagnosis of a rare disease and literature review","volume":"57","author":"Momanu","year":"2021","unstructured":"Momanu ACaba LGorduza NCGorham-Stout disease with multiple bone involvement-challenging diagnosis of a rare disease and literature review. Medicina (Kaunas). 2021;57(7):681."},{"issue":"5","key":"bib18","series-title":"J Thorac Cardiovasc Surg","first-page":"1205","article-title":"Duropleural fistula as a consequence of Gorham-Stout syndrome: a combination of 2 rare conditions","volume":"131","author":"Agrawal","year":"2006","unstructured":"Agrawal RMohammed IReilly PG.Duropleural fistula as a consequence of Gorham-Stout syndrome: a combination of 2 rare conditions. J Thorac Cardiovasc Surg. 2006;131(5):1205-1206."},{"key":"bib19","series-title":"J Clin Neurosci","first-page":"486","article-title":"Multifocal Gorham-Stout disease associated with Chiari I malformation and recurrent aseptic meningitis: case report and review of literature","volume":"72","author":"Stephens","year":"2020","unstructured":"Stephens SSquires LCampbell RDavies JChaseling R.Multifocal Gorham-Stout disease associated with Chiari I malformation and recurrent aseptic meningitis: case report and review of literature. J Clin Neurosci. 2020;72:486-492."},{"issue":"3","key":"bib20","series-title":"Bol Med Hosp Infant Mex","first-page":"217","article-title":"Clinical and radiological improvement in Gorham-Stout disease after sirolimus treatment","volume":"80","author":"Barnes-Salda\u00f1a","year":"2023","unstructured":"Barnes-Salda\u00f1a FVenegas-Andrade AColin-Mart\u00ednez \u00d3Lizardo-Rodr\u00edguez AGarc\u00eda-Romero MTDur\u00e1n-McKinster C.Clinical and radiological improvement in Gorham-Stout disease after sirolimus treatment. Bol Med Hosp Infant Mex. 2023;80(3):217-221."},{"issue":"1","key":"bib21","series-title":"J Otolaryngol Head Neck Surg","article-title":"A case report of Gorham-Stout disease diagnosed during the course of recurrent meningitis and cholesteatoma","volume":"49","author":"Hosoya","year":"2020","unstructured":"Hosoya MOishi NNishiyama JOgawa K.A case report of Gorham-Stout disease diagnosed during the course of recurrent meningitis and cholesteatoma. J Otolaryngol Head Neck Surg. 2020;49(1):18."},{"issue":"2","key":"bib22","series-title":"Childs Nerv Syst","first-page":"455","article-title":"Gorham Stout disease of the temporal bone with cerebrospinal fluid leak","volume":"38","author":"Aouad","year":"2022","unstructured":"Aouad PYoung NMSaratsis AMReynolds MARyan ME.Gorham Stout disease of the temporal bone with cerebrospinal fluid leak. Childs Nerv Syst. 2022;38(2):455-460."},{"issue":"2","key":"bib23","series-title":"J Bone Miner Res","first-page":"207","article-title":"Gorham-Stout syndrome: a monocyte-mediated cytokine propelled disease","volume":"21","author":"Colucci","year":"2006","unstructured":"Colucci STaraboletti GPrimo LGorham-Stout syndrome: a monocyte-mediated cytokine propelled disease. J Bone Miner Res. 2006;21(2):207-218."},{"issue":"3","key":"bib24","series-title":"Clin Rheumatol","first-page":"813","article-title":"Gorham-Stout disease: radiological, histological, and clinical features of 12 cases and review of literature","volume":"35","author":"Liu","year":"2016","unstructured":"Liu YZhong DRZhou PRGorham-Stout disease: radiological, histological, and clinical features of 12 cases and review of literature. Clin Rheumatol. 2016;35(3):813-823."},{"issue":"1","key":"bib25","series-title":"Radiology","first-page":"28","article-title":"Observations on massive osteolysis; a review of the literature and report of a case","volume":"71","author":"Johnson","year":"1958","unstructured":"Johnson PMMcClure JG.Observations on massive osteolysis; a review of the literature and report of a case. Radiology. 1958;71(1):28-42."},{"key":"bib26","series-title":"Clin Neurol Neurosurg","first-page":"83","article-title":"Chiari I malformation associated with Gorham's disease of the skull base","volume":"116","author":"Coulter","year":"2014","unstructured":"Coulter ICKhan SAFlanagan AMMarks SM.Chiari I malformation associated with Gorham's disease of the skull base. Clin Neurol Neurosurg. 2014;116:83-86."},{"issue":"4","key":"bib27","series-title":"Oral Surg Oral Med Oral Pathol","first-page":"331","article-title":"Perspectives on massive osteolysis. Report of a case and review of the literature","volume":"55","author":"Heffez","year":"1983","unstructured":"Heffez LDoku HCCarter BLFeeney JE.Perspectives on massive osteolysis. Report of a case and review of the literature. Oral Surg Oral Med Oral Pathol. 1983;55(4):331-343."},{"issue":"6","key":"bib28","series-title":"Adv Wound Care (New Rochelle)","first-page":"230","article-title":"Generalized lymphatic anomaly and Gorham-Stout disease: overview and recent insights","volume":"8","author":"Ozeki","year":"2019","unstructured":"Ozeki MFukao T.Generalized lymphatic anomaly and Gorham-Stout disease: overview and recent insights. Adv Wound Care (New Rochelle). 2019;8(6):230-245."},{"issue":"3","key":"bib29","series-title":"Int J Radiat Oncol Biol Phys","first-page":"491","article-title":"Gorham\u2019s massive osteolysis: the role of radiation therapy and a review of the literature","volume":"26","author":"Dunbar","year":"1993","unstructured":"Dunbar SFRosenberg AMankin HRosenthal DSuit HD.Gorham\u2019s massive osteolysis: the role of radiation therapy and a review of the literature. Int J Radiat Oncol Biol Phys. 1993;26(3):491-497."},{"issue":"1","key":"bib30","series-title":"Adv Radiat Oncol","article-title":"A case of Gorham-Stout disease of the skull base treated with intensity modulated radiation therapy","volume":"7","author":"Roy","year":"2021","unstructured":"Roy AAndruska NBrenneman RA case of Gorham-Stout disease of the skull base treated with intensity modulated radiation therapy. Adv Radiat Oncol. 2021;7(1):100809."},{"issue":"6","key":"bib31","series-title":"Spine (Phila Pa 1976)","first-page":"545","article-title":"Massive osteolysis of the cervical spine. A case report of bone graft failure","volume":"6","author":"Woodward","year":"1981","unstructured":"Woodward HRChan DPLee J.Massive osteolysis of the cervical spine. A case report of bone graft failure. Spine (Phila Pa 1976). 1981;6(6):545-549."},{"issue":"8","key":"bib32","series-title":"Acta Neurochir (Wien)","first-page":"909","article-title":"Gorham\u2019s disease of skull base and cervical spine\u2014confusing picture in a two year old","volume":"148","author":"Girn","year":"2006","unstructured":"Girn HRTowns GChumas PHolland PChakrabarty A.Gorham\u2019s disease of skull base and cervical spine\u2014confusing picture in a two year old. Acta Neurochir (Wien). 2006;148(8):909-913."},{"issue":"5","key":"bib33","series-title":"J Clin Diagn Res","first-page":"PD17","article-title":"Gorham\u2019s syndrome associated with Chiari I malformation and recurrent meningitis without fistula: case report","volume":"12","author":"Costa","year":"2018","unstructured":"Costa FANeto OMGibbon FLSilva GGAlam LA.Gorham\u2019s syndrome associated with Chiari I malformation and recurrent meningitis without fistula: case report. J Clin Diagn Res. 2018;12(5):PD17-PD19."},{"issue":"4","key":"bib34","series-title":"Pediatr Neurosurg","first-page":"212","article-title":"A rare case of lymphangiomatosis of the craniocervical spine in conjunction with a Chiari I malformation","volume":"39","author":"Jea","year":"2003","unstructured":"Jea AMcNeil ABhatia SA rare case of lymphangiomatosis of the craniocervical spine in conjunction with a Chiari I malformation. Pediatr Neurosurg. 2003;39(4):212-215."},{"issue":"3","key":"bib35","series-title":"J Neuroophthalmol","first-page":"419","article-title":"Vanishing act: Gorham-Stout disease leading to dynamic cerebrospinal fluid abnormalities","volume":"38","author":"Peragallo","year":"2018","unstructured":"Peragallo JHSoares BP.Vanishing act: Gorham-Stout disease leading to dynamic cerebrospinal fluid abnormalities. J Neuroophthalmol. 2018;38(3):419-421."},{"issue":"5","key":"bib36","series-title":"Neurol Neurochir Pol","first-page":"427","article-title":"Cerebrospinal fluid leakage and Chiari I malformation with Gorham\u2019s disease of the skull base: a case report","volume":"51","author":"Nagashima","year":"2017","unstructured":"Nagashima HMizukawa KTaniguchi MYamamoto YKohmura E.Cerebrospinal fluid leakage and Chiari I malformation with Gorham\u2019s disease of the skull base: a case report. Neurol Neurochir Pol. 2017;51(5):427-431."},{"issue":"1","key":"bib37","series-title":"J Biochem","first-page":"207","article-title":"Mapping and sequencing of RNAs without recourse to molecular cloning: application to RNAs of the Sabin 1 strain of poliovirus and its defective interfering particles","volume":"99","author":"Omata","year":"1986","unstructured":"Omata THorie HKuge SImura NNomoto A.Mapping and sequencing of RNAs without recourse to molecular cloning: application to RNAs of the Sabin 1 strain of poliovirus and its defective interfering particles. J Biochem. 1986;99(1):207-217."},{"issue":"11","key":"bib38","series-title":"AJNR Am J Neuroradiol","first-page":"E198","article-title":"Intraosseous CSF fistula in a patient with Gorham disease resulting in intracranial hypotension","volume":"32","author":"Adler","year":"2011","unstructured":"Adler FGupta NHess CPDowd CFDillon WP.Intraosseous CSF fistula in a patient with Gorham disease resulting in intracranial hypotension. AJNR Am J Neuroradiol. 2011;32(11):E198-E200."},{"issue":"6","key":"bib39","series-title":"J Neurosurg Spine","first-page":"956","article-title":"Cerebrospinal fluid leakage in Gorham-Stout disease due to dura mater involvement after progression of an osteolytic lesion in the thoracic spine","volume":"21","author":"Suero Molina","year":"2014","unstructured":"Suero Molina EJNiederstadt TRuland VCerebrospinal fluid leakage in Gorham-Stout disease due to dura mater involvement after progression of an osteolytic lesion in the thoracic spine. J Neurosurg Spine. 2014;21(6):956-960."},{"issue":"1","key":"bib40","series-title":"Pediatr Neurosurg","first-page":"58","article-title":"Disappearing bone disease and Chiari I malformation","volume":"46","author":"Hughes","year":"2010","unstructured":"Hughes BDGrant GACummings TJFuchs HE.Disappearing bone disease and Chiari I malformation. Pediatr Neurosurg. 2010;46(1):58-61."},{"issue":"5","key":"bib41","series-title":"J Neurosurg Pediatr","first-page":"508","article-title":"Intracranial hypotension and hypertension: reversible Chiari malformation due to dynamic cerebrospinal fluid abnormalities in Gorham-Stout disease. Case report","volume":"22","author":"Yoshimoto","year":"2018","unstructured":"Yoshimoto STakai KTakahashi KYasui TTaniguchi M.Intracranial hypotension and hypertension: reversible Chiari malformation due to dynamic cerebrospinal fluid abnormalities in Gorham-Stout disease. Case report. J Neurosurg Pediatr. 2018;22(5):508-512."},{"issue":"5","key":"bib1","series-title":"World J Orthop","first-page":"694","article-title":"Vanishing bone disease (Gorham-Stout syndrome): a review of a rare entity","volume":"5","author":"Nikolaou","year":"2014","unstructured":"Nikolaou VSChytas DKorres DEfstathopoulos N.Vanishing bone disease (Gorham-Stout syndrome): a review of a rare entity. World J Orthop. 2014;5(5):694-698."},{"issue":"5","key":"bib2","series-title":"Pediatr Blood Cancer","first-page":"e27614","article-title":"Efficacy of systemic sirolimus in the treatment of generalized lymphatic anomaly and Gorham-Stout disease","volume":"66","author":"Ricci","year":"2019","unstructured":"Ricci KWHammill AMMobberley-Schuman PEfficacy of systemic sirolimus in the treatment of generalized lymphatic anomaly and Gorham-Stout disease. Pediatr Blood Cancer. 2019;66(5):e27614."},{"issue":"1","key":"bib3","series-title":"J Oral Maxillofac Surg","first-page":"199","article-title":"Vanishing bone disease: a review","volume":"69","author":"Kiran","year":"2011","unstructured":"Kiran DNAnupama A.Vanishing bone disease: a review. J Oral Maxillofac Surg. 2011;69(1):199-203."},{"issue":"3","key":"bib4","series-title":"J Oral Maxillofac Pathol","first-page":"411","article-title":"Gorham's disease: a diagnostic challenge","volume":"18","author":"Saify","year":"2014","unstructured":"Saify FYGosavi SR.Gorham's disease: a diagnostic challenge. J Oral Maxillofac Pathol. 2014;18(3):411-414."},{"issue":"1","key":"bib5","series-title":"EFORT Open Rev","first-page":"35","article-title":"Current concepts from diagnosis to management in Gorham-Stout disease: a systematic narrative review of about 350 cases","volume":"7","author":"Angelini","year":"2022","unstructured":"Angelini AMosele NPagliarini ERuggieri P.Current concepts from diagnosis to management in Gorham-Stout disease: a systematic narrative review of about 350 cases. EFORT Open Rev. 2022;7(1):35-48."},{"issue":"1","key":"bib6","series-title":"BMC Neurol","article-title":"Gorham-Stout disease affecting the spine with cerebrospinal fluid leakage and Chiari-like tonsillar herniation: a rare case report and review of literature","volume":"23","author":"Xing","year":"2023","unstructured":"Xing QQMiao MZhang QWWu YHe FF.Gorham-Stout disease affecting the spine with cerebrospinal fluid leakage and Chiari-like tonsillar herniation: a rare case report and review of literature. BMC Neurol. 2023;23(1):59."},{"issue":"5","key":"bib7","series-title":"Pediatr Blood Cancer","first-page":"832","article-title":"Clinical features and prognosis of generalized lymphatic anomaly, kaposiform lymphangiomatosis, and Gorham-Stout disease","volume":"63","author":"Ozeki","year":"2016","unstructured":"Ozeki MFujino AMatsuoka KNosaka SKuroda TFukao T.Clinical features and prognosis of generalized lymphatic anomaly, kaposiform lymphangiomatosis, and Gorham-Stout disease. Pediatr Blood Cancer. 2016;63(5):832-838."},{"issue":"5","key":"bib8","series-title":"J Bone Joint Surg Am","first-page":"985","article-title":"Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis","volume":"37-A","author":"Gorham","year":"1955","unstructured":"Gorham LWStout AP.Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis. J Bone Joint Surg Am. 1955;37-A(5):985-1004."},{"key":"bib9","series-title":"Front Immunol","article-title":"The molecular mechanism of Gorham syndrome: an update","volume":"14","author":"Xiang","year":"2023","unstructured":"Xiang JZhong W.The molecular mechanism of Gorham syndrome: an update. Front Immunol. 2023;14:1165091."},{"key":"bib10","series-title":"Bone","first-page":"47","article-title":"Viewpoints on vessels and vanishing bones in Gorham-Stout disease","volume":"63","author":"Dellinger","year":"2014","unstructured":"Dellinger MTGarg NOlsen BR.Viewpoints on vessels and vanishing bones in Gorham-Stout disease. Bone. 2014;63:47-52."},{"key":"bib11","series-title":"Ann N Y Acad Sci","first-page":"203","article-title":"Gorham\u2019s disease: an osseous disease of lymphangiogenesis?","volume":"1131","author":"Radhakrishnan","year":"2008","unstructured":"Radhakrishnan KRockson SG.Gorham\u2019s disease: an osseous disease of lymphangiogenesis? Ann N Y Acad Sci. 2008;1131:203-205."},{"issue":"14","key":"bib12","series-title":"Circulation","first-page":"1413","article-title":"Podoplanin-expressing cells derived from bone marrow play a crucial role in postnatal lymphatic neovascularization","volume":"122","author":"Lee","year":"2010","unstructured":"Lee JYPark CCho YPPodoplanin-expressing cells derived from bone marrow play a crucial role in postnatal lymphatic neovascularization. Circulation. 2010;122(14):1413-1425."},{"issue":"1","key":"bib13","series-title":"Lymphology","first-page":"18","article-title":"Primary versus trauma-induced Gorham-Stout disease","volume":"51","author":"Tanoue","year":"2018","unstructured":"Tanoue NMoedano LWitte MMontague MLukefahr ABernas M.Primary versus trauma-induced Gorham-Stout disease. Lymphology. 2018;51(1):18-27."},{"issue":"6","key":"bib14","series-title":"Am J Med","first-page":"1151","article-title":"Gorham\u2019s syndrome: a case report and review of the literature","volume":"83","author":"Choma","year":"1987","unstructured":"Choma NDBiscotti CVBauer TWMehta ACLicata AA.Gorham\u2019s syndrome: a case report and review of the literature. Am J Med. 1987;83(6):1151-1156."},{"key":"bib15","series-title":"Front Surg","article-title":"Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham-Stout syndrome: a case report and literature review","volume":"9","author":"Zhang","year":"2022","unstructured":"Zhang HHan CPang DSurgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham-Stout syndrome: a case report and literature review. Front Surg. 2022;9:981025."},{"issue":"4","key":"bib16","series-title":"Childs Nerv Syst","first-page":"695","article-title":"Gorham-Stout disease of skull base leading to cranial settling and rhinorrhea: a case-based review","volume":"38","author":"Maroufi","year":"2022","unstructured":"Maroufi SFHabibi ZDabbagh Ohadi MAMohammadi ENejat F.Gorham-Stout disease of skull base leading to cranial settling and rhinorrhea: a case-based review. Childs Nerv Syst. 2022;38(4):695-703."},{"issue":"7","key":"bib17","series-title":"Medicina (Kaunas)","first-page":"681","article-title":"Gorham-Stout disease with multiple bone involvement-challenging diagnosis of a rare disease and literature review","volume":"57","author":"Momanu","year":"2021","unstructured":"Momanu ACaba LGorduza NCGorham-Stout disease with multiple bone involvement-challenging diagnosis of a rare disease and literature review. Medicina (Kaunas). 2021;57(7):681."},{"issue":"5","key":"bib18","series-title":"J Thorac Cardiovasc Surg","first-page":"1205","article-title":"Duropleural fistula as a consequence of Gorham-Stout syndrome: a combination of 2 rare conditions","volume":"131","author":"Agrawal","year":"2006","unstructured":"Agrawal RMohammed IReilly PG.Duropleural fistula as a consequence of Gorham-Stout syndrome: a combination of 2 rare conditions. J Thorac Cardiovasc Surg. 2006;131(5):1205-1206."},{"key":"bib19","series-title":"J Clin Neurosci","first-page":"486","article-title":"Multifocal Gorham-Stout disease associated with Chiari I malformation and recurrent aseptic meningitis: case report and review of literature","volume":"72","author":"Stephens","year":"2020","unstructured":"Stephens SSquires LCampbell RDavies JChaseling R.Multifocal Gorham-Stout disease associated with Chiari I malformation and recurrent aseptic meningitis: case report and review of literature. J Clin Neurosci. 2020;72:486-492."},{"issue":"3","key":"bib20","series-title":"Bol Med Hosp Infant Mex","first-page":"217","article-title":"Clinical and radiological improvement in Gorham-Stout disease after sirolimus treatment","volume":"80","author":"Barnes-Salda\u00f1a","year":"2023","unstructured":"Barnes-Salda\u00f1a FVenegas-Andrade AColin-Mart\u00ednez \u00d3Lizardo-Rodr\u00edguez AGarc\u00eda-Romero MTDur\u00e1n-McKinster C.Clinical and radiological improvement in Gorham-Stout disease after sirolimus treatment. Bol Med Hosp Infant Mex. 2023;80(3):217-221."},{"issue":"1","key":"bib21","series-title":"J Otolaryngol Head Neck Surg","article-title":"A case report of Gorham-Stout disease diagnosed during the course of recurrent meningitis and cholesteatoma","volume":"49","author":"Hosoya","year":"2020","unstructured":"Hosoya MOishi NNishiyama JOgawa K.A case report of Gorham-Stout disease diagnosed during the course of recurrent meningitis and cholesteatoma. J Otolaryngol Head Neck Surg. 2020;49(1):18."},{"issue":"2","key":"bib22","series-title":"Childs Nerv Syst","first-page":"455","article-title":"Gorham Stout disease of the temporal bone with cerebrospinal fluid leak","volume":"38","author":"Aouad","year":"2022","unstructured":"Aouad PYoung NMSaratsis AMReynolds MARyan ME.Gorham Stout disease of the temporal bone with cerebrospinal fluid leak. Childs Nerv Syst. 2022;38(2):455-460."},{"issue":"2","key":"bib23","series-title":"J Bone Miner Res","first-page":"207","article-title":"Gorham-Stout syndrome: a monocyte-mediated cytokine propelled disease","volume":"21","author":"Colucci","year":"2006","unstructured":"Colucci STaraboletti GPrimo LGorham-Stout syndrome: a monocyte-mediated cytokine propelled disease. J Bone Miner Res. 2006;21(2):207-218."},{"issue":"3","key":"bib24","series-title":"Clin Rheumatol","first-page":"813","article-title":"Gorham-Stout disease: radiological, histological, and clinical features of 12 cases and review of literature","volume":"35","author":"Liu","year":"2016","unstructured":"Liu YZhong DRZhou PRGorham-Stout disease: radiological, histological, and clinical features of 12 cases and review of literature. Clin Rheumatol. 2016;35(3):813-823."},{"issue":"1","key":"bib25","series-title":"Radiology","first-page":"28","article-title":"Observations on massive osteolysis; a review of the literature and report of a case","volume":"71","author":"Johnson","year":"1958","unstructured":"Johnson PMMcClure JG.Observations on massive osteolysis; a review of the literature and report of a case. Radiology. 1958;71(1):28-42."},{"key":"bib26","series-title":"Clin Neurol Neurosurg","first-page":"83","article-title":"Chiari I malformation associated with Gorham's disease of the skull base","volume":"116","author":"Coulter","year":"2014","unstructured":"Coulter ICKhan SAFlanagan AMMarks SM.Chiari I malformation associated with Gorham's disease of the skull base. Clin Neurol Neurosurg. 2014;116:83-86."},{"issue":"4","key":"bib27","series-title":"Oral Surg Oral Med Oral Pathol","first-page":"331","article-title":"Perspectives on massive osteolysis. Report of a case and review of the literature","volume":"55","author":"Heffez","year":"1983","unstructured":"Heffez LDoku HCCarter BLFeeney JE.Perspectives on massive osteolysis. Report of a case and review of the literature. Oral Surg Oral Med Oral Pathol. 1983;55(4):331-343."},{"issue":"6","key":"bib28","series-title":"Adv Wound Care (New Rochelle)","first-page":"230","article-title":"Generalized lymphatic anomaly and Gorham-Stout disease: overview and recent insights","volume":"8","author":"Ozeki","year":"2019","unstructured":"Ozeki MFukao T.Generalized lymphatic anomaly and Gorham-Stout disease: overview and recent insights. Adv Wound Care (New Rochelle). 2019;8(6):230-245."},{"issue":"3","key":"bib29","series-title":"Int J Radiat Oncol Biol Phys","first-page":"491","article-title":"Gorham\u2019s massive osteolysis: the role of radiation therapy and a review of the literature","volume":"26","author":"Dunbar","year":"1993","unstructured":"Dunbar SFRosenberg AMankin HRosenthal DSuit HD.Gorham\u2019s massive osteolysis: the role of radiation therapy and a review of the literature. Int J Radiat Oncol Biol Phys. 1993;26(3):491-497."},{"issue":"1","key":"bib30","series-title":"Adv Radiat Oncol","article-title":"A case of Gorham-Stout disease of the skull base treated with intensity modulated radiation therapy","volume":"7","author":"Roy","year":"2021","unstructured":"Roy AAndruska NBrenneman RA case of Gorham-Stout disease of the skull base treated with intensity modulated radiation therapy. Adv Radiat Oncol. 2021;7(1):100809."},{"issue":"6","key":"bib31","series-title":"Spine (Phila Pa 1976)","first-page":"545","article-title":"Massive osteolysis of the cervical spine. A case report of bone graft failure","volume":"6","author":"Woodward","year":"1981","unstructured":"Woodward HRChan DPLee J.Massive osteolysis of the cervical spine. A case report of bone graft failure. Spine (Phila Pa 1976). 1981;6(6):545-549."},{"issue":"8","key":"bib32","series-title":"Acta Neurochir (Wien)","first-page":"909","article-title":"Gorham\u2019s disease of skull base and cervical spine\u2014confusing picture in a two year old","volume":"148","author":"Girn","year":"2006","unstructured":"Girn HRTowns GChumas PHolland PChakrabarty A.Gorham\u2019s disease of skull base and cervical spine\u2014confusing picture in a two year old. Acta Neurochir (Wien). 2006;148(8):909-913."},{"issue":"5","key":"bib33","series-title":"J Clin Diagn Res","first-page":"PD17","article-title":"Gorham\u2019s syndrome associated with Chiari I malformation and recurrent meningitis without fistula: case report","volume":"12","author":"Costa","year":"2018","unstructured":"Costa FANeto OMGibbon FLSilva GGAlam LA.Gorham\u2019s syndrome associated with Chiari I malformation and recurrent meningitis without fistula: case report. J Clin Diagn Res. 2018;12(5):PD17-PD19."},{"issue":"4","key":"bib34","series-title":"Pediatr Neurosurg","first-page":"212","article-title":"A rare case of lymphangiomatosis of the craniocervical spine in conjunction with a Chiari I malformation","volume":"39","author":"Jea","year":"2003","unstructured":"Jea AMcNeil ABhatia SA rare case of lymphangiomatosis of the craniocervical spine in conjunction with a Chiari I malformation. Pediatr Neurosurg. 2003;39(4):212-215."},{"issue":"3","key":"bib35","series-title":"J Neuroophthalmol","first-page":"419","article-title":"Vanishing act: Gorham-Stout disease leading to dynamic cerebrospinal fluid abnormalities","volume":"38","author":"Peragallo","year":"2018","unstructured":"Peragallo JHSoares BP.Vanishing act: Gorham-Stout disease leading to dynamic cerebrospinal fluid abnormalities. J Neuroophthalmol. 2018;38(3):419-421."},{"issue":"5","key":"bib36","series-title":"Neurol Neurochir Pol","first-page":"427","article-title":"Cerebrospinal fluid leakage and Chiari I malformation with Gorham\u2019s disease of the skull base: a case report","volume":"51","author":"Nagashima","year":"2017","unstructured":"Nagashima HMizukawa KTaniguchi MYamamoto YKohmura E.Cerebrospinal fluid leakage and Chiari I malformation with Gorham\u2019s disease of the skull base: a case report. Neurol Neurochir Pol. 2017;51(5):427-431."},{"issue":"1","key":"bib37","series-title":"J Biochem","first-page":"207","article-title":"Mapping and sequencing of RNAs without recourse to molecular cloning: application to RNAs of the Sabin 1 strain of poliovirus and its defective interfering particles","volume":"99","author":"Omata","year":"1986","unstructured":"Omata THorie HKuge SImura NNomoto A.Mapping and sequencing of RNAs without recourse to molecular cloning: application to RNAs of the Sabin 1 strain of poliovirus and its defective interfering particles. J Biochem. 1986;99(1):207-217."},{"issue":"11","key":"bib38","series-title":"AJNR Am J Neuroradiol","first-page":"E198","article-title":"Intraosseous CSF fistula in a patient with Gorham disease resulting in intracranial hypotension","volume":"32","author":"Adler","year":"2011","unstructured":"Adler FGupta NHess CPDowd CFDillon WP.Intraosseous CSF fistula in a patient with Gorham disease resulting in intracranial hypotension. AJNR Am J Neuroradiol. 2011;32(11):E198-E200."},{"issue":"6","key":"bib39","series-title":"J Neurosurg Spine","first-page":"956","article-title":"Cerebrospinal fluid leakage in Gorham-Stout disease due to dura mater involvement after progression of an osteolytic lesion in the thoracic spine","volume":"21","author":"Suero Molina","year":"2014","unstructured":"Suero Molina EJNiederstadt TRuland VCerebrospinal fluid leakage in Gorham-Stout disease due to dura mater involvement after progression of an osteolytic lesion in the thoracic spine. J Neurosurg Spine. 2014;21(6):956-960."},{"issue":"1","key":"bib40","series-title":"Pediatr Neurosurg","first-page":"58","article-title":"Disappearing bone disease and Chiari I malformation","volume":"46","author":"Hughes","year":"2010","unstructured":"Hughes BDGrant GACummings TJFuchs HE.Disappearing bone disease and Chiari I malformation. Pediatr Neurosurg. 2010;46(1):58-61."},{"issue":"5","key":"bib41","series-title":"J Neurosurg Pediatr","first-page":"508","article-title":"Intracranial hypotension and hypertension: reversible Chiari malformation due to dynamic cerebrospinal fluid abnormalities in Gorham-Stout disease. Case report","volume":"22","author":"Yoshimoto","year":"2018","unstructured":"Yoshimoto STakai KTakahashi KYasui TTaniguchi M.Intracranial hypotension and hypertension: reversible Chiari malformation due to dynamic cerebrospinal fluid abnormalities in Gorham-Stout disease. Case report. J Neurosurg Pediatr. 2018;22(5):508-512."}],"container-title":["Journal of Neurosurgery: Case Lessons"],"original-title":[],"link":[{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-case-lessons\/9\/6\/article-CASE24298.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-case-lessons\/9\/6\/article-CASE24298.xml","content-type":"text\/html","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/thejns.org\/downloadpdf\/journals\/j-neurosurg-case-lessons\/9\/6\/article-CASE24298.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,2,2]],"date-time":"2026-02-02T23:36:38Z","timestamp":1770075398000},"score":1,"resource":{"primary":{"URL":"https:\/\/thejns.org\/view\/journals\/j-neurosurg-case-lessons\/9\/6\/article-CASE24298.xml"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,2,10]]},"references-count":82,"journal-issue":{"issue":"6"},"URL":"https:\/\/doi.org\/10.3171\/case24298","relation":{},"ISSN":["2694-1902"],"issn-type":[{"value":"2694-1902","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,2,10]]}}}