{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T01:00:51Z","timestamp":1790125251042,"version":"4.0.1"},"reference-count":24,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"12","license":[{"start":{"date-parts":[[2026,9,21]],"date-time":"2026-09-21T00:00:00Z","timestamp":1789948800000},"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 Neurosurg Case Lessons"],"published-print":{"date-parts":[[2026,9,21]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Gorham-Stout disease (GSD), also referred to as vanishing bone disease, is a rare lymphatic malformation in which bone is progressively destroyed and replaced by abnormal lymphatic vessels. Skull base involvement is uncommon but may result in CSF leakage, meningitis, and intracranial hypotension.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>\n                      The authors report the case of a 4-year-old boy with temporal bone GSD who presented with\n                      <jats:italic>Streptococcus pneumoniae<\/jats:italic>\n                      meningitis secondary to CSF otorrhea. After treatment with sirolimus, pamidronate, and initial surgical repair of the CSF leak, he developed a prolonged and unusual course marked by multiple recurrent transitions between intracranial hypotension and intracranial hypertension over many years. The first episode of intracranial hypertension responded to acetazolamide. Recurrent intracranial hypotension subsequently prompted repeat surgical leak repair. Later, recurrent intracranial hypertension with severe papilledema and visual field involvement required ventriculoperitoneal shunt placement. To the authors\u2019 knowledge, this is the first reported case of GSD with repeated transitions between intracranial hypotension and intracranial hypertension.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>These observations suggest that CSF dynamics in skull base GSD may remain fragile despite successful leak repair and ongoing medical therapy. Early recognition and prolonged surveillance of intracranial pressure disturbances are essential, with timely escalation to medical or neurosurgical intervention to prevent irreversible neurological and visual morbidity.<\/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\/CASE26531\">https:\/\/thejns.org\/doi\/10.3171\/CASE26531<\/jats:ext-link>\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case26531","type":"journal-article","created":{"date-parts":[[2026,9,16]],"date-time":"2026-09-16T00:00:37Z","timestamp":1789516837000},"source":"Crossref","is-referenced-by-count":0,"title":["Repeated transitions between intracranial hypotension and intracranial hypertension in Gorham-Stout disease: illustrative case"],"prefix":"10.3171","volume":"12","author":[{"given":"Lucas G.","family":"Westerink","sequence":"first","affiliation":[{"name":"Division of Neurosurgery, Department of Surgery, The Hospital for Sick Children, University of Toronto, Ontario"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5350-1763","authenticated-orcid":true,"given":"Manuel","family":"Carcao","sequence":"additional","affiliation":[{"name":"Division of Haematology\/Oncology, Department of Paediatrics, The Hospital for Sick Children, University of Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"George M.","family":"Ibrahim","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Department of Surgery, The Hospital for Sick Children, University of Toronto, Ontario"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"issue":"3","key":"bib1","series-title":"Clin Rheumatol","first-page":"813","article-title":"Gorham\u2013Stout disease: radiological, histological, and clinical features of 12 cases and review of literature","volume":"35","author":"Liu","year":"2016","unstructured":"Liu YZhong DRZhou PRGorham\u2013Stout disease: radiological, histological, and clinical features of 12 cases and review of literature. Clin Rheumatol. 2016;35(3):813-823. doi:10.1007\/s10067-014-2780-2"},{"issue":"1","key":"bib2","series-title":"Orphanet J Rare Dis","first-page":"134","article-title":"Clinical features and current management experience in Gorham\u2013Stout disease: a systematic review","volume":"20","author":"Zhou","year":"2025","unstructured":"Zhou ZQiu TZhou JClinical features and current management experience in Gorham\u2013Stout disease: a systematic review. Orphanet J Rare Dis. 2025;20(1):134. doi:10.1186\/s13023-025-03649-9"},{"issue":"3","key":"bib3","series-title":"Yonago Acta Med","first-page":"318","article-title":"A case of Gorham\u2013Stout disease treated with fistula closure by transmeatal approach","volume":"64","author":"Watanabe","year":"2021","unstructured":"Watanabe TYazama HKunimoto YKoyama SFujiwara K.A case of Gorham\u2013Stout disease treated with fistula closure by transmeatal approach. 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