Practical Algorithm for the Management of Multisutural Craniosynostosis with Associated Chiari Malformation and/or Hydrocephalus Article Swipe
Gelsomina Aruta
,
Pietro Fiaschi
,
Marco Ceraudo
,
Gianluca Piatelli
,
Valeria Capra
,
Andrea Bianconi
,
Andrea Rossi
,
Francesca Secci
,
Marco Pavanello
·
YOU?
·
· 2023
· Open Access
·
· DOI: https://doi.org/10.1159/000529129
YOU?
·
· 2023
· Open Access
·
· DOI: https://doi.org/10.1159/000529129
Introduction: The association between multisutural craniosynostosis with Chiari malformation (CM), venous hypertension, and hydrocephalus is widely described in the literature, especially in children with paediatric craniofacial syndromes. Some efforts have been done in the last years to understand the complex pathogenetic mechanisms underlying this association, and several theories have been proposed. In particular, it is now accepted that the hypothesis of the overcrowding of the posterior fossa due to precocious suture fusion is the cause of the cerebellar herniation in syndromic and non-syndromic patients, against the theory of intrinsic cerebellar anomalies, ventriculomegaly, and venous hypertension. However, whatever the pathophysiological mechanism, it is still unclear what the best management and treatment of CM and hydrocephalus are in multisutural craniosynostosis patients. The aim of this study was to report our 25 years’ experience in treating paediatric patients affected by these rare pathologies in order to propose a simple and effective therapeutic flow chart for their management. Materials and Methods: We retrospectively collected data of each patient who underwent a cranial vault remodelling (CVR) for complex multisutural craniosynostosis in our institution in the last 25 years, while monosutural craniosynostosis was excluded. We recorded data concerning type of craniosynostosis and craniofacial syndromes, presence of ventriculomegaly, and CM at presentation and clinical and radiological follow-up. Therefore, we evaluated the final outcomes (improved, stable, deteriorated) of these patients and created a practical flow chart that could help physicians choose the best surgical treatment when different pathological conditions, as Chiari malformation I (CMI) or hydrocephalus, affect complex craniosynostosis children. Results: Thirty-nine patients (39 out of 55; 70.9%), with an isolated multisutural craniosynostosis at presentation, underwent a two-step CVR as first surgery; 36 patients (92.3%) had an improved outcome, 2 patients (5.1%) had a stable outcome, and 1 patient (2.56%) had a deteriorated outcome. Other eight children (8 out of 55; 14.5%) had a radiological evidence of asymptomatic CMI at presentation. In this group, we performed CVR as first surgery. As for the final outcome, 7 patients had an improved outcome (87.5%) with good aesthetic result and stability or resolution of CMI. Finally, 7 patients (7 out of 55; 12.7%) presented a various combination of CMI and ventriculomegaly or hydrocephalus at presentation. Among them, 3 patients had an improved outcome (42.8%), and 4 patients had a deteriorated outcome (57.1%). Discussion: The prevalence of one pathological condition with associated symptoms over the others was the key factor leading our therapeutic strategy. When craniosynostosis is associated with a radiological CM, the assessment of clinical symptoms is of capital importance. When asymptomatic or pauci-symptomatic, we suggest a CVR as first step, for its efficacy in reducing tonsillar herniation and solving CM symptoms. When craniosynostosis is associated with ventricular enlargement, the presence of intracranial hypertension signs and symptoms forces physicians to first treat hydrocephalus with a ventriculo-peritoneal shunt or endoscopic third ventriculostomy. For patients with various degrees and severity of ventriculomegaly and associated CM, the outcomes were very heterogeneous, even when the same therapeutic strategy was applied to patients with similar starting conditions and symptoms. This is maybe the most unexpected and least clear part of our results. Despite the proposed algorithm comes from a clinical experience on 85% successfully treated patients with multiple craniosynostosis, more extensive and deep studies are needed to better understand CM and hydrocephalus development in such conditions.
Related Topics
Concepts
Craniosynostosis
Medicine
Hydrocephalus
Chiari malformation
Ventriculomegaly
Surgery
Craniofacial
Cranial vault
Synostosis
Pediatrics
Syringomyelia
Radiology
Magnetic resonance imaging
Skull
Psychiatry
Pregnancy
Genetics
Biology
Fetus
Metadata
- Type
- article
- Language
- en
- Landing Page
- https://doi.org/10.1159/000529129
- https://www.karger.com/Article/Pdf/529129
- OA Status
- hybrid
- Cited By
- 10
- References
- 35
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W4318694142
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
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https://openalex.org/W4318694142Canonical identifier for this work in OpenAlex
- DOI
-
https://doi.org/10.1159/000529129Digital Object Identifier
- Title
-
Practical Algorithm for the Management of Multisutural Craniosynostosis with Associated Chiari Malformation and/or HydrocephalusWork title
- Type
-
articleOpenAlex work type
- Language
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enPrimary language
- Publication year
-
2023Year of publication
- Publication date
-
2023-01-01Full publication date if available
- Authors
-
Gelsomina Aruta, Pietro Fiaschi, Marco Ceraudo, Gianluca Piatelli, Valeria Capra, Andrea Bianconi, Andrea Rossi, Francesca Secci, Marco PavanelloList of authors in order
- Landing page
-
https://doi.org/10.1159/000529129Publisher landing page
- PDF URL
-
https://www.karger.com/Article/Pdf/529129Direct link to full text PDF
- Open access
-
YesWhether a free full text is available
- OA status
-
hybridOpen access status per OpenAlex
- OA URL
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https://www.karger.com/Article/Pdf/529129Direct OA link when available
- Concepts
-
Craniosynostosis, Medicine, Hydrocephalus, Chiari malformation, Ventriculomegaly, Surgery, Craniofacial, Cranial vault, Synostosis, Pediatrics, Syringomyelia, Radiology, Magnetic resonance imaging, Skull, Psychiatry, Pregnancy, Genetics, Biology, FetusTop concepts (fields/topics) attached by OpenAlex
- Cited by
-
10Total citation count in OpenAlex
- Citations by year (recent)
-
2025: 6, 2024: 3, 2023: 1Per-year citation counts (last 5 years)
- References (count)
-
35Number of works referenced by this work
- Related works (count)
-
10Other works algorithmically related by OpenAlex
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| abstract_inverted_index.first | 272, 320, 428, 459 |
| abstract_inverted_index.fossa | 66 |
| abstract_inverted_index.least | 510 |
| abstract_inverted_index.maybe | 505 |
| abstract_inverted_index.order | 141 |
| abstract_inverted_index.shunt | 465 |
| abstract_inverted_index.signs | 453 |
| abstract_inverted_index.step, | 429 |
| abstract_inverted_index.still | 102 |
| abstract_inverted_index.study | 123 |
| abstract_inverted_index.their | 152 |
| abstract_inverted_index.them, | 365 |
| abstract_inverted_index.these | 137, 220 |
| abstract_inverted_index.third | 468 |
| abstract_inverted_index.treat | 460 |
| abstract_inverted_index.vault | 168 |
| abstract_inverted_index.while | 183 |
| abstract_inverted_index.years | 35 |
| abstract_inverted_index.(5.1%) | 283 |
| abstract_inverted_index.12.7%) | 351 |
| abstract_inverted_index.14.5%) | 303 |
| abstract_inverted_index.Chiari | 7, 242 |
| abstract_inverted_index.affect | 248 |
| abstract_inverted_index.better | 541 |
| abstract_inverted_index.choose | 232 |
| abstract_inverted_index.factor | 397 |
| abstract_inverted_index.forces | 456 |
| abstract_inverted_index.fusion | 71 |
| abstract_inverted_index.group, | 315 |
| abstract_inverted_index.needed | 539 |
| abstract_inverted_index.others | 393 |
| abstract_inverted_index.report | 126 |
| abstract_inverted_index.result | 337 |
| abstract_inverted_index.simple | 145 |
| abstract_inverted_index.stable | 286 |
| abstract_inverted_index.suture | 70 |
| abstract_inverted_index.theory | 86 |
| abstract_inverted_index.venous | 10, 93 |
| abstract_inverted_index.widely | 15 |
| abstract_inverted_index.years, | 182 |
| abstract_inverted_index.(2.56%) | 291 |
| abstract_inverted_index.(87.5%) | 333 |
| abstract_inverted_index.(92.3%) | 276 |
| abstract_inverted_index.70.9%), | 259 |
| abstract_inverted_index.Despite | 516 |
| abstract_inverted_index.against | 84 |
| abstract_inverted_index.applied | 494 |
| abstract_inverted_index.between | 3 |
| abstract_inverted_index.capital | 417 |
| abstract_inverted_index.complex | 39, 172, 249 |
| abstract_inverted_index.cranial | 167 |
| abstract_inverted_index.created | 223 |
| abstract_inverted_index.degrees | 474 |
| abstract_inverted_index.efforts | 28 |
| abstract_inverted_index.leading | 398 |
| abstract_inverted_index.outcome | 332, 371, 379 |
| abstract_inverted_index.patient | 163, 290 |
| abstract_inverted_index.propose | 143 |
| abstract_inverted_index.several | 46 |
| abstract_inverted_index.similar | 498 |
| abstract_inverted_index.solving | 438 |
| abstract_inverted_index.stable, | 217 |
| abstract_inverted_index.studies | 537 |
| abstract_inverted_index.suggest | 424 |
| abstract_inverted_index.treated | 528 |
| abstract_inverted_index.unclear | 103 |
| abstract_inverted_index.various | 354, 473 |
| abstract_inverted_index.(42.8%), | 372 |
| abstract_inverted_index.(57.1%). | 380 |
| abstract_inverted_index.Finally, | 344 |
| abstract_inverted_index.However, | 95 |
| abstract_inverted_index.Methods: | 156 |
| abstract_inverted_index.Results: | 252 |
| abstract_inverted_index.accepted | 56 |
| abstract_inverted_index.affected | 135 |
| abstract_inverted_index.children | 22, 298 |
| abstract_inverted_index.clinical | 206, 413, 523 |
| abstract_inverted_index.efficacy | 432 |
| abstract_inverted_index.evidence | 307 |
| abstract_inverted_index.improved | 279, 331, 370 |
| abstract_inverted_index.isolated | 262 |
| abstract_inverted_index.multiple | 531 |
| abstract_inverted_index.outcome, | 280, 287, 326 |
| abstract_inverted_index.outcome. | 295 |
| abstract_inverted_index.outcomes | 215, 483 |
| abstract_inverted_index.patients | 134, 221, 254, 275, 282, 328, 346, 367, 375, 471, 496, 529 |
| abstract_inverted_index.presence | 198, 449 |
| abstract_inverted_index.proposed | 518 |
| abstract_inverted_index.recorded | 189 |
| abstract_inverted_index.reducing | 434 |
| abstract_inverted_index.results. | 515 |
| abstract_inverted_index.severity | 476 |
| abstract_inverted_index.starting | 499 |
| abstract_inverted_index.strategy | 492 |
| abstract_inverted_index.surgery. | 321 |
| abstract_inverted_index.surgery; | 273 |
| abstract_inverted_index.surgical | 235 |
| abstract_inverted_index.symptoms | 390, 414, 455 |
| abstract_inverted_index.theories | 47 |
| abstract_inverted_index.treating | 132 |
| abstract_inverted_index.two-step | 269 |
| abstract_inverted_index.whatever | 96 |
| abstract_inverted_index.years’ | 129 |
| abstract_inverted_index.Materials | 154 |
| abstract_inverted_index.aesthetic | 336 |
| abstract_inverted_index.algorithm | 519 |
| abstract_inverted_index.children. | 251 |
| abstract_inverted_index.collected | 159 |
| abstract_inverted_index.condition | 387 |
| abstract_inverted_index.described | 16 |
| abstract_inverted_index.different | 238 |
| abstract_inverted_index.effective | 147 |
| abstract_inverted_index.evaluated | 212 |
| abstract_inverted_index.excluded. | 187 |
| abstract_inverted_index.extensive | 534 |
| abstract_inverted_index.intrinsic | 88 |
| abstract_inverted_index.patients, | 83 |
| abstract_inverted_index.patients. | 118 |
| abstract_inverted_index.performed | 317 |
| abstract_inverted_index.posterior | 65 |
| abstract_inverted_index.practical | 225 |
| abstract_inverted_index.presented | 352 |
| abstract_inverted_index.proposed. | 50 |
| abstract_inverted_index.stability | 339 |
| abstract_inverted_index.strategy. | 401 |
| abstract_inverted_index.symptoms. | 440, 502 |
| abstract_inverted_index.syndromic | 80 |
| abstract_inverted_index.tonsillar | 435 |
| abstract_inverted_index.treatment | 109, 236 |
| abstract_inverted_index.underwent | 165, 267 |
| abstract_inverted_index.(improved, | 216 |
| abstract_inverted_index.Therefore, | 210 |
| abstract_inverted_index.anomalies, | 90 |
| abstract_inverted_index.assessment | 411 |
| abstract_inverted_index.associated | 389, 405, 444, 480 |
| abstract_inverted_index.cerebellar | 77, 89 |
| abstract_inverted_index.concerning | 191 |
| abstract_inverted_index.conditions | 500 |
| abstract_inverted_index.endoscopic | 467 |
| abstract_inverted_index.especially | 20 |
| abstract_inverted_index.experience | 130, 524 |
| abstract_inverted_index.follow-up. | 209 |
| abstract_inverted_index.herniation | 78, 436 |
| abstract_inverted_index.hypothesis | 59 |
| abstract_inverted_index.management | 107 |
| abstract_inverted_index.mechanism, | 99 |
| abstract_inverted_index.mechanisms | 41 |
| abstract_inverted_index.paediatric | 24, 133 |
| abstract_inverted_index.physicians | 231, 457 |
| abstract_inverted_index.precocious | 69 |
| abstract_inverted_index.prevalence | 383 |
| abstract_inverted_index.resolution | 341 |
| abstract_inverted_index.syndromes, | 197 |
| abstract_inverted_index.syndromes. | 26 |
| abstract_inverted_index.underlying | 42 |
| abstract_inverted_index.understand | 37, 542 |
| abstract_inverted_index.unexpected | 508 |
| abstract_inverted_index.Discussion: | 381 |
| abstract_inverted_index.Thirty-nine | 253 |
| abstract_inverted_index.association | 2 |
| abstract_inverted_index.combination | 355 |
| abstract_inverted_index.conditions, | 240 |
| abstract_inverted_index.conditions. | 549 |
| abstract_inverted_index.development | 546 |
| abstract_inverted_index.importance. | 418 |
| abstract_inverted_index.institution | 177 |
| abstract_inverted_index.literature, | 19 |
| abstract_inverted_index.management. | 153 |
| abstract_inverted_index.monosutural | 184 |
| abstract_inverted_index.particular, | 52 |
| abstract_inverted_index.pathologies | 139 |
| abstract_inverted_index.remodelling | 169 |
| abstract_inverted_index.therapeutic | 148, 400, 491 |
| abstract_inverted_index.ventricular | 446 |
| abstract_inverted_index.association, | 44 |
| abstract_inverted_index.asymptomatic | 309, 420 |
| abstract_inverted_index.craniofacial | 25, 196 |
| abstract_inverted_index.deteriorated | 294, 378 |
| abstract_inverted_index.enlargement, | 447 |
| abstract_inverted_index.hypertension | 452 |
| abstract_inverted_index.intracranial | 451 |
| abstract_inverted_index.malformation | 8, 243 |
| abstract_inverted_index.multisutural | 4, 116, 173, 263 |
| abstract_inverted_index.overcrowding | 62 |
| abstract_inverted_index.pathogenetic | 40 |
| abstract_inverted_index.pathological | 239, 386 |
| abstract_inverted_index.presentation | 204 |
| abstract_inverted_index.radiological | 208, 306, 408 |
| abstract_inverted_index.successfully | 527 |
| abstract_inverted_index.Introduction: | 0 |
| abstract_inverted_index.deteriorated) | 218 |
| abstract_inverted_index.hydrocephalus | 13, 113, 361, 461, 545 |
| abstract_inverted_index.hypertension, | 11 |
| abstract_inverted_index.hypertension. | 94 |
| abstract_inverted_index.non-syndromic | 82 |
| abstract_inverted_index.presentation, | 266 |
| abstract_inverted_index.presentation. | 312, 363 |
| abstract_inverted_index.heterogeneous, | 486 |
| abstract_inverted_index.hydrocephalus, | 247 |
| abstract_inverted_index.retrospectively | 158 |
| abstract_inverted_index.craniosynostosis | 5, 117, 174, 185, 194, 250, 264, 403, 442 |
| abstract_inverted_index.ventriculomegaly | 359, 478 |
| abstract_inverted_index.ventriculostomy. | 469 |
| abstract_inverted_index.craniosynostosis, | 532 |
| abstract_inverted_index.ventriculomegaly, | 91, 200 |
| abstract_inverted_index.pathophysiological | 98 |
| abstract_inverted_index.pauci-symptomatic, | 422 |
| abstract_inverted_index.ventriculo-peritoneal | 464 |
| cited_by_percentile_year.max | 99 |
| cited_by_percentile_year.min | 89 |
| countries_distinct_count | 1 |
| institutions_distinct_count | 9 |
| citation_normalized_percentile.value | 0.92622236 |
| citation_normalized_percentile.is_in_top_1_percent | False |
| citation_normalized_percentile.is_in_top_10_percent | True |