Postoperative speech impairment and surgical approach to posterior fossa tumours in children: a prospective European multicentre cohort study

Grønbæk, Jonathan Kjær; Wibroe, Morten; Toescu, Sebastian; Frič, Radek; Thomsen, Birthe Lykke; Möller, Luciana M.; Grillner, Pernilla; Gustavsson, Bengt; Mallucci, Conor; Aquilina, Kristian; Molinari, Emanuela; Hjort, Magnus Aasved; Westerholm-Ormio, Mia; Kiudelienė, Rosita; Mudra, K; Hauser, Péter; Baarsen, Kirsten van; Hoving, Eelco W.; Zipfel, Julian; Nysom, Karsten; Schmiegelow, Kjeld; Sehested, Astrid; Juhler, Marianne; Mathiasen, René; Kjaersgaard, Mimi; Bøgeskov, Lars; Skjøth‐Rasmussen, Jane; Tamm, John Hauerberg; Poulsgaard, Lars; Gudrunardottir, Thora; Grønbæk, Sylvester Kløcker; Blichfeldt, Alberte; Raben-Levetzau, Felix Nicolai; Callesen, Michael; Rathe, Mathias; Klokker, Rikke Bassø; Öettingen, Gorm von; Mikkelsen, Torben Stamm; Henriksen, Louise Tram; Cortnum, Søren Ole Stigaard; Tofting-Olesen, Kamilla; Karppinen, Atte; Solem, Kristin; Torsvik, Ingrid Kristin; Mosand, Ann-Karin; Simonsen, Line Rapp; Ehrstedt, Christoffer; Kristiansen, Ingela; Fritzson, Karin; Haga, Line Balestrand; Fagerholt, Hege Kristine; Blixt, Helene Stømqvist; Sundgren, Hanna; Håkansson, Yvonne; Castor, Charlotte; Nyman, Per Olof; Wretman, Anne; Nilsson, Pelle; Bjørklund, Ann-Christin; Sabel, Magnus; Haij, Inga-Lill; Nilsson, Frans; Olausson, Hanna; Cummings, Claire; Flemming, Jade; Afolabi, Deborah; Phipps, Kim; Kamaly, Ian; Williams, Sharon E.; Jeelani, Noor-ul-Owase; McArthur, Donald; Wiles, Elisabeth; Walker, David; Cooper, Rebecca; Fellows, Greg; Hoole, Lizzy; Slater, Kirsty; Kandasamy, Jothy; McAndrew, Rachel; McLaughlin, Katie A.; Schumann, Martin; Avula, Shivaram; Pizer, Barry; Rutkauskienė, Giedrė; Matukevičius, Algimantas; Abbeele, Leonie van den; Markia, Balázs; Pálmafy, Beatrix; Clausen, Niels; Møller, Karen Margrethe Ottosen; Thomassen, Harald; Cappelen, Johan; Stensvold, Einar; Devennay, Irene; Lönnqvist, Tuula; Nordfors, Kristiina; Lähteenmäki, Päivi

doi:10.1016/s2352-4642(21)00274-1

Cited by 31 publications

(8 citation statements)

References 31 publications

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“… 4 A prospective study would also permit the integration and analysis of recently identified surgical risk factors excluded in this retrospective study, namely, surgical experience and extent of resection. 74 , 75 …”

Section: Discussionmentioning

confidence: 99%

Improved prediction of postoperative pediatric cerebellar mutism syndrome using an artificial neural network

Sidpra

Marcus

Löbel

et al. 2022

Neuro-Oncology Advances

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Background Postoperative paediatric cerebellar mutism syndrome (pCMS) is a common but severe complication which may arise following the resection of posterior fossa tumours in children. Two previous studies have aimed to preoperatively predict pCMS, with varying results. In this work, we examine the generalisation of these models and determine if pCMS can be predicted more accurately using an artificial neural network (ANN). Methods An overview of reviews was performed to identify risk factors for pCMS, and a retrospective dataset collected as per these defined risk factors from children undergoing resection of primary posterior fossa tumours. The ANN was trained on this dataset and its performance evaluated in comparison to logistic regression and other predictive indices via analysis of receiver operator characteristic curves. Area under the curve (AUC) and accuracy were calculated and compared using a Wilcoxon signed rank test, with p<0.05 considered statistically significant. Results 204 children were included, of whom 80 developed pCMS. The performance of the ANN (AUC 0.949; accuracy 90.9%) exceeded that of logistic regression (p<0.05) and both external models (p<0.001). Conclusion Using an ANN, we show improved prediction of pCMS in comparison to previous models and conventional methods.

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Section: Discussionmentioning

confidence: 99%

Improved prediction of postoperative pediatric cerebellar mutism syndrome using an artificial neural network

Sidpra

Marcus

Löbel

et al. 2022

Neuro-Oncology Advances

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“…Our findings help understand CMS pathophysiology and guide surgical strategy, but do not prove causal relationships and do not allow CMS prediction. The impact of avoidance of DN and SCP injury on the incidence of CMS has to be investigated in adequately powered prospective studies, since previous pathophysiological concepts (e.g., the role of the vermis) resulted in equivocal surgical study results [7,13,25,27]. In our opinion, such studies would need to include detailed preand postoperative neuroimaging, as the surgical approach itself does not necessarily mean that deep cerebellar structure remains intact: While the telovelar approach "lacks incision of any part of the cerebellum," retraction or surgical manipulation can of course still cause injury for instance to the DN [37].…”

Section: Categorymentioning

confidence: 99%

“…It is defined as a postoperative syndrome clinically characterized by delayed onset of mutism and emotional lability, which can occur in combination with muscular hypotonia, dysphagia, and cerebellar cognitive affective syndrome and cerebellar motor syndrome, and is transient, although residual sequelae can persist [14]. Additionally, some authors differentiate between paucity and absence of speech as well as symptom severity and temporal course [13,15,16,27].…”

Section: Introductionmentioning

confidence: 99%

Structural damage burden and hypertrophic olivary degeneration in pediatric postoperative cerebellar mutism syndrome

et al. 2022

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Cerebellar mutism syndrome (CMS) occurs in one out of four children after posterior fossa tumor surgery, with open questions regarding risk factors, pathophysiology, and prevention strategies. Because of similarities between several cerebellar syndromes, a common pathophysiology with damage to the dentato-thalamo-cortical and dentato-rubro-olivary pathways has been proposed. Hypertrophic olivary degeneration (HOD) is an imaging correlate of cerebellar injury observed for instance in stroke patients. Aim of this study was to investigate whether the occurrence and severity of CMS correlates with the extent of damage to the relevant anatomical structures and whether HOD is a time-dependent postoperative neuroimaging correlate of CMS. We performed a retrospective single center study of CMS patients compared with matched non-CMS controls. CMS occurred in 10 children (13% of the overall cohort) with a median age of 8 years. Dentate nucleus (DN) injury significantly correlated with CMS, and superior cerebellar peduncle (SCP) injury was associated by tendency. HOD was observed as a dynamic neuroimaging phenomenon in the postoperative course and its presence significantly correlated with CMS and DN injury. Children who later developed HOD had an earlier onset and tended to have longer persistence of CMS. These findings can guide surgical measures to protect the DN and SCP during posterior fossa tumor resections and to avoid a high damage burden (i.e., bilateral damage). Development of intraoperative neuromonitoring of the cerebellar efferent pathways as well as improved preoperative risk stratification could help to establish a patient-specific strategy with optimal balance between degree of resection and functional integrity.

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“…Data on surgical morbidity are particularly rare in children and often restricted to tumors of the posterior fossa. 44 The currently largest series refers to ependymomas rather than gliomas. However, sufficient data exist on functional impairments, such as vision, and consecutive negative effects on quality of life (QOL) in children.…”

Section: Morbiditymentioning

confidence: 99%

Neurosurgery for eloquent lesions in children: state-of-the-art rationale and technical implications of perioperative neurophysiology

Krieg

Bernhard

Ille

et al. 2022

Neurosurgical Focus

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OBJECTIVE In adult patients, an increasing group of neurosurgeons specialize entirely in the treatment of highly eloquent tumors, particularly gliomas. In contrast, extensive perioperative neurophysiological workup for pediatric cases has been limited essentially to epilepsy surgery. METHODS The authors discuss radio-oncological and general oncological considerations based on the current literature and their personal experience. RESULTS While several functional mapping modalities facilitate preoperative identification of cortically and subcortically located eloquent areas, not all are suited for children. Direct cortical intraoperative stimulation is impractical in many young patients due to the reduced excitability of the immature cortex. Behavioral requirements also limit the utility of functional MRI and magnetoencephalography in children. In contrast, MRI-derived tractography and navigated transcranial magnetic stimulation are available across ages. Herein, the authors review the oncological rationale of function-guided resection in pediatric gliomas including technical implications such as personalized perioperative neurophysiology, surgical strategies, and limitations. CONCLUSIONS Taken together, these techniques, despite the limitations of some, facilitate the identification of eloquent areas prior to tumor surgery and radiotherapy as well as during follow-up of residual tumors.

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Postoperative speech impairment and surgical approach to posterior fossa tumours in children: a prospective European multicentre cohort study

Cited by 31 publications

References 31 publications

Improved prediction of postoperative pediatric cerebellar mutism syndrome using an artificial neural network

Improved prediction of postoperative pediatric cerebellar mutism syndrome using an artificial neural network

Structural damage burden and hypertrophic olivary degeneration in pediatric postoperative cerebellar mutism syndrome

Neurosurgery for eloquent lesions in children: state-of-the-art rationale and technical implications of perioperative neurophysiology

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