Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage

Beckhaus, Julia; Friedrich, Carsten; Boekhoff, Svenja; Calaminus, Gabriele; Bison, Brigitte; Eveslage, Maria; Timmermann, Beate; Flitsch, Jörg; Müller, Hermann L.

doi:10.1093/ejendo/lvad027

Cited by 17 publications

(8 citation statements)

References 25 publications

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“…We reported that not only for the subgroup of patients younger than 6 years of age at CP diagnosis but also in the older age groups (at CP diagnosis), younger age was a risk factor for events in terms of progression and relapses. On the other hand, overall survival rates were similar in all age subgroups, indicating that radio-oncological treatment and/or surgical reinterventions were efficient rescue treatment modalities in case of relapse or progression of CP ( 22 ).…”

Section: Discussionmentioning

confidence: 95%

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Perinatally diagnosed congenital craniopharyngiomas in the KRANIOPHARYNGEOM trials

Beckhaus,

Boekhoff,

Scheinemann

et al. 2023

Endocrine Connections

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Background: Craniopharyngiomas (CP) are rare embryonic tumors. Clinical presentation and outcome of patients perinatally diagnosed with congenital CP (cCP) are not clear and refer mainly to a few case reports in the literature. The aim of this study was to analyse clinical presentation and outcome in patients with cCP. Study design: Three hundred and sixty-one patients diagnosed with adamantinomatous CP were recruited 2007–2022 in KRANIOPHARYNGEOM 2007/Registry 2019 and prospectively observed. In two cases, cCP was diagnosed prenatally and in one case on the 2nd day of life. Pre- and perinatal diagnostic findings, postnatal evaluation, therapeutic interventions and outcome in these three cases of cCP were analysed. Results: All patients survived. One patient developed psychomotor retardation and a mild hemiparesis. Prenatal routine ultrasound examination led to the diagnosis of cCP. Tumor resection was performed during the early postnatal period (range: 11–51 days of age). Functional capacity, measured by Fertigkeitenskala-Münster-Heidelberg (FMH) was reduced in three and behavioural parameters, measured by the Strength and Difficulties Questionnaire (SDQ) were abnormal in two cases. Conclusion: cCP is a rare diagnosis with a prevalence of 0.83% in our study group. Compared to cases reported in the literature, the presented cases were treated immediately and had a better prognosis. Based on improvements of diagnostic and therapeutic techniques, prenatal diagnosis of cCP should lead to transfer prior to delivery of cCP patients to a specialized centre for delivery and postnatal treatment of newborns with sellar masses by a multidisciplinary team to secure the improved prognosis of these patients.

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Section: Discussionmentioning

confidence: 95%

“…We have previously reported on the negative effect of young age at diagnosis on prognoses and outcome after CP ( 21 , 22 ). CPs are rarely diagnosed in the perinatal period.…”

Section: Introductionmentioning

confidence: 99%

Perinatally diagnosed congenital craniopharyngiomas in the KRANIOPHARYNGEOM trials

Beckhaus,

Boekhoff,

Scheinemann

et al. 2023

Endocrine Connections

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“…Morbid obesity is a frequent sequela in patients with childhood-onset craniopharyngioma [ 12 ]. Patients with anterior and posterior HI and HL are at the highest risk of developing obesity [ 94 ]. Morbid obesity can result in MetS, a well-known risk factor for CVD.…”

Section: Discussionmentioning

confidence: 99%

Vascular Morbidity and Mortality in Craniopharyngioma Patients—A Scoping Review

Beckhaus,

Friedrich,

Müller

2024

Cancers

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Craniopharyngioma (CP) treatment, including surgery and radiotherapy, can have short- and long-term vascular side effects. Hypothalamic damage is related to morbid obesity and may increase the lifelong risk of experiencing vascular events in CP patients. This review summarized the available evidence regarding vascular complications in adamantinomatous or papillary CP patients, whatever their age at diagnosis. Three databases (Medline, CINAHL, Web of Science) were searched (06/2023) to retrieve eligible articles. The search was limited to peer-reviewed articles. Titles, abstracts, and full texts were screened by two independent reviewers, and data were extracted using a self-developed grid. Seventy-two studies were included in this review; the majority were case reports. Reported vascular sequela that occurred due to surgery were fusiform dilation of the carotid artery, stroke, vasospasm, hemorrhage, and aneurysm. Related conditions that emerged due to radiotherapy included Moyamoya syndrome and cavernoma. Cardiovascular morbidity and mortality often lead to hypothalamic obesity and metabolic syndrome in CP patients. Vascular damage is a rare complication of CP treatment. Surgical strategies should protect the surrounding hypothalamic and vascular structures. Patients receiving radiotherapy, particularly at a young age, should undergo magnetic resonance angiography monitoring to identify possible neurovascular sequela during post-treatment care.

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“…The Kraniopharygeom 2007 project can be mentioned as a summary of this period. Such a German project, indeed, had/has the goal to study AC from 2007 in all its main features to provide information and increase the knowledge on the outcome [ 111 ] and about very specific aspects, like cerebral infarction [ 112 ] or cardiac remodeling in AC [ 113 ] or pregnancy after childhood onset AC [ 114 ].…”

Section: Introductionmentioning

confidence: 99%

Adamantinomatous craniopharyngioma: evolution in the management

Massimi,

Palombi,

Musarra

et al. 2023

Childs Nerv Syst

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Background In spite of the continuous progresses in pediatric neurosurgery, adamantinomatous craniopharyngioma (AC) remains a challenging tumor due to its proximity to optic pathways, pituitary gland, hypothalamus, and Willis’ circle, which can result in significant endocrine, cognitive, and neurological morbidity after treatment with subsequent impact on the patient’s quality of life (QoL). The relevance that QoL has today explains the changes in the management of AC observed over the time. The goal of the present article is to provide a historical background, to show the milestones in the changes of the AC treatment, and to analyze the current main options to manage such a challenging tumor. Material and methods The pertinent literature has been reviewed. Moreover, a comparison between the past and recent personal series is reported. Results Three main eras have been identified. The first (named Cushing era) was characterized by the need to realize a harmless surgery and to define the best way to approach AC; the second (microscope era) was characterized by a tremendous technical and technological development, with remarkable results in term of safe tumor resection and control but relatively poor QoL outcomes; and the third one (current period) is characterized by an increasing integration between surgery and adjuvant treatments, with relatively minor tumor control but significant improvement of QoL (comparable overall survival). The authors’ experience reflects these changes. Two groups of children were compared: 52 cases (mean follow-up: 17.5 years) belong to the historical series (group 1, 1985–2003, aggressive surgical management) and 41 (mean follow-up: 8.5 years) to the current one (Group 2, 2004–2021, integrated management). No significant differences between the two groups were detected about recurrence rate, surgical mortality, and overall survival. However, Group 2 showed significant lower rates of postoperative panhypopituitarism, obesity, and visual deterioration. Conclusions Radical surgery allows for a good AC control with a low rate of recurrence but high risk of permanent morbidity. Despite the greater number of recurrences and surgeries, the more conservative policy, based on a combination of treatments, seems to provide the same tumor control with a better QoL. The advances in trans-nasal and trans-ventricular endoscopy, in proton therapy and in the management of the AC cyst are the main factors that allowed such an improvement.

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Outcome after pediatric craniopharyngioma: the role of age at diagnosis and hypothalamic damage

Cited by 17 publications

References 25 publications

Perinatally diagnosed congenital craniopharyngiomas in the KRANIOPHARYNGEOM trials

Perinatally diagnosed congenital craniopharyngiomas in the KRANIOPHARYNGEOM trials

Vascular Morbidity and Mortality in Craniopharyngioma Patients—A Scoping Review

Adamantinomatous craniopharyngioma: evolution in the management

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