-
Child's Nervous System : ChNS :... Jul 2024Pediatric optic pathway/hypothalamic gliomas (OPHG) pose challenges in treatment due to their location and proximity to vital structures. Surgical resection plays a key... (Meta-Analysis)
Meta-Analysis Review
Pediatric optic pathway/hypothalamic gliomas (OPHG) pose challenges in treatment due to their location and proximity to vital structures. Surgical resection plays a key role in the management of OPHG especially when the tumor exhibits mass effect and causes symptoms. However, data regarding outcomes and complications of surgical resection for OPHG remains heterogenous. The authors performed a systematic review on pediatric OPHG in four databases: PubMed, EMBASE, Cochrane Library, and Google Scholar. We included studies that reported on the visual outcomes and complications of OPHG resection. A meta-analysis was performed and reported per the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. A total of 26 retrospective studies were included. Seven hundred ninety-seven pediatric patients with OPHG undergoing surgical resection were examined. A diagnosis of NF1 was confirmed in 9.7%. Gross total resection was achieved in 36.7%. Intraorbital optic pathway gliomas showed a significantly higher gross total resection rate compared to those located in the chiasmatic/hypothalamic region (75.8% vs. 9.6%). Postoperatively, visual acuity improved in 24.6%, remained unchanged in 68.2%, and worsened in 18.2%. Complications included hydrocephalus (35.4%), anterior pituitary dysfunction (19.6%), and transient diabetes insipidus (29%). Tumor progression post-resection occurred in 12.8%, through a mean follow-up of 53.5 months. Surgical resection remains an essential strategy for treating symptomatic and large pediatric OPHG and can result in favorable vision outcomes in most patients. Careful patient selection is critical. Patients should be monitored for hydrocephalus development postoperatively and followed up to assess for tumor progression and adjuvant treatment necessity.
Topics: Humans; Child; Postoperative Complications; Hypothalamic Neoplasms; Glioma; Optic Nerve Glioma; Neurosurgical Procedures; Treatment Outcome; Child, Preschool
PubMed: 38649470
DOI: 10.1007/s00381-024-06407-7 -
Clinical Case Reports Mar 2024Primary GBM of the optic nerve and chiasma should be included in the differential diagnosis of progressive lesions despite initial treatment; clinicians should avoid...
KEY CLINICAL MESSAGE
Primary GBM of the optic nerve and chiasma should be included in the differential diagnosis of progressive lesions despite initial treatment; clinicians should avoid delay in confirming the histology to initiate proper treatment and improve prognosis.
ABSTRACT
Primary GBM of the optic nerve or chiasma is very rare. The characteristics of this condition have not been well-described, which poses difficulties in establishing the correct diagnosis, affecting the treatment and the prognosis. We present a case of GBM of the optic chiasma diagnosed through an open biopsy at our centre. Following the PRISMA statement, we also conducted a systematic review after protocol registration in PROSPERO (CRD42021285855). We searched Medline and Embase through Ovid from inception until December 31, 2021. Two reviewers independently screened the studies. Studies were eligible for inclusion if they reported cases of primary GBM confined to the optic nerve or chiasma as the initial radiological diagnosis. A 77-year-old female was referred for progressive visual loss lasting 8 weeks. MRI revealed a suspected lesion in the left chiasma. The patient's vision deteriorated further despite initially diagnosing an inflammatory process and empirical treatment with corticosteroids. Subsequently, the patient underwent an open biopsy and surgical debulking. Histology, including epigenetic analysis, confirmed GBM grade IV. Radiochemotherapy was administered. The patient died 19 months after surgery. We identified 45 similar cases (22 female) reported in 35 studies between 1949 and 2020. The mean age of the cases was 61 (SD = 14.6). Most cases were misdiagnosed and mistreated accordingly, so there was a median delay of 8 weeks (IQR: 5-14 weeks) in obtaining histological confirmation of the diagnosis, delaying the initiation of appropriate treatment. Five cases became no treatment since the patients died shortly after the delayed histologic diagnosis. The Kaplan-Meier estimate indicated that most patients died within 20 months of presentation, with a 1-year survival rate of 50%, and untreated cases had very low survival rates compared to treated cases. Primary GBM of the optic nerve and chiasma is a rare condition primarily affecting adults. The rarity of this condition contributes to initial misdiagnosis, mistreatment, and delays in confirming the histology and initiating appropriate treatment. The prognosis remains poor, but treatment, including surgery and radiochemotherapy, improves survival.
PubMed: 38515994
DOI: 10.1002/ccr3.8636 -
Thyroid Research Feb 2024Coexistence of TSH-secreting pituitary adenoma (TSHoma) and Graves' disease (GD) is rare and complicates the management decision.
BACKGROUND
Coexistence of TSH-secreting pituitary adenoma (TSHoma) and Graves' disease (GD) is rare and complicates the management decision.
METHODS
We present a case of the co-existence of TSHoma and GD. In addition, we systematically searched articles describing TSHoma and GD in the same patient published until 20th March 2023, using Pubmed, Scopus and Embase.
CASE PRESENTATION
A 46-year-old man presented with symptoms of thyrotoxicosis. His thyroid function tests showed serum TSH 3.35 (reference range 0.3-4.2) mIU/L, FT3 19.7 (3.7-6.4) pmol/L, and FT4 68.9 (11-23.3) pmol/L. The serum TSH receptor antibody was 11.5 mIU/L (positive at ≥ 1.75 mIU/L). Pituitary magnetic resonance imaging showed macroadenoma compressing the optic chiasm. The patient underwent trans-sphenoidal resection of pituitary adenoma. Postoperatively, he remained on maintenance carbimazole and octreotide.
RESULTS
Fourteen articles comprising 15 patients were identified from the systemic search. A total of 16 patients (including the current case) were included in the systematic review. The mean (± SD) age at diagnosis was 41 ± 13.6 years. The majority were females (75%). The median (IQR) TSH was 1.95 (0.12-5.5) mIU/L, the median (IQR) free T3 was 11.7 (7.6-19.7) pmol/L and the median (IQR) free T4 level was 47.6 (33.3-64.4) pmol/L. Ten (76.9%) patients had positive TSH receptor antibody levels. 84.6% had pituitary macroadenoma. Pituitary surgery was performed in 12 (75%) patients. At the last follow-up, 4 (25%) patients had complete resolution of symptoms after pituitary surgery, 3 (18.7%) were on maintenance treatment with thionamides for GD, 1 (6.25%) on beta-blockers and 1 (6.25%) on somatostatin analog.
CONCLUSION
TSHoma and GD can co-exist, and it is essential to identify this rare association as it can significantly impact treatment strategies.
PubMed: 38311752
DOI: 10.1186/s13044-023-00184-2 -
Neurosurgical Review Jan 2024Cavernomas are histologically benign vascular malformations found at different sites in the brain. A rare site for such cavernomas, however, is the anterior optic... (Review)
Review
Cavernomas are histologically benign vascular malformations found at different sites in the brain. A rare site for such cavernomas, however, is the anterior optic pathway, comprising the optic nerve, chiasma, and optic tract-called optochiasmatic cavernomas (OCC). These lesions usually present with sudden onset or progressive vision loss, headache, and features mimicking pituitary apoplexy. In this paper, we describe a case of OCC operated at our center. We carry out an updated review of literature depicting cases of OCC, their clinical presentation, management, and postoperative complications. We also propose a novel classification system based on lesion location and further analyze these cavernoma types with respect to the surgical approach used and visual outcome. A 30-year-old lady had presented with a 3-week history of progressive bilateral vision loss and headache. Based on imaging, she was suspected to have a cavernous angioma of the chiasma and left optic tract. Due to progressive vision deterioration, the lesion was surgically excised using pterional craniotomy. Postoperatively, her visual symptoms improved, but she developed diabetes insipidus. Clinical and radiological follow-up has been done for 18 months after surgery. A total of 81 cases have been described in the literature, including the present case. Chiasmal apoplexy is the most common presentation. Surgical excision is the standard of care. Our analysis based on lesion location shows the most appropriate surgical approach to be used for each cavernoma type. Visual outcome correlates with the preoperative visual status. Visual outcome is good in patients presenting with acute chiasmal apoplexy, and when complete surgical excision is performed. The endonasal endoscopic approach was found to provide the best visual outcome. In addition to preoperative visual status, complete surgical excision predicts favorable visual outcomes in OCC. Our proposed classification system guides the appropriate surgical approach required for a particular location of the cavernoma.
Topics: Adult; Female; Humans; Headache; Hemangioma, Cavernous; Optic Chiasm; Optic Nerve; Stroke; Vision Disorders
PubMed: 38238497
DOI: 10.1007/s10143-024-02288-1 -
Biomedical Engineering Online Nov 2023The contouring of organs at risk (OARs) in head and neck cancer radiation treatment planning is a crucial, yet repetitive and time-consuming process. Recent studies have... (Meta-Analysis)
Meta-Analysis Review
PURPOSE
The contouring of organs at risk (OARs) in head and neck cancer radiation treatment planning is a crucial, yet repetitive and time-consuming process. Recent studies have applied deep learning (DL) algorithms to automatically contour head and neck OARs. This study aims to conduct a systematic review and meta-analysis to summarize and analyze the performance of DL algorithms in contouring head and neck OARs. The objective is to assess the advantages and limitations of DL algorithms in contour planning of head and neck OARs.
METHODS
This study conducted a literature search of Pubmed, Embase and Cochrane Library databases, to include studies related to DL contouring head and neck OARs, and the dice similarity coefficient (DSC) of four categories of OARs from the results of each study are selected as effect sizes for meta-analysis. Furthermore, this study conducted a subgroup analysis of OARs characterized by image modality and image type.
RESULTS
149 articles were retrieved, and 22 studies were included in the meta-analysis after excluding duplicate literature, primary screening, and re-screening. The combined effect sizes of DSC for brainstem, spinal cord, mandible, left eye, right eye, left optic nerve, right optic nerve, optic chiasm, left parotid, right parotid, left submandibular, and right submandibular are 0.87, 0.83, 0.92, 0.90, 0.90, 0.71, 0.74, 0.62, 0.85, 0.85, 0.82, and 0.82, respectively. For subgroup analysis, the combined effect sizes for segmentation of the brainstem, mandible, left optic nerve, and left parotid gland using CT and MRI images are 0.86/0.92, 0.92/0.90, 0.71/0.73, and 0.84/0.87, respectively. Pooled effect sizes using 2D and 3D images of the brainstem, mandible, left optic nerve, and left parotid gland for contouring are 0.88/0.87, 0.92/0.92, 0.75/0.71 and 0.87/0.85.
CONCLUSIONS
The use of automated contouring technology based on DL algorithms is an essential tool for contouring head and neck OARs, achieving high accuracy, reducing the workload of clinical radiation oncologists, and providing individualized, standardized, and refined treatment plans for implementing "precision radiotherapy". Improving DL performance requires the construction of high-quality data sets and enhancing algorithm optimization and innovation.
Topics: Humans; Deep Learning; Organs at Risk; Head; Head and Neck Neoplasms; Algorithms; Image Processing, Computer-Assisted
PubMed: 37915046
DOI: 10.1186/s12938-023-01159-y -
European Radiology Nov 2023We conducted a systematic review and individual participant data meta-analysis of publications reporting the ophthalmologic presentation, clinical exam, and orbital MRI... (Meta-Analysis)
Meta-Analysis
OBJECTIVES
We conducted a systematic review and individual participant data meta-analysis of publications reporting the ophthalmologic presentation, clinical exam, and orbital MRI findings in patients with giant cell arteritis and ocular manifestations.
METHODS
PubMed and Cochrane databases were searched up to January 16, 2022. Publications reporting patient-level data on patients with ophthalmologic symptoms, imaged with orbital MRI, and diagnosed with biopsy-proven giant cell arteritis were included. Demographics, clinical symptoms, exam, lab, imaging, and outcomes data were extracted. The methodological quality and completeness of reporting of case reports were assessed.
RESULTS
Thirty-two studies were included comprising 51 patients (females = 24; median age, 76 years). Vision loss (78%) and headache (45%) were commonly reported visual and cranial symptoms. Ophthalmologic presentation was unilateral (41%) or bilateral (59%). Fundus examination most commonly showed disc edema (64%) and pallor (49%). Average visual acuity was very poor (2.28 logMAR ± 2.18). Diagnoses included anterior (61%) and posterior (16%) ischemic optic neuropathy, central retinal artery occlusion (8%), and orbital infarction syndrome (2%). On MRI, enhancement of the optic nerve sheath (53%), intraconal fat (25%), and optic nerve/chiasm (14%) was most prevalent. Among patients with monocular visual symptoms, 38% showed pathologic enhancement in the asymptomatic orbit. Six of seven cases reported imaging resolution after treatment on follow-up MRIs.
CONCLUSIONS
Vision loss, pallid disc edema, and optic nerve sheath enhancement are the most common clinical, fundoscopic, and imaging findings reported in patients diagnosed with giant cell arteritis with ocular manifestations, respectively. MRI may detect subclinical inflammation and ischemia in the asymptomatic eye and may be an adjunct diagnostic tool.
CLINICAL RELEVANCE STATEMENT
Brain and orbital MRIs may have diagnostic and prognostic roles in patients with suspected giant cell arteritis who present with ophthalmic symptoms.
Topics: Female; Humans; Aged; Giant Cell Arteritis; Vision Disorders; Magnetic Resonance Imaging; Optic Neuropathy, Ischemic; Edema
PubMed: 37256352
DOI: 10.1007/s00330-023-09770-2