Atypical pituitary abscess lacking rim enhancement and diffusion restriction with an unusual organism, Moraxella catarrhalis: A case report and review of the literature

Background: Pituitary abscess (PA) can be fatal if diagnosed late. Rim enhancement is a typical radiological finding of PA on postgadolinium T1-weighted magnetic resonance imaging (MRI). Diffusion-weighted imaging is helpful in distinguishing PA from other sellar cystic lesions. Herein, we report the first atypical case of PA showing neither rim enhancement nor diffusion restriction with an unusual organism, Moraxella catarrhalis. Case Description: A 77-year-old woman presented with headache, polyuria, polydipsia, and fatigue for a month before presenting to a local hospital. MRI showed pituitary enlargement with contrast enhancement. She had neither fever nor visual deficits and was followed up with hormonal replacement. Six months later, she complained of visual impairment, and MRI showed further pituitary enlargement with a thickened stalk compressing the optic chiasma. Neither rim enhancement nor diffusion restriction was observed. Endoscopic endonasal transsphenoidal surgery was performed based on the radiological diagnosis of lymphocytic hypophysitis or pituitary tumors. A thick, creamy yellow pus was drained from the sellar lesion. Intraoperative rapid histopathological findings revealed polymorphonuclear leukocytes infiltrating the pituitary gland. PA was diagnosed, and irrigation and open drainage of the abscess was performed. Bacterial culture of the pus detected M. catarrhalis by mass spectrometer, confirming the diagnosis. She underwent appropriate antibiotic administration, and her visual deficits improved. Conclusion: We report the first atypical case of PA showing neither rim enhancement nor diffusion restriction with M. catarrhalis. Even if preoperative findings are not suggestive of PA, it should be considered as a differential diagnosis. Intraoperative rapid histopathological findings are useful for accurately diagnosing PA and initiating appropriate surgical treatment.

Retinoblastoma Extending up to the Optic Chiasma and Presenting as a Suprasellar Mass

Retinoblastoma is the most common ocular malignancy of childhood. It is present in childhood with leukocoria and strabismus. Most patients are diagnosed under 3 years of age. Funduscopic examination can reveal an intraocular mass, but imaging is essential for complete evaluation of the lesion. Although ultrasound is a non-invasive and relatively inexpensive screening tool, cross-sectional imaging is required to assess the involvement of optic nerve and intracranial spread. We report a case of retinoblastoma in a 5 year old male child who presented with headache diminution of vision in both eyes. Contrast enhanced magnetic resonance imaging (MRI) revealed an enhancing soft tissue mass in the right eye with involvement of optic nerve and optic chiasma and contiguous with a large suprasellar mass causing hydrocephalus. Non-contrast computed tomography (CT) showed extensive calcifications in the mass lesion.

A long-term survival case with proton beam therapy for advanced sphenoid sinus cancer with hypopituitarism

Sphenoid sinus malignancies are rare diseases. Secondary hypopituitarism associated with sphenoid sinus malignancy is not well known. A 41-year-old male complained of right ptosis. Neurological findings revealed right oculomotor, trochlear and glossopharyngeal nerve palsy. Imaging diagnosis suggested a tumor that had spread bilaterally from the sphenoid sinus to the ethmoid sinus, nasopharynx and posterior pharyngeal space. Biopsy revealed squamous cell carcinoma (SCC). Based on these findings, a clinical diagnosis of SCC of the sphenoid sinus was made. Removal of the tumor without damaging nearby organs would have been difficult because the tumor extended to the bilateral optic nerves, optic chiasma and internal carotid artery, and surgeons, therefore, recommended proton beam therapy (PBT). Before PBT, the hypopituitarism occurred in the patient and we administered hydrocortisone and levothyroxine. During treating for hypopituitarism, we performed PBT with nedaplatin and 5-fluorouracil. The daily PBT fractions were 2.2 relative biological effectiveness (RBE) for the tumor received total dose of 81.4 Gy RBE. The acute side effect of grade 2 dermatitis according to the National Cancer Institute Common Terminology Criteria for Adverse Events version 4.0. Occurred after PBT. The patient needs to take hydrocortisone and levothyroxine, but he remains in complete remission 8 years after treatment without surgery or chemotherapy. Visual function is gradually declining, but there is no evidence of severe radiation-induced optic neuropathy.

Treatment Practices in Optic Nerve Glioma

Optic nerve glioma (OPG) is a rare tumor in children and adolescents. It comprises 1–5% of central nervous system tumors. It can be sporadic or associated with the neurofibromatosis 1 (NF1) gene. These are usually slow-growing tumors and may remain localized to the optic nerve or can have encroached upon adjoining structures like optic chiasma, opposite optic nerve, and hypothalamus. So, there may be decreased or loss of vision, proptosis, focal neurological symptoms, precocious puberty, and short stature. Due to the involvement of these critical structures, its treatment should be based on multidisciplinary consensus. The treatment modalities include surgery, RT, and chemotherapy. The aim of the treatment should be to preserve vision. However, the timing and selection of optimal treatment modalities are always a clinical dilemma. Recently, there have been promising results with newer techniques of radiotherapy and chemotherapy.

Shrinkage of a pituitary metastasis of melanoma induced by pembrolizumab: a case report

Background Pituitary metastases are rare, often deriving from lung or breast cancer owing to the upper vena cava proximity. Pituitary metastases can manifest with signs and symptoms of pituitary tumors, consequent to mass effect (headache, visual impairment) and/or hormonal alterations (hyperprolactinemia, hypopituitarism, and diabetes insipidus). Immune checkpoint inhibitors burst immunity against tumors, significantly increasing patients’ survival, but their autoimmune side effects frequently involve the skin, the gastrointestinal tract, and the endocrine glands (pituitary, thyroid, pancreas). Case presentation A 77-year-old Caucasian man had undergone trans-nasal sphenoidal surgery for a nonsecreting pituitary macroadenoma in 2001, without remnant or endocrine deficits. In 2016, he was operated for a shoulder melanoma. In February 2018, imaging evaluation demonstrated metastases in lung, liver, and femur. Therefore, treatment with pembrolizumab (anti-programmed death 1) was scheduled in May 2018, but, before starting this therapy, a brain computed tomography performed for a sudden loss of consciousness detected a sellar mass of 17 × 12 mm, which extended to the pituitary stalk and compressed the optic chiasma. Focused magnetic resonance imaging confirmed the size and characteristics of the lesion, while emergency evaluation of the hormonal profile demonstrated an impairment of adrenal and thyroid function. The pituitary lesion demonstrated a remarkable shrinkage (8 × 6 mm), which was confirmed by subsequent imaging evaluations. Conclusions This is the first case reporting on effectiveness of immune checkpoint inhibitors in a patient with pituitary metastasis from a melanoma.

A Study of Dose Painting Radiotherapy Guided by Three-Dimensional Arterial Spin labeling Perfusion Magnetic Resonance Imaging for Non-Enhancing Low-Grade Gliomas

Background: The purpose of this study was to investigate the feasibility and dosimetric characteristics of dose painting for non-enhancing low-grade gliomas (NE-LGGs) guided by three-dimensional arterial spin labeling (3D-ASL) perfusion magnetic resonance imaging.Methods: Eighteen patients with NE-LGGs were enrolled. 3D-ASL, T2 fluid attenuated inversion recovery (T2 FLAIR) and contrast-enhanced T1-weight magnetic resonance images were obtained. The gross tumor volume (GTV) was delineated on the T2 FLAIR. The hyper-perfusion region of the GTV (GTV-ASL) was determined by 3D-ASL, and the GTV-SUB was obtained by subtracting the GTV-ASL from the GTV. The planning target volume (PTV), PTV-ASL and PTV-SUB were developed by expanding the external margins of the GTV, GTV-ASL and GTV-SUB, respectively. Three plans were established for each patient: in plan 1, the traditional homogeneous prescription dose to the PTV was 45-60 Gy; in plan 2 and plan 3, the dose to the PTV-ASL increased by 10-20% based on plan 1, without the maximum dose constraint to the PTV-ASL in plan 3. The dosimetric differences among the three plans were compared.Results: Compared with plan 1, the dose to 2% (D2%), 98% (D98%) and 50% (Dmean) of PTV-ASL volumes increased by 14.67%, 16.17% and 14.31% in plan 2 and 19.84%, 15.52% and 14.27% in plan 3, respectively (P < 0.05); the D2% of the PTV, PTV-SUB increased by 11.89% and 8.34% in plan 2, 15.89% and 8.49% in plan 3, respectively (P< 0.05). The PTV coverages were comparable among the three plans (P > 0.05). In Plan 2 and plan 3, the conformity indexs decreased by 18.60% and 12.79%; while the homogeneity indexs increased by 1.43 and 2 times (P<0.05). Compared with plan 1, the D0.1cc of brain stem and Dmax of optic chiasma slightly increased in plan 2 and plan 3, but the absolute doses met the dose constraint. The other organs at risk were similar among the three plans (P > 0.05).Conclusions: The sub-volume with hyper-perfusion for NE-LGG radiotherapy can be segmented based on the perfusion difference guided by 3D-ASL. And the radiation dose to the hyper-perfusion area increased safely by 10-20% through dose painting.

Baseline MRI findings as predictors of hypopituitarism in patients with non-functioning pituitary adenomas

Hypopituitarism tends to occur in large pituitary adenomas. However, similar tumors could present with strikingly different hormonal deficiencies. In this study, we looked at MRI characteristics in non-functioning pituitary adenomas (NFPA), which could predict secondary adrenal insufficiency (SAI) and central hypothyroidism (CHT). We reviewed the files of patients with NFPA attending our clinic. Tumor size, invasiveness, MR-signal intensity, and gadolinium enhancement in preoperative MRI were recorded along with documented presurgical hypopituitarism profile. Logistic regression was used to predict SAI, CHT or both (SAI/CHT) based on MRI and demographic parameters. Receiver operating characteristic curves were used to determine their diagnostic utility. 121 patients were included. Older age (P=0.021), male sex (P=0.043), stalk deviation (P<0.0001), contrast enhancement (P=0.029) and optic chiasma compression (P=0.012) were associated with SAI/CHT. Adenoma vertical height, largest diameter, and estimated volume were also strongly associated with SAI/CHT (P<0.0001). These associations remained significant in a multivariate analysis. No tumor smaller than 12mm in vertical height, 17mm in largest diameter, or 0.9 cm3 in volume was associated with SAI/CHT. At cutoff >18mm for vertical height, >23 mm for largest diameter, and >3.2 cm3 the sensitivity was around 90-92% for detecting SAI/CHT. Only vertical height was significantly associated with any one or more pituitary hormonal deficit (P=0.001). In conclusion, adenoma size, independent of the measurement used, remains the best predictor of SAI/CHT in NFPA. Dynamic testing to rule out SAI is probably indicated in adenomas larger than 18 mm vertical height, 23 mm largest diameter and 3.2cm3 adenoma volume.

Juvenile Xanthogranuloma of the Sellar Region with a 5-Year Medical History: Case Report and Literature Review

<b><i>Introduction:</i></b> Xanthogranuloma of the sellar region is a rare benign lesion, and there are few cases reported in children. Its histogenesis is controversial, and it is difficult to strictly differentiate it from craniopharyngioma (CP), Rathke’s cleft cyst, or pituitary adenoma. <b><i>Case Presentation:</i></b> A 16-year-old boy presented with a rare xanthogranuloma of the sellar region after complaining of retardation of growth 5 years previously. The ophthalmologic evaluation revealed no visual field disturbance. Endocrinological examination revealed hypopituitarism. Magnetic resonance imaging showed an intrasellar mass extending into the suprasellar region and compressing the optic chiasma, which appeared mixed signals on T1-weighted images. Endonasal transsphenoidal resection of the tumor was performed. Histological analysis of the tumor sections demonstrated granulomatous tissue with cholesterol clefts, hemosiderin deposits, fibrous tissues, multinucleated giant cells, and lymphocyte. Thus, the tumor was pathologically diagnosed as xanthogranuloma of the sellar region, which is different from adamantinomatous CP. There was no epithelial tissue in any part of the tumor including tumor capsule but have focal necrosis and calcification. His endocrinological dysfunction did not recover, so a hormonal replacement was continuously required. <b><i>Conclusion:</i></b> Xanthogranuloma of the sellar region is a rare entity but must be considered in the differential diagnosis of lesions of the sellar region, even in pediatric population. We should think about this disease when dealing with children with stunted growth accompanied by a long medical history. Our case demonstrates the natural progression of the disease, suggesting that xanthogranuloma of the sellar region without epithelial components may be an independent disease.

Development and Evaluation of Deep Learning-based Automated Segmentation of Pituitary Adenoma in Clinical Task

Purpose To create an automated segmentation method for the sellar region, several tools to extract invasiveness-related features, and evaluate their clinical usefulness by predicting the tumor consistency. Materials and Methods Patients included were diagnosed with pituitary adenoma at Peking Union Medical College Hospital. A deep convolutional neural network, called Gated-Shaped U-Net (GSU-Net), was created to automatically segment the sellar region into eight classes. Five MRI features were extracted from the segmentation results, including tumor diameters, volume, optic chiasma height, Knosp grading system, and degree of ICA contact. The clinical usefulness of proposed methods was evaluated by the diagnostic accuracy of the tumor consistency. Results 163 patients confirmed with pituitary adenoma were included as the first group and were randomly divided into a training dataset and test dataset (131 and 32 patients, respectively). 50 patients confirmed with acromegaly were included as the second group. The Dice coefficient of pituitary adenoma in important image slices was 0.940. The proposed methods achieved accuracies of over 80% for the prediction of five invasive-related MRI features. Methods derived from the automatic segmentation showing better performances than original methods and achieved AUCs of 0.840 and 0.920 for clinical models and radiomics models, respectively. Conclusion The proposed methods could automatically segment the sellar region and extract features with high accuracies. The outstanding performance of the prediction of the tumor consistency indicates their clinical usefulness for supporting neurosurgeons in judging patients’ conditions, predicting prognosis, and other downstream tasks during the preoperative period.

Cystic lesion of the sella turcica: interesting case report and review of literature

<p class="western"><span><span><strong>ABSTRACT</strong></span></span></p><p class="western"><span><span>Intrasellar arachnoid cysts are rare entities. High index of suspicion and precise radiological evaluation are the cornerstone for appropriate diagnosis and treatment. We present a symptomatic case of a sellar cystic lesion in a 52-year-old lady, who had complaints of decreased vision associated with diplopia of three months duration. Clinical evaluation showed bitemporal hemianopia. Magnetic Resonance Imaging (MRI) of brain revealed features of sellar arachnoid cyst with the intracystic component similar to cerebrospinal fluid (CSF). She underwent endoscopic transnasal transsphenoidal excision of lesion. Her visual symptoms improved well on subsequent follow up.However infrequent, sellar arachnoid cyst should be borne in mind as differential diagnosis for any cystic sellar lesions, as they can mimic pituitary adenomas, either by impairment of pituitary hormonal function or by exerting pressure effects on optic chiasma. Evaluation modalities, therapeutic challenges and literature review are presented in this case report.</span></span></p><p class="western"> </p>