Synchronized multiple regression of diagnostic radiation-induced rather than spontaneous: disseminated primary intracranial germinoma in a woman: a case report

<p>Abstract</p> <p>Introduction</p> <p>Examples of the spontaneous regression of primary intracranial germinomas can be found in the literature. We present the case of a patient with disseminated lesions of primary intracranial germinoma which synchronously shrunk following diagnostic irradiation. We will discuss whether this regression was spontaneous or radiation-induced.</p> <p>Case presentation</p> <p>A 43-year-old Japanese woman presented to our hospital complaining of memory problems over a period of one year and blurred vision over a period of three months. Following magnetic resonance imaging, she was found to have a massive lesion in the third ventricle and small lesions in the pineal region, fourth ventricle, and in the anterior horn of the left lateral ventricle. Prior to an open biopsy to confirm the pathology of the lesions, she underwent a single cranial computed tomography scan and a single cranial digital subtraction angiography for a transcranial biopsy. Fourteen days after the first magnetic resonance image - 12 and eight days after the computed tomography scan and digital subtraction angiography, respectively - a pre-operative magnetic resonance image was taken, which showed a notable synchronous shrinkage of the third ventricle tumor, as well as shrinkage of the lesions in the pineal region and in the fourth ventricle. She did not undergo steroid administration until after a biopsy that confirmed the pathological diagnosis of pure germinoma. She then underwent whole craniospinal irradiation and went into a complete remission.</p> <p>Conclusions</p> <p>In our case report, we state that diagnostic radiation can induce the regression of germinomas; this is the most reasonable explanation for the synchronous multiple regression observed in this case of germinoma. Clinicians should keep this non-spontaneous regression in mind and monitor germinoma lesions with minimal exposure to diagnostic radiation before diagnostic confirmation, and also before radiation treatment with or without chemotherapy begins.</p

Prolonged Postoperative Pyrexia and Transient Nonnephrogenic Vasopressin-Analogue-Resistant Polyuria following Endoscopic Transsphenoidal Resection of an Infundibular Epidermoid Cyst

Prolonged postoperative pyrexia (PPP) due to Mollaret’s meningitis following endoscopic transsphenoidal surgery (eTSS) for an intracranial epidermoid cyst can be confused with postoperative meningeal infection after transsphenoidal resection, especially in the middle of the COVID-19 pandemic. Anosmia, as well as dysgeusia, cannot be evaluated in patients of eTSS for a while after surgery. We report a case of an infundibular epidermoid cyst with post-eTSS Mollaret’s meningitis (MM). The post-eTSS MM caused vasopressin-analogue-resistant polyuria (VARP) in synchronization with PPP. A 59-year-old man experiencing recurrent headaches and irregular bitemporal hemianopsia over three months was diagnosed with a suprasellar tumor. The suprasellar tumor was an infundibular cyst from the infundibular recess to the posterior lobe of the pituitary, which was gross-totally resected including the neurohypophysis via an extended eTSS. Since awakening from general anesthesia after the gross total resection (GTR) of the tumor, the patient continuously had suffered from headache until the 13th postoperative day (POD13). The patient took analgesics once a day before the surgery and three times a day after the surgery until POD11. Pyrexia (37.5–39.5 degree Celsius) in synchronization with nonnephrogenic VARP remitted on POD18. Intravenous antibiotics had little effect on changes of pyrexia. Serum procalcitonin values (reference range <0.5 ng/mL) are 0.07 ng/mL on POD12 and 0.06 ng/mL on POD18. His polyuria came to react with sublingual desmopressin after alleviation of pyrexia. He left the hospital under hormone replacement therapy without newly added neurological sequelae other than hypopituitarism. After GTR of an infundibular epidermoid cyst, based on values of serum procalcitonin, post-eTSS MM can be distinguished from infection and can be treated with symptomatic treatments. The postoperative transient nonnephrogenic VARP that differs from usual central diabetes insipidus can react with sublingual desmopressin after alleviation of PPP in the clinical course of post-eTSS MM. An infundibular epidermoid cyst should be sufficiently resected in one sitting to minimize comorbidities, its recurrence, or postoperative MM to the utmost

Acute hypopituitarism associated with periorbital swelling and cardiac dysfunction in a patient with pituitary tumor apoplexy: a case report

Abstract Background Pituitary tumor apoplexy is a rare clinical syndrome caused by acute hemorrhage or infarction in a preexisting pituitary adenoma. It typically manifests as an acute episode of headache, visual disturbance, mental status changes, cranial nerve palsy, and endocrine pituitary dysfunction. However, not all patients present with classical symptoms, so it is pertinent to appreciate the clinical spectrum of pituitary tumor apoplexy presentation. We report an unusual case of a patient with pituitary tumor apoplexy who presented with periorbital edema associated with hypopituitarism. Case presentation An 83-year-old Japanese man developed acute anterior hypopituitarism; he showed anorexia, fatigue, lethargy, severe bilateral periorbital edema, and mild cardiac dysfunction in the absence of headache, visual disturbance, altered mental status, and cranial nerve palsy. Magnetic resonance imaging showed a 2.5-cm pituitary tumor containing a mixed pattern of solid and liquid components indicating pituitary tumor apoplexy due to hemorrhage in a preexisting pituitary adenoma. Replacement therapy with oral hydrocortisone and levothyroxine relieved his symptoms of central adrenal insufficiency, central hypothyroidism, periorbital edema, and cardiac dysfunction. Conclusions Common causes of periorbital edema include infections, inflammation, trauma, allergy, kidney or cardiac dysfunction, and endocrine disorders such as primary hypothyroidism. In the present case, the patient’s acute central hypothyroidism was probably involved in the development of both periorbital edema and cardiac dysfunction. The present case highlights the need for physicians to consider periorbital edema as an unusual predominant manifestation of pituitary tumor apoplexy

Isolated adrenocorticotropic hormone deficiency and thyroiditis associated with nivolumab therapy in a patient with advanced lung adenocarcinoma: a case report and review of the literature

Abstract Introduction Immune checkpoint inhibitors are a promising class of anticancer drugs. The clinical benefits afforded by immune checkpoint inhibitors can be accompanied by immune-related adverse events that affect multiple organs, and endocrine immune-related adverse events include thyroiditis and hypophysitis. Hypophysitis is less frequent and has a less severe clinical presentation in patients treated with other immune checkpoint inhibitors, such as nivolumab, pembrolizumab, and atezolizumab, than in those treated with ipilimumab. However, studies have described isolated adrenocorticotropic hormone deficiency cases associated with nivolumab, pembrolizumab, and atezolizumab therapy, most of which occurred during the course of immune checkpoint inhibitor therapy. We report a rare case of patient with isolated adrenocorticotropic hormone deficiency that occurred after nivolumab therapy. Case presentation A 69-year-old Japanese woman with advanced lung adenocarcinoma developed painless thyroiditis with transient elevations of serum thyroid hormones during 3 months of cancer treatment with nivolumab and began thyroid hormone replacement therapy for subsequent primary hypothyroidism. Four months after nivolumab therapy was discontinued, she developed isolated adrenocorticotropic hormone deficiency; corticosteroid replacement therapy relieved her secondary adrenal insufficiency symptoms, such as anorexia and fatigue. Human leukocyte antigen typing revealed the presence of DRB1*04:05-DQB1*04:01-DQA1*03:03 and DRB1*09:01-DQB1*03:03-DQA1*03:02 haplotypes, which increase susceptibility to autoimmune polyendocrine syndrome associated with thyroid and pituitary disorders in the Japanese population. Conclusions Our patient developed thyroiditis during cancer treatment with nivolumab and subsequently exhibited isolated adrenocorticotropic hormone deficiency 4 months after discontinuing the drug. Administration of nivolumab in combination with a genetic predisposition to polyglandular autoimmunity probably caused both the thyroiditis and hypophysitis, resulting in primary hypothyroidism and isolated adrenocorticotropic hormone deficiency, respectively, in our patient. The present case highlights the need for physicians to be aware that endocrine immune-related adverse events, including hypophysitis, can occur more than several months after discontinuing a drug

Ectopic pituitary null cell adenoma arising from the infundibulum in the third ventricle: A successful endonasal transsphenoidal resection after long-term follow-up MR imaging – A technical note

Background: Since the origin and growth pattern of third ventricle ectopic pituitary adenoma (ectPA) remain unclear, its optimal surgical approach is debatable. Clinical presentation: We present a rare case of null cell pituitary adenoma arising from the pituitary infundibulum with long-term preoperative follow-up images. The tumor was resected gross-totally via an extended transsphenoidal approach. Conclusion: To our best knowledge, this is the first case with long-term preoperative follow-up images, which can bridge the knowledge gap in operations of third ventricle ectPA as following: (1) Truly third ventricle ectPA can exist, (2) the third ventricle ectPA extended into the sella turcica along the pituitary stalk, (3) this ectPA can arise from the suprasellar peri-infundibular ectopic pituitary cells or the pars tuberalis of the adenohypophysis, and therefore adhere to the optic chiasm, (4) thus neurosurgeons should take great care in resection of ectPA arising from the infundibulum, and (5) it can be resected through an endoscopic extended transsphenoidal approach. Keywords: Craniopharyngioma, Ectopic pituitary adenoma, Extended endoscopic transsphenoidal approach, Germ cell tumor, Neuroepithelial tumor, Null cell adenoma, Pars tuberalis, Suprasellar peri-infundibular ectopic pituitary cell

Unruptured medial paraclinoid internal carotid artery aneurysm embedded within symptomatic nonfunctioning pituitary giant adenoma: A high-risk comorbid pathology in transsphenoidal surgery and its countermeasures

Background: Injury of the internal carotid artery (ICA) in the cavernous portion is one of the most critical complications of transsphenoidal surgery (TSS), especially in cases of coexistence with a pituitary adenoma (PA) and ICA aneurysm. Case description: We present a rare case of unruptured medial paraclinoid ICA aneurysm (ICA-An) associated with symptomatic nonfunctioning giant PA. After endovascular coil embolization of the unruptured 4-mm saccular medial paraclinoid ICA-An, the patient underwent adenomectomy through an endoscopic endonasal TSS. During the bone resection over the right sellar floor near the right cavernous sinus, a tangle of packed coils in the treated medial paraclinoid ICA-An was observed immediately after a bite of a Kerrison rongeur. The dural layer over the coiled aneurysm had become thin to the point of transparency or complete absence. Careful inspection revealed that the bone hillock was formed by the medial paraclinoid ICA-An. Gross total resection of the adenoma was achieved without vascular injuries related to the coiled aneurysm despite postoperative transient right oculomotor paresis. Conclusions: This case conveys three important lessons about TSS: 1) coil embolization will manage a medial paraclinoid ICA-An as a sufficient preoperative procedure for TSS; 2) a medial paraclinoid ICA-An can appear directly under the sellar floor as an apparent extradural aneurysm; and; 3) surgeons should take great care in procedures near a coil-embolized medial paraclinoid ICA-An because the aneurysmal wall can be thin to the point of transparency. Keywords: Medial paraclinoid internal carotid artery aneurysm, Coil embolization, Extradural unruptured aneurysm, Pituitary giant adenoma, Endoscopic endonasal transsphenoidal surger

Technical note: Endoscopic resection of a dermoid cyst anchored to the anterior optic chiasm

Introduction: Supratentorial dermoid cysts arise due to the misplacement of embryonic inclusions in the vicinity of the developing neural tube up to the third week of life, when the neural groove begins to close. This report describes a case of a suprasellar dermoid cyst anchored to the anterior optic chiasm, which was accurately located by endoscopic observation, removed gross-totally without visual disturbance, and confirmed pathologically. Although the cyst wall was gross-totally resected, the patient’s impaired visual field returned to a normal level. The resection procedure under endoscopic vision is demonstrated. Materials and Methods: A 22-year-old man gradually developed bitemporal hemianopsia with retrobulbar pain over two months. Magnetic resonance imaging demonstrated a suprasellar cyst with intracystic fat contents forming a fluid level within the lesion. The suprasellar cyst was gross-totally removed with endoscopic endonasal transsphenoidal procedures. Intraoperative inspection confirmed that this cyst arose from the optic chiasm. Pathological examination showed the lesion as a dermoid cyst. Results: After gross-totally resection of the cyst, the patient presented a further, transient impairment of bitemporal hemianopsia; at four month follow-up, his visual disturbance was not evident any longer. Conclusion: To the best of our knowledge, ours is the only case of a dermoid cyst anchored to the anterior optic chiasma, which was visually confirmed under endoscopic observation. After surgery, the patient presented a transient impairment of the visual field, which was not evident at four month follow-up. It will contribute to a similar case, in which surgeons hesitate to make an incision in the optic chiasm. A subtotal excision should be considered in cases of dermoid cysts anchored to the anterior optic chiasm, because all the previously reported cases of suprasellar dermoid cysts are young people or those who have a relatively long life expectancy