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Open access

Diplopia associated with loop routing in deep brain stimulation: illustrative case

Yasushi Miyagi and Eiichirou Urasaki

BACKGROUND

Deep brain stimulation (DBS) is a powerful surgical option for drug-resistant movement disorders; however, electromagnetic interference (EMI) from external sources poses a potential risk for implanted electronics.

OBSERVATIONS

A 61-year-old woman with Parkinson’s disease originally had two implantable pulse generators (IPGs) for bilateral subthalamic DBS, which were then replaced with one dual-channel IPG routed in a loop. After the replacement surgery, with the same DBS programming as before the IPG replacement (bipolar setting for right, unipolar setting for left), the patient began to complain of transient paroxysmal diplopia. After multiple attempts to adjust the stimulation parameters, the diplopia was resolved by changing the left unipolar setting to a bipolar setting. At the authors’ institution, before the present case, four other patients had undergone IPG replacement with loop routing. None of these previous patients complained of diplopia; however, two of the four presented with diplopia in an experimental unipolar setting.

LESSONS

Clinicians should be aware that loop-routed circuits may generate distortion of the stimulus field in DBS, even in the absence of external EMI sources.

Open access

Intrathecal baclofen therapy for Lesch-Nyhan disease: illustrative case

Takeshi Satow, Masafumi Ogawa, and Taro Komuro

BACKGROUND

Lesch-Nyhan disease (LND) is a very rare metabolic disorder involving the purine salvage pathway. LND manifests hyperuricemia, self-mutilation, cognitive impairment, and movement disorders such as spasticity and dystonia, whose control is difficult pharmaceutically.

OBSERVATIONS

Intrathecal baclofen (ITB) therapy was received by a 22-year-old male for generalized dystonia. His paroxysmal abnormal dystonic posturing reduced after surgery, making the task of caregivers easier despite the unchanged assignment on the dystonia scale during a follow-up period of 4 years.

LESSONS

ITB may be a safe and feasible option for dystonic symptoms and difficulty with nursing care in patients with LND.

Open access

Negative-pressure hydrocephalus in the course of a complex postoperative intracranial pressure disturbance: illustrative case

Tomoya Suzuki, Shogo Kaku, Kostadin Karagiozov, and Yuichi Murayama

BACKGROUND

Negative-pressure hydrocephalus (NePH) is a rare clinical entity that presents on the background of ventriculomegaly with atypical symptoms. Its diagnosis is difficult, and some patients experience several shunt revisions until the proper solution is found.

OBSERVATIONS

The authors present a patient who developed acute deterioration due to iatrogenic NePH after surgery for a vertebral artery thrombosed giant aneurysm. The deterioration occurred after the insertion of a lumbar drain by which the authors intended to reduce a postoperative subcutaneous cerebrospinal fluid (CSF) collection. The drainage created an unexpected negative-pressure gradient in the CSF spaces, which resulted in NePH. Interventions, such as extraventricular drainage and blood patch, corrected the negative transmantle pressure and stabilized the patient’s condition.

LESSONS

Because the pathophysiology of NePH is theoretically considered to be caused by negative transmantle pressure, the intervention should be performed in order to deal with the coexistence of obstruction in the CSF pathways and a CSF leak. A blood patch would be an effective option in treating the CSF leak when the site of leakage is certain. This is the first case in which a blood patch was effectively applied in the treatment for NePH with a favorable outcome without any permanent CSF diversion.

Open access

Pituitary stone resulting in visual dysfunction and spontaneous rhinorrhea in nonfunctioning pituitary adenoma: illustrative case

Takuya Kanemitsu, Naokado Ikeda, Masao Fukumura, Satoshi Sakai, Hidehiro Oku, Motomasa Furuse, Naosuke Nonoguchi, Ryo Hiramatsu, Shinji Kawabata, Akihisa Imagawa, Tsunehiko Ikeda, and Masahiko Wanibuchi

BACKGROUND

Calcifications in pituitary adenomas are rare, being found in only 5.4%–25% of reported cases. These are divided into eggshell-like calcifications around the tumor and nodular calcifications at the center of the tumor, the latter of which are called “pituitary stones” (PSs).

OBSERVATIONS

The authors report the case of a 60-year-old male with a nonfunctional pituitary adenoma with PSs and asymptomatic ventricular dilatation who presented with spontaneous cerebrospinal fluid (CSF) rhinorrhea and rapid visual aggravation without an increase in tumor size over the course of 4 years. After endoscopic transnasal surgery, his visual acuity immediately improved temporarily. It was believed that the increased intracranial pressure due to secondary hydrocephalus resulted in visual aggravation; thus, a ventriculoperitoneal (VP) shunt was created. After creation of the VP shunt, the patient’s visual acuity improved gradually and completely. Histological findings showed that adenoma cells were observed among the lamellar bone trabeculae. To the best of the authors knowledge, this is the first report of osteoid metaplasia–type PSs in nonfunctioning pituitary adenoma.

LESSONS

PSs formed near the sellar floor and caused spontaneous CSF rhinorrhea due to direct mechanical stress on the dura mater and optic nerves, which may have caused meningitis and secondary hydrocephalus that resulted in visual impairment independent of tumor size.

Open access

Rapid contrast-induced encephalopathy after a small dose of contrast agent: illustrative case

Zhouyang Zhao, Lijin Huang, Jinhua Chen, and Hongshen Zhu

BACKGROUND

Contrast-induced encephalopathy is a rare complication of cerebral angiography with only few cases reported to date. This paper reports on contrast-induced encephalopathy mimicking meningoencephalitis following cerebral angiography with iopromide, a subhypertonic nonionic contrast agent.

OBSERVATIONS

A 50-year-old woman underwent cerebral angiography for assessment of recurrent nasopharyngeal carcinoma with invasion of internal carotid artery. The patient experienced symptoms including a disturbance of consciousness, seizures, frequent blinking, and stiffness in the extremities immediately after angiography of the left common carotid artery using iopromide (4 ml/s, total 6 ml). Computed tomography scans of the brain showed no obvious abnormalities, whereas brain magnetic resonance imaging showed swelling of the left cerebral cortex without signs of ischemia or hemorrhage. The patient was treated with intravenous rehydration, mannitol dehydration, and other supportive treatment. With this treatment, neurological status progressively improved, with complete resolution of symptoms at day 10.

LESSONS

This observation highlights that even a small dose of subhypertonic nonionic contrast agent can rapidly induce contrast encephalopathy.

Open access

Validation of the newly conceived Surgical Swedish ICH grading scale for surgically treated patients with intracerebral hemorrhage: patient series

Johan A. Haga, Frantz R. Poulsen, and Axel Forsse

BACKGROUND

The authors sought to externally validate a newly developed clinical grading scale, the Surgical Swedish ICH (SwICH) score. Patients surgically treated for spontaneous supratentorial intracerebral hemorrhage (ICH) from 2009 to 2019 in a single center in Denmark were identified. Data were retrospectively collected from patient records and neuroimaging. Surgical SwICH and ICH scores were calculated for each patient, and the validity of the Surgical SwICH was assessed and compared.

OBSERVATIONS

The 126 patients included had an overall 30-day mortality rate of 23%. All patients with a Surgical SwICH score of 0 survived past one year. No patient scored the maximum Surgical SwICH score of 6. The 30-day mortality rates for Surgical SwICH scores 1, 2, 3, and 4 were 0%, 20%, 53%, and 25%, respectively (p <0.0001 for trend). Mortality rates for ICH scores 1, 2, 3, and 4 were 0%, 11%, 33%, and 76%, respectively (p <0.001 for trend). Receiver operator characteristics showed an area under curve of 0.78 for the Surgical SwICH score and 0.80 for the ICH score (p = 0.21 difference).

LESSONS

The Surgical SwICH score was a good predictor of 30-day mortality in patients surgically treated for spontaneous supratentorial ICH. However, the Surgical SwICH score did not outperform the previously established ICH score in predicting 30-day mortality.

Open access

[68Ga]-DOTATATE PET/CT and PET/MRI in the diagnosis and management of esthesioneuroblastoma: illustrative cases

Michelle Roytman, Andrew B. Tassler, Ashutosh Kacker, Theodore H. Schwartz, Georgiana A. Dobri, Sara B. Strauss, Alyssa M. Capalbo, Rajiv S. Magge, Marissa Barbaro, Eaton Lin, Joseph R. Osborne, and Jana Ivanidze

BACKGROUND

Esthesioneuroblastoma (ENB), also known as olfactory neuroblastoma, is a rare sinonasal neuroectodermal malignancy with a slow onset of symptoms, favorable 5-year survival, and a propensity for delayed locoregional recurrence. Current treatment options include resection, adjuvant radiotherapy, and/or chemotherapy; however, because of its rarity and location, determining the optimal treatment for ENB has been challenging.

OBSERVATIONS

ENBs strongly express somatostatin receptors (SSTRs), particularly SSTR2, providing a molecular target for imaging and therapy.

LESSONs

The authors present a case series of ENBs imaged with [68Ga]-DOTATATE PET/MRI and PET/CT and discuss the emerging role of [68Ga]-DOTATATE PET for ENB diagnosis, staging, and treatment response monitoring.

Open access

Endoscopic third ventriculostomy for VP shunt malfunction during the third trimester of pregnancy: illustrative case

Ahmad K. Alhaj, Tariq Al-Saadi, Marie-Noëlle Hébert-Blouin, Kevin Petrecca, and Roy W. R. Dudley

BACKGROUND

Endoscopic third ventriculostomy (ETV) is a successful procedure for treating noncommunicating hydrocephalus as an alternative to initial ventriculoperitoneal (VP) shunt placement and as a salvage procedure when a VP shunt fails. Physiological changes of pregnancy can lead to VP shunt failure and complicate the management of shunt malfunction, particularly in the third trimester.

OBSERVATIONS

The authors present a case in which an ETV was successfully used in the third trimester (31 weeks of gestation) of pregnancy for acute hydrocephalus due to VP shunt malfunction, and the patient went on to deliver a healthy baby at term; the patient remained well in the long-term follow-up. An English-language PubMed literature review revealed four cases of VP shunt failure successfully treated with an ETV in the first or second trimester but no such reports in the third trimester of pregnancy.

LESSONS

ETV appears to be a safe and effective alternative to VP shunt replacement in the late prenatal period of pregnancy.

Open access

Endovascular treatment of a ruptured posterior fossa pure arterial malformation: illustrative case

Melissa M. J. Chua, Saksham Gupta, Walid Ibn Essayed, Dustin J. Donnelly, Habibullah Ziayee, Juan Vicenty-Padilla, Alvin S. Das, Rosalind P. M. Lai, Saef Izzy, and Mohammad Ali Aziz-Sultan

BACKGROUND

Pure arterial malformations (PAMs) are rare vascular anomalies that are commonly mistaken for other vascular malformations. Because of their purported benign natural history, PAMs are often conservatively managed. The authors report the case of a ruptured PAM leading to subarachnoid hemorrhage (SAH) with intraventricular extension that was treated endovascularly.

OBSERVATIONS

A 38-year-old man presented with a 1-day history of headaches and nausea. A computed tomography scan demonstrated diffuse SAH with intraventricular extension, and angiography revealed a right posterior inferior cerebellar artery–associated PAM. The PAM was treated with endovascular Onyx embolization.

LESSONS

To the authors’ knowledge, only 2 other cases of SAH associated with PAM have been reported. In those 2 cases, surgical clipping was pursued for definitive treatment. Here, the authors report the first case of a ruptured PAM treated using an endovascular approach, showing its feasibility as a treatment option particularly in patients in whom open surgery is too high a risk.

Open access

Functional neurological disorders in patients undergoing spinal surgery: illustrative case

Ketan Yerneni, Harsh Wadhwa, Parastou Fatemi, and Corinna C. Zygourakis

BACKGROUND

“Conversion disorder” refers to bodily dysfunction characterized by either sensory or motor neurological symptoms that are unexplainable by a medical condition. Given their somatosensory context, such disorders often require extensive medical evaluation, and the diagnosis can only be made after structural disease is excluded or fails to account for the severity and/or spectrum of the patient’s deficits.

OBSERVATIONS

The authors briefly review functional psychiatric disorders and discuss the comprehensive workup of a patient with a functional postoperative neurological deficit, drawing from their recent experience with a patient who presented with conversion disorder immediately after undergoing anterior cervical discectomy and fusion.

LESSONS

Conversion disorder has been found to be associated with bodily stress, requiring surgeons to be aware of this condition in the postoperative setting. This is especially true in neurosurgery, given the overlap of true neurological pathology, postoperative complications, and manifestations of conversion disorder. Although accurately diagnosing and managing patients with conversion disorder remains challenging, an understanding of the multifactorial nature of its etiology can help clinicians develop a methodical approach to this condition.