Ictal Coprolalia: Three Cases with Nondominat Frontal Lobe Involvement and Review of the Literature.

Objective: Coprolalia is defined as the involuntary use of obscene, socially unacceptable, and derogatory words. Ictal coprolalia is a rare presentation of epilepsy. This study aimed to determine the localizing and lateralizing value and frequency of ictal coprolalia in epilepsy patients. Methods: Medical files, discharge summaries, and electroencephalography (EEG) reports of 2238 patients were reviewed retrospectively. We identified patients who suffered from ictal coprolalia. Electroencephalography reports, neuroimaging [brain magnetic resonance imaging (MRI), fluorodeoxyglucose positron emission tomography (FDG-PET), single-photon emission computerized tomography (SPECT)] records, F-18 FDG fused on MRI images, and ictal SPECT fused on MRI images were evaluated. Also, original and review articles were identified through a systematic search of Pubmed, Scopus, and Clarivate Analytics. Results: Ictal coprolalia was detected in 3 male (0.15%) patients. In all patients, ictal semiology was extratemporal-frontal type, and potential/proven epileptic focus was non-dominant hemisphere frontal lobe. Topectomy was done in one of the patients, including the suspected dysplastic area plus the area where the electroencephalographic ictal and interictal changes occur, on the left frontal lobe, and the patient had an Engel's classification class IIA. The data depending on the published cases showed that ictal coprolalia was dominant in the male gender and the responsible epileptic area tended to be located in the non-dominant hemisphere frontotemporal region. Conclusion: The rate of ictal coprolalia in the Turkish population is lower compared to other series. Our results are consistent with previous studies in which reported that male preponderance for ictal coprolalia and involvement of non-dominant frontal lobe.

A rare involvement in skin cancer: Merkel cell carcinoma with bone marrow infiltration in a kidney transplant recipient.

A 60-year-old man presented with a painless, rapidly growing, haemorrhagic pink nodule on the posterior of his thigh that had developed 1 month previously. He had a diagnosis of IgA nephropathy and had received a renal allograft 7 years before. An excisional biopsy was performed and the diagnosis of Merkel cell carcinoma (MCC) was made. No distant metastases was detected. 10 months after first presentation, due to the development of acute pancytopenia and concomitant FDG PET/CT findings compatible with disease progression, bone marrow biopsy was performed which revealed metastasis of MCC. Dermatologists and oncologists should be aware that MCC could potentially involve the bone marrow in organ transplant recipients. In the follow-up period, a complete blood count should be carried out; FDG PET/CT can be obtained to follow up the metabolic status of the disease and bone marrow biopsy should be performed if necessary.

Lesional resective epilepsy surgery in childhood: Comparison of two decades and long-term seizure outcome from a single center.

OBJECTIVE: Epilepsy surgery has shown efficacy in children. We aimed to assess long-term seizure outcome in children who underwent epilepsy surgery and determine predictive factors for seizure freedom. METHODS: This is a retrospective study of 196 children who underwent epilepsy surgery between 1994 and 2015 and had a minimum postoperative follow-up of 5 years. RESULTS: The median age at the time of surgery was 9.5 (0.08-19.8) years; 110 (56.1%) had temporal, 62 (31.6%) had extratemporal resections, and 24 (12.2%) had hemispheric surgery. The duration of postsurgical follow-up was between 5 and 20 years (mean±SD: 7 ± 3.2). Overall, 129 of 196 (65.8%) patients had Engel class I outcome at final visit. Among patients who underwent temporal, extratemporal and hemispheric surgery; 84 of 110 (76.4%), 34 of 62 (54.8%), and 11 of 24 (45.8%) patients had complete seizure freedom, respectively (p: 0.016). Patients with tumors had the best outcome, with 83.1% seizure freedom. The number of preoperative antiseizure medications (OR 3.19, 95% CI 1.07-9.48), the absence of postoperative focal epileptiform discharges (OR 8.98, 95% CI 4.07-19.79) were independent predictors of seizure freedom. Across two decades, the age at surgery was decreased (p: 0.003), overall seizure freedom (61.8% vs 68%) did not differ. In the past decade, a higher proportion of malformations of cortical development was operated (14.7% vs 35.9%, p: 0.007). SIGNIFICANCE: Our findings showed favorable long-term seizure outcome in children who underwent epilepsy surgery. The results are encouraging for developing centers with limited resources to establish pediatric epilepsy programs.

Evaluation of SISCOM in routine regional cerebral blood flow alterations after clozapine, in schizophrenia.

Subtraction ictal single photon emission tomography (SPET) co-registered to magnetic resonance imaging (SISCOM) is an ictal-interictal brain perfusion SPET subtraction method, developed for evaluation of brain perfusion changes applied for the identification of epileptic foci. The aim of this study was to test whether regional cerebral blood flow (rCBF) alterations due to clozapine in schizophrenic patients could also be detected with SISCOM. We have studied the brain perfusion SPET data obtained both before (pre-SPET) and 8 weeks after (post-SPET) clozapine treatment, in 20 patients with schizophrenia. These data were used for SISCOM processing. In order to identify any alterations in the perfusion pattern using SISCOM, pre- and post-SPET data were subtracted from each other. Activation maps were created and merged on either pre- or post-SPET images. Visual interpretation of brain perfusion SPET studies were performed and compared with SISCOM findings. We found that final SISCOM images and visual evaluation of pre- and post-SPET studies were well concordant in 17/20 patients. Discordance was observed in 3 patients. In 1 of these 3 patients alterations observed with SISCOM were confirmed as subtle changes on visual re-evaluation of the images. In the remaining 2 of these 3 patients, SISCOM did not confirm the changes observed by visual analysis. Additionally, SISCOM depicted perfusion alteration in occipital cortex in 5 patients. In conclusion, the algorithm of SISCOM seemed to be useful and complementary to visual evaluation, to assess rCBF changes due to clopazine in outpatient schizophrenic patients who had treatment refractoriness or intolerance of previous antipsychotics and to provide additional information when both pre- and post-SPET data were subtracted from each other.

The effect of clozapine on regional cerebral blood flow and brain metabolite ratios in schizophrenia: relationship with treatment response.

The purpose of this study was to investigate the effect of clozapine on regional cerebral blood flow (rCBF) and its relationship with response to treatment. In addition, we aimed to study the influence of clozapine on proton magnetic resonance spectroscopy ((1)H-MRS) findings in the dorsolateral prefrontal cortex (DLPFC) in a subgroup of patients. Psychopathology, neurocognitive functioning, and SPECT imaging of 22 patients were assessed at the baseline and 8 weeks after the initiation of clozapine treatment. In 10 of these patients intermediate-echo (TE: 135 ms) single-voxel (1)H-MRS was also performed at the baseline and after 8 weeks. Clozapine treatment increased the right frontal (superior and medial)/caudate perfusion ratio in the whole group, while it increased bilateral frontal (superior and medial)/caudate perfusion ratios in treatment responders. In addition, percentage changes in left and right frontal (superior and medial)/caudate perfusion ratios compared to the baseline were higher in treatment responders than in non-responders. The improvement in attention was related to the increase in percentage change in the right frontal (superior and medial)/caudate perfusion ratio, while the improvement in verbal fluency was related to the increase in percentage changes in both right and left frontal (superior and medial)/caudate perfusion ratios and to right frontal (superior and medial)/thalamus perfusion. Baseline frontal (superior and medial)/thalamus perfusion could explain 32% of the variability of percentage improvements in psychopathology. (1)H-MRS showed that the baseline PANSS general psychopathology score was inversely correlated with the baseline NAA/Cre ratio. An increased NAA/Cre ratio in DLPFC after 8 weeks of clozapine treatment was also revealed by (1)H-MRS. Our SPECT imaging results suggest the presence of an imbalance in fronto-striato-thalamic circuitry that changes with clozapine, especially in the responders, while (1)H-MRS results indicate a supportive effect of clozapine on neuronal integrity.

Unusual extracardiac findings detected on myocardial perfusion single photon emission computed tomography studies with Tc-99m sestamibi.

The authors describe the incidence and various uptake patterns of Tc-99m sestamibi (MIBI) in the extracardiac area due to unusual causes on myocardial perfusion single photon emission computed tomography (SPECT) studies. Seven patients are presented in whom incidental extracardiac findings were observed during the interpretation of the raw data besides the routine evaluation of myocardial reconstructed SPECT slices. These 7 patients were detected out of 582 consecutive patients (1.2%) who had myocardial perfusion SPECT with Tc-99m MIBI. The findings on the raw data led to additional reconstruction of thoracic SPECT images and eventually detailed examination of the extracardiac area. Two of the patients underwent surgery because of incidental extracardiac findings (thymoma and multinodular goiter) on cardiac scintigraphy. Other causes of increased extracardiac activity were the intestine protruded through the left hemithorax, uptake in the pulmonary arterial wall, and pulmonary interstitial fibrosis due to sarcoidosis. The reasons for decreased Tc-99m MIBI accumulation in the extracardiac area in the 2 other patients were significantly dilated pulmonary arteries and hydatic cyst, which were not defined before to our knowledge. Familiarity with the normal biodistribution and variable uptake patterns in the raw images becomes necessary during the interpretation of myocardial SPECT in order not to miss very unusual incidental extracardiac uptake or information that could lead to alteration in patient management. Potential underlying mechanisms of extracardiac Tc-99m MIBI accumulation are discussed, and the literature about noncardiac Tc-99m MIBI findings detected on myocardial perfusion SPECT studies was reviewed.

Simultaneous surface registration of ictal and interictal SPECT and magnetic resonance images for epilepsy studies.

BACKGROUND: Subtraction of ictal and interictal single photon emission computed tomography (SPECT) images is known to be successful in localizing the seizure focus in the pre-surgical evaluation of patients with partial epilepsy. A computer-aided methods for producing subtraction ictal SPECT co-registered to the magnetic resonance image (MRI) (the SISCOM method) is commonly used. The two registrations involved in SISCOM are (1) between the ictal-interictal SPECT images, which was shown to be the more critical, and (2) between the ictal image and MRI. OBJECTIVE: To improve the accuracy of ictal-interictal registration in SISCOM by registering all three images (ictal, interictal SPECT, MRI) simultaneously. METHODS: The registration problem is formulated as the minimization of a cost function between three surfaces. Then, to achieve a global minimum of this cost function, the Powell algorithm with randomly distributed initial configurations is used. This technique is tested by a realistic simulation study, a phantom study and a patient study. RESULTS: The results of the simulation study demonstrate that, in surface-based registration, the triple-registration method results in a smaller ictal-interictal SPECT registration error than the pair-wise registration method (P<0.05) for a range of values of the cost-function parameter. However, the improved registration error is still larger than that obtained by the normalized mutual information method (P<0.001), which is a voxel-based registration algorithm. The phantom and patient studies reveal no observable difference between registration results. CONCLUSIONS: Although the improved accuracy of triple registration is slightly worse than voxel-based registration, it will soon be possible to apply the results of this study in research utilizing the triple-registration principle to improving voxel-based results of ictal-interictal registration.

SPECT in periodic lateralized epileptiform discharges (PLEDs): a case report on PLEDs.

Periodic lateralized epileptiform discharges (PLEDs), which are known as unusual electroencephalogram (EEG) patterns, are described in a patient who had stroke and seizures. This patient underwent Tc-99m HMPAO (hexamethyl propylene amine oxime) brain single photon emission computed tomography (SPECT) imaging both during PLEDs on EEG and after the cessation of PLEDs. The initial SPECT study revealed increased CBF in the left frontal and parietal cortex extending through the left temporal region and in the left basal ganglium. After the PLEDs disappeared, the second SPECT study showed decreased perfusion on the left frontal and parietal region in the brain. Brain SPECT findings supported the contention that PLEDs may be an ictal phenomenon. Here we also present a review on PLEDs and contributions of brain SPECT as a functional imaging modality to investigate the underlying mechanism of this interesting EEG pattern.

SPECT-PET in Epilepsy and Clinical Approach in Evaluation.

In epilepsy, a detailed history, blood chemistry, routine electroencephalography, and brain MRI are important for the diagnosis of seizure type or epilepsy syndrome for the decision of appropriate drug treatment. Although antiepileptic drugs are mostly successful for controlling epileptic seizures, 20%-30% patients are resistant to medical treatment and continue to have seizures. In this intractable patient group, surgical resection is the primarily preferred treatment option. This particular group of patients should be referred to the epilepsy center for detailed investigation and further treatment. When the results of electroencephalography, MRI, and clinical status are discordant or there is no structural lesion on MRI, ictal-periictal SPECT, and interictal PET play key roles for lateralization or localization of epileptic region and guidance for the subsequent subdural electrode placement in intractable epilepsy. SPECT and PET show the functional status of the brain. SPECT and PET play important roles in the evaluation of epilepsy sydromes in childhood by showing abnormal brain regions. Most of the experience has been gained with (18)FDG-PET, in this respect. (11)C-flumazenil-PET usually deliniates the seizure focus more smaller than (18)FDG-PET and is sensitive in identifying medial temporal sclerosis. (11)C-alpha-methyl-l-tryptophan is helpful in the differentiation of epileptogenic and nonepileptogenic regions in children especially in tuberous sclerosis and multifocal cortical dysplasia for the evaluation of surgery. Finally, when there is concordance among these detailed investigations, resective surgery or palliative procedures can be discussed individually.

Reflex Seizures Triggered by Exposure to Characters With Numerical Value: A Case With Right Temporal Cortical Dysplasia.

Reflex seizures can be triggered by a variety of stimuli. We present a case with drug-resistant complex partial seizures originating in right temporal lobe triggered extensively by visual, auditory, and mental exposure to multidigit numbers. The patient was investigated in video-EEG monitoring unit and seizures were triggered by numerical stimuli. Scalp EEG findings suggested a right temporal focus but ictal semiological findings suspicious for an extratemporal area necessitated the invasive EEG study. A right anterior temporal seizure focus was established with invasive monitoring and cortical stimulation studies. Magnetic resonance imaging showed a cortical dysplasia in right anterior temporal lobe and ictal single-photon emission computed tomography confirmed the epileptogenic focus, leading to a right temporal lobectomy and amygdalohippocampectomy and a pathological diagnosis of focal cortical dysplasia type Ia. The patient is seizure-free at the end of the second postoperative year despite repeated exposures to numbers. To our knowledge, this is the first report of seizures triggered by numbers. It is also of particular importance as the reflex seizures are associated with a cortical lesion and it may suggest involvement of right anterior temporal lobe in numerical processing.

The seizure semiology consistent with frontal lobe symptomatogenic zone in children.

The aim of this study is to analyze the seizure semiology consistent with frontal lobe symptomatogenic zone in childhood. We analyzed 549 videotaped seizures from 79 patients (mean age 9.9 ± 3.8 years). Magnetic resonance imaging was normal in 30 patients. The seizures in the time interval of 10 p.m. to 6 a.m. were considered as nocturnal. The mean number of seizures per patient was 6.8 ± 7.3. The mean seizure duration was 25.7 ± 26.9 sec; postictal confusion was 27 ± 16.1 sec (7-92 seconds). The seizures were observed in sleep with a rate of 56.8%; 43.1% of them were during wakefulness. Overall 50.4% of the seizures occured during night-time sleep. Tonic seizure (77.2%) was the most frequent simple motor seizure. Versive seizures were the second most frequent type of simple motor seizure (26.7%). Clonic seizures were 17.7%, complex motor seizures were 20.5%, and dialeptic seizures were 3% of all the seizures. Epileptic spasm, myoclonic seizures, aphasia, and akinetic semiologies were not observed. Vocalization was observed in 16% of the seizures. Frontal lobe seizures in childhood have a short duration, occur frequently, especially during night time sleep, and have a brief postictal period. Tonic semiology, versive semiology are the most frequent seizure semiologies; hypermotor and secondary generalized tonic clonic seizures and vocalizations are observed less in children compared to adults.

Scintigraphic imaging of radiolabelled drug delivery systems in rabbits with arthritis.

This paper describes a novel approach for designing drug delivery systems for intra-articular (i.a.) treatment of rheumatoid arthritis. Retention of these systems was evaluated by radiolabeling with Tc-99m and gamma scintigraphy in arthritic rabbits. Liposome, niosome, lipogelosome and niogelosome formulations of Diclofenac Sodium (DFNa) have been prepared and drug release properties and in vitro characterisation studies have been carried out. According to characterisation results L1 (DMPC: CHOL: DCP (7:1:2)), L1J1 (DMPC: CHOL: DCP (7:1:2) in C-940 1:1 (w/w)), N (SUR I: CHOL: DCP (7:1:2)) and NJ1 (SUR I: CHOL: DCP (7:1:2) in C-940 1:1 (w/w)) formulations were chosen for the further studies. Retention time of these formulations was evaluated by gamma scintigraphic imaging studies. Rabbits with antigen-induced arthritis were injected intra-articularly with Tc-99m labelled drug delivery systems. Serial scintigraphic images were obtained to investigate the retentions of labelled drug delivery systems in the arthritic joints and choose a suitable formulation for the treatment protocol of arthritis. At the end of the scintigraphic imaging studies it was observed that radiolabelled lipogelosome formulation containing DFNa (L1J1) retained much longer in the experimentally arthritic knee joints of the rabbits. This formulation was used for the treatment protocol of arthritis. Mono articular arthritis was induced in the knee joints of rabbits and it was monitored at regular time intervals by measuring changes in knee joint diameter. Also macroscopic and histopathologic evaluations were performed for further evaluation of arthritis. Great retention of DFNa in the arthritic joint might reduce potential adverse systemic effects of the drug because of local administration into the diseased area. It appeared to be a promising drug delivery system for intra-articular drug delivery.

Lacrimation as an Ictal Autonomic Event in a Patient With Seizures Originating From the Right Hemisphere.

In dacrystic seizures lacrimation has previously been reported as an ictal autonomic event accompanying emotional and somatic signs. However, it has not been observed as an isolated event in autonomic seizures. We report a patient experiencing complex partial seizures characterized by lacrimation as the initial and most prominent ictal event, with no sign of crying. The patient's ictal electroencephalogram (EEG) suggested a focus in the right hemisphere, along with interictal single-photon emission computed tomographic (SPECT) and positron emission tomographic (PET) studies demonstrating hypoperfusion-hypometabolism in the right temporoparietal lobe. Magnetic resonance imaging (MRI) did not show any structural abnormalities. Involvement of the temporal lobe has been suggested for dacrystic seizures, and both dominant and nondominant hemispheric foci were detected in such cases. Therefore, partially in concordance with previous reports, our case may point to neurobiologic interaction, between the temporoparietal region and the hypothalamus, for mediating lacrimation.

High 2-deoxy-2-[18F]fluoro-D-glucose accumulation in a case of retroperitoneal fibrosis following resection of carcinoid tumor.

Positron emission tomography (PET) using 2-deoxy-2-[18F]fluoro-D-glucose (FDG) has been shown to be a highly sensitive diagnostic tool to stage, restage, and monitor the progress of various neoplasms. A number of physiological and non-neoplastic conditions, however, also may be associated with focal accumulation of FDG and can cause false-positive results. This work reports a 52-year-old man who had marked FDG accumulation in carcinoid tumor of the distal ileum. The tumor was resected. A follow-up FDG-PET scan one and a half years later revealed intense FDG accumulation in the abdominal periaortic region, suggesting recurrent malignancy. Computerized tomography (CT) of the abdomen showed soft tissue mass surrounding the aorta. Fine-needle aspiration biopsy of the periaortic soft tissue confirmed benign retroperitoneal fibrosis. This case shows that retroperitoneal fibrosis can cause intense FDG accumulation giving false impression of malignancy. In interpretation of whole-body FDG-PET, various physiological and benign causes of FDG accumulation must be considered in order to avoid pitfalls. The authors have reviewed the literature and discussed association of carcinoid tumor and retroperitoneal fibrosis.

Stripe sign in pulmonary embolism: a review of the causes.

The most common indication for radionuclide imaging of lungs is the evaluation of suspected pulmonary thromboembolism (PE). Scintigraphically PE classically produces ventilation/perfusion mismatch, that is perfusion defects in areas showing normal ventilation. Stripe sign refers to the visualization a stripe at normally perfused lung interposed between a defect and adjacent pleural surfaces as originally described by Gottschalk. In the present case the authors describe a patient with a ventilation/perfusion scan suggesting PE. She also had a stripe sign on the perfusion scan with normal ventilation. On the follow-up perfusion scintigraphy, normal perfusion was seen in the corresponding area. The literature on stripe sign is also reviewed.

Metastatic insular thyroid carcinoma: visualized on Tc-99m pertechnetate, Tc-99m MDP and iodine-131 scintigraphy; a review of the literature for other radionuclide agents.

Poorly differentiated insular thyroid carcinoma is classified as a separate entity among other tumors of the thyroid gland. Its histological pattern and clinical course are regarded as intermediate between well-differentiated and anaplastic thyroid cancer. The authors report Tc-99m pertechnetate, Tc-99m MDP and radioiodine imaging features in a 33-year-old male patient with metastatic insular carcinoma of the thyroid. The extent of involvement was almost identical in all three studies. Insular carcinoma of the thyroid was shown by biopsy, and the patient received a cumulative dose of 14,800 MBq (400 mCi) radioactive I-131. Other radionuclide imaging agents are also reviewed.

Diaphragmatic and crural FDG uptake in hyperventilating patients: a rare pattern important to recognize.

The authors describe various patterns of F-18 fluorodeoxyglucose (FDG) accumulation in the diaphragm and crura. They present 6 patients in whom incidental diaphragmatic and crural uptake of FDG was observed during positron emission tomography (PET). Hyperventilation is thought to be the potential underlying mechanism of this condition.

Increased intra-abdominal pressure results in both obstructive and nonobstructive types of impairments of renal pelvic excretion.

UNLABELLED: An experimental study has been conducted to evaluate the effects of increased intra-abdominal pressure (IAP) on the excretion function from renal pelvis into the ureter. METHODS: Sixteen adult male New Zealand rabbits were randomly allocated in two groups after obtaining basal scintigraphies. Group I consisted of 8 rabbits which were subjected to an increased IAP for ten day period. In Group II an other 8 rabbits were subjected to an increased IAP for 60 day period. At the end of the periods of increased IAP the post-pressure scintigraphic evaluations of both groups were carried out. Time to maximum activity (Tmax), T1/2 and T2/3 values of the radiopharmaceutical in the kidneys and down slope parameters (DS1/2, DS2/3) were calculated from the renogram curves of both right and left kidneys. Comparison of basal and post-pressure scintigrapies and the mean difference of the renogram parameters between groups were performed. RESULTS: All basal scintigraphies were normal. Post-pressure scintigraphies revealed an obvious delay in the excretion phase in both groups. Both kidneys of a rabbit in group I revealed a non-obstructive pattern through responding well to administration of diuretic. Furthermore both kidneys of a rabbit from group II have revealed an obstructive pattern. T1/2 and T2/3 values were significantly elevated and mean DS1/2, DS2/3 were significantly decreased in post-pressure scintigraphies of the both groups without revealing a statistically significant difference between groups I and II. The mean difference of Tmax was significantly increased in group II. CONCLUSION: The increases in IAP, through the compressive effect upon ureters together with a concomitant increase in intravesical pressure, may impair the passage of urine from renal pelvis into ureter. The impaired passage may play a role in the development of hydronephrosis which may either reveal a nonobstructive and/or obstructive pattern.