OpenMed/multicare-articles huggingface dataset:
Light cupula of the horizontal semicircular canal occurring alternately on both sides: a case report
Authors: Jung Eun Shin, Chang-Hee Kim
Journal: BMC Ear Nose Throat Disord (2015)
Keywords: direction-changing positional nystagmus, head-roll test, light cupula, positional vertigo
MeSH terms: Case Reports

Background:
The light cupula is a condition wherein the cupula of the semicircular canal has a lower specific gravity than its surrounding endolymph. It is characterized by a persistent geotropic direction-changing positional nystagmus in the supine head-roll test, and the identification of a null plane with slight head-turning to either side.
Case presentation:
This study describes a case of recurring light cupula that occurred alternately on both sides. At the first episode, a null plane was identified on the right side, which led to the diagnosis of a light cupula on the right side. At the second episode, a null plane was identified on the left side, leading to the diagnosis of a light cupula on the left side.
Conclusion:
This is the first case report of recurring light cupula alternately involving both sides. Although the pathophysiology is not entirely understood yet, the light cupula should be considered as one of causes of recurrent positional vertigo.
Subacute drug-eluting stent thrombosis caused by stent underexpansion: evaluation by optical coherence tomography
Authors: Roberto Martín-Reyes, Santiago Jiménez-Valero, Felipe Navarro, Raúl Moreno
Journal: Case Rep Med (2011)
MeSH terms: Case Reports

We present the case report of a patient presenting with ST segment elevation myocardial infarction due to a subacute drug-eluting stent trombosis within the proximal segment of the left circumflex artery (LCX). Six days before a total chronic occlusion was treated at the mid segment of the LCX by overlapping two drug-eluting stents. Optical coherence tomography (OCT) was helpful to demonstrate stent underexpansion of the overlaping segment as the main mechanism of early stent thrombosis. This case is illustrative about the potential role of OCT to identify the mechanisms of ST and thus guiding the PCI procedure. Moreover, our case shows the capability of the Imagewire to cross a severe stenosis due to stent underexpansion that could not be crossed by the IVUS catheter.
Simple and easy surgical technique for infantile hemangiomas: intralesional excision and primary closure
Authors: Tadashi Nomura, Takeo Osaki, Hiroyoshi Ishinagi, Hirotaka Ejiri, Hiroto Terashi
Journal: Eplasty (2015)
Keywords: dog ear, infantile hemangioma, intralesional excision, primary closure, scar

Objective:
Infantile hemangioma (IH) is a benign vascular tumor that gradually shrinks over several years. Involuting or involuted IHs usually retain their shape, however, and result in redundant skin or conspicuous scarring due to ulceration in the proliferating phase. We present a case series of 12 patients who underwent intralesional excision and primary closure for treatment of involuting or involuted IH.
Methods:
Twelve patients (5 boys, 7 girls) underwent our treatment method for involuting or involuted IH. A blinded assessor evaluated clinical result of each patient.
Results:
Surgical results were excellent in 4 patients, good in 6, and fair in 2. A small dog ear was prominent in 1 patient; nevertheless, all parents were satisfied with the results.
Conclusions:
Intralesional excision and primary closure for treatment of involuting or involuted IH is an easy and simple procedure that does not result in dog-ear formation or elongated residual scarring.
Transcortical sensory aphasia heralding SARS-Cov-2-induced autoimmune encephalitis with gyral restricted diffusion hyperintensities: a novel case report
Authors: Arka Prava Chakraborty, Alak Pandit, Ajitava Dutta, Shambaditya Das, Goutam Ganguly, Souvik Dubey
Journal: Egypt J Neurol Psychiatr Neurosurg (2022)
Keywords: covid-19, covid-19 autoimmune encephalitis, cognitive dysfunction, gyral restricted diffusion hyperintensities, transcortical sensory aphasia
MeSH terms: Case Reports

Background:
Coronavirus disease 2019 (CoVID-19), primarily thought of as a respiratory system disease is actually a multi-system disease with immunological implications. CNS involvement in COVID has been explained in recent literature mainly for stroke, encephalopathy, encephalitis, acute disseminated encephalomyelitis and myelopathy. There are few studies characterizing clinical spectrum of COVID autoimmune encephalitis. We present a unique case of post-COVID autoimmune encephalitis in a diabetic male presenting with language dysfunction and novel radiologic findings.
Case presentation:
Patient admitted to inpatient department of a tertiary care hospital of India was evaluated by bedside clinical examination, routine blood tests, CSF study with intrathecal SARS-Cov-2 antibody detection, commercially available tests for autoimmune encephalitis, neuroviral panel with HSV PCR, EEG, 3-Tesla MRI and PET scan. Patient was found to have personality change and transcortical sensory aphasia in the outset of COVID encephalitis. MRI findings like temporal involvement and insular ribboning are also being reported. The patient was treated with IV immunoglobulin and is on an improving course.
Conclusions:
This case reports dysphasia due to COVID-mediated injury to the language networks, with novel radiologic findings. Role of parainfectious versus immune etiology is also discussed. Further studies are needed to elucidate the mechanism and clinical spectrum of post-COVID autoimmune encephalitis.
Autoantibodies to a Nodal Isoform of Neurofascin in Pediatric Chronic Inflammatory Demyelinating Polyneuropathy
Authors: Eline Chauvet, Geraldine Blanchard Rohner, Véroniqu Manel, Emilien Delmont, Joseph Boucraut, Stephanie Garcia-Tarodo
Journal: Child Neurol Open (2023)
Keywords: paranodal antibodies, pediatric chronic inflammatory demyelinating polyradiculoneuropathy, rituximab
MeSH terms: Case Reports

Pediatric chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) is an acquired immune-mediated disorder of the peripheral nervous system with a number of diagnostic pitfalls. A subset of treatment-resistant CIDP adult patients have been found with antibodies against paranodal proteins. We report the first pediatric case in a 14 year-old adolescent with a severe CIDP phenotype in whom positive anti-neurofascin 155 antibodies were found in his serum. Resistant to conventional therapies, he showed dramatic improvement when treated with Rituximab with mild to moderate functional motor disability at 24 month follow-up. In pediatric CIDP patients that remain refractory to conventional treatments, the presence of antibodies to paranodal proteins warrants investigation as it can have potential therapeutic guidance.
Primary hepatic neuroendocrine tumours-Case series of a rare malignancy
Authors: Nelson Chen, Kellee Slater
Journal: Int J Surg Case Rep (2019)
Keywords: case series, hepatobiliary cancers, neuroendocrine tumours, primary hepatic cancers, primary hepatic neuroendocrine tumours, primary liver cancers

Introduction:
Primary hepatic neuroendocrine tumours (PHNET) were first described by Edmondson et al. in 1958 and are rare, accounting for only 0.3% of all neuroendocrine tumours. Only several hundred cases have been reported.
Presentation of case:
We present two cases. The first is a 65-year-old asymptomatic male referred with a liver lesion on ultrasound performed to investigate a mildly elevated Alanine Aminotransferase (ALT). Hepatitis serology and tumour markers were normal. He had an unremarkable colonoscopy and gastroscopy. CT and MRI revealed a single liver lesion adjacent to the gallbladder suspicious for malignancy. He underwent a segment IVb/V liver resection. Histology was consistent with a 65 mm grade 2 PHNET. Subsequent Dotatate PET/CT scans have been normal at 5 years. The second is an asymptomatic 73-year-old male referred with fluctuating hepatic enzymes and a history of alcohol overuse. Imaging revealed a suspicious lesion in segment III of the liver. He underwent a left lateral liver resection. Histology revealed an 18 mm grade 1 PHNET. A subsequent Dotatate PET/CT was normal with no new disease at six months.
Discussion:
PHNET, albeit rare are in the differential diagnosis for primary hepatic malignancies. Tumour markers are usually normal and radiological imaging can mimic other hypervascular hepatic tumours. Surgery is the only curative treatment for localised disease to date.
Conclusion:
PHNET needs to be considered in asymptomatic patients with hypervascular hepatic lesions. More research is required before other adjunct treatment options can be suggested.
Isolated pan-pontine posterior reversible encephalopathy syndrome in a patient with uncontrolled hypertension
Authors: Jacqueline Marie Gowan, Antonio Liu
Journal: Clin Case Rep (2018)
Keywords: mri, albuminocytologic csf, central variant pres, encephalopathy, hypertension, pan‐pontine
MeSH terms: Case Reports

Recognition of central variant PRES is key to avoiding detrimental treatment. The pons may be the most vulnerable area in central PRES, and delayed presentation may result in greater damage. CSF reporting may lead to eventual recognition of a common profile, and possible aid in future diagnosis.
Hybrid Desmoplastic/Follicular Ameloblastoma of the Mandible: A Case Report and Review of the Literature
Authors: Masayasu Iwase, Airi Fukuoka, Yoko Tanaka, Naoyuki Saida, Eriko Onaka, Sanae Bando, Gen Kondo
Journal: Case Rep Pathol (2017)
MeSH terms: Case Reports

Desmoplastic ameloblastoma (DA) is one of the 6 histopathological subtypes of ameloblastoma. Hybrid lesions in which histopathologically conventional ameloblastoma coexists with areas of DA are rare. A 40-year-old male was referred to our hospital complaining of a swelling in the right premolar region of the mandible. A panoramic radiograph showed an area of radiolucency with a well-defined corticated border, whereas computed tomography revealed a unilocular radiolucent lesion and buccal expansion together with cortical perforation. The lesion was treated via enucleation and curettage of the marginal bone and fenestration. A histopathological examination showed a hybrid ameloblastoma with a pronounced desmoplastic pattern and follicular changes. The patient's postoperative course has been favorable up to now, and no marked changes have been observed. We presented a case of hybrid ameloblastoma and reviewed the 36 reported cases of hybrid ameloblastoma that have been reported in the English literature.
Inflammatory myofibroblastic tumor of the transverse colon with synchronous gastrointestinal stromal tumor in a patient with ulcerative colitis: a case report
Authors: Eugenia Raffaeli, Luca Cardinali, Maurizio Fianchini, Donatella Brancorsini, Piergiorgio Mosca, Cristina Marmorale
Journal: Int J Surg Case Rep (2019)
Keywords: gist, gastrointestinal stromal tumor, imt, inflammatory myofibroblastic tumor, inflammatory pseudotumor, ulcerative colitis

Introduction:
Inflammatory myofibroblastic tumor (IMT) is a rare proliferative disease of uncertain etiology, characterized by the proliferation of fusate or epithelioid myofibroblasts admixed with predominantly mononuclear inflammatory cells. IMT is generally considered a benign lesion, although in some cases this neoplasm has shown an aggressive behavior in terms of local recurrence and metastasis. We report the case of a patient with a ten-year history of ulcerative colitis affected by IMT of the transverse colon and by synchronous gastrointestinal stromal tumor (GIST) of stomach.
Presentation of case:
A 59-year-old woman with a ten-year history of ulcerative colitis has been admitted to our hospital with signs and symptoms of acute recurrence of ulcerative colitis: abdominal pain, diarrhea, hematochezia and rectal tenesmus. Colonoscopy showed a left colon with diffuse hyperemia, mucosal erosions and a 2-cm, irregularly shaped, polypoid lesion at the level of the transverse colon. Histopathological examination of the specimen obtained via biopsy of the polypoid lesion has revealed a mesenchymal neoplasm with uncertain characters of malignancy. Due to the severity of the inflammatory bowel disease resistant to immunosuppressive and steroid drug treatment, surgical indication was given.
Discussion:
Although the relationship between IMT and Crohn's disease has been widely reported in literature, the relationship between IMT and ulcerative colitis has never been previously described.
Conclusion:
To the best of our knowledge, this is the first case of IMT associated with ulcerative colitis reported in literature and the synchronous association with a gastric GIST represents another primacy.
Interlaminar stabilization and decompression for the treatment of bilateral juxtafacet cysts: Case report and literature review
Authors: Iahn Cajigas, Alberto Varon, Howard B Levene
Journal: Int J Surg Case Rep (2019)
Keywords: coflex, interlaminar stabilization device, laminectomy, prostheses, spinal stenosis, synovial cysts

Introduction:
Lumbar juxtafacet cysts (JFCs) are a common cause of lumbar radiculopathy which tend to occur in areas of increased facet mobility. While resection alone is a possible treatment, recent publications suggest that laminectomy alone for JFCs may not yield as favorable an outcome as laminotomies reinforced with posterior dynamic hardware. The Coflex® is a novel interlaminar stabilization device that has been shown to achieve comparable results to rigid fusion in the management of lumbar stenosis in patients with no more than grade one anterolisthesis, and superior performance compared to laminectomy alone when a combined outcome score was used. We describe the combined use of dynamic posterior element fusion with primary cyst resection in the management of bilateral JFCs.
Presentation of case:
A 71-year-old man who developed a progressive left L4 radiculopathy along with new urinary incontinence was found to have bilateral L3/4 JFCs causing significant lumbar stenosis and neurogenic claudication. After treatment with primary cyst resection and interlaminar stabilization, the patient experienced complete symptom resolution and was discharged to inpatient-rehabilitation on post-operative day 1.
Discussion:
While current recommendations for the management of juxtafacet cysts causing progressive neurologic symptoms include surgical cyst removal and lumbar decompression with or without fusion, the role of dynamic interlaminar stabilization has not been explored.
Conclusion:
Direct decompression followed by interlaminar stabilization may represent an alternative for patients to simultaneously benefit from a decompression of their juxtafacet cysts while affording posterior element reconstruction.
Bi-Allelic Mutations in STXBP2 Reveal a Complementary Role for STXBP1 in Cytotoxic Lymphocyte Killing
Authors: Jamie A Lopez, Tahereh Noori, Adrian Minson, Lu Li Jovanoska, Kevin Thia, Michael S Hildebrand, Hedieh Akhlaghi, Phillip K Darcy, Michael H Kershaw, Natasha J Brown, Andrew Grigg, Joseph A Trapani, Ilia Voskoboinik
Journal: Front Immunol (2018)
Keywords: munc18-1, munc18-2, apoptosis, cytotoxic t cells, cytotoxic lymphocytes, familial haemophagocytic lymphohistiocytosis, immunodeficiency, natural killer cells
MeSH terms: Munc18 Proteins / genetics, Munc18 Proteins / immunology, T-Lymphocytes, Cytotoxic / immunology, Case Reports, Research Support, Non-U.S. Gov't, Alleles, Cell Line, Cytotoxicity, Immunologic, Female, Humans, Killer Cells, Natural / immunology, Leukocytes, Mononuclear / immunology, Middle Aged, Mutation, Munc18 Proteins, STXBP1 protein, human, STXBP2 protein, human

The ability of cytotoxic lymphocytes (CL) to eliminate virus-infected or cancerous target cells through the granule exocytosis death pathway is critical to immune homeostasis. Congenital loss of CL function due to bi-allelic mutations in PRF1, UNC13D, STX11, or STXBP2 leads to a potentially fatal immune dysregulation, familial haemophagocytic lymphohistiocytosis (FHL). This occurs due to the failure of CLs to release functional pore-forming protein perforin and, therefore, inability to kill the target cell. Bi-allelic mutations in partner proteins STXBP2 or STX11 impair CL cytotoxicity due to failed docking/fusion of cytotoxic secretory granules with the plasma membrane. One unique feature of STXBP2- and STX11-deficient patient CLs is that their short-term in vitro treatment with a low concentration of IL-2 partially or completely restores natural killer (NK) cell degranulation and cytotoxicity, suggesting the existence of a secondary, yet unknown, pathway for secretory granule exocytosis. In the current report, we studied NK and T-cell function in an individual with late presentation of FHL due to hypomorphic bi-allelic mutations in STXBP2. Intriguingly, in addition to the expected alterations in the STXBP2 and STX11 proteins, we also observed a concomitant significant reduction in the expression of homologous STXBP1 protein and its partner STX1, which had never been implicated in CL function. Further analysis of human NK and T cells demonstrated a functional role for the STXBP1/STX1 axis in NK and CD8+ T-cell cytotoxicity, where it appears to be responsible for as much as 50% of their cytotoxic activity. This discovery suggests a unique and previously unappreciated interplay between STXBP/Munc proteins regulating the same essential granule exocytosis pathway.
The Successful Treatment of a Case of Linear Psoriasis with Ixekizumab
Authors: Sara Ghoneim, Alvaro J Ramos-Rodriguez, Fernando Vazquez de Lara, Lauren Bonomo
Journal: Case Rep Dermatol Med (2017)
MeSH terms: Case Reports

Linear psoriasis is an unusual clinical variation of psoriasis that manifests segmentally along the lines of Blaschko. A major differential diagnosis is inflammatory linear verrucous epidermal nevus (ILVEN). The treatment of linear psoriasis is often challenging, with inadequate response to biological agents reported in the literature. We report a case of a 25-year-old African-American female who presented with asymptomatic hyperkeratotic papules along the lines of Blaschko and was subsequently diagnosed with linear psoriasis. After failing conventional treatment regimens, the patient received a trial of ixekizumab with complete resolution of cutaneous lesions reported after 4 months and only 8 doses of the anti-IL-17 biologic agent.
Q fever endocarditis in HIV-infected patient
Authors: Miguel G Madariaga, Joseph Pulvirenti, Marin Sekosan, Christopher D Paddock, Sherif R Zaki
Journal: Emerg Infect Dis (2004)
MeSH terms: Endocarditis / complications, HIV Infections / complications, Q Fever / complications, Case Reports, Research Support, Non-U.S. Gov't, Review, Anti-Infective Agents / therapeutic use, Humans, Male, Middle Aged, Q Fever / drug therapy, Anti-Infective Agents

We describe a case of Q fever endocarditis in an HIV-infected patient. The case was treated successfully with valvular replacement and a combination of doxycycline and hydroxychloroquine. We review the current literature on Q fever endocarditis, with an emphasis on the co-infection of HIV and Coxiella burnetii.
Therapeutic hypothermia in cerebral air embolism: a case report
Authors: Jochen Bäuerle, Andreas Fischer, Tobias Hornig, Karl Egger, Tobias Wengenmayer, Jürgen Bardutzky
Journal: Springerplus (2013)
Keywords: ct, cerebral air embolism, mri, therapeutic hypothermia

Background:
Cerebral air embolism is a life-threatening complication of various diagnostic and therapeutic procedures. Hyperbaric oxygenation is considered to be the cornerstone of its treatment.
Case description:
We report a patient with cerebral air embolism after endoscopy of a perineal abscess. Immediate CT imaging confirmed the diagnosis and MRI showed cortically localized areas of restricted diffusion along the gyri. Since hyperbaric oxygenation was not available, moderate hypothermia was applied for neuroprotection.
Conclusion:
This case illustrates a rare complication of endoscopic interventions, and imaging characteristics of cerebral air embolism were described. Furthermore, we discuss the potential utility of therapeutic hypothermia in cerebral air embolism.
Necrosis of the 4th and 5th digits after intra-articular injection of diazepam into the wrist
Authors: Niklas Iblher, Gerd Bjoern Stark, Vincenzo Penna
Journal: Case Rep Surg (2011)
MeSH terms: Case Reports

A 75-year-old woman presented with progressing pain, cyanosis, and hypaesthesia in her left hand after an intra-articular injection with diazepam into the wrist for osteoarthritis-related pain. Due to an iatrogenic intra-arterial injection, malperfusion of the ulnar digits developed. Angiography revealed blockage of perfusion of the 4th and 5th digits. Despite intra-arterial lysis, heparinisation, and vasodilatation, perfusion could not be reinstalled. Necrosis of the distal phalanges of the 4th and 5th digits developed, which had to be treated with amputation. The pathomechanism of tissue damage and the treatment options after intra-arterial injections are reviewed and discussed.
Acute abdomen in a patient with cancer pain on oxycodone
Authors: Naomi Kishine, Atsunobu Tsunoda, Seiji Kishimoto, Tomohisa Shoko
Journal: Case Rep Med (2011)
MeSH terms: Case Reports

Opioids are a mainstay of treatment for moderate to severe cancer pain. At present, oxycodone has fewer adverse effects compared to morphine and is widely used for cancer pain therapy. The adverse effects of oxycodone are similar to morphine and include constipation, nausea, and sedation. However, acute abdominal pain is rarely seen. Here, we describe a cancer patient presenting with acute abdomen with stercoral diarrhea. A 54-year-old man with squamous cell carcinoma of the external auditory canal had been taking oxycodone for pain relief. The patient had taken oxycodone for several months and had never complained of either diarrhea or constipation. After an increase in the dosage of oxycodone, he complained of abdominal distension and constipation. After being administered a laxative, he complained of diarrhea and severe abdominal pain. He visited the emergency department and was diagnosed with acute colonic obstruction caused by severe constipation. He self-medicated with oxycodone at dosages of up to 180 mg/day, and this abrupt increase of oxycodone caused stercoral diarrhea. Finally, total blockage of stool developed, resulting in acute abdomen.