OpenMed/multicare-articles huggingface dataset:
Hydrogen Gas Inhalation Regressed Coronary Artery Aneurysm in Kawasaki Disease-Case Report and Article Review
Authors: Ho-Chang Kuo
Journal: Front Cardiovasc Med (2022)
Keywords: kawasaki disease, aneurysm, hydrogen gas, inhalation, regression
MeSH terms: Case Reports

Kawasaki disease (KD) is a systemic vasculitis that primarily affects children under the age of 5 years old and is among the most common acquired heart disease in developed countries, particularly in Asia. No effective treatment is currently available for aneurysm formation in KD. In this report, we showed a KD patient with an aneurysm over the right coronary artery with a size of 6.08 mm in diameter and 35 mm in length, which completely regressed to within normal range after hydrogen inhalation within 4 months after disease onset. This 10-year-old KD patient was diagnosed on the 12th day of disease onset with incomplete presentation of KD symptoms. Intravenous immunoglobulin was prescribed after KD diagnosis was confirmed by the formation of a coronary artery aneurysm. Once discharged from the hospital, the family used hydrogen inhalation (77% hydrogen and 23% oxygen) at home with nasal cannula 1 h per day. The aneurysm was found to be completely regressed at the 4-month follow-up (day 138 of the illness). The follow-up laboratory data showed complete blood cell count, differential count, electrolytes, liver enzyme, and renal function to all be within normal range. This is the first study to report an aneurysm from KD with regression under supplementary therapy with hydrogen gas inhalation and no other complications. Therefore, hydrogen gas inhalation may be an alternative anti-free radical or anti-oxidant therapy for KD, but further study is still required.
Gigantic submandibular pleomorphic adenoma: A rare case report
Authors: Mohamed Abdelkhalek, Mohamed Elmetwally, Alaa Mazy, Mona Gad, Ahmed Elsaid, Shadi Awny, Basel Refky, Ahmed Abdallah, Amr F Elalfy, Farida A Shokeir, Samar Abdallah, Abeer Elfeky, Rowaa Aboelamayem, Mohamed A Hegazy
Journal: Int J Surg Case Rep (2019)
Keywords: giant, pleomorphic adenoma, submandibular gland

Introduction:
Pleomorphic adenoma (PA) is the most common benign salivary gland tumor. When neglected on some occasions, its size and weight can enormously augment.
Case presentation:
We report a case of a giant submandibular pleomorphic adenoma in a 75 years old female patient which measured 34 × 26 × 20 cm and weighed 8.1 kg.
Conclusion:
Neglecting such tumors without treatment, can cause severe facial disfigurement and can even hinder the airway.
Acquired perforating collagenosis associated with ranibizumab injection and succesfully switched to aflibercept
Authors: Ayse Yagmur Kanra, Sevil Ari Yaylali, Ayse Serap Karadag, Aylin Ardagil Akçakaya, Itir Ebru Zemheri
Journal: GMS Ophthalmol Cases (2018)
MeSH terms: Case Reports

Objective: To report a case of acquired reactive perforating collagenosis (ARPC) triggered by an intravitreal ranibizumab injection that was successfully treated by switching to aflibercept (AFL). Methods: A 73-year-old Caucasian man with an occult choroidal neovascular membrane in the right eye received three-monthly intravitreal ranibizumab injections. Two weeks after the second ranibizumab injection, he complained about a generalized, excessively pruriginous eruption that was further exacerbated by the third injection. On the basis of clinical and histological findings, he was diagnosed with ARPC and treated with narrow band ultraviolet-B (NBUVB) phototherapy. Results: He was subsequently switched to intravitreal AFL injections administered according to a pro re nata regimen. Following NBUVB phototherapy, three additional AFL injections were required. Still, the reactive perforating collagenosis was in remission and the choroidal neovascular membrane was inactive. Conclusions: Our case is the first report of ARPC after ranibizumab injections. Both the skin lesions and the choroidal neovascular membrane were successfully treated after switching to AFL.
Utility of Intracardiac Echocardiography to Diagnose Infective Endocarditis
Authors: Ying Chi Yang, Thein Tun Aung, Sarah Khan, Abdul Wase
Journal: J Investig Med High Impact Case Rep (2019)
Keywords: infective endocarditis, intracardiac echography, prosthetic valve
MeSH terms: Cardiac Imaging Techniques, Echocardiography, Endocarditis / diagnostic imaging, Heart Valve Prosthesis / microbiology, Case Reports, Aged, Endocarditis / etiology, Humans, Male

Infective endocarditis (IE) can lead to significant morbidity and mortality without appropriate treatment. Modified Duke Criteria are accepted by many professional societies to establish the diagnosis of IE, and cardiac imaging is one of the major diagnostic criteria. Transesophageal echocardiography is an algorithmic escalation to diagnose IE when transthoracic echo does not appreciate a positive finding. In patients with contraindications to transesophageal echocardiography, cardiac magnetic resonance imaging, cardiac computed tomography (CT), cardiac CT angiography, and fluorodeoxyglucose positron emission tomography with CT or CT angiography may be alternative diagnostic tools. However, these imaging modalities have their own limitations such as local unavailability, the presence of non-magnetic resonance imaging compatible implants, or impaired renal function. Intracardiac echocardiography could be a considerable alternative under those circumstances.
The Application of MR Spectroscopy and MR Perfusion in Cerebral Syphilitic Gumma: A Case Report
Authors: Linyang Cui, Jie Liu, Wenjun Zhang, Zushan Xu, Hongjun Hou
Journal: Front Neurosci (2020)
Keywords: mr perfusion, mr spectroscopy cerebral syphilitic gumma, brain tumor, cerebral syphilitic gumma, magnetic resonance imaging

Cerebral syphilitic gumma is a rare disease and can be misdiagnosed as a common brain tumor when only conventional imaging results are adopted. Differentiating between syphilitic gumma and tumors may be achieved by applying advanced magnetic resonance (MR) techniques, such as MR spectroscopy and MR perfusion. However, the MR perfusion characteristics of cerebral syphilitic gumma have not been reported yet. Here, we report a case of cerebral syphilitic gumma in a 52-year-old woman and discuss the imaging features of conventional MR, MR spectroscopy, and MR perfusion. The results suggest that the application of MR spectroscopy and MR perfusion could provide additional information that contributes to the diagnosis of cerebral syphilitic gumma.
Pyriform sinus perforation during nasotracheal intubation: a case report
Authors: Mohammed Elrabie Ahmed, Bahaa Mohammed Refaie, Farghali Abdelrahman, Abdul-Rahman Abdul-Majeed Ragab
Journal: Int J Surg Case Rep (2020)
Keywords: complication, intubation, perforation, pyriform
MeSH terms: Case Reports

Introduction:
Endotracheal intubation is the gold standard to protect the airway during general anesthesia. It is the commonest technique in medical practice with a low complication rate.
Presentation of case:
We report a case of left pyriform fossa perforation in a child after apparently uneventful nasotracheal intubation for tonsillectomy.
Discussion:
This is a rare complication. Physicians performing endotracheal intubation or dealing with intubated patients should be aware of the clinical symptoms of such complications. A rational management approach has been discussed.
Conclusions:
Early diagnosis is the mainstay to improve the outcome and avoid the morbidity and potentially life-threatening course of such trauma.
Three Chinese pedigrees of A20 haploinsufficiency: clinical, cytokine and molecular characterization
Authors: Yi Tian, Bingxuan Wu, Linyi Peng, Jian Wang, Min Shen
Journal: Front Immunol (2022)
Keywords: tnfaip3, autoantibody, familial behçet’s syndrome, haploinsufficiency of a20, whole-exon gene sequencing
MeSH terms: Cytokines / genetics, Haploinsufficiency, Case Reports, Systematic Review, Research Support, Non-U.S. Gov't, Asian People / genetics, Female, Humans, Pedigree, Phenotype, Cytokines

Objective:
Haploinsufficiency of A20 (HA20) is a newly described rare autoinflammatory disease caused by TNFAIP3 gene mutations. HA20 has seldom been documented in the Chinese population. Herein, we report eight patients with HA20 from three unrelated families in China.
Methods:
Eight Chinese Han patients were diagnosed with HA20 in our department from 2018 to 2021. Their clinical data and genotypes were carefully documented and studied. The newly identified variants were functionally verified. We also conducted a systematic literature review of HA20, and the clinical characteristics and genotype of HA20 between the Chinese population and other populations were compared.
Results:
Eight HA20 patients from three families comprised six adults and two children. There was one man and seven women. The clinical characteristics included recurrent oral ulcers (8/8, 100%), fever (4/8, 50%), perianal ulcer (3/8, 38%), skin lesions (2/8, 25%), arthritis (1/8, 13%), and uveitis (1/8, 13%). Three TNFAIP3 variants, A547T, c.1906+2T>G, and R271X, were identified. Two novel variants, A547T and c.1906+2T>G, were validated to be pathogenic in our study. In a literature review a total of 126 patients with HA20 reported by 35 articles were included. The clinical phenotype of Chinese HA20 patients was similar to that of patients from other populations except for a lower frequency of genital ulcers (16.7% vs. 54.4%, p < 0.01). Autoantibodies were detectable in approximately one-third of the 126 patients, among which ANA and anti-thyroid antibodies were commonly seen.
Conclusion:
The rarity and diversity of phenotypes make the diagnosis of HA20 a huge challenge to physicians. HA20 should be considered in child-onset patients with manifestations that resemble Behçet's syndrome, especially those whose family members have similar symptoms. Gene testing is critically helpful for the diagnosis of HA20. Two novel TNFAIP3 variants, A547T and c.1906+2T>G, were identified in this study.
Steroid cell ovarian neoplasm, not otherwise specified: a case report and review of the literature
Authors: Paul Singh, Frank Deleon, Ralph Anderson
Journal: Case Rep Obstet Gynecol (2012)

Background. Steroid cell ovarian tumors, not otherwise specified, represent a unique cause of female virilization. Most commonly encountered in premenopausal women, these tumors can exist throughout a women's lifetime, from before puberty until after menopause. Case. Steroid cell, not otherwise specified, was diagnosed in a 70-year-old female significant for hirsutism. The patient demonstrated elevated total testosterone levels with normal gonadotropins, DHEA, and DHEA-S levels. CT imaging revealed a right ovarian mass and subsequent laparoscopic right oophorectomy yielded clinical improvement promptly. Conclusion. Virilization in females can occur based on ovarian or adrenal pathology. In terms of ovarian-based female virilization, many tumors exist that may induce women to demonstrate masculine features, such as pure Sertoli, pure Leydig, Sertoli-Leydig combinations, and gynandroblastomas. Each of these tumor types possesses a unique histologic pattern that allows for pathologic identification after removal. A rare source of ovarian-based female virilization is steroid cell neoplasms, not otherwise specified, that do not demonstrate these specific histologic characteristics and thus represent a diagnosis of exclusion after other causes of ovarian-based female virilization have been ruled out.
Mitochondrial Disorder Aggravated by Metoprolol
Authors: Cheryl Samuels, Mary Kay Koenig, Mariana Hernandez, Aravind Yadav, Ricardo A Mosquera
Journal: Case Rep Pediatr (2016)

Beta-adrenergic blocking agents or beta-blockers are a class of medications used to treat cardiac arrhythmias and systemic hypertension. In therapeutic dosages, they have known adverse outcomes that can include muscular fatigue and cramping, dizziness, and dyspnea. In patients with mitochondrial disease, these effects can be amplified. Previous case reports have been published in the adult population; however, their impact in pediatric patients has not been reported. We describe a pediatric patient with a mitochondrial disorder who developed respiratory distress after metoprolol was prescribed for hypertension. As the patient improved with discontinuation of medication and no alternative etiology was found for symptoms, we surmise that administration of metoprolol aggravated his mitochondrial dysfunction, thus worsening underlying chest wall weakness.
A Great Masquerader: Nasal Type, Extranodal Natural Killer/T-cell Lymphoma Presenting as Recalcitrant Bacterial Sinusitis and Periorbital Cellulitis
Authors: Zaw Min, Nitin Bhanot
Journal: Case Rep Med (2020)
MeSH terms: Case Reports

Nasal extranodal natural killer/T-cell lymphoma (ENKL) is a rare clinical entity. It may, however, masquerade as a commonly encountered disease, such as sinusitis. A high index of clinical suspicion of nasal ENKL should be raised when there is inadequate clinical response despite appropriate therapeutic intervention of sinusitis. Biopsy would be warranted and crucial in those instances to make an accurate and timely diagnosis.
Plasmablastic Lymphoma, a Rare Entity in Bone Marrow with Unusual Immunophenotype and Challenging Differential Diagnosis
Authors: Majd Al Shaarani, Rodney E Shackelford, Samip R Master, Glenn M Mills, Yasir AlZubaidi, Ahmed Mamilly, Eric X Wei
Journal: Case Rep Hematol (2019)
MeSH terms: Case Reports

Plasmablastic lymphoma (PBL) is an aggressive malignancy that usually occurs in the setting of immunosuppression. The immunohistochemical profile of PBL is that of terminally differentiated B lymphocytes. CD138, CD38, and MUM1 are usually immunopositive. However, pan B-cell markers such as CD20 and PAX-5 are usually negative. MYC rearrangement is the most commonly encountered genetic alteration, with immunoglobulin (IG), especially immunoglobulin heavy (IGH) chain, being the most frequent partner. We report a case of PBL in a 48-year-old human immunodeficiency virus- (HIV-) positive male who was admitted to the hospital with signs and symptoms suspicious for tumor lysis syndrome. Bone marrow examination revealed hypercellular marrow with trilineage hypoplasia and sheets of intermediate to large neoplastic cells with basophilic vacuolated cytoplasm comprising the majority of cellular elements of the bone marrow. The neoplastic cells were negative for conventional B-cell, T-cell, plasma cell, and myeloid markers, while flow cytometric analysis revealed an abnormal CD45-dim population that was partially weakly positive for CD71 and CD79b. The diagnosis was initially thought to be a high-grade primitive hematopoietic neoplasm, possibly an acute undifferentiated leukemia. BOB-1, however, was immunopositive in the neoplastic cells, confirming its B-cell origin. MYC was positive by immunohistochemistry and break-apart FISH, as were CD45, MUM-1, and EMA immunostains. There was immunoglobulin kappa (IGK) light chain gene rearrangement by polymerase chain reaction (PCR). Additionally, Epstein-Barr virus- (EBV-) encoded small RNAs (EBER) were positive by in situ hybridization (ISH). The tumor proliferation index by Ki-67 immunostaining approached 95%. Although the tumor cells were negative for CD38 and CD138, the diagnosis of PBL was still rendered. We recommend using a broad spectrum of B-cell markers, including BOB-1 and OCT-2, in such challenging cases of B-cell lymphomas with no expression of conventional B-cell markers. We also emphasize that the negative CD38 and CD138 should not exclude PBL from the differential diagnosis.
Spontaneous Recovery from Unresponsive Wakefulness Syndrome to a Minimally Conscious State: Early Structural Changes Revealed by 7-T Magnetic Resonance Imaging
Authors: Xufei Tan, Jian Gao, Zhen Zhou, Ruili Wei, Ting Gong, Yuqin Wu, Kehong Liu, Fangping He, Junyang Wang, Jingqi Li, Xiaotong Zhang, Gang Pan, Benyan Luo
Journal: Front Neurol (2018)
Keywords: cuneus, disorders of consciousness, recovery, structural changes, temporoparietal junction, ultra-high field (7 t)
MeSH terms: Case Reports

Background:
Determining the early changes of brain structure that occur from vegetative state/unresponsive wakefulness syndrome (VS/UWS) to a minimally conscious state (MCS) is important for developing our understanding of the processes underlying disorders of consciousness (DOC), particularly during spontaneous recovery from severe brain damage.
Objective:
This study used a multi-modal neuroimaging approach to investigate early structural changes during spontaneous recovery from VS/UWS to MCS.
Methods:
The Coma Recovery Scale-Revised (CRS-R) score, 24-h electroencephalography (EEG), and ultra-high field 7-T magnetic resonance imaging were used to investigate a male patient with severe brain injury when he was in VS/UWS compared to MCS. Using white matter connectometry analysis, fibers in MCS were compared with the same fibers in VS/UWS. Whole-brain analysis was used to compare all fibers showing a 10% increase in density with each other as a population.
Results:
Based on connectometry analysis, the number of fibers with increased density, and the magnitude of increase in MCS compared to VS/UWS, was greatest in the area of the temporoparietal junction (TPJ), and was mostly located in the right hemisphere. These results are in accordance with the active areas observed on 24-h EEG recordings. Moreover, analysis of different fibers across the brain, showing at least a 10% increase in density, revealed that altered white matter connections with higher discriminative weights were located within or across visual-related areas, including the cuneus_R, calcarine_R, occipital_sup_R, and occipital_mid_R. Furthermore, the temporal_mid_R, which is related to the auditory cortex, showed the highest increase in connectivity to other areas. This was consistent with improvements in the visual and auditory components of the CRS-R, which were greater than other improvements.
Conclusion:
These results provide evidence to support the important roles for the TPJ and the visual and auditory sensory systems in the early recovery of a patient with severe brain injury. Our findings may facilitate a much deeper understanding of the mechanisms underlying conscious-related processes and enlighten treatment strategies for patients with DOC.
Synchronous bilateral breast cancer in a male
Authors: María Caridad Rubio Hernández, Yenia Ivet Díaz Prado, Suanly Rodríguez Pérez, Ronald Rodríguez Díaz, Zaili Gutiérrez Aleaga
Journal: Ecancermedicalscience (2013)
Keywords: bilateral, male breast cancer, multifocal, synchronous
MeSH terms: Case Reports

Male breast cancer, which represents only 1% of all breast cancers, is occasionally associated with a family history of breast cancer. Sporadic male breast cancers presenting with another primary breast cancer are extremely rare. In this article, we report on a 70-year-old male patient with bilateral multifocal and synchronous breast cancer and without a family history of breast cancer.
Percutaneous holmium laser fulguration of calyceal diverticula
Authors: Amjad Alwaal, Raed A Azhar, Sero Andonian
Journal: Case Rep Urol (2012)
MeSH terms: Case Reports

Introduction. Calyceal diverticular stones are uncommon findings that represent a challenge in their treatment, due to the technical difficulty in accessing the diverticulum, and the high risk of their recurrence. Current percutaneous technique for calyceal diverticular stones involves establishing a renal access, clearing the stone, and fulguration of the diverticular lining with a roller-ball cautery electrode using hypotonic irrigation solution such as sterile water or glycine solution which may be associated with the absorption of hypotonic fluids with its inherent electrolyte disturbances. Case Report. In this paper, we present for the first time percutaneous holmium laser fulguration of calyceal diverticula in 2 patients using normal saline. Their immediate postoperative sodium was unchanged and their follow-up imaging showed absence of stones. Both patients remain asymptomatic at 30 months post-operatively. Conclusion. This demonstrates that holmium laser is a safe alternative method to fulgurate the calyceal diverticulum after clearing the stone percutaneously.
Brentuximab vedotin related bilateral Purtscher-like retinopathy unresponsive to pulse steroid therapy and intravitreal aflibercept injection
Authors: Ziya Ayhan, Sureyya Yigit Kaya, Mehmet Ali Ozcan, Ali Osman Saatci
Journal: GMS Ophthalmol Cases (2017)
Keywords: brentixumab vedotin, hodgkin’s lymphoma, purtscher-like retinopathy
MeSH terms: Case Reports

We describe a 36-year-old woman with a relapsing Hodgkin's lymphoma who developed a severe bilateral sudden visual loss almost three weeks after the initiation of brentuximab therapy. Ancillary fundus tests yielded bilateral severe retinal arteriolar occlusion 360° and serous macular retinal detachment. No visual improvement could be achieved despite the pulse corticosteroid therapy and a single bilateral intravitreal aflibercept administration cessation of the brentuximab therapy. Unfortunately, she succumbed to respiratory failure almost six weeks after the diagnosis of Purtscher-like retinopathy.
Hypoperfusion cerebral infarction after carotid artery stenting: A case report
Authors: Yuerong Ma, Renwei Zhang, Yumin Liu
Journal: Front Surg (2023)
Keywords: acute cerebral infarction, carotid artery stent implantation, cerebral artery stenosis, hyperperfusion syndrome, hypoperfusion syndrome
MeSH terms: Case Reports

Carotid artery stent implantation (CAS) plays an important role in preventing cerebral infarction associated with carotid stenosis. The postoperative complications of CAS include cerebral hyperperfusion syndrome (CHS), cerebral infarction, vascular injury, carotid sinus reaction, and stent restenosis. Hyperperfusion syndrome (CHS) is a serious complication that arises after the performance of carotid endarterectomy (CEA) or CAS and is characterized by high blood pressure, headache, epilepsy, and focal neurological deficit. Therefore, it is very important to evaluate and diagnose CHS. Cerebral infarction after CAS is often caused by distal embolism due to the shedding of microemboli. With the application of distal brain protection devices, the risk of distal embolism is significantly reduced. In this study, we report a rare case of hypoperfusion cerebral infarction after carotid artery stenting in a patient with severe carotid stenosis complicated with contralateral common carotid artery occlusion.
Case report: Joint diagnosis and treatment of intrathoracic gastric duplication with a gastric communication in a child by laparoscopy and gastroscopy
Authors: Huashan Zhao, Yunpeng Zhai, Rui Guo, Hongxiu Xu, Longfei Lv, Shisong Zhang
Journal: Front Pediatr (2023)
Keywords: child, diagnosis, gastroscopy, intrathoracic gastric duplication, laparoscopy, treatment
MeSH terms: Case Reports

Intrathoracic gastric duplication has rarely been reported. A 5-year-old child with gastric duplication located in the left thorax was diagnosed and treated successfully using laparoscopy combined with gastroscopy. Preoperative computed tomography, upper gastrointestinal contrast study, ultrasound, and other imaging methods were insufficient for accurate diagnosis in this case. Laparoscopy combined with gastroscopy is more suitable for the diagnosis and treatment of gastric duplication.
Bisoprolol Transdermal Patch Is Effective for the Treatment of AF Tachycardia
Authors: Yoh Arita, Hajime Saeki, Miwa Miyoshi, Shinji Hasegawa
Journal: Case Rep Cardiol (2018)
MeSH terms: Case Reports

Atrial fibrillation (AF) is an irregular and often rapid heart rate that can increase the risk of stroke, heart failure, and other heart-related complications. Its incidence increases with age and the presence of concomitant heart disease. We present the cases of a 93-year-old woman, an 82-year-old man, and an 87-year-old woman who developed AF tachycardia. This report highlights the use of a bisoprolol transdermal patch to treat AF tachycardia in 3 adult elderly patients. In this paper, we report an initial treatment strategy using a bisoprolol transdermal patch and show heart rate trends for 24 hours.