OpenMed/multicare-articles huggingface dataset:
Ventricular Fibrillation following Varicella Zoster Myocarditis
Authors: Adam Ioannou, Irene Tsappa, Sofia Metaxa, Constantinos G Missouris
Journal: Case Rep Cardiol (2017)
MeSH terms: Case Reports

Varicella-zoster virus (VZV) infection can rarely lead to serious cardiac complications and life-threatening arrhythmias. We present a case of a 46-year-old male patient who developed VZV myocarditis and presented with recurrent syncopal episodes followed by a cardiac arrest. He had a further collapse eight years later, and cardiac magnetic resonance imaging (MRI) demonstrated mild mid-wall basal and inferolateral wall fibrosis. He was treated with an implantable cardioverter defibrillator (ICD) and represented two years later with ICD shocks, and interrogation of the device revealed ventricular fibrillation episodes. This case demonstrates the life-threatening long-term sequelae of VZV myocarditis in adults. We suggest that VZV myocarditis should be considered in all patients who present with a syncopal event after VZV infection. In these patients, ICD implantation is a potentially life-saving procedure.
Sengstaken Tube Removal under Direct Hemodynamic Monitoring after Post Transplantation Venous Occlusion
Authors: A L Colón, L Rodríguez-Bachiller, E Velasco, B Díaz-Zorita, D Rincón, J A López-Baena, J Ferreiroa
Journal: Case Rep Surg (2019)
MeSH terms: Case Reports

The surgical procedure for orthotopic liver transplantation (OLT) is well standardized, and most groups use the retrohepatic caval preservation or piggyback technique to improve hemodynamic tolerance. However, when a discrepancy between the site in the right upper quadrant of the liver recipient and a small graft is present, this technique can provoke a rotation on the axis of the vena cava and cause an occlusion of the suprahepatic vein drainage. This problem can be detected intraoperatively, and several methods have been described to resolve it by placing different devices to correct the position. Early withdrawal may cause the development of clinical hepatic congestion with ascites unresponsive to medical treatment. We present three cases of OLT who developed obstruction of the venous drainage solved intraoperatively with the placement of a Sengstaken-Blakemore tube. As a novelty, prior to the withdrawal of the device, a transjugular hemodynamic study was performed to ensure the correct position of the liver with adequate venous drainage.
Differences in Healing of a Horizontal Root Fracture as Seen on Conventional Periapical Radiography and Cone-Beam Computed Tomography
Authors: Ronnachat Rothom, Patchanee Chuveera
Journal: Case Rep Dent (2017)
MeSH terms: Case Reports

Different locations and healing patterns of horizontal root fractures bear different prognoses. Conventional periapical radiographs have been routinely used for the evaluation of the locations and healing of horizontal root fractures, with the limitation of presenting two-dimensional images. The three-dimensional imaging technology, cone-beam computed tomography (CBCT), has recently gained interest in dental traumatology, in particular for locating and diagnosing root fractures. However, the assessment of healing patterns of horizontal root fracture using CBCT compared to conventional radiographs has not been established. This case report describes the different healing patterns evaluated by two-dimensional radiographs and CBCT of a horizontally root-fractured maxillary right central incisor treated with mineral trioxide aggregate (MTA) with a two-year follow-up. The findings suggest that the healing patterns of horizontal root fractures seen on conventional radiographs and CBCT may be different.
Paroxysmal Nocturnal Hemoglobinuria in the Context of a Myeloproliferative Neoplasm: A Case Report and Review of the Literature
Authors: Juri Alessandro Giannotta, Bruno Fattizzo, Wilma Barcellini
Journal: Front Oncol (2021)
Keywords: bone marrow failure, case report, complement inhibitors, myeloproliferative neoplasm, paroxysmal nocturnal hemoglobinuria, thrombosis
MeSH terms: Case Reports

Paroxysmal nocturnal hemoglobinuria (PNH) is characterized by intravascular hemolytic anemia and thrombosis and is notoriously associated with aplastic anemia and myelodysplastic syndromes. Rarer associations include myeloproliferative neoplasms (MPNs), which are also burdened by increased thrombotic tendency. The therapeutic management of this rare combination has not been defined so far. Here, we describe a 62-year-old man who developed a highly hemolytic PNH more than 10 years after the diagnosis of MPN. The patient started eculizumab, obtaining good control of intravascular hemolysis but without amelioration of transfusion-dependent anemia. Moreover, we performed a review of the literature regarding the clinical and pathogenetic significance of the association of PNH and MPN. The prevalence of PNH clones in MPN patients is about 10%, mostly in association with JAK2V617F-positive myelofibrosis. Thrombotic events were a common clinical presentation (35% of subjects), sometimes refractory to combined treatment with cytoreductive agents, anticoagulants, and complement inhibitors. The latter showed only partial effectiveness in controlling hemolytic anemia and, due to the paucity of data, should be taken in consideration after a careful risk/benefit evaluation in this peculiar setting.
Case report: Open bite as an extrapyramidal side effect with aripiprazole, a dopamine partial agonist
Authors: Satoko Sumi, Takahiko Nagamine, Koji Sumi, Reona Aijima, Kyoko Oka, Akira Toyofuku
Journal: Front Psychiatry (2022)
Keywords: adolescent, aripiprazole, extrapyramidal side effect, open bite, orthodontic treatment
MeSH terms: Case Reports

Long-term, fixed-point posttreatment observation of orthodontically treated patients provided us with the opportunity to capture the onset, development, and improvement of open bite, a type of malocclusion. Based on the chronological sequence of events, i.e., a tendency for open bite to worsen with increasing aripiprazole dosage and to improve with decreasing dosage, it was inferred that the onset of malocclusion was caused by extrapyramidal symptoms related to aripiprazole dosage. Physicians should be aware of this side effect when prescribing aripiprazole to children and adolescents. Careful consideration of medication history is necessary when dentists treat open bite in children and adolescents.
Acuphagia on the Obsessive-Compulsive Spectrum in an Adolescent Male
Authors: William Butler, Luis Allen
Journal: Case Rep Psychiatry (2020)
MeSH terms: Case Reports

Background. Pica is a condition that is commonly missed in childhood. This condition occurs worldwide and is considered normal in children from ages 18- to- 36 months. It is also commonly seen in pregnant women due to associated nutritional deficiencies. Acuphagia is a subtype of pica which has been briefly described in the literature. Its classification has been speculated to belong on a spectrum of obsessive-compulsive disorders (OCD). This case involves Mr. C, a 16-year-old male with a history of depression, anxiety, and ten previous intentional foreign body ingestions involving sharp objects such as needles, forks, and thumbtacks. He states that he recently ate a nail and denies any current obsessions. He was admitted from a local involuntary receiving facility due to decreased bowel movements in the last week. Learning points and recommendations for practitioners are described.
Ectopic ovary with torsion: uncommon diagnosis made by ultrasound
Authors: Adham do Amaral E Castro, Fernando Morandini, Caroline Paludo Calixto, Wagner Haese Barros, Edson Tetsuya Nakatani, Allan do Amaral E Castro
Journal: Radiol Bras (2017)
Keywords: ectopic ovary, ovary torsion, ultrasonography
MeSH terms: Case Reports

Ultrasound is an important diagnostic tool in inguinal hernia and in the evaluation of the contents of the hernia sac. This report presents a case in which ultrasound revealed a herniated ectopic ovary, complicated by torsion of its vascular pedicle, in the right labia majora. We also present a brief discussion of ovarian hernia, its potential complications, and the treatments available.
Case Report: Diagnosis of Petrous Apex IgG4-Related Disease by Middle Cranial Fossa Craniotomy and Temporal Bone Biopsy
Authors: Louis Hofmeyr, Gerrida Herbst, Elias Pretorius, Brian Sarembock, Kathryn Taylor, David Roytowski
Journal: Front Neurol (2022)
Keywords: gradenigo syndrome, igg4-related disease, case report, diplopia, middle cranial fossa, temporal bone
MeSH terms: Case Reports

Introduction:
Primary IgG4-related disease (IgG4-RD) of the temporal bone is a rare condition. Unlike typical petrous apicitis or Gradenigo syndrome, our patient presented exclusively with unilateral cranial nerve VI palsy and symptoms of diplopia. Skull base imaging demonstrated a destructive bony lesion in the petrous apex. Imaging and systemic investigations were insufficient to support a diagnosis. The diagnosis was achieved histologically after acquiring the specimen by middle cranial fossa craniotomy and temporal bone biopsy. This case report is thought to be the first published description of a diagnosis of IgG4-RD proven with the middle cranial fossa approach.
Case report:
We describe a 29-year-old female with primary IgG4-RD of the petrous apex of the temporal bone. This patient presented with a few-month history of left-sided headache and recent-onset diplopia due to paralysis of cranial nerve VI. Imaging demonstrated a petrous apex lesion, and comprehensive systemic investigations could not reach a diagnosis. A middle cranial fossa craniotomy and a biopsy of the temporal bone lesion were undertaken to establish the diagnosis. Histological confirmation of IgG4-RD was proven. Following treatment with corticosteroids, the patient experienced complete recovery and resolution of her symptoms.
Conclusion:
This study describes a case of primary IgG4-RD of the petrous apex of the temporal bone that presented with diplopia and was diagnosed by middle fossa craniotomy and temporal bone biopsy. To the best of our knowledge, this is the first case description where primary diagnosis was made based on middle cranial fossa craniotomy and temporal bone biopsy.
A case report of melioidosis complicated by infective sacroiliitis in Sri Lanka
Authors: A K T M Karunarathna, S A Mendis, W P D P Perera, Geethika Patabendige, A S Pallewatte, Aruna Kulatunga
Journal: Trop Dis Travel Med Vaccines (2018)
Keywords: burkholderia pseudomallei, eradication phase, intensive phase, melioidosis, multiple intramuscular abscesses, musculoskeletal manifestations of melioidosis, siadh, sacroiliitis
MeSH terms: Case Reports

Background:
Melioidosis is an infection caused by a facultative intracellular Gram-negative bacterium, Burkholderia pseudomallei. It can present as septicemia, localized infection with/without septicemia, asymptomatic infections, ulcers, pneumonia, visceral abscesses, neurological infection, musculoskeletal infections and can involve any organ.
Case presentation:
A 56 year old Sri Lankan diabetic female presented with fever, chills and rigors for 2 weeks. She also had malaise and loss of appetite, but no other features. On examination, she was febrile (temperature was 101.4 0 F) and rest of the examination was unremarkable. Her blood culture was positive for Burkholderia pseudomallei and she was started on IV antibiotics, on day 3. During her 2nd week of hospital stay, she developed right sided low back pain with buttock pain, right hip joint pain and restricted hip joint movements suggestive of right sacroiliitis. CE CT and MRI scans confirmed the diagnosis of right iliopsoas abscesses and right sacroiliitis.Incision and drainage was performed and a pigtail catheter was left in place for continuous drainage of abscesses. Her intensive phase was initiated with IV ceftazidime 2 g every 6 h for 12 days, then changed over to IV meropenem 2 g every 8 h together with oral co-trimoxazole. 2 weeks later, oral co-trimoxazole was replaced by oral doxycycline for another 6 weeks (due to transient pancytopaenia). She made a complete and uneventful recovery with oral co-trimoxazole for another 6 months, in her eradication phase.We report this case to show the importance in early diagnosis of melioidosis, and to consider it in the differential diagnosis of multiple abscesses and to emphasize the importance in suspecting melioidosis as a causative agent in infective sacroiliitis.
Discussion:
Melioidosis can have 2 major presentations; acute infection (symptoms lasting less than 2 months) and chronic infection (symptoms lasting more than 2 months). Musculoskeletal melioidosis is a well-recognized manifestation of the disease, which can manifest as soft tissue abscesses, septic arthritis, spondylitis, sacroiliitis and osteomyelitis.Management of melioidosis consists of 2 phases. The intensive phase and the eradication phase. These are aimed at the importance of rapidly treating the septicemia, the need of eradication of the persistent disease and the prevention of recurrent infections or relapses. The intensive phase consists of minimum 10-14 days of IV antibiotics: IV ceftazidime or IV carbapenem (meropenem/ imipenem). Eradication phase should be followed by 3-6 months of oral co-trimoxazole alone or in combination with oral doxycycline/ oral amoxiciliin-clavulanic acid.
Ectopic ACTH Syndrome Emerging 5 Years after the Diagnosis of Neuroendocrine Tumor
Authors: Minghao Liu, Diane Hamele-Bena, John Ausiello, Gabrielle Page-Wilson
Journal: Case Rep Endocrinol (2019)
MeSH terms: Case Reports

Ectopic ACTH syndrome (EAS) arising years after the diagnosis of a neuroendocrine tumor (NET) is exceedingly rare. We describe a case of EAS occurring five years after the diagnosis of a metastatic lung NET in a 61-year-old woman. She presented with severe hypokalemia but was not overtly Cushingoid on exam. Serum cortisol was 61mcg/dL after an overnight 1mg dexamethasone suppression test (<1.8mcg/dL) and urinary free cortisol was 7544 mcg/24h (<45mcg/24h), establishing the diagnosis of Cushing's syndrome. Plasma levels of peptides which have been associated with EAS, Agouti-related peptide (AgRP) and the ACTH precursors POMC (31-kDa) and pro-ACTH (22-kDa), were elevated. Metyrapone was initiated, but hypercortisolism persisted and the patient succumbed to pneumonia shortly after presentation. Retrospective examination of biopsy tissues showed rare ACTH immunoreactivity at the time of initial diagnosis, followed by staining in a greater proportion of cells as the disease progressed, consistent with EAS arising years after the diagnosis of NET. Given the increase in mortality associated with EAS, this unusual case highlights the importance of early detection and raises the possibility that early immunohistochemical stains for ACTH and measurements of ACTH precursors may facilitate the identification of NETs at high risk for EAS.
Silent Fistula of the Ascending Aorta to Pericardium by Brucella Endocarditis
Authors: Feridoun Sabzi, Siavoosh Vaziri, Reza Faraji
Journal: J Cardiovasc Thorac Res (2015)
Keywords: brucella, endocarditis, heart
MeSH terms: Case Reports

We report the case of a 26-year-old male patient with 2-week history of Brucella aortic valve endocarditis that was referred from general hospital to our hospital emergency room with pallor of the skin and mucous membranes accompanied by systemic hypotension and chest pain. Trans esophageal echocardiography (TEE) revealed a 30-mm ascending aorta at the pulmonary trunk with no evidence of the false lumen or intimal flap. TEE also showed a large vegetation of the aortic valve that limited to noncoronary sinus with moderate pericardial effusion. TEE did not showed fistula tract of nonaortic coronary sinus ring to intra mural of aorta and to pericardial cavity. The patient underwent open heart surgery with resection of destructed aortic valve and vegetation and replacement of aortic valve with prosthetic valve (Carbomedics, Sorin group. 23 mm sizes) with separated pledged suture. Debridement of aortic intra mural fistula tract and its replacement with fresh pericardial patch than performed. The pericardial cavity had moderate bloody effusion. The patient recovered uneventfully and was discharged in the 15th postoperative day. In this case, we report a rare silent clinical presentation of aortic wall fistula by vegetation and aortic ring abscess and periaortic wall hematoma, and reviewed its medical and surgical treatment.
Single ventricle, bicuspid aorta and interatrial wall aneurysm as a rare complex adult congenital heart disease: a case report
Authors: Blerim Berisha, Xhevdet Krasniqi, Agim Thaqi, Masar Gashi, Dardan Koçinaj
Journal: Cases J (2009)

Background:
Single ventricle, bicuspid aortic valve and interatrial wall aneurysm in adulthood are a rare and unique case in medical literature. This presented case with congenital heart disease has never been treated surgically and clinical consequences seriously presented in adulthood.
Case presentation:
A 27 year old man with complex congenital heart disease presented. At the age of six, the single ventricle was ultrasonographly diagnosed, but at age 27 clinical consequences started to be seriously present. We explored his history, clinical course, physical examination, laboratory findings, medical treatments and actual patient condition.
Conclusion:
The possibilities for surgical evaluation are presented.
Case Report: Mosaicism of a novel nonsense variant in the neurofibromin gene underlies a mosaic generalized NF1 phenotype
Authors: Hui Li Kwong, Yong-Kwang Tay, Ene-Choo Tan
Journal: F1000Res (2021)
Keywords: mosaicism, mutation, neurofibromatosis 1, sequencing
MeSH terms: Neurofibromatosis 1 / genetics, Neurofibromin 1 / genetics, Case Reports, Research Support, Non-U.S. Gov't, Adult, Codon, Nonsense, Genes, Neurofibromatosis 1, Humans, Male, Middle Aged, Mosaicism, Phenotype, Codon, Nonsense, Neurofibromin 1

Neurofibromatosis 1 (NF1) is a neurocutaneous syndrome characterized by multiple café-au-lait macules, cutaneous neurofibromas or plexiform neurofibromas, iris Lisch nodules, axillary and inguinal freckling. Mosaicism in NF1 can either present as a generalized disease, or in a localized (segmental) manner. Mosaic generalized NF1 may have presentations that are similar to generalized NF1 or have a milder phenotype and hence may be under-recognised in clinical practice. We report a nonsense mutation in the NF1 gene in a 55-year old Chinese male with the mosaic generalized phenotype. He reported noticing increasing numbers of skin-colored papules over his face, neck, back and abdomen when he was about 40 years old. From both next-generation and Sanger sequencing data, the variant appeared to be mosaic and present at about 24%. It is in exon 39 and has not been reported in any database or published literature.
Alleviating a Nomad's Anguish: Successful Treatment of a Case of Leg Mycetoma-A Case Report
Authors: Anthony Muchiri Maina, Joseph Theuri Macharia
Journal: Case Rep Orthop (2012)

Introduction. Mycetoma is a localized, chronic, progressive, granulomatous, inflammatory, non contagious, tumour-like lesion with sinuses discharging different types of granules. The organisms are usually inoculated into any body part subject to trauma-usually the foot. Treatment is medical and/or surgical. Prognosis is good in early cases with high recurrences in late cases or those inadequately treated. The authors describe the successful treatment of a severe case of leg mycetoma, with combined surgical and medical therapies.
Large intra-abdominal mucinous cystic adenoma: is it of ovarian or mesenteric origin
Authors: Radisnay Guzmán Lambert, Barbara Yordanis Hernandez Cervantes, Mariuska Rodríguez Gonzalez, Duniesky Martínez Lopez, Frank Edwin
Journal: Pan Afr Med J (2020)
Keywords: cystadenoma, conundrum, histophatology
MeSH terms: Cystadenoma, Mucinous / diagnosis, Mesentery / pathology, Ovarian Neoplasms / diagnosis, Case Reports, Constipation / etiology, Cystadenoma, Mucinous / pathology, Cystadenoma, Mucinous / surgery, Female, Humans, Laparotomy, Middle Aged, Ovarian Neoplasms / pathology

A 54-year-old female presented with a six year history of increasing abdominal swelling and discomfort and two months of intermittent constipation and difficulty with micturition. She was referred from the gynecological service having been investigated for a pelvic pathology without any positive findings. Her medical history was otherwise unremarkable. Physical examination revealed a non-tender intra-abdominal mass extending from epigastrium to the pelvis with a smooth surface. A large intra-abdominal multi-loculated cyst, separate from the ovaries, was seen on imaging. At laparotomy, the cystic tumour was discovered to arise from the mesentery of the terminal ileum and was resected en bloc. Histopathology revealed the tumour to be a benign mucinous cystadenoma, possibly of ovarian origin. This report aims to raise awareness of the difficulty of distinguishing ovarian from extra-ovarian mucinous cystadenomas on histopathological examination alone.
Rapid Destructive Arthropathy of the Knee in Parkinson's Disease with Pisa Syndrome: A Case of Knee-Spine Syndrome
Authors: Hirokazu Takai, Masato Kitajima, Seiko Takai, Tomoki Takahashi, Ken-Ichi Katsura, Makoto Tokunaga, Susumu Watanabe
Journal: Case Rep Orthop (2021)
MeSH terms: Case Reports

The changes occurring in knee osteoarthritis often cause alterations in the spinal loading condition, which further lead to degenerative changes. This close relationship of the knee and spine has been reported as knee-spine syndrome. A 60-year-old woman with Parkinson's disease (PD; Hoehn-Yahr stage IV) had severe knee pain with moderate lateral osteoarthritis of the knee (Kellgren-Lawrence classification grade II). Conservative therapy had no effect at all, and the knee developed destructive osteoarthritis rapidly without any traumatic episodes. The radiographic findings progressed to Kellgren-Lawrence grade IV within a month. Magnetic resonance imaging revealed partial depression of the joint surface, including shredded ossicles and substantial amounts of synovial fluid. The imaging findings were considered to be caused by a subchondral insufficiency fracture (SIF). Total knee arthroplasty was performed using a semiconstrained prosthesis. The alignment of her lower extremity improved, and the patient could walk without knee pain. The patient had Pisa syndrome, a lateral flexion of the trunk, which is a postural deformity of the trunk secondary to long-standing PD. The postural deformity in PD is not based on spinal deformity itself but on the loss of postural reflexes and the imbalance of muscle tonus. Her left knee pain appeared 1 month after L1-L4 posterior lumbar interbody fusion (PLIF) as the Pisa syndrome to her left side worsened. The more the trunk tilts to the lateral side, the center of the gravity axis will shift and pass through more lateral points of the knee and result in higher knee load. The stress concentration from the spine to the lateral joint of the knee caused lateral knee osteoarthritis, namely, knee-spine syndrome. When patients undergo correction surgery for adult spinal disorder with impairment of postural reflexes, they need to be followed up carefully regarding not only the spinal alignment but also the lower extremities.
MIR137 is the key gene mediator of the syndromic obesity phenotype of patients with 1p21.3 microdeletions
Authors: Arianna Tucci, Claudia Ciaccio, Giulietta Scuvera, Susanna Esposito, Donatella Milani
Journal: Mol Cytogenet (2016)
Keywords: 1p213, array-cgh, autism spectrum disorder, genetics, intellectual disability, mir137, obesity
MeSH terms: Case Reports

Background:
Deletions in the long arm of chromosome 1 have been described in patients with a phenotype consisting primarily of obesity, intellectual disability and autism-spectrum disorder. The minimal region of overlap comprises two genes: DPYD and MIR137.
Case presentation:
We describe a 10-year-old boy with syndromic obesity who carries a novel 1p21.3 deletion overlapping the critical region with the MIR137 gene only.
Conclusions:
This study suggests that MIR137 is the mediator of the obesity phenotype of patients carrying 1p21.3 microdeletions.