OpenMed/multicare-articles huggingface dataset:
Pulmonary Malakoplakia by Rhodococcus equi in an HIV-Infected Patient in Mexico: A Case Report
Authors: Victor H Ahumada, Ivan Ortiz-Monasterio, Jose L Hernandez, Amy B Peralta
Journal: Case Rep Infect Dis (2020)
MeSH terms: Case Reports

Background:

Rhodococcus equi-related pulmonary malakoplakia is a rare condition with few reported cases; hereby, we present a case associated with advanced human immunodeficiency virus (HIV) infection, and thus far to our knowledge, the first report in Mexico. It is estimated that approximately 10% of the infections occur in immunocompetent patients, whereas the rest are immune deficient, targeting virtually any organ. Histologically, malakoplakia is characterized by the buildup of infiltrated inflammatory tissue as a consequence of the gathering of histiocytes embedded with concentric inclusions. The diagnosis relies on the cultures and the susceptibility testing as well as the pathologic findings compatible with the disease. Case Presentation. We present a 25-year-old male patient with persistent nonproductive cough for over a year and with weight loss, who comes to the emergency department with recent fever, swollen and tender lymph nodes, and hemoptysis. The patient gets diagnosed and treated for Rhodococcus equi-related pulmonary malakoplakia.
Conclusion:
Knowing the involvement of Rhodococcus in HIV is fundamental for the diagnosis and optimal treatment, which although unknown, a combination of antibiotics with intracellular penetration, on-time resection, and a proper immune reconstitution represents the best approach. Prognosis varies with mortality rates from 34% to 54%.
Atypical right diaphragmatic hernia (hernia of Morgagni), spigelian hernia and epigastric hernia in a patient with Williams syndrome: a case report
Authors: Farhan Rashid, Ramakrishna Chaparala, Javed Ahmed, Syed Y Iftikhar
Journal: J Med Case Rep (2009)

Introduction:
Williams syndrome is rare genetic disorder resulting in neurodevelopmental problems. Hernias of the foramen of Morgagni are rare diaphragmatic hernias and they mostly present on the right side, in the anterior mediastinum. They are usually asymptomatic and are difficult to diagnose, especially in patients with learning disabilities.
Case presentation:
This 49-year-old woman with Williams syndrome, cognitive impairment and aortic stenosis presented to physicians with right-sided chest pain. She had previously undergone repair of her right spigelian and epigastric hernia. Her abdominal examination was unremarkable. Chest X-ray suggested right-sided diaphragmatic hernia and pleural effusion for which she received treatment. The computed tomography scan showed a diaphragmatic hernia with some collapse/consolidation of the adjacent lung. Furthermore, the patient had aortic stenosis and was high risk for anaesthesia (ASA grade 3). She underwent successful laparoscopic repair of her congenital diaphragmatic hernia leading to a quick and uneventful postoperative recovery.
Conclusion:
These multiple hernias suggest that patients with Williams syndrome may have some connective tissue disorder which makes them prone to develop hernias especially associated with those parts of the body which may have intracavity pressure variations like the abdomen. Diaphragmatic hernia may be the cause of chest pain in these patients. A computed tomography scan helps in early diagnosis, and laparoscopic repair helps in prevention of further complications, and leads to quick recovery especially in patients with learning disabilities. In the presence of significant comorbidities, a less invasive operative procedure with quick recovery becomes advisable.
Combined NK-CIK and PD-1 inhibitor (nivolumab), an effective immunotherapy for treating intrahepatic lymphoepithelioma-like cholangiocarcinoma unassociated with EBV infection: Two case reports and a literature review
Authors: Alen Sam Saji, Biao Yang, Wan Ting Hou, Xia Liu, Qiu Ping Ren, Yuan Feng Wei, Yu Zu Zhang, Xi Yang
Journal: Front Oncol (2023)
Keywords: ebv, cholangiocarcinoma, liver, lymphoepithelioma-like carcinoma, nivolumab
MeSH terms: Case Reports

Intrahepatic lymphoepithelioma-like cholangiocarcinoma (LELCC) is a very rare malignant tumor arising from the biliary epithelium. To date, there has been a lack of evidence on the radiographical features, clinicopathological features, and treatment modalities of LELCC, with less than 28 cases of LELCC without Epstein-Barr virus (EBV) infection having been reported worldwide. The treatment of LELCC remains unexplored. Here, we present two cases of patients with LELCC without EBV infection who were treated by liver resection, chemotherapy, and immunotherapy and who achieved long survival time. The patients received surgery to remove the tumors and then adjuvant chemotherapy using the GS regimen and combined immunotherapy involving natural killer-cytokine-induced killer (NK-CIK) and nivolumab were performed. Both patients had a good prognosis with a survival time of more than 100 months and 85 months.
Is Estrogen a Missing Culprit in Thyroid Eye Disease? Sex Steroid Hormone Homeostasis Is Key to Other Fibrogenic Autoimmune Diseases - Why Not This One?
Authors: Amy M FitzPatrick
Journal: Front Immunol (2022)
Keywords: graves’ disease, graves’ orbitopathy, ted, estrogen, estrogen receptor, sex steroids, steroid homeostasis, thyroid eye disease
MeSH terms: Acquired Immunodeficiency Syndrome, Arthritis, Rheumatoid, Autoimmune Diseases, Graves Disease, Graves Ophthalmopathy, Lupus Erythematosus, Systemic, Scleroderma, Systemic, Research Support, Non-U.S. Gov't, Estrogens, Gonadal Steroid Hormones, Homeostasis, Humans, Steroids, Estrogens, Gonadal Steroid Hormones, Steroids

Sex bias in autoimmune disease (AID) prevalence is known, but the role of estrogen in disease progression is more complex. Estrogen can even be protective in some AIDs; but in systemic lupus erythematosus (SLE), rheumatoid arthritis (RA), and systemic sclerosis (SSc), estrogen, its metabolites, and its receptors have been demonstrated to play critical, localized inflammatory roles. Estrogen is instrumental to the fibrosis seen in RA, SLE, SSc and other disease states, including breast cancer and uterine leiomyomas. Fibrotic diseases tend to share a common pattern in which lymphocyte-monocyte interactions generate cytokines which stimulate the deposition of fibrogenic connective tissue. RA, SLE, SSc and thyroid eye disease (TED) have very similar inflammatory and fibrotic patterns-from pathways to tissue type. The thorough investigations that demonstrated estrogen's role in the pathology of RA, SLE, and SSc could, and possibly should, be carried out in TED. One might even expect to find an even greater role for estrogen, and sex steroid homeostasis in TED, given that TED is typically sequalae to Graves' disease (GD), or Hashimoto's disease (HD), and these are endocrine disorders that can create considerable sex steroid hormone dysregulation. This paper highlights the pathophysiology similarities in 4 AIDs, examines the evidence of sex steroid mediated pathology across 3 AIDs and offers a case study and speculation on how this may be germane to TED.
Fracture of the Tibial Baseplate in Bicompartmental Knee Arthroplasty
Authors: Bart Stuyts, Melanie Vandenberghe, Hans Van der Bracht, Yves Fortems, Elke Van den Eeden, Luc Cuypers
Journal: Case Rep Orthop (2015)

Introduction. Bicompartmental knee arthroplasty (BKA) addresses combined medial and patellofemoral compartment osteoarthritis, which is relatively common, and has been proposed as a bridge between unicompartmental and total knee arthroplasty (TKA). Case Presentation. We present the case report of a young active man treated with BKA after unsuccessful conservative therapy. Four years later, loosening with fracture of the tibial baseplate was identified and the patient was revised to TKA. Discussion. Although our case is only the second fractured tibial baseplate to be reported, we believe that the modular titanium design, with two fixation pegs, is too thin to withstand daily cyclic loading powers. Light daily routine use, rather than high-impact sports, is therefore advised. Failures may also be related to the implant being an early generation and known to be technically complex, with too few implant sizes. We currently use TKA for the treatment of medial and patellofemoral compartment osteoarthritis.
Surgical delay in reverse sural artery flap prevents congestion of the flap: a case report of the stepwise delay method
Authors: Yuta Izawa, Hiroko Murakami, Tetsuya Shirakawa, Kentaro Futamura, Masayuki Hasegawa, Yoshihiko Tsuchida
Journal: Case Reports Plast Surg Hand Surg (2023)
Keywords: reverese sural artery flap, congestion, surgical delay
MeSH terms: Case Reports

We performed reverse sural artery flap (RSAF) with the stepwise delay method, cutting the vascular pedicle step by step, as the patient had a high risk of flap necrosis. Surgical delay in RSAF is anticipated to prevent not only flap cyanosis but also flap congestion.
Pulmonary alveolar proteinosis in association with congenital dyserythropoietic anemia: a case report
Authors: Marcus A Carden, Ashish Barman, Gita Massey
Journal: Case Rep Pediatr (2012)
MeSH terms: Case Reports

A two-year-old girl with congenital dyserythropoietic anemia (CDA) acutely developed fever, tachypnea, and increased oxygen requirement. Chest X-ray revealed bilateral interstitial infiltrates and mild cardiomegaly. Blood cultures grew no infectious agents, while pulmonary specimens grew cytomegalovirus (CMV). Treatment with intravenous ganciclovir was initiated but without response. Final cytologic preparations of bronchoalveolar lavage (BAL) fluid revealed eosinophilic amorphous material consistent with pulmonary alveolar proteinosis (PAP). CDA and PAP are extremely rare disorders in pediatrics. PAP should be considered in patients with hematological disorders who present with acute interstitial pneumonia, after infectious causes are ruled out.
Guillain-Barré syndrome following snake bite: An unusual complication
Authors: Abhishek Srivastava, A B Taly, Anupam Gupta, Aumir Moin, T Murali
Journal: Ann Indian Acad Neurol (2010)
Keywords: guillain-barrésyndrome, neuropathy, snake bite, tetanus toxoid
MeSH terms: Case Reports

A 40-year-old man presented with a nonhealing wound on the left ankle for the last 5 weeks, a tingling sensation in both hands for 20 days, and weakness in all four limbs for 10 days. He had been bitten by a snake while working in a sugarcane field 6 weeks earlier and had received tetanus toxoid and anti-snake venom on the day of the bite. He had clinical, biochemical, and electrophysiological features of Guillain-Barré syndrome, with motor and sensory neuropathy-primarily suggestive of demyelination with secondary axonal degeneration. Recognition of this unusual complication following snake bite or use of anti-snake venom / tetanus toxoid has considerable epidemiological, therapeutic, and prognostic significance.
Nonsyndromic familial oligodontia with multiple dens invaginatus: a case report of an unusual case
Authors: D P Vinuth, Poonam Agarwal, Gunjan Dube, S Abhilash, Pallavi Dube
Journal: Case Rep Dent (2013)

Oligodontia is a rare dental anomaly with a prevalence of 0.3% in permanent teeth and much less frequency in the primary dentition. Familial oligodontia represents an absence of varying numbers of primary and/or secondary teeth as an isolated trait. It is a complex and multifactorial condition. Many explanations-evolutionary, genetic, and environmental-have been proposed as the etiology. Simultaneous with oligodontia are often the different positional changes of the existing teeth, their morphology, size, and growth disturbances of the maxillofacial skeleton. Early recognition is vital to provide adequate treatment and prevent squeal. Multidisciplinary referral or consultation is thus important in treatment planning to improve function and esthetics. The present paper reports a rare case of familial oligodontia associated with multiple dense invaginatus and microdontia.
Perforated sigmoid colon cancer presenting as an incarcerated inguinal hernia: A case report
Authors: Hassan Sabra, Mersad Alimoradi, Etienne El-Helou, Rawan Azaki, Maysaloun Khairallah, Tony Kfoury
Journal: Int J Surg Case Rep (2020)
Keywords: colorectal cancer, incarcerated hernia, inguinal hernia, perforated colon cancer
MeSH terms: Case Reports

Background:
Inguinal hernias are common among the population and usually contain part of the omentum or small bowel, however, other unusual contents have been reported. We report a case of a patient who presented for an incarcerated left inguinal hernia and was found to have an underlying perforated sigmoid cancer.
Summary:
An 87 years old man presented with typical signs of an incarcerated left inguinal area. During surgery, we dissected free a herniated hard inflammatory mass until it was found to be covering an underlying perforated sigmoid tumor. Inguinal hernia is a common condition affecting many individuals, and colorectal cancer is one of the rare contents reported inside these hernias. Due to the rarity of such a presentation, the surprise encounter during groin surgery may lead to suboptimal treatment. Perforated colorectal cancer, like in our case, may signify a more aggressive disease, and hence a correct diagnosis is crucial to improve outcomes. When underlying colon cancer is suspected during hernia surgeries, proper exploration and oncologic resection might be the optimal choice.
Conclusion:
Surgeons should keep the possibility of underlying colorectal cancer in mind when operating on inguinal hernias and opt for oncologic resection of the identified tumor along with proper lymph node dissection.
A case report of Pasteurella multocida meningitis in a patient with non-traumatic skull base defect
Authors: Mohamed Kamal Sabra, Adeel Ahmad Khan, Musaed Al Samawi, Yasser El Deeb
Journal: IDCases (2020)
Keywords: csf rhinorrhea, meningitis, pasteurella multocida, skull defect
MeSH terms: Case Reports

Background:

Pasteurella multocida is a gram-negative coccobacillus that is primarily found in oropharynx of dogs, cats and other animals. It causes infections in human beings through contact with animal saliva in the form of licks, bites and scratches of animals colonized by the bacteria. Meningitis due to Pasteurella multocida is rare in immunocompetent individuals. We report a case of meningitis due to Pasteurella multocida in an immunocompetent patient.
Case report:
A 30-year-old gentleman presented with 2-day history of fever and neck stiffness. 6 weeks earlier, he was treated as a case of bacterial meningitis. During that hospital stay, he was diagnosed to have bony defect in the sellar floor based on MRI head performed to evaluate for a prolonged history of CSF rhinorrhea. He was discharged and scheduled for an elective endoscopic endonasal/open repair of the skull base defect after resolution of meningitis. CSF findings during current admission also showed features of bacterial meningitis. CSF culture showed Pasteurella multocida sensitive to penicillin, ampicillin and ceftriaxone. Retrospective history revealed patient's contact with stray cats as he used to feed them but there was no history of licks, bites. He was treated with intravenous ceftriaxone 2 g twice a day for 14 days with complete resolution of his symptoms.
Conclusion:

Pasteurella multocida is an important cause of bacterial meningitis in patients with skull defect. Patients with traumatic or non-traumatic bony defect of skull should avoid contact with dogs and cats to prevent the spread of infection the central nervous system.
Case Report: Multisystem Autoimmune and Overlapping GAD65-Antibody-Associated Neurological Disorders With Beneficial Effect of Epilepsy Surgery and Rituximab Treatment
Authors: Petia Dimova, Krassimir Minkin
Journal: Front Neurol (2022)
Keywords: gad, autoimmune, cerebellar, diabetes, epilepsy, insular, surgery, temporal
MeSH terms: Case Reports

Glutamic acid decarboxylase (GAD) antibodies are associated with disabling conditions such as stiff person syndrome, temporal lobe epilepsy (TLE), limbic encephalitis, cerebellar ataxia (CA), and ocular movement disorders, which are usually chronic and difficult to treat. GAD-related TLE has poor response to anti-seizure medications and immune therapies, and epilepsy surgery is rarely successful. We report on a 47-year-old female with history of migraine, autoimmune thyroid disease, ankylosing spondylitis, and drug-resistant TLE. A video electroencephalography recorded frequent seizures with temporo-insular semiology, correlating to left temporal epileptiform activity and left mesiotemporal hyperintensity on magnetic resonance imaging. GAD autoimmunity was confirmed by very high GAD antibody titers in serum and cerebrospinal fluid. Steroids, immunoglobulins, and cyclophosphamide had no effect, and selective left amygdalectomy was performed based on very restricted hypermetabolism on positron-emission tomography. After transient seizure freedom, significant epilepsy improvement was observed in spite of memory decline. Transient worsening was noted 1 year later during diabetes mellitus manifestation and 5 years later during presentation of progressive CA, which stabilized on rituximab treatment. We believe this case illustrates the diversity and the frequent overlap of GAD-associated disorders, the need of early and aggressive immunotherapy in severe patients, as well as the possible benefit from epilepsy surgery in some GAD-TLE.
A Rare Presentation of a Duodenal Neuroendocrine Tumor
Authors: Aboud Kaliounji, Sami Alkoutami, Michael Farraj, Haya Kaliounji, Kristen L Farraj
Journal: Cureus (2023)
Keywords: duodenum, gastroenterology tumor, gist, neuroendocrine tumor, small bowel
MeSH terms: Case Reports

Neuroendocrine tumors (NETs), which are a rare type of tumor, are defined as epithelial cells with predominantly neuroendocrine differentiation and consist of a spectrum of tumors emerging from stem cells throughout the body and can occur anywhere in the body. While they are rare, the incidence over the past few decades has increased. Here we present a case of a 64-year-old female who was incidentally found to have a duodenal neuroendocrine tumor. The patient initially presented to the emergency department secondary to syncope and collapse. During her trauma evaluation, an incidental lobulated soft tissue mass inferior to the distal stomach was seen on complete computed tomography (CT) scans. The surgery team was consulted for resection of the mass and an octreotide scan was performed prior to resection to further evaluate the mass and to check for any signs of metastatic disease. The octreotide scan demonstrated intense radiotracer accumulation within the duodenal mass consistent with a neuroendocrine tumor and no areas of abnormal radiotracer accumulation suspicious for metastatic disease. Pathology of the resected mass was positive for a well-differentiated neuroendocrine tumor with an organoid pattern and homogenous oval-round neoplastic cells with a salt-pepper nuclear and pseudo glandular arrangement that was well-circumscribed and partially encapsulated with negative margins. Immunohistochemistry was positive for AE ⅓, CD56, Synaptophysin, and chromogranin and negative for CD117, DOG-1, CD34, and CD45. The prevalence of NETs has increased over the years due to the improvement in diagnostic tools, such as upper gastrointestinal endoscopy. In addition to the fact that the duodenum is a rare location for such tumors, neuroendocrine tumors are also typically found in those under 50 years old. However, our patient was found to have both a duodenal mass and was over the age of 50 at the time of presentation and diagnosis. To date, a consensus on a conclusive treatment of duodenal NETs (D-NETs) has not been reached. This case brings to light the importance of further research in diagnosing and treating neuroendocrine tumors and also raises awareness for clinicians to have this in their differential.
Case Report: Testicular Torsion in Unilateral Supernumerary Testis
Authors: Lu Xiaofei, Zhou Benzhang
Journal: Front Pediatr (2022)
Keywords: case report, pediatrics–children, polyorchidism, supernumerary testis, testicular torsion
MeSH terms: Case Reports

Supernumerary testis (SNT), or polyorchidism, is a rare congenital anomaly of the genitourinary tract, described as the presence of more than two testicles. Testicular torsion (TT) in unilateral SNT is extremely rare. We report a case of unilateral SNT in a 16-year-old boy accompanied by TT, which was diagnosed preoperatively based on the outcomes of physical examination and ultrasound and confirmed intraoperatively. We opted for orchiectomy of this SNT because of the long-time hypoperfusion. And as for the normal testis, orchidopexy was performed. His clinical presentation subsided after surgery, and the patient was discharged 3 days later.
Dexamethasone as Abortive Treatment for Refractory Seizures or Status Epilepticus in the Inpatient Setting
Authors: Alexander B Ramos, Roberto A Cruz, Nicole R Villemarette-Pittman, Piotr W Olejniczak, Edward C Mader Jr
Journal: J Investig Med High Impact Case Rep (2019)
Keywords: anti-inflammatory, antiepileptic drugs, dexamethasone, inflammation, refractory seizures, status epilepticus, steroid
MeSH terms: Dexamethasone / administration & dosage, Glucocorticoids / administration & dosage, Status Epilepticus / drug therapy, Case Reports, Administration, Intravenous, Aged, Epilepsia Partialis Continua / drug therapy, Epilepsy, Partial, Motor / drug therapy, Female, Humans, Inpatients, Male, Middle Aged, Treatment Outcome, Glucocorticoids, Dexamethasone

Refractory seizures or status epilepticus (RS/SE) continues to be a challenge in the inpatient setting. Failure to abort a seizure with antiepileptic drugs (AEDs) may lead to intubation and treatment with general anesthesia exposing patients to complications, extending hospitalization, and increasing the cost of care. Studies have shown a key role of inflammatory mediators in seizure generation and termination. We describe 4 patients with RS/SE that was aborted when dexamethasone was added to conventional AEDs: a 61-year-old female with temporal lobe epilepsy who presented with delirium, nonconvulsive status epilepticus, and oculomyoclonic status; a 56-year-old female with history of traumatic left frontal lobe hemorrhage who developed right face and hand epilepsia partialis continua followed by refractory focal clonic seizures; a 51-year-old male with history of traumatic intracranial hemorrhage who exhibited left-sided epilepsia partialis continua; and a 75-year-old female with history of breast cancer who manifested nonconvulsive status epilepticus and refractory focal clonic seizures. All patients continued experiencing RS/SE despite first- and second-line therapy, and one patient continued to experience RS/SE despite third-line therapy. Failure to abort RS/SE with conventional therapy motivated us to administer intravenous dexamethasone. A 10-mg load was given (except in one patient) followed by 4.0- 5.2 mg q6h. All clinical and electrographic seizures stopped 3-4 days after starting dexamethasone. When dexamethasone was discontinued 1-3 days after seizures stopped, all patients remained seizure-free on 2-3 AEDs. The cessation of RS/SE when dexamethasone was added to conventional antiseizure therapy suggests that inflammatory processes are involved in the pathogenesis of RS/SE.