OpenMed/multicare-articles huggingface dataset:
Case Report: Autoimmune Pemphigus Vulgaris in a Patient Treated With Cemiplimab for Multiple Locally Advanced Cutaneous Squamous Cell Carcinoma
Authors: Rosalba Buquicchio, Valentina Mastrandrea, Sabino Strippoli, Davide Quaresmini, Michele Guida, Raffaele Filotico
Journal: Front Oncol (2021)
Keywords: pemphigus vulgaris, anti-programmed-death-1, cemiplimab, cutaneous squamous cell carcinoma, desmoglein, immune check-point inhibitors
MeSH terms: Case Reports

Background:
Pemphigus vulgaris (PV) is a rare and severe autoimmune blistering disorder affecting the skin and mucous membranes, characterized by the production of autoantibodies against two desmosomal adhesion proteins, desmoglein 1 and 3. In patients with advanced squamous cell carcinoma of the skin unfit for surgery and radiotherapy, immune check-point inhibitors, including the anti-Programmed Death-1 (PD-1) agent cemiplimab have been successfully employed proving relevant clinical outcomes. Cemiplimab is a monoclonal antibody capable of inhibiting PD-1 signalling that has recently been approved for the treatment of patients with metastatic or locally advanced cutaneous squamous cell carcinoma. Although the peculiar setting of advanced CSCC involving elderly patients, rare and unusual skin immune-related adverse events such as PV could be observed in cemiplimab treated patients.
Case report:
A 95-year-old man without a history of autoimmune disease was treated with cemiplimab for multiple and advanced squamous cell carcinomas of the head obtaining a complete response to therapy. After seven cycles of cemiplimab administered every 21 days, the patient developed a mucocutaneous blistering eruption. Clinical diagnosis of PV was suspected on the basis of the diffuse involvement of trunk and extremities with large blisters and necrotic eschar. It was carried out an ELISA test, that showed high level of circulating antibodies against desmoglein 1, thus confirming the diagnosis of PV. For this reason, cemiplimab infusion was discontinued and complete resolution of skin lesions was obtained using oral prednisone 0,8 mg/kg/daily for four weeks. Once remission was achieved, a maintenance dose of 10 mg/day was administered, observing a good control of bullous disease and low value of desmoglein 1. Response to CSCC persisted also during cemiplimab discontinuation, until obtaining a complete remission still persisting at 9 months after the last cycle of therapy.
Conclusion:
The case we observed is the first description of PV revealed from cemiplimab therapy, thus suggesting that cemiplimab could allow the arise of underlying autoimmune PV, through a mechanism both T and B-cell-mediated.
Rescue surgery for advanced anal gland adenocarcinoma: A case report
Authors: Sonaira Francisca Alves da Silva Bernardes, Dirceu de Castro Rezende Junior, Natasha Sa Gille Rissin, Tânia Rosa Pereira da Mota, Alexandre de Brito Borges Pimentel
Journal: Int J Surg Case Rep (2019)
Keywords: anal gland neoplasms, case report, neoadjuvant therapy, surgical oncology

Introduction:
Extramucosal anal canal adenocarcinomas can arise in anorectal fistulas and the anal glands, the latter being rare. We present a rare case of anal gland adenocarcinoma treated with a combination of neoadjuvant therapy and radical surgical resection.
Presentation of case:
A 56-year-old man presented with rectal bleeding and irritation, and a nodule that had been enlarging for 10 months. Rectal examination revealed a bleeding ulceroproliferative growth at the left lateral edge of the anus without apparent invasion of the anal mucosa. Histopathology and immunohistochemistry confirmed the diagnosis of anal canal adenocarcinoma CK7+,CDX2-, and focalCK20+). Endoanal ultrasound showed a lesion involving the anal canal, extending into the transition zone with the lower rectum, invading the external anal sphincter, with no cleavage plane with the urethra, measuring 89 × 33 × 57 mm, associated with lymphadenopathy in the lower mesorectum (uT4N1). PET/CT confirmed a hypermetabolic lesion on the anal edge and bilateral hypermetabolic inguinal lymph nodes suggestive of secondary involvement. Colonoscopy was normal. The patient was started on neoadjuvant therapy with oral capecitabine and radiotherapy (57.6 Gy). Twelve weeks, the patient underwent extralevator abdominoperineal excision, cystoscopy (free urethral mucosa), skeletonization of the urethra with partial resection of the corpus cavernosum, and pelvic floor reconstruction with a vertical rectus abdominis myocutaneous flap.
Discussion:
Treatment of anal gland adenocarcinoma remains to be established. A combination of radical surgical resection and neoadjuvant/adjuvant chemoradiotherapy has been suggested, as performed here.
Conclusion:
Patients with advanced anal gland adenocarcinoma may benefit from neoadjuvant therapy followed by rescue surgery.
An Unusual Case of Asystole following Penetrating Neck Trauma and Anoxic Brain Injury
Authors: Matthew Nayor, Alissa J Berliner, Grant V Chow, David D Spragg
Journal: Case Rep Cardiol (2011)
MeSH terms: Case Reports

Bradycardia and transient asystole are well-described sequelae of a myriad of neurologic insults, ranging from focal to generalized injuries. Increased vagal tone also predisposes many individuals, particularly adolescents, to transient neurally mediated bradyarrhythmia. However, prolonged periods of sinus arrest without junctional or ventricular escape are quite rare, even after significant neurologic injury. We describe the case of a 17-year-old man who presented with anoxic brain injury secondary to hemorrhagic shock from a stab wound to the neck. His recovery was complicated by prolonged periods of sinus arrest and asystole, lasting over 60 seconds per episode. This case illustrates that sustained asystolic episodes may occur following significant neurologic injury, and may continue to recur even months after an initial insult. Pacemaker implantation for such patients should be strongly considered.
Late-Stage Löffler's Endocarditis Mimicking Cardiac Tumor: A Case Report
Authors: Takafumi Koyama, Hiroyuki Yamamoto, Manabu Matsumoto, Jun Isogai, Tadashi Isomura, Shinji Tanaka
Journal: Front Cardiovasc Med (2020)
Keywords: löffler's endocarditis, cardiac magnetic resonance, cardiac thrombus, endomyocardial fibrosis, hypereosinophilic syndrome
MeSH terms: Case Reports

Löffler's endocarditis (cardiac involvement in hypereosinophilic syndrome) is rare yet life-threatening if left untreated. We describe a case of hypereosinophilic syndrome presenting as a cardiac mass with an abnormal electrocardiogram. Diagnostic studies of the cardiac mass strongly suggested a malignant cardiac tumor invading the papillary muscle. Thus, excision of the cardiac mass and endomyocardial resection with mitral valve replacement were successfully performed. Pathology revealed various stages of thrombosis and irreversible myocardial damage caused by eosinophilic infiltration with no malignancy, leading to the correct diagnosis of late-stage Löffler's endocarditis. The subsequent combination of anticoagulation and corticosteroids was effective with a favorable outcome. This case highlights pitfalls in multimodality imaging of cardiac thrombus and the clinical significance of considering Löffler's endocarditis in the diagnostic work-up of a cardiac mass.
Case Report and Systematic Review: Sarcomatoid Parathyroid Carcinoma-A Rare, Highly Malignant Subtype
Authors: Yongchao Yu, Yue Wang, Qingcheng Wu, Xuzi Zhao, Deshun Liu, Yongfu Zhao, Yuguo Li, Guangzhi Wang, Jingchao Xu, Junzhu Chen, Ning Zhang, Xiaofeng Tian
Journal: Front Endocrinol (Lausanne) (2021)
Keywords: pth, diagnosis, parathyroid carcinoma, sarcomatoid, surgery
MeSH terms: Adenoma / diagnostic imaging, Adenoma / surgery, Neoplasm Recurrence, Local / diagnostic imaging, Parathyroid Neoplasms / diagnostic imaging, Parathyroid Neoplasms / surgery, Case Reports, Research Support, Non-U.S. Gov't, Systematic Review, Female, Humans, Middle Aged

Background:
Parathyroid carcinoma (PC) is a rare malignancy, the incidence of which is less than 1/1 million per year. Sarcomatoid parathyroid carcinoma (SaPC) is an extremely peculiar subtype; only three cases have been reported internationally. It consists of both malignant epithelial components and sarcomatoid components (mesenchymal origin) simultaneously. This "confusing" cancer exhibits higher invasiveness, and traditional surgery does not appear to achieve the expectation, which differs significantly from that of general PC.
Objective:
To characterize the clinicopathologic features of SaPC and explore similarities and differences between SaPC and general PC.
Materials and methods:
We collected clinical data of SaPC cases from our center and literature. The SaPC case in our center was presented. To better understand the characteristics of SaPC, we also reviewed clinical information in general PC cases from our center and literature within the last 5 years, and a systematic review was performed for further comparison.
Results:
A 60-year-old woman was admitted for a neck mass and hoarseness. After the surgery, she was confirmed as SaPC and ultimately developed local recurrence at 3 months. Together with the reported cases from literature, four cases of SaPC (three cases from literature) and 203 cases of general PC (200 cases from literature) were reviewed. Both tumors showed obvious abnormalities in parathormone (PTH) level and gland size. Compared to general PC, SaPC has a later age of onset (60.50 ± 7.42 vs. 51.50 ± 8.29), relatively low levels of PTH (110.28 ± 59.32 vs. 1,156.07 ± 858.18), and a larger tumor size (6.00 ± 1.63 vs. 3.14 ± 0.70). For SaPC, all four cases were initially misdiagnosed as thyroid tumors (4/4). Spindle cell areas or transitional zones were common pathological features in SaPC cases (3/4).
Conclusion:
SaPC is a very rare pathologic subtype of PC and appears to be much more easily misdiagnosed as a thyroid tumor. Spindle cell areas or transitional zones are highly possible to be pathological features in its sarcomatoid components. Despite many similarities, there are some differences between SaPC and general PC-SaPC does not show the obvious endocrine feature but stronger aggressiveness. Surgical treatment of SaPC does relieve life-threatening symptoms and improve quality of life even with recurrence in the short term.
Isolated Polycystic Liver Disease: An Unusual Cause of Recurrent Variceal Bleed
Authors: Mohammad Saud Khan, Zubair Khan, Toseef Javaid, Jamal Akhtar, Abdelmoniem Moustafa, Amos Lal, Abhinav Tiwari, Mohammad Taleb
Journal: Case Rep Gastrointest Med (2018)
MeSH terms: Case Reports

Isolated polycystic liver disease is a rare disorder. Majority of the patients with isolated polycystic liver disease are asymptomatic with incidental detection of liver cysts on imaging studies done for other purposes. Minority of patients develop symptoms which are mostly secondary to enlarging cysts size and hepatomegaly. Rarely, these patients develop portal hypertension and can present with its clinical manifestations and consequences in the form acute variceal bleeding or recurrent ascites. We present a rare case of 67-year-old female patient with significant history of polycystic liver disease who presented to the hospital with recurrent hematemesis and melena. She underwent esophagogastroduodenoscopy which showed multiple large esophageal varices requiring banding.
Use of a Superficial Abdominal Wall Vein in a Gravida Patient for Emergency Vascular Access
Authors: Christine Ha, Rotem Naftalovich, Faraz Chaudhry, Jean Eloy, Erica Spano, George Tewfik
Journal: Case Rep Anesthesiol (2023)
MeSH terms: Case Reports

Obtaining vascular access through a superficial vein of the abdominal wall of a gravida patient is an option in an emergency Cesarean surgery when other means fail. Such superficial veins may be mistaken for striae gravidarum on physical exam. A small intravenous (IV) cannula is not ideal but could save valuable time and avoid delaying induction of general anesthesia. Once the airway is secured, a larger bore IV can then be inserted while surgical exposure is undergoing. Analysis of the risks and benefits of inducing general anesthesia with a small gauge IV for a gravida patient should take into consideration risk factors for massive peripartum hemorrhage such as placental disorders (accreta, increta, precreta, abruption, or previa), presence of uterine fibroids, preeclampsia, hemolysis, elevated liver enzymes, low platelet count (HELP syndrome), severe polyhydramnios, history of grand multiparty, and bleeding disorders such as Von Willibrands and hemophilia.
Case Report: Huge Dumbbell-Shaped Primary Hydatid Cyst Across the Intervertebral Foramen
Authors: Yu Tian, Mei Jiang, Xin Shi, Yujun Hao, Lei Jiang
Journal: Front Neurol (2020)
Keywords: case report, echinococcosis, hydatid cyst, intervertebral foramen, spinal canal
MeSH terms: Case Reports

Primary hydatid cyst of the spinal canal is extremely rare. We reported a 42-year-old Kazakh man with right lower back pain and weakness in both lower limbs for 2 months, who lived in the pastoral area. Clinical examination revealed that the patient had no cysts on other organs and no previous medical history except for a huge cyst inside and next to the vertebrae. MRI examination revealed a huge dumbbell-shaped primary cyst across the intervertebral foramen. Pathological examination after operation confirmed a fine-grained hydatid cyst disease. Therefore, in the pastoral area, doctors should be alert to the occurrence of hydatid cyst disease if patients complained about progressive back pain and lower limb weakness or other spinal cord compression symptoms. Once hydatid cysts in other organs or systems were detected, the occurrence of the disease could be highly suspected. Complete resection is an effective treatment for hydatid cyst disease.
Case Report: Refractory Hypotension of GABA B Receptor Autoimmune Encephalitis
Authors: Kong Hanxin, Wang Xiaomin, Ying Hao, Zheng Shuihong
Journal: Front Neurol (2020)
Keywords: gaba b receptor autoimmune encephalitis, autoimmune disease, autonomic nerve function, case report, refractory hypotension
MeSH terms: Case Reports

Introduction: With the development of antibody detection technology, Gamma-Aminobutyric Acid (GABA) B receptor encephalitis is a known autoimmune disease. This paper describes a patient with refractory hypotension who suffered GABA B receptor autoimmune encephalitis. Case Report: We describe a 63-year-old man with GABA B receptor autoimmune encephalitis who had hypotension on day 17 of the disease onset. Despite two rounds of immunoglobulin administration, high-dose intravenous steroid injections and immunosuppressive therapy on day 35 of hospitalization, psychiatric symptoms and seizures were significantly improved; however, the patient's blood pressure remained low. Conclusion: This case study and literature review investigated the impairment of autonomic nerve function and its subsequent management in patients with GABA B receptor autoimmune encephalitis.
Diffuse Gastric Ganglioneuromatosis: Novel Presentation of PTEN Hamartoma Syndrome-Case Report and Review of Gastric Ganglioneuromatous Proliferations and a Novel PTEN Gene Mutation
Authors: Alexander J Williams, Emily S Doherty, Michael H Hart, Douglas J Grider
Journal: Case Rep Med (2018)
MeSH terms: Case Reports

Gastrointestinal ganglioneuromatous proliferations are rare, most often found in the colon, and are three types: polypoid ganglioneuromas, ganglioneuromatous polyposis, and diffuse ganglioneuromatosis. We present a case of diffuse ganglioneuromatosis in the posterior gastric wall in a nine-year-old female. To our knowledge, this is the first reported case of diffuse ganglioneuromatosis located in the stomach. Only six cases of gastric ganglioneuromatous proliferations have previously been reported, two in English and none were diffuse ganglioneuromatosis. A diagnosis of diffuse ganglioneuromatosis is relevant for patient care because, unlike sporadic polypoid ganglioneuromas or ganglioneuromatous polyposis, most are syndromic. Diffuse ganglioneuromatosis is commonly associated with neurofibromatosis type 1, multiple endocrine neoplasia type 2b, and Cowden Syndrome, one of the phenotypes of PTEN hamartoma tumor syndrome. The patient had the noted gastric diffuse ganglioneuromatosis, as well as other major and minor criteria for Cowden syndrome. Genetic testing revealed a novel frameshift mutation in the PTEN gene in the patient, her father, paternal aunt, and the aunt's son who is a paternal first cousin of the patient.
Case report: Bladder preserving after maximal transurethral resection of the bladder tumor combined with chemotherapy and immunotherapy in recurrent muscle-invasive bladder cancer patients: A report of two cases
Authors: Jiaquan Mao, Chunguang Yang, Sheng Xin, Kai Cui, Zheng Liu, Tao Wang, Zhiquan Hu, Shaogang Wang, Jihong Liu, Xiaodong Song, Wen Song
Journal: Front Med (Lausanne) (2022)
Keywords: bladder-preserving therapy, case report, immunotherapy, muscle-invasive bladder cancer (mibc), programmed death-ligand 1 (pd-l1)
MeSH terms: Case Reports

Background:
Cisplatin-based neoadjuvant chemotherapy combined with radical cystectomy (RC) plus pelvic lymph node dissection (PLND) is the preferred treatment option for muscle-invasive bladder cancer (MIBC). However, some patients are unable to tolerate RC or may have postoperative complications after RC. And most patients have a strong desire for bladder-preserving treatment. There are no reports on the efficacy of maximal transurethral resection of the bladder tumor (TURBT) in combination with chemotherapy plus tislelizumab for bladder-preserving in recurrent MIBC patients.
Case presentation:
We report two cases diagnosed with recurrent MIBC who achieved pathological complete response (pCR) and bladder-preserving after maximal TURBT combined with chemotherapy plus tislelizumab.
Conclusion:
Postoperative immunotherapy should be considered for all patients with recurrent MIBC who are eligible for immunotherapy. In addition, high programmed death ligand-1 (PD-L1) expression, high tumor mutation burden (TMB), and TP53 mutation level can be combined to predict tislelizumab efficacy.
Bilateral multicenter pseudohemangiomatous interstitial hyperplasia of the breast: a case report
Authors: Chengcai Yao, Changchun Liu
Journal: Case Reports Plast Surg Hand Surg (2023)
Keywords: pseudohemangiomatous interstitial hyperplasia, breast reconstruction, case report, mammary gland
MeSH terms: Case Reports

Bilateral multicenter breast pseudohemangiomatous stromal hyperplasia (PASH) is a rare, benign breast disease. Here, we report on a female patient with bilateral multicenter PASH who underwent a mastectomy and prosthetic reconstruction. The surgery was successful, and no recurrence was observed during the 18 months of follow up.
Castleman's Disease: A Case Report of the Unicentric Type
Authors: J Gomez-Ramirez, M Posada, L Sanchez-Urdazpal, E Martin-Perez, L Del Campo, I Garcia, J L Martin, E Larrañaga
Journal: Case Rep Surg (2012)

Castleman's disease, or angiofollicular lymph node hyperplasia, is a relatively rare disorder characterized by the benign proliferation of lymphoid tissue related to the chronic human herpes virus 8 (HHV-8) infection and the human immunodeficiency virus (HIV). Two clinical entities have been described: a unicentric presentation with the disease confined to a single anatomic lymph node and a multicentric presentation characterized by generalized lymphadenopathy and a more aggressive clinical course. Also, three histopathological subtypes have been described: hyaline-vascular, plasma cell, and a mixed variant. Preoperative diagnosis of hyaline-vascular Castleman's disease is difficult, and the definitive result is based on postoperative pathological findings. The gold standard therapy is the complete surgical excision.
Case series: Continued remission of PTSD symptoms after discontinuation of prazosin
Authors: Christie Richardson, Jonathan Yuh, Jing Su, Martin Forsberg
Journal: Front Psychiatry (2023)
Keywords: flashbacks, nightmares, post-traumatic stress disorder, prazosin, psychopharmacology, trauma
MeSH terms: Case Reports

Post-traumatic stress disorder is a debilitating chronic illness that affects 6 out of 100 adults after a severe trauma. The alpha-adrenergic antagonist prazosin, which is prescribed off-label for flashbacks and nightmares due to trauma, is often continued indefinitely due to reports of symptoms returning upon discontinuation. There is no standard guidance for a trial of discontinuation of prazosin due to intolerance or side effects. In this case series, three patients are started on prazosin leading to remission of trauma-related symptoms, and symptoms continue to remit after treatment for an average of about 2 years followed by discontinuation of the medication. There are many similarities in these case reports which serve to provide guidance as to when a trial of prazosin discontinuation may be warranted.
Asymptomatic deep vein thrombosis in a patient with major depressive disorder
Authors: Takuto Ishida, Takeshi Katagiri, Hiroyuki Uchida, Takefumi Suzuki, Koichiro Watanabe, Masaru Mimura
Journal: Case Rep Psychiatry (2012)

Pulmonary embolism is a serious, life-threatening condition and most commonly derives from deep vein thrombosis of the lower extremities. Once deep vein thrombosis (DVT) reaches a proximal vein (i.e., popliteal vein or higher), pulmonary embolism reportedly occurs in up to 50% of patients. Case Presentation. We report on an inpatient with major depressive disorder in a catatonic state in whom an asymptomatic proximal deep vein thrombosis of 11 × 70 mm was detected through routine screening, using doppler ultrasound scanning. Anticoagulant therapy was immediately started and continued for three months, which resulted in resolution of the deep vein thrombosis. Discussion. To our knowledge, this is the first description of asymptomatic proximal DVT that was detected in a psychiatric inpatient setting. In light of the reported causal relationship between DVT and pulmonary embolism, screening for DVT can be of high clinical value in patients with psychiatric disorders, especially when their physical activity is highly compromised.