OpenMed/multicare-articles huggingface dataset:
Endoscopic Mucosal Resection of a Proximal Esophageal Pyogenic Granuloma
Authors: Elias Estifan, Varun Patel, Matthew Grossman
Journal: Case Rep Gastrointest Med (2019)
MeSH terms: Case Reports

Pyogenic Granuloma (PG), also known as lobular capillary hemangioma, is usually seen as a polypoid red lesion found on the skin or the mucosal surface of the oral cavity. PG of the gastrointestinal tract is rare, in particular involving the esophagus, only 14 cases have been reported in the English literature. We present an 80-year-old male who underwent endoscopy for evaluation of dysphagia and was found to have a single, red, bilobed 10 mm polyp with adherent white exudate approximately 19 cm from the incisors. Endoscopic ultrasound was performed with a 20 mHz miniprobe which showed the lesion contained to the mucosal layer with no muscularis propria invasion. A decision was made to perform endoscopic mucosal resection (EMR). A mixture of saline and methylene blue was injected into the submucosal plane to raise the lesion with subsequent successful mucosal hot snare resection. The resection defect was then approximated and closed with a hemostatic clip to prevent bleeding. Pathology of the specimen revealed small capillary vessels growing in a lobular architecture with an edematous stroma and a florid inflammatory infiltrate representing a pyogenic granuloma. EMR allows for an en bloc resection of mucosal lesions with tumor-free margins, thereby providing both diagnostic and prognostic information. Comparing EMR with the novel technique of endoscopic submucosal dissection (ESD), the incidence of bleeding and perforation is much lower; making EMR the best and safest resection option for this rare hemangioma. In this case, we demonstrate that EMR is a safe technique in removing a pyogenic granuloma in the esophagus.
Bilateral ectopic third molars in maxillary sinus associated with dentigerous cyst-A rare case report
Authors: Sarwpriya Sharma, Jaideep Singh Chauhan
Journal: Int J Surg Case Rep (2019)
Keywords: dentigerous cyst, ectopic, impacted, maxillary molars, maxillary sinus

Introduction:
Ectopic eruptions of a tooth in a non-dentate region is a rare entity and such cases have been reported to occur in the nasal cavity, orbital floor, maxillary sinus, mandibular condyle, chin, palate and coronoid process.
Case presentation:
We report a case of bilateral ectopic eruption of maxillary third molars associated dentigerous cyst. A 27 years old female patient reported to us with chief complaint of purulent discharge form nose and recurrent facial swelling for last 2 years. Complete removal of cystic lining and extraction of ectopic maxillary third molars along with mandibular third molars was carried out.
Results:
Post-operative healing was uneventful.
Discussion:
Bilateral ectopic eruptions of maxillary third molars in Antrum of Highmore (Maxillary sinus) are rarely seen and scantly documented in the literature. Dental ectopia may occur in antrum and present with local sino-nasal symptoms attributed to chronic sinusitis. Due to its rarity and lack of consensus over its management, the incidence deserves to be added to the literature & discussed.
Ondansetron-related hemorrhagic posterior reversible encephalopathy syndrome (PRES) following gastric bypass
Authors: M Alain Babi, Mark J Gorman, Marilyn J Cipolla, Gilman Allen, Salman Al Jerdi, Ryan Clouser, Christopher Commichau
Journal: Springerplus (2016)
Keywords: cerebral autoregulation, imaging, intracerebral hemorrhage, intracranial hypertension, intracranial pressure, ondansetron, pres (posterior reversible encephalopathy syndrome), stroke

Background:
Posterior reversible encephalopathy syndrome (PRES) is a clinical-radiographic syndrome formally recognized in 1996, which describes specific changes noted on neuroimaging thought to be related to impaired cerebral blood flow autoregulation and endothelial dysfunction. We report a case of PRES in the setting of increased ingestion of ondansetron; complicated by hemorrhagic transformation and refractory intracranial hypertension. We hypothesize an association of 5-HT3 antagonism and PRES.
Findings:
This is a case study report; with review of previously published literature through PubMed search. We describe the case of a 25 year old man following bariatric surgery who increased his ingestion of ondansetron, taking up to 40 tablets/day due to excessive nausea and vomiting. The patient was hospitalized for progressively more severe headache of 1 week's duration. Computed tomography (CT) revealed bilateral cerebral edema in the parietal and occipital lobes in the setting of elevated blood pressure (BP). Three days into his admission, following improvement in his BP with oral anti-hypertensive but continued use of the ondansetron, the patient developed near complete blindness. CT head imaging revealed progression of the posterior cerebral edema and intraparenchymal hemorrhage. He was admitted to our ICU and despite supportive treatment, his neurological examination worsened while CT head imaging findings remained stable. Invasive multimodality monitoring revealed elevated intracranial pressure. The patient was aggressively treated and after a prolonged hospitalization and rehabilitation course, made a significant recovery.
Conclusion:
This case highlights a very rare potential neurological complication of ondansetron, a commonly used medication. We hypothesize an underlying association between PRES and 5-HT3 antagonism, via the latter's potential role in endothelial dysfunction. Prompt recognition and treatment of PRES is essential, in order to prevent secondary cerebral injury and the associated potentially grave consequences.
Orbital Swelling and Ptosis as an Initial Presentation of Childhood Acute Lymphoblastic Leukaemia: A Case Report and Review of the Literature
Authors: Ahmed Alsalem, Bayan Almasoudi, Ghaida Alzahrani, Lama Sindi, Joud Alwan
Journal: Case Rep Pediatr (2021)
MeSH terms: Case Reports

We are reporting the case of a 3-year-old-girl who initially presented with unilateral eyelid swelling and ptosis. A diagnosis of acute lymphoblastic leukaemia (ALL) was eventually made based on an orbital incisional biopsy and a bone marrow examination. Historically, orbital involvement had been linked to myeloid leukaemia; however, in lymphoid leukaemia, they are increasingly being implicated and had been reported as the sole presentation of the disease. These findings stress the importance of conducting ophthalmologic assessments in cases diagnosed with ALL in order to prevent delays in proper assessment and treatment. Management options in orbital disease are fortunately not significantly different than well-established treatment protocols.
Unusual Presentation of Follicular Dendritic Cell Sarcoma as a Cystic Neck Swelling
Authors: G Chidananda-Murthy, P Babu, J Chandran, G Raja
Journal: Case Rep Oncol Med (2018)
MeSH terms: Case Reports

Follicular dendritic cell sarcoma is a very rare neoplasm that most commonly involves cervical lymph nodes and usually presents as a solid mass. Presentation as a cystic neck mass is very rare. Radiological studies and aspiration cytology are often unreliable, and diagnosis is usually made after thorough clinical and pathological examination along with immunohistochemical analysis. In this article, we report a case of a 42-year-old man who presented with right-sided neck swelling of a 2-month duration. Contrast-enhanced CT showed a cystic lesion in the right side of the neck inferomedial to parotid gland located between medial border of sternocleidomastoid muscle and internal jugular vein. Fine needle aspiration cytology was inconclusive. Patient underwent excision biopsy. Histological examination showed a solid-cystic tumor composed of spindle cells arranged in storiform pattern and showed a positive staining for CD23, CD35, and CD21 that confirmed the diagnosis of follicular dendritic cell sarcoma.
Bilateral Ovarian Endometriomas Presenting as Nonprogress of Labor: First Case Report in the Literature Is Concomitant Surgical Excision during Cesarean Section Advisable?
Authors: Shashank Shekhar, Chanderdeep Sharma, Vinay Somya, Meghna Thusoo, Nidhi Raina
Journal: Case Rep Obstet Gynecol (2012)

Objective. To report the first case of bilateral ovarian endometriomas, leading to nonprogress of labour, successfully excised during cesarean section. Design. Case report. Setting. Department of Obstetrics & Gynecology of Dr. RPGMC Tanda, Kangra, India. Patients. A primigravida in labour at term gestation. Interventions. Surgical management. Main Outcome Measures. Description and treatment of a pregnant woman with bilateral ovarian endometriomas during cesarean section. Results. Successful excision of ovarian endometriomas and reconstruction of the ovaries during cesarean section. Conclusion. Management of incidentally detected endometriomas during cesarean section should be individualized, taking into account the symptoms, size, bilaterality, and adhesion with adjacent organs.
Very High Dehydroepiandrosterone Sulfate (DHEAS) in Serum of an Overweight Female Adolescent Without a Tumor
Authors: Daniel I Iliev, Regina Braun, Alberto Sánchez-Guijo, Michaela Hartmann, Stefan A Wudy, Doreen Heckmann, Gernot Bruchelt, Anika Rösner, Gary Grosser, Joachim Geyer, Gerhard Binder
Journal: Front Endocrinol (Lausanne) (2020)
Keywords: dehydroepiandrosterone (dhea), dehydroepiandrosterone sulfate (dheas), steroid sulfatase, transporter proteins, tumor
MeSH terms: Codon, Nonsense, Dehydroepiandrosterone Sulfate / blood, Pediatric Obesity / pathology, Steryl-Sulfatase / genetics, Case Reports, Research Support, Non-U.S. Gov't, Adolescent, Case-Control Studies, Female, Humans, Multidrug Resistance-Associated Protein 2, Pediatric Obesity / blood, Pediatric Obesity / genetics, Prognosis, Young Adult, ABCC2 protein, human, Codon, Nonsense, Multidrug Resistance-Associated Protein 2, Dehydroepiandrosterone Sulfate, Steryl-Sulfatase

Introduction: An increase of serum dehydroepiandrosterone (DHEA) sulfate (DHEAS) is observed in premature adrenarche and congenital adrenal hyperplasia. Very high DHEAS levels are typical for adrenal tumors. Approximately 74% of DHEAS is hydrolyzed to DHEA by the steroid sulfatase (STS). The reverse reaction is DHEA sulfation. Besides these two enzyme reactions, the DHEAS transported through the cell membrane is important for its distribution and excretion. Case Presentation: We present a female adolescent with overweight and a very high DHEAS. The presence of a DHEAS-producing tumor was rejected using ultrasonography, Magnetic Resonance Tomography (MRT), and dexamethasone suppression. STS deficiency was suspected. Sequence analysis revealed a heterozygous nonsense mutation which predicts a truncation of the carboxyl region of the STS that is implicated in substrate binding. No partial gene deletion outside exon 5 was detected by multiplex ligation-dependent probe amplification. The bioassay revealed normal enzyme activity in the patient's leukocytes. A defect of transporter proteins was suggested. Both efflux [multidrug-resistance protein (MRP)2 and breast cancer-resistance protein (BCRP)] and uptake [organic anion-transporting polypeptide (OATP) and organic anion transporter (OAT) carriers] transporters were studied. Sequence analysis of exons revealed a heterozygous Q141K variant for BCRP. Conclusions: A novel heterozygous nonsense mutation in the STS gene and a known heterozygous missense variant in the BCRP gene were found. The heterozygous nonsense mutation in the STS gene is not supposed to be responsible for STS deficiency. The BCRP variant is associated with reduced efflux transport activity only in its homozygous state. The combination of the two heterozygous mutations could possibly explain the observed high levels of DHEAS and other sulfated steroids.
Case Report: Severe osteoporosis misunderstood by bone metastasis after total gastrectomy and multiple metastasectomy
Authors: Seong Ho Hwang, Dong Jin Kim
Journal: Front Oncol (2023)
Keywords: bone metastasis, gastric cancer, local recurrence, metastasectomy, nutritional screening, osteoporosis, total gastrectomy
MeSH terms: Case Reports

After radical gastrectomy for gastric cancer, patients should be monitored from two perspectives. One is local recurrence or metastasis, and the other is nutritional and metabolic side effects. Herein, we report a case of severe osteoporosis that was misunderstood for bone metastasis due to increased bone scan and positron emission tomography-computed tomography uptake in the patient who underwent total gastrectomy and consecutive multivisceral metastasectomy. She was administered bisphosphonates, calcium carbonate, and cholecalciferol. After 3 months, a follow-up bone scan revealed decreased intensity of hot-uptake lesions, healed fracture lesions, and eventually improved bone pain. This study supports the need for careful nutritional screening as well as cancer surveillance after gastrectomy for gastric cancer and the need for screening guidelines for bone metabolic diseases.
An Adrenocortical Carcinoma Associated with Non-Islet Cell Tumor Hypoglycemia and Aberrant ACTH Production
Authors: M D S A Dilrukshi, A W Wickramarachchi, D D K Abeyaratne, Brian Shine, Bahram Jafar-Mohammadi, N P Somasundaram
Journal: Case Rep Endocrinol (2020)
MeSH terms: Case Reports

Introduction. Adrenocortical carcinomas (ACCs) are infrequently reported to present with severe hypoglycemia syndrome resulting from the secretion of insulin-like growth factor II (IGF-II) by tumor cells. Adrenocorticotropic hormone- (ACTH) independent hypercortisolism is the norm of hormonally active ACCs, but aberrant ACTH production by tumor cells can theoretically cause ACTH-dependent hypercortisolism. The purpose of this report was to present a case of an ACC manifested with the co-occurrence of two extremely rare presentations. Case Description. We present a rare case of a 43-year-old male patient admitted with recurrent episodes of severe non-ketotic and non-insulin-mediated hypoglycemia due to IGF-II mediated disease and ACTH-dependent Cushing's syndrome. He was diagnosed with a diffusely disseminated adrenocortical carcinoma with immunohistochemistry of tumor cells showing focal ACTH immunostain positivity. Conclusion. Non-islet cell tumor hypoglycemia and ACTH-dependent Cushing's syndrome are extremely rare presentations of an ACC, and co-occurrence of these entities in a single patient is never reported in the literature.
Iatrogenic Aortocoronary Arteriovenous Fistula following Coronary Artery Bypass Surgery: A Case Report and Complete Review of the Literature
Authors: Jonathan D Gardner, William R Maddox, Joe B Calkins Jr
Journal: Case Rep Cardiol (2012)

The case of a patient who presented with angina following a coronary artery bypass (CABG) operation during which the left internal mammary artery was inadvertently anastomosed to a cardiac vein is presented. The literature concerning previously reported cases of aortocoronary arteriovenous fistulas (ACAVF) due to inadvertent grafting of a coronary vein is reviewed and the significance of this complication is discussed. ACAVF due to inadvertent grafting of a coronary vein is a rare complication of CABG and may be a more common cause of graft failure than has previously been recognized. Distortion of cardiac anatomy, the presence of epicardial fat, and an intramyocardial course of the artery intended for grafting are predisposing factors. Some patients present with angina pectoris and heart failure whereas others have no symptoms. The diagnostic test of choice is coronary angiography. Cardiac MRI and CT have a limited role due to the smaller size and the more clearly defined course of these fistulas. Asymptomatic patients are simply observed since spontaneous closure of these fistulas is reported. Symptomatic patients can be treated with combined medical management and percutaneous methods.
Case report: Aromatic L-amino acid decarboxylase deficiency in three patient cases from the Kingdom of Saudi Arabia
Authors: Musaad Abukhaled, Laila Alrakaf, Hesham Aldhalaan, Suad Al Yamani
Journal: Front Pediatr (2023)
Keywords: aadc deficiency, saudi arabia, case report, delayed diagnosis, developmental delay, hypotonia, oculogyric crises, seizures
MeSH terms: Case Reports

Aromatic L-amino acid decarboxylase (AADC) deficiency is an ultra-rare and often severe neurometabolic disorder resulting from variants in the dopa decarboxylase (DDC) gene. A timely diagnosis is critical to prevent secondary complications, promote development, and optimize outcomes from future innovative treatment options, such as gene therapy. This article describes three patients with AADC deficiency managed in the Kingdom of Saudi Arabia (KSA). All three patients had homozygous variants within the DDC gene, including one novel gene variant (c.245G > A, p.Arg82Glu), and presented with symptoms from birth. In all cases, a diagnostic delay was observed owing to non-specific signs and symptoms, a lack of disease awareness among primary care physicians, and delays associated with outsourcing of genetic tests. All three patients were managed by a multidisciplinary team at a specialist tertiary center. Clinical outcomes for all three cases were poor, with one patient passing away at 3 years of age and the other two patients continuing to experience substantial disability and poor quality of life. There is an urgent need to raise awareness and improve diagnostic testing for rare diseases such as AADC deficiency in the KSA in order to improve outcomes, particularly as innovative disease-targeting therapies become available.
High Efficacy of Preoperative Low-Dose Radiotherapy with Sanazole (AK-2123) for Extraskeletal Ewing's Sarcoma: A Case Report
Authors: Tomoya Sakabe, Hiroaki Murata, Eiichi Konishi, Kazutaka Koto, Naoyuki Horie, Takaaki Matsui, Yasushi Sawai, Hideya Yamazaki, Tsutomu V Kagiya, Toshikazu Kubo
Journal: Sarcoma (2011)
MeSH terms: Case Reports

Extraskeletal Ewing's sarcoma is a rare soft tissue tumor that is morphologically indistinguishable from Ewing's sarcoma of bone. We report a case of extraskeletal Ewing's sarcoma with several systemic problems. A 69-year-old man presented with a 5-month history of a rapidly enlarging mass in the right thigh. Because preoperative radiotherapy with sanazole (AK-2123) contributed the tumor mass reduction down to 40% in size, the tumor was successfully resected with clear surgical margins and repaired with a musculocutaneous flap. The high efficacy of pre-operative low-dose radiotherapy with sanazole was histologically confirmed that the resected tumor specimen involved no viable tumor cells and showed 100% necrosis. Based on clinical outcomes in this case, the combined modality of pre-operative low-dose radiotherapy with hypoxic cell radiosensitizer and adequate surgical resection might provide for the useful clinical application of extraskeletal Ewing's sarcoma treatment.
Three Metachronous Osteosarcomas within 22 Years without Pulmonary Metastases: A Case Report and Review of the Literature
Authors: Ulrike Michaela Pirker-Frühauf, Jörg Friesenbichler, Katharina Rabitsch, Bernadette Liegl-Atzwanger, Thomas Bauernhofer, Reinhard Windhager, Andreas Leithner
Journal: Case Rep Orthop (2013)

Introduction. We present the extremely rare case of a patient with three metachronous osteosarcomas within 22 years without evident pulmonary manifestation of disease 30 years after first diagnosis. Case Presentation. In 1983, a high-grade osteosarcoma of the left distal femur was diagnosed in an 18-year-old Caucasian male. He received rotationplasty accompanied by pre- and postoperative chemotherapy. Ten years later, an osteoblastic osteosarcoma occurred in TH12. En bloc resection and pre- and postoperative chemotherapy followed. In 2005, the patient developed another high-grade osteosarcoma in his right distal femur. Treatment included a wide resection and reconstruction with a tumour endoprosthesis as well as (neo)adjuvant chemotherapy. After the third tumour occurrence, cytogenetic and molecular genetic examinations (p53, rb1) were performed, showing a normal genetic pattern. Screening for metastases never showed clinical evidence of extraskeletal tumour manifestation. Discussion. In patients presenting metachronous osteosarcoma, identification of their lesions clonality (second primary tumour or metastases) could lead to a better understanding of tumour development and help to filter patients who need extended long-term followup due to a higher risk of late occurring sarcoma recurrence.
Intraosseous ganglion cyst of the lunate
Authors: Shahram Nazerani, Adel Ebrahimpour, Arvin Najafi, Ehsan Shams Koushki
Journal: Trauma Mon (2012)
Keywords: bone cysts, ganglion cysts, lunate bone, wrist
MeSH terms: Case Reports

Intraosseous ganglia can affect the carpal bones of the hand and must be considered in the differential diagnosis of wrist pain. A 38-year-old female presented with a 14-month history of left wrist pain and a radiolucent cystic lesion was seen computed tomography (CT) scanning. Characteristic radiographic findings of a cyst in association with a fine sclerotic rim was apparent. We report an unusual presentation of a ganglion cyst in the lunate bone with excellent treatment outcome.
Simultaneous surgical treatment tactics of acute destructive cholecystitis combined with choledocholithiasis: A case report
Authors: Dauren T Zhumatayev, Аbilay N Baimakhanov, Mazhit K Abdykadyrov, Dauren A Nurmakov, Aidar D Raimkhanov, Аlibek M Smagulov, Nurken M Abdiyev
Journal: Int J Surg Case Rep (2020)
Keywords: case report, cholecystitis, cholelithiasis, endoscopic papillosphincterotomy, laparoscopic cholecystectomy
MeSH terms: Case Reports

Introduction:
The widespread development of minimally invasive treatment methods have expanded the choices and options available to surgeons to manage patients suffering from cholelithiasis and its complications, including choledocholithiasis. As new surgical strategies are developed and become widely available, there is an ongoing debate as to which surgical strategies are optimal for the management of acute cholecystitis and concomitant choledocholithiasis. The treatment of patients in this category should be carried out according to clear criteria, taking into consideration the patient's condition, concomitant diseases, the size of calculi in the common bile duct as well as the resources of the medical institution.
Case presentation:
We present the clinical case of a 65-year-old female with cholelithiasis, acute gangrenous cholecystitis, choledocholithiasis, and obstructive jaundice. She was treated with endoscopic retrograde cholangiopancreatography (ERCP) with endoscopic papillosphincterotomy (EPST) and laparoscopic cholecystectomy (LCE) in a one-stage operation.
Discussion:
Several sources recommended performing LCE after ERCP with EPST in acute cholecystitis when complicated by choledocholithiasis to reduce the conversion rate and the risk of recholedocholithiasis.
Conclusion:
Simultaneous resolution of acute destructive cholecystitis complicated by choledocholithiasis in a one-stage operation is accompanied by a decrease in emotional and psychological trauma, also leads to early rehabilitation of patients and, thereby, reduces overall postoperative disability and morbidity.