OpenMed/multicare-articles huggingface dataset:
Osteonecrosis of the distal tibia in systemic lupus erythematosus: A rare case report
Authors: Ihsan Oesman, Danarto Hari Adhimukti
Journal: Int J Surg Case Rep (2020)
Keywords: distal tibia, osteonecrosis, systemic lupus erythematosus
MeSH terms: Case Reports

Introduction:
Osteonecrosis (ON) is characterized by cellular death of bone components due to interruption of blood supply that leads to bone ischemia and potential joint destruction. There are multiple risk factors and medical condition associated with ON, including systemic lupus erythematosus (SLE). The most common sites of ON are the femoral head, distal femur, proximal humerus, talus and lumbar spine. Very few cases of nontraumatic ON in distal tibia have been reported in the literature.
Case illustration:
We present a case of 23-year-old female diagnosed with osteonecrosis of distal tibia and history of SLE. The patient also had history of avascular necrosis of right hip and underwent right total hip arthroplasty. We treated the patients with conservative treatment for intial management.
Discussion:
The risk of ON in SLE patients is likely due to the results of both the SLE itself and use of corticosteroids. Systemic inflammation in SLE reduces the development of osteoblasts, increases osteoclast maturation and activity and increases protohrombotic agents that can lead to rapid bone loss. Corticosteroids are the most consistent risk factor associated with the development of ON in SLE. Conservative medical management is effective in the early stages of the disease before bone collapse.
Conclusion:
Despite advances in the diagnosis and treatment of SLE, symptomatic ON continues to be a significant comorbidity. Strategies to detect and manage early stage ON is necessary to prevent the progression of this serious complication.
Massive gastrointestinal haemorrhage caused by pancreatic pseudocyst complicated with Dieulafoy's disease in a child: A case report and review of the literature
Authors: Lintao Liu, Lichao Zhang, Xiaoli Zhu, Meng Li, Juan Cao, Likang Ji, Xiaoyang Qi, Weili Xu
Journal: Front Pediatr (2022)
Keywords: child, injury, massive gastrointestinal haemorrhage, pancreatic pseudocyst, stomach dieulafoy's disease
MeSH terms: Case Reports

Background:
Pancreatic pseudocyst (PPC) with massive gastrointestinal bleeding is rare, especially in children. Inadvertent intraoperative examination and damage to the gastric mucosa and malformed blood vessels by the fluid content of PPC can lead to massive bleeding, which may endanger the patient's life.
Case presentation:
Here, we present a case of an 8-year-old boy who was diagnosed with a massive gastrointestinal haemorrhage caused by PPC complicated with Dieulafoy's disease. At his first admission, his complaint was being hit to the stomach by the handlebar while riding bicycle 24 h before admission. After being hospitalized, he was diagnosed with pancreatic injury by abdominal CT. Conservative treatment lasted for 1 month in the Department of Pediatric Surgery. Then, a pancreatic pseudocyst was formed. Under the guidance of ultrasonic endoscopy, cyst puncture and drainage of pseudocysts through the gastric wall were performed. Unexplained hematemesis occurred 8 days after surgery. Emergency gastroscopy was performed, and abnormal submucosal vascular haemorrhage was found at the gastric fundus. Gastric Dieulafoy's disease was diagnosed. The boy underwent gastroscopic titanium clipping of abnormal arteries. He had no complications during the 3-month follow-up. Then, the patient returned to the hospital, and the stent was removed under endoscopy. No bleeding was found, and the patient was discharged. The patient recovered smoothly and was followed up for half a year without any complications, and hematological indicators were normal.
Conclusion:
Endoscopic ultrasonography-guided gastric puncture and internal drainage of cysts is a safe and effective surgical method for the treatment of pancreatic pseudocysts. However, at the same time, it is necessary to thoroughly and carefully explore the stomach cavity to prevent adverse consequences caused by a missed diagnosis of gastric Dieulafoy's disease or other abnormal abnormalities.
Neuromuscular Coordination Deficit Persists 12 Months after ACL Reconstruction But Can Be Modulated by 6 Weeks of Kettlebell Training: A Case Study in Women's Elite Soccer
Authors: Mette K Zebis, Christoffer H Andersen, Jesper Bencke, Christina Ørntoft, Connie Linnebjerg, Per Hölmich, Kristian Thorborg, Per Aagaard, Lars L Andersen
Journal: Case Rep Orthop (2017)
MeSH terms: Case Reports

The aim of the present single-case study was to investigate the effect of 6 weeks' kettlebell training on the neuromuscular risk profile for ACL injury in a high-risk athlete returning to sport after ACL reconstruction. A female elite soccer player (age 21 years) with no previous history of ACL injury went through neuromuscular screening as measured by EMG preactivity of vastus lateralis and semitendinosus during a standardized sidecutting maneuver. Subsequently, the player experienced a noncontact ACL injury. The player was screened again following postreconstruction rehabilitation, then underwent 6-week kettlebell training, and was subsequently screened again at 6-week follow-up. Prior to and after postreconstruction rehabilitation the player demonstrated a neuromuscular profile during sidecutting known to increase the risk for noncontact ACL injury, that is, reduced EMG preactivity for semitendinosus and elevated EMG preactivity for vastus lateralis. Subsequently, the 6-week kettlebell training increased semitendinosus muscle preactivity during sidecutting by 38 percentage points to a level equivalent to a neuromuscular low-risk profile. An ACL rehabilitated female athlete with a high-risk neuromuscular profile changed to low-risk in response to 6 weeks of kettlebell training. Thus, short-term kettlebell exercise with documented high levels of medial hamstring activation was found to transfer into high medial hamstring preactivation during a sidecutting maneuver.
Case Report: Mucous Membrane Pemphigoid With IgG and IgA Anti-Laminin γ1 Antibodies and IgA Anti-Laminin α5 Antibodies
Authors: Wenjing Kuang, Hua Qian, Qiyue Zhang, Wei Li, Takashi Hashimoto, Xin Zeng, Xiaoguang Li
Journal: Front Immunol (2022)
Keywords: autoantibody, laminin α5, laminin γ1, mucous membrane pemphigoid, oral lesion
MeSH terms: Pemphigoid, Benign Mucous Membrane / diagnosis, Pemphigoid, Bullous, Skin Diseases, Vesiculobullous, Case Reports, Research Support, Non-U.S. Gov't, Autoantibodies, Autoantigens, Humans, Immunoglobulin A, Immunoglobulin G, Autoantibodies, Autoantigens, Immunoglobulin A, Immunoglobulin G

Mucous membrane pemphigoid (MMP) and anti-laminin (LM) γ1 pemphigoid, two subtypes of subepidermal autoimmune bullous diseases characterized by autoantibodies against epidermal basement membrane zone proteins, mainly show mucosal and skin lesions, respectively. The known autoantigens of MMP includes BP180, BP230, LM332, integrin α6β4 and type VII collagen, and anti-LMγ1 pemphigoid targets LMγ1. In this study, we present an unique MMP case with oral mucosal lesions, which showed positive IgA signals on basement membrane zone in indirect immunofluorescence using normal human skin and on dermal side in indirect immunofluorescence using salt-split skin, positive IgA autoantibodies against LMγ1 by immunoblotting of epidermal extracts, positive IgA autoantibodies against LMα5 by immunoblotting of LM521 recombinant protein (rLM521) and positive IgG autoantibodies against LMγ1 by immunoblotting of rLM111 and rLM521 at first visit (Day 0). After therapy, further serological analyses of serum samples collected at Day 30 and Day 50 indicated that IgA autoantibodies against LMγ1 were likely to be pathogenic. These results suggest that LMγ1 is another autoantigen of MMP, and our patient might be the first reported case of anti-LMγ1 MMP.
Giant Lipoma Presents from the Buccal Vestibule
Authors: Yuki Sakamoto, Gohei Oyama
Journal: Case Rep Dent (2020)
MeSH terms: Case Reports

A lipoma is a benign tumor, where the parenchyma is composed of adipose tissue. Lipomas comprise 0.1%-5% of all benign tumors in the oral cavity. A 43-year-old man, with a known masticatory disorder, visited the Department of Oral Surgery at our hospital because of a facial swelling. The swelling, which had worsened over 15 years, was not painful, but the patient had discomfort while eating and talking. A detailed examination revealed a smooth, elastic, soft, circular, and yellowish pink pedunculated tumor-like lesion, with a diameter of approximately 40 mm, on the right buccal mucosa. An oval-shaped neoplastic lesion with a well-defined border of 40 mm × 20 mm was noted on MRI. Based on these results, the lesion was characterized as being a benign lipoma. The tumor was resected under local anesthesia. The pedunculated tumor was excised with an electric knife, and the wound was sutured. No trismus or paresthesia was noted on the postoperative follow-up.
Delayed diagnosis of celiac stenosis causing hepatic transplant ischaemic necrosis: diagnosis by spectral Doppler findings
Authors: Mark Sun, R Brooke Jeffrey, Michael A DiMaio, Eric W Olcott
Journal: BJR Case Rep (2016)
MeSH terms: Case Reports

Following presentation with abnormal liver function enzymes, confusion and fatigue, a 65-year-old male with alcoholic cirrhosis underwent spectral Doppler sonography that showed tardus parvus-like morphology in the main and left hepatic arteries, although peak systolic velocities and resistive indices remained normal. The patient's continuing clinical symptoms prompted CT angiography, which demonstrated an unexpected, haemodynamically significant stenosis of the celiac artery. Although the stenosis was successfully stented and the hepatic arterial waveforms normalized, the transplanted liver had already undergone ischaemic necrosis, with resulting failure and the need for retransplantation. Recognition of abnormal waveforms, despite normal peak systolic velocities and resistive indices, with prompt definitive imaging evaluation of the arterial tree beyond just the main hepatic artery, may lead to the diagnosis of unexpected flow-limiting lesions in time to allow revascularization and thus prevent ischaemic transplant failure.
A Case of Common Variable Immunodeficiency with CREBP Gene Mutation without Rubinstein Taybi Syndrome Features
Authors: Ugur Musabak, Serdar Ceylaner, Tuba Erdogan, Ebru Sebnem Ayva
Journal: Case Reports Immunol (2022)
MeSH terms: Case Reports

Hypogammaglobulinemias, based on inborn errors of immunity, are primary immunodeficiencies (PIDs) that can also be diagnosed for the first time in adulthood. Common variable immunodeficiency (CVID) is a multifactorial disease often symptomatic due to antibody deficiency. In addition, some PIDs are classified into the category of immunodeficiencies with syndromic features due to their accompanying clinical findings unrelated to immunity. In this article, a patient with CVID who was diagnosed in adulthood and who was revealed to have a mutation specific to Rubinstein-Taybi syndrome and clinical features reminiscent of this syndrome only after molecular tests was presented.
Mechanisms Involving Myocardial Injury in Tropical Stings and Bites
Authors: Thirunavukarasu Kumanan, Mahesan Guruparan, Ratnasamy Vithiya, Indika Gawarammana
Journal: Case Rep Emerg Med (2017)
MeSH terms: Case Reports

It is known that a number of toxic substances produce myocardial injury by several mechanisms involving interruption of coronary blood flow due to stimulation of clotting mechanism and coronary vasospasm. Number of toxic substances may cause direct myocardial toxicity independent of coronary blood flow. Acute myocardial injury due to stings and bites is a rare entity and not well understood. Here we illustrate a case of myocardial injury due to Russell's viper envenomation.
Uterine Rupture on MRI Presenting as Nonspecific Abdominal Pain in a Primigravid Patient with 28-Week Twins Resulting in Normal Neurodevelopmental Outcomes at Age Two
Authors: Kathryn L Ponder, Rosa Won, Laurel Clymer
Journal: Case Rep Obstet Gynecol (2019)
MeSH terms: Case Reports

Background:
Uterine rupture is a rare occurrence that requires a high index of suspicion, particularly in a primigravid patient who presents prior to the onset of labor. Mortality rates are particularly high in primigravid patients.
Case:
A 36-year-old gravida 1, para 0 patient with dichorionic diamniotic twins presented at 28-weeks of gestation with abdominal pain. The pain was initially intermittent and felt to be musculoskeletal in origin. Ultrasound imaging after 3 days of worsening abdominal pain revealed extrauterine fluid, prompting an urgent MRI. MRI diagnosed the uterine rupture with hemoperitoneum and herniation of both amniotic sacs outside of the uterus, including one twin's torso and extremities, prompting emergency cesarean section. The premature twins required 2-month hospitalizations and had no neurodevelopmental impairments at 2-year follow-up.
Conclusion:
We present a unique case of rupture of an unscarred uterus in a primigravid patient prior to the onset of labor. Multiple gestation is a risk factor. This report adds to a handful of cases in which a history of endometriosis or extrauterine pelvic surgery was also present. The use of ultrasound and MRI to evaluate nonspecific abdominal pain led to the diagnosis and survival of both the mother and her premature twins.
Rare Case of Multiple Perirenal, Extra-Adrenal Myelolipoma: Case Report, Current Management Options, and Literature Review
Authors: Goran Benko, Andrina Kopjar, Marin Plantak, Danijel Cvetko, Vicko Glunčić, Anita Lukić
Journal: Case Rep Urol (2021)
MeSH terms: Case Reports

Extra-adrenal myelolipomas are rare, asymptomatic entities, although large tumors may cause local symptoms or hemorrhage. When these lesions occur outside the adrenals in the retroperitoneum, they are radiographically easily confused with both primary and secondary retroperitoneal tumors, which tend to be aggressive. Although myelolipomas are benign and can be managed conservatively, if malignancy is suspected, a surgical procedure is an option. We report a case of a 68-year-old patient with multiple perirenal extra-adrenal myelolipomas. Initial abdominal ultrasound reviled an inhomogeneous mass surrounding the left kidney. Subsequent CT examination of the abdomen showed four separate, extrarenal, well-circumscribed, round-shaped, fat-containing retroperitoneal tumors. Given the significant size of the masses, that compressed major abdominal vessels and the suspicion of liposarcoma, a surgical excision of the lesions was performed. The tumors were easily separated, all surrounding structures were spared, and they were removed completely. Histologically, all masses consisted of hematopoietic and mature fat tissue and the final diagnosis was extra-adrenal myelolipoma. The patient was released from the hospital 7th day after surgery in good condition and at his baseline. Since myelolipomas are, by definition, nonfunctional benign tumors, there was no need for further follow-up. The radiological evaluation and fine needle biopsy are usually sufficient to establish the diagnosis, but in some cases of well-differentiated liposarcoma, the differentiation between myelolipoma and liposarcoma can be challenging. Therefore, considering that myelolipomas and liposarcomas have opposite prognoses, which affects the surgeon's decision on the extent of surgical procedure and further treatment, we also emphasize the importance of intraoperative assessment of the tumor, both by the surgeon and by intraoperative pathology consultation.
Double-lumen catheter in the right jugular vein induces two sub-endothelial abscesses in an unusual place, the transition between the superior vena cava and the right atrium: a case report
Authors: João Kennedy Teixeira Lima, Sylvia Rannyelle Teixeira Lima, Antonio Leonel de Lima Jr, Cícero Valdizébio Pereira Agra, Vitor Engrácia Valenti, Rayana Loch Gomes, Luciano Miller Rodrigues, João Antonio Correa, Rodrigo D Raimundo, Luiz Carlos de Abreu
Journal: Int Arch Med (2014)
Keywords: central venous catheters, endocarditis, hospitalisation, renal dialysis
MeSH terms: Case Reports

Endocarditis is a type of infection that is common in internal medicine wards and in haemodialysis clinics. The location that is most affected are the heart valves. Herein, we report a case of an uncommon abscess, a sub-endothelial abscess between the transition of the superior vena cava and the right atrium. There were several emboli to the lung and foot, and the agent was related to Staphylococcus aureus and a double-lumen catheter. Usually, this type of abscess is located in valves, either the tricuspid valve if related to catheters or injection drug use or the mitral valve if related to other causes. An exhaustive review was made, but we found no information about the location of this abscess and the rarity of the event motivating the report of infection.
Metastatic Renal Cell Carcinoma to the Testis: A Clinicopathologic Analysis of Five Cases
Authors: Gang Wang, Chen Zhou, Carlos F Villamil, Alan So, Ren Yuan, John C English, Edward C Jones
Journal: Case Rep Pathol (2020)
MeSH terms: Case Reports

The testicular spread of renal cell carcinoma is extremely rare. Five cases of renal cell carcinoma metastatic to the testis are described. The patients ranged from 45 to 81 years of age. Four of the five patients had known renal cell carcinoma. The time intervals between the partial and radical nephrectomies for the primary kidney tumors and the occurrence of testicular metastases ranged from 29 to 34 months. In one patient, the testicular mass was the initial presentation leading to a diagnosis of renal cell carcinoma. There were three ipsilateral metastases, one contralateral metastasis, and one bilateral metastasis. The metastatic deposits ranged in size from 2.0 to 5.7 cm. One case had multiple metastatic tumor nodules. All of the metastatic tumors had clear cell histological features, microscopically concordant with the primary renal cell carcinoma subtype. Three patients died of the disease 17 to 42 months after orchiectomy. One patient is alive with additional metastatic lesions 13 months after orchiectomy. One patient had been free of disease at 87 months after orchiectomy but is now on targeted therapy for an additional metastasis at 93 months after orchiectomy. To date, this report is one of the largest single series of patients with renal cell carcinoma metastatic to the testis, and it has the longest follow-up and survival among all the reported cases.
Unanticipated diagnosis of skeletal muscle Kaposi sarcoma: a case report
Authors: Harshini Udayakumar, Venkatraman Indiran, Prabhakaran Madurai Muthu
Journal: Pan Afr Med J (2020)
Keywords: kaposi sarcoma, atypical kaposi sarcoma, skeletal muscle sarcoma
MeSH terms: Muscle Neoplasms / diagnostic imaging, Muscle, Skeletal / diagnostic imaging, Sarcoma, Kaposi / diagnostic imaging, Case Reports, Aged, Humans, Male, Muscle Neoplasms / pathology, Muscle, Skeletal / pathology, Sarcoma, Kaposi / pathology

Kaposi sarcoma (KS) is a cancer, characteristically manifesting as red or purple patches of abnormal tissue growing subcutaneously around the mouth, nose, and throat. Primary musculoskeletal KS is a never reported as skeletal muscles sarcomas are first differentials. Pertaining to the musculoskeletal system complicity of KS, African and classic KS lesions are inclined to manifest lesion in the peripheral skeleton. On the other hand AIDS-related KS routinely involves the maxillofacial bones and/or axial skeleton. KS distinguishably involves the tempo-parietal bones, paranasal sinus, hands and feet and other facial bones. Asymmetric involvement of the bones by KS is the rule. Though reported, involvement of the joints in KS is unusual. Skeletal muscle involvement has only sparingly been reported in AIDS-related KS patient. A primary KS of the skeletal muscle in an otherwise normal patient with no skin manifestations has never been reported thus far. The occurrence of KS in any atypical site may pose as difficulty to diagnose it. It is important for the radiologist to acquaintance with the spectrum of imaging manifestations of KS in various affected organs. Particularly in asymptomatic patients, lesions go unrecognized on routine imaging studies (e.g. KS on plain x-ray films) and clinicians are unwary of their existence. Awareness that KS can occur in any of these unusual locations may help avoid potential misdiagnosis with serious consequences (e.g. spinal cord compression) and/or mis-management.
A prenatal case with discrepant findings between non-invasive prenatal testing and fetal genetic testings
Authors: Qiong Pan, Baojuan Sun, Xiaoli Huang, Xin Jing, Hailiang Liu, Fuman Jiang, Jie Zhou, Mengmeng Lin, Hongni Yue, Ping Hu, Ying Ning
Journal: Mol Cytogenet (2014)
Keywords: massively parallel sequencing, mosaicism, non-invasive prenatal testing
MeSH terms: Case Reports

At 17(+4) week, non-invasive prenatal testing (NIPT) results of a 24-years-old mother showed high risk of monosomy X (45, X). Abnormally shaped head and cardiac defects were observed in prenatal ultrasound scan at 19(+3) week. Amniocentesis conducted at 19(+3) week identified karyotype 47, XX, +18, which suggested that the NIPT failed to detect trisomy 18 (T18) in this case. With a further massively parallel sequencing (MPS) of maternal blood, fetal and placental tissues, we found a confined placental mosaicism (CPM) with non-mosaic T18 fetus and multiclonal placenta with high prevalence of 45, X and low level of T18 cells. FISH and SNP-array evidence from the placental tissue confirmed genetic discrepancy between the fetus and placenta. Because the primary source of the fetal cell-free DNA that NIPT assesses is mostly originated from trophoblast cells, the level of T18 placental mosaicism may cause false negative NIPT result in this rare case of double aneuploidy.
Osteomalacia as a late metabolic complication of Ifosfamide chemotherapy in young adults: illustrative cases and review of the literature
Authors: D N Church, A B Hassan, S J Harper, C J Wakeley, C G A Price
Journal: Sarcoma (2007)
MeSH terms: Case Reports

Purpose. Ifosfamide is a drug commonly used in the management of sarcomas and other solid tumours. One potential toxicity of its use is renal tubular damage, which can lead to skeletal abnormalities; rickets in children and osteomalacia in adults. We aimed to characterise this rare complication in adults. Patients. Three illustrative patient cases treated in our institution are presented. All were treated for sarcoma, and received varying doses of ifosfamide during their therapy. Methods. We performed a review of the literature on the renal tubular and skeletal complications of ifosfamide in adults. Papers were identified by searches of PubMed using the terms "osteomalacia," "nephrotoxicity," "Fanconi syndrome," "ifosfamide," and "chemotherapy" for articles published between 1970 and 2006. Additional papers were identified from review of references of relevant articles. Results. There are only four case reports of skeletal toxicity secondary to ifosfamide in adults; the majority of data refer to children. Risk factors for development of renal tubular dysfunction and osteodystrophy include platinum chemotherapy, increasing cumulative ifosfamide dose, and reduced nephron mass. The natural history of ifosfamide-induced renal damage is variable, dysfunction may not become apparent until some months after treatment, and may improve or worsen with time. Discussion. Ifosfamide-induced osteomalacia is seldom described in adults. Clinicians should be vigilant for its development, as timely intervention may minimise complications.