OpenMed/multicare-articles huggingface dataset:
Imeglimin Improved Plasma Glucose Levels in Patients With Latent Autoimmune Diabetes of Adults: Report of 2 Cases
Authors: Shuichi Okada, Kazuya Okada, Junichi Okada, Eijiro Yamada
Journal: JCEM Case Rep (2023)
Keywords: diabetic ketoacidosis, glutamic acid decarboxylase antibody, glycated hemoglobin, imeglimin, latent autoimmune diabetes of adults
MeSH terms: Case Reports

Imeglimin has not been well studied as an oral agent for the treatment of latent autoimmune diabetes of adults (LADA). We treated 2 cases of LADA with imeglimin. The case 1 patient was originally diagnosed with type 2 diabetes (T2D) at age 50 years and was treated with sulfonylurea, biguanide, canagliflozin, imeglimin, and dulaglutide. Before imeglimin, his glycated hemoglobin A1c (HbA1c) change was 94.0 mmol/mol (8.6%) in November 2022, but it dropped to 71.0 mmol/mol (6.5%) in May 2023 after imeglimin was added. The case 2 patient was originally diagnosed with T2D when she was aged 48 years. She was treated with vildagliptin, biguanide, luseogliflozin, and imeglimin. Her HbA1c before imeglimin was 92.9 mmol/mol (8.5%) in January 2023, which decreased to 75.4 mmol/mol (6.9%) in July 2023 after imeglimin was added. Although imeglimin has not been approved for treating type 1 diabetes and LADA, adding imeglimin to the current medication was effective in improving and controlling the patients' plasma glucose.
Beta-HCG Secretion by a Pulmonary Choriocarcinoma in a Male Patient
Authors: Brecht Devos, Cedric Willemse, Mira Sterckx, Johan Debruyne, Inge Stappaerts, Tom Van den Mooter, Marjan Hertoghs, Pascale Abrams
Journal: Case Rep Oncol Med (2024)
MeSH terms: Case Reports

Background:
Paraneoplastic secretion of beta-subunit of human chorionic gonadotropin (β-HCG) in pulmonary carcinoma is rare. Case Presentation. A 65-year-old man presented with bilateral gynaecomastia with abnormally high levels of β-hCG and elevated oestradiol, progesterone, and testosterone levels on April 7, 2023. After excluding testicular malignancy, CT scan of the chest and abdomen revealed bilateral pulmonary lesions. Transthoracic biopsy confirmed malignancy with choriocarcinoma. MRI of the brain showed a solitary brain metastasis, while on a subsequent 18F-FDG PET/CT, no other metastatic lesions were seen. The patient was treated with chemoimmunotherapy carboplatin-etoposide-pembrolizumab with good partial response.
Conclusion:
Our case of a presumably stage IV dedifferentiated mNSCLC presenting as an extragonadal β-hCG secreting pulmonary choriocarcinoma is a very rare tumor with a poor prognosis. Its biology, origin, and treatment remain to be elucidated. Cancer genome sequencing is necessary for the identification of the origin and seeking treatment.
Minimally Invasive Beaded Electrosurgical Dissectors, Basic Science, and Pilot Studies
Authors: Taiyo C Weber, Mark Jewell, Carl I Schulman, Jefferson Morgan, Alison M Lee, Alicia K Olivier, Elizabeth A Swanson
Journal: Aesthet Surg J Open Forum (2024)
MeSH terms: Case Reports

Background:
Minimally invasive beaded electrosurgical dissectors ("BEED devices") provide simultaneous sharp dissection, blunt dissection, and electrosurgical coagulation while performing 100 cm2 porcine tissue plane dissections in 0.8 to 3 min with minimal bleeding and no perforations.
Objectives:
The aim of the study was to report the basic science and potential clinical applications and to video document the speed and quality of planar dissections in in vivo and ex vivo porcine models with thermal damage quantified by thermal and histopathologic measurements. Additionally, in vivo porcine specimens were followed for 90 days to show whether adverse events occurred on a gross or macroscopic basis, as evidenced by photography, videography, physical examination, and dual ultrasonography.
Methods:
Ex vivo porcine models were subjected to 20, 30, and 50 W in single-stroke passages with BEED dissectors (granted FDA 510(k) clearance (K233002)) with multichannel thermocouple, 3 s delay recordation combined with matching hematoxylin and eosin (H&E) histopathology. In vivo porcine models were subjected to eight 10 × 10 cm dissections in each of 2 subjects at 20, 30, and 50 W and evaluated periodically until 90 days, wherein histopathology for H&E, collagen, and elastin was taken plus standard and Doppler ultrasounds prior to euthanasia.
Results:
Five to 8 mm width dissectors were passed at 1 to 2 cm/s in ex vivo models (1-10 cm/s in vivo models) with an average temperature rise of 5°C at 50 W. Clinically evidenced seromas occurred in the undressed, unprotected wounds, and resolved well prior to 90 days, as documented by ultrasounds and histopathology.
Conclusions:
In vivo and ex vivo models demonstrated thermal values that were below levels known to damage subcutaneous adipose tissue or skin. Tissue histopathology confirmed healing parameters while Doppler ultrasound demonstrated normal blood flow in posttreatment tissues.
Urgent Cesarean Section in a Patient on Venovenous Extracorporeal Membrane Oxygenation
Authors: Daniela Havens-Lastarria, Sara K Biladeau, Daniel Haines, Ryan Grell
Journal: Cureus (2024)
Keywords: acute hypoxemic respiratory failure, cesarian section, complications and outcomes on gravid and post-partum patients on ecmo, delivery during ecmo support, placental abruption, vv ecmo
MeSH terms: Case Reports

A G7P6 40-year-old female at 20 weeks gestation, with a history of polysubstance use disorder and hepatitis C, presented to the emergency department with severe shortness of breath and hypoxia requiring intubation. After a thorough workup, she was diagnosed with aspiration pneumonitis and was treated with a course of antibiotics. After progressing well, she was soon extubated and transferred to a subacute rehabilitation facility (SAR). There, she acutely decompensated, requiring readmission, reintubation, and venovenous extracorporeal membrane oxygenation (ECMO) cannulation. After a brief period of improvement, the patient became increasingly unstable with hypotension, anemia, and downtrending fibrinogen. Bedside imaging indicated a possible placental abruption. After extensive discussion among the care teams and patient's healthcare proxy, an urgent cesarean section was performed. Although the fetus was determined to be nonviable, the patient tolerated the procedure well and was eventually decannulated from ECMO and transferred to a SAR.
Integrated cognitive behavioral intervention for functional tics (I-CBiT): case reports and treatment formulation
Authors: Amanda Maxwell, Jade-Jocelyne Zouki, Valsamma Eapen
Journal: Front Pediatr (2023)
Keywords: tourette syndrome, autism spectrum disorder (asd), functional movement disorders, functional tic like behavior, integrated intervention, premonitory urge
MeSH terms: Case Reports

Introduction:
The onset of the COVID-19 pandemic saw a global surge in functional tic-like behaviors (FTLBs). FTLBs are unique from primary tic disorders. They are thought to manifest through a complex interplay between environmental and personal factors, including the stress-arousal system, and are characterized by their sudden and explosive onset. Accordingly, common interventions for tic disorders show limited efficacy in this population. We present an Integrated Cognitive Behavioral Intervention for Functional Tics (I-CBiT) that uses an urge acceptance model to manage tics and related stress and anxiety.
Methods:
We describe the treatment outcomes of eight young people presenting with new and sudden onset FTLBs who underwent I-CBiT, which integrates traditional behavioral tic interventions with third-wave cognitive behavioral therapies. All cases completed the three-phase intervention involving core components of psychoeducation, exposure and response prevention with urge acceptance, sensory grounding strategies, and cognitive behavioral intervention targeting the stress-arousal system. Tic severity and impairment were assessed prior to treatment and at completion.
Results:
All cases showed a significant reduction in tic severity post I-CBiT and an improvement in overall daily living function. These cases highlight the role of urge acceptance in managing both tic urges and the underlying stress-arousal system to bring about long-term change.
Conclusion:
We demonstrated the efficacy of I-CBiT for managing FTLBs. Our findings illustrate the importance of treating underlying stress and anxiety in this population and, therefore, a need for greater interaction between multidisciplinary services in managing FTLBs to comprehensively cover the varied symptom presentations linked to thoughts, emotions, bodily sensations, and stress responses.
Case report: Olanzapine-associated water retention, high blood pressure, and subsequent preterm preeclampsia
Authors: Julia Izsak, Dimitra Falari, Pia Arnbert, Daniel Pouragheli, Jenny M Kindblom, Daina Lasaitiene
Journal: Front Psychiatry (2023)
Keywords: hypertension, olanzapine, preeclampsia, pregnancy, water retention
MeSH terms: Case Reports

Olanzapine is one of the most frequently used antipsychotic medications during pregnancy, but information about its safety and adverse effects profile during pregnancy is scarce. We herein describe a case of a pregnant woman with several psychiatric disorders who developed water retention, hypertension, and subsequent preterm preeclampsia 3 weeks after initiation of treatment with olanzapine. To the best of our knowledge, this is the first case of olanzapine-associated preeclampsia described in literature.
Bilateral spontaneous quadriceps tendon rupture: a case report and literature review
Authors: Mohammad Alkhatatba, Yazan Anaqreh, Suhaib Bani Essa, Ala'a Alma'aiteh, Hamzeh Ziad Audat, Naser Obeidat, Marwan Ahmed
Journal: SICOT J (2023)
Keywords: bilateral, diagnosis, quadriceps tendon rupture, rehabilitation, spontaneous, surgical repair

Bilateral spontaneous quadriceps tendon rupture is a rare condition characterized by the simultaneous tear of the fibrous tissue connecting the quadriceps muscle to the patella bone. Prompt diagnosis is crucial for appropriate treatment and optimal outcomes. We present a case of a 70-year-old male with bilateral knee pain and an inability to walk, resulting from a trivial fall. Despite initial misdiagnosis, a thorough evaluation, including physical examination and imaging, revealed bilateral quadriceps tendon rupture. Surgical repair was performed, followed by a comprehensive rehabilitation program. At the four-month follow-up, the patient showed significant improvement in pain and function. This article provides a comprehensive review of the existing literature on bilateral quadriceps tendon rupture, emphasizing the challenges in the diagnosis and management of this rare condition. Early diagnosis, prompt surgical intervention, and a tailored rehabilitation program are crucial for successful outcomes.
The Challenges of Distinguishing Different Causes of TMA in a Pregnant Kidney Transplant Recipient
Authors: A Krelle, S Price, M M Law, S Kranz, P Shamdasani, S Kane, J Unterscheider, P Champion de Crespigny
Journal: Case Rep Nephrol (2024)
MeSH terms: Case Reports

Thrombotic microangiopathy (TMA) reflects a syndrome of endothelial injury characterised by microangiopathic haemolytic anaemia (nonimmune), thrombocytopenia, and often end-organ dysfunction. TMA disorders are well-recognised in kidney transplant recipients, often due to an underlying genetic predisposition related to complement dysregulation, or de novo due to infection, immunosuppression toxicity, or antibody-mediated rejection. In pregnancy, TMA disorders are most commonly due to severe pre-eclampsia or HELLP, but may also be due to thrombotic thrombocytopenic purpura (TTP) or complement-mediated (atypical) haemolytic uremic syndrome (aHUS). Complement dysregulation is being recognised as playing a role in the development of preeclampsia and HELLP syndrome in addition to aHUS. Due to overlapping clinical and laboratory features, diagnosis can be difficult and delays in treatment can be life-threatening for both mother and fetus. This report describes a 32 year-old female who had two successive wanted pregnancies. The first pregnancy was terminated at 22 weeks gestation due to presumed severe preeclampsia and fetal growth restriction in the context of known chronic kidney failure due to reflux nephropathy. A living-related kidney transplant was performed to improve the chances of pregnancy resulting in a live birth. A subsequent pregnancy was complicated by progressive kidney impairment and hypertension at 22 weeks gestation. Kidney biopsy showed TMA, but the etiology was unclear. This report highlights the diagnostic dilemma of TMA in a pregnant kidney transplant recipient and a role for the anti-C5 terminal complement blockade monoclonal antibody eculizumab, in pregnancy-associated TMA, especially at a peri-viable gestation.
Case report: Interleukin-17 targeted biological therapy in netherton syndrome
Authors: Rahul Mahajan, Shirin Bakshi, Anoop Kumar, Dipankar De, Sanjeev Handa
Journal: Front Pediatr (2023)
Keywords: il-17, genodermatology, ichthyosis, netherton syndrome, secukinumab
MeSH terms: Case Reports

Netherton syndrome (NS) is rare and multisystemic congenital skin disorder classically distinguied as a triad of congenital ichthyosiform erythroderma, trichorrhexis invaginata (TI), and an atopic diathesis. Recent advances in pathogenesis have explored the role of IL-23/Th17 pathway in NS. Herein, we present a 17 years old girl harbouring homozygous four base pair deletion in exon 26 of the SPINK5 gene, presented with pruritus, scaling, dry skin and generalized eczematous lesions. She was administered anti IL17A (subcutaneous secukinumab) therapy. The treatment was well tolerated and resulted in a favourable clinical response, reduction of the IL17A gene expression and CD4 + Th17 cell population after 6 months which revealed an abrogation of Th17-skewing during therapy.
The Safety and Efficacy of Resolving Burr Entrapment by the Ping-Pong and Mother-in-Child Techniques
Authors: Cong Thanh Nguyen, Lam Truong Hoai, Duc Nguyen Hung, Minh Tran Duc
Journal: Cureus (2024)
Keywords: complex pci, complication of treatment, coronary artery intervention, coronary artery lesion (cal), rotablator
MeSH terms: Case Reports

Burr entrapment is a serious risk when performing rotational atherectomy on specific anatomical features of lesions such as tortuosity, calcification, and acute angulation. This occurrence, known as the Kokeshi phenomenon in Japanese, is caused by the burr's proximal section being unable to ablate while pulling back the burr, leaving the distal end of the burr covered in diamond crumbs capable of lesion ablation following rotation. There are reports of different approaches used to retrieve an entrapped rotablator burr. In this case, we demonstrate that the ping-pong and mother-in-child techniques, which use separate guide catheters to engage the same coronary artery wiring across the lesion afterward and deep engagement of guide extension catheter manual traction, are highly effective and secure methods for retrieval.
Case report: SAF-189s is a potent inhibitor in a lorlatinib-resistant NSCLC patient with acquired compound mutations ALK L1196M and D1203N
Authors: Na Li, Huihui Li, Ding Wang, Xiaoling Xu
Journal: Front Pharmacol (2023)
Keywords: alk d1203n, alk l1196m, saf-189s, lorlatinib resistance, treatment
MeSH terms: Case Reports

Acquired anaplastic lymphoma kinase (ALK) mutation is the major resistant mechanism to ALK tyrosine kinase inhibitors (TKIs) in non-small cell lung cancer (NSCLC) patients. At present, treatment options after acquiring secondary ALK mutations are still limited. Here, we report on a patient with metastatic ALK-rearranged NSCLC who was sequentially treated with ALK TKIs, from crizotinib to lorlatinib, and developed rare acquired compound ALK mutations (L1196M and D1203N) that confer resistance to lorlatinib. Moreover, our report describes the clinical response of an NSCLC patient with these compound mutations to multiple anti-tumor therapies. Among them, the patient was treated with SAF-189s 120 mg daily and had a stable disease lasting 3 months. Chemotherapy (pemetrexed-carboplatin) combined with bevacizumab was then administered. She achieved a partial response, which was maintained for 7 months as the best response. Since both SAF-189s and chemotherapy have shown a clear antitumor effect, they may be viable therapeutic options for these patients. Thus, our study can provide some reference in the treatment of NSCLC patients with ALK L1196M/D1203N compound mutations.
Congenital toxoplasmosis and audiological outcome: from a case series to a suggestion of patient-based schedule
Authors: S Salomè, R Malesci, V Delle Cave, A Amitrano, R Gammella, F Fanelli, E Capone, L Capasso, A R Fetoni, F Raimondi
Journal: Front Pediatr (2024)
Keywords: ct, snhl (sensorineural hearing loss), audiologic assessment, audiological outcome, congenital toxoplasmosis

Introduction:
Sensorineural hearing loss (SNHL) has been suggested to be possibly related to congenital toxoplasmosis (CT), although its prevalence varies from 0% to 26%. This variance appears to be dependent especially on early timing of treatment. However, the available data are based on outdated studies conducted on small groups of patients that lack homogeneity. Therefore, to establish evidence-based guidelines for audiologic monitoring in CT, we conducted a comprehensive evaluation of a large case series over a long period of time.
Patients and methods:
This is a single-center, retrospective cohort that enrolled all infants and children who were exposed in utero to Toxoplasma gondii and/or congenitally infected between September 1980 and December 2022. They underwent standard serial audiological evaluations to detect possible SNHL at an early stage. The first evaluation was performed during the initial assessment to define the onset of congenital toxoplasmosis, with another evaluation conducted at least at 12 months of life.
Results:
We collected data from 1,712 patients, and 183 (10.7%) were diagnosed with CT. Among these cases, 78 children (42.6%) presented with symptomatic CT at the onset, exhibiting ocular findings (21.1%), clinical cerebral manifestations (6.1%), and/or abnormal findings on neuroimaging (35.5%). Therapy was administrated at the onset in 164 patients (89.6%) with 115 of them starting treatment prior to 2.5 months of age (0-388, median 32.00 ± 92.352 days of life). Only one patient presented with SNHL at the onset, but this was apparently unrelated to CT. The median number of audiological assessments was 2.2 ± 1.543 (2-10). No patients developed any grade of delayed hearing loss, both in treated and untreated groups. The median age at last audiological evaluation was 2.3 ± 2.18 years (1-8), although the median follow-up period was 12.4 years (±6.3), ranging from 1 to 27 years.
Conclusions:
Based on these data, it appears that SNHL may be less frequent in CT than previously assumed. We recommend conducting an audiological assessment at the onset (within the first 2.5 months of life) to comprehensively define the type of CT onset, and then conducting another evaluation within 9 months of life.
A case of intra-abdominal gossypiboma diagnosed preoperatively by endoscopic ultrasound fine-needle-aspiration
Authors: Katsuyuki Dainaka, Yoshida Juichiro, Yutaka Inada, Akifumi Fukui, Takeshi Nishimura, Hideki Fujii, Naoya Tomatsuri, Yusuke Okuyama, Hideki Sato, Yoji Urata
Journal: DEN Open (2024)
Keywords: endoscopic ultrasound fine‐needle‐aspiration, gossypiboma, polarized light microscope, pseudosubmucosal tumor, retained surgical sponge
MeSH terms: Case Reports

Gossypiboma is an extremely rare adverse event occurring post-surgery, where surgical gauze is left within the body. If aseptically retained, it can lead to the formation of granulation tissue through chronic inflammation and adhesion with surrounding tissues, potentially persisting asymptomatically for many years. While diagnosis of this condition has been reported through various imaging modalities such as abdominal ultrasound and computed tomography, cases not presenting with typical findings are difficult for preoperative diagnosis, and instances where it is discovered postoperatively exist. Particularly when in contact with the gastrointestinal tract within the abdominal cavity, differentiation from submucosal tumors of the digestive tract becomes problematic. This report describes the imaging characteristics of endoscopic ultrasound and the usefulness of endoscopic ultrasound-fine-needle-aspiration for tissue diagnosis in the preoperative diagnosis of intra-abdominal gossypiboma.
Malignant phyllodes tumor of the breast with predominant osteosarcoma and chondrosarcomatous differentiation: a rare case report and review of literature
Authors: Wenfang Li, Qin Ou, Yingdong Li, Linlin Yuan Yuan
Journal: Front Oncol (2024)
Keywords: breast tumor, chondrosarcoma, malignant phyllodes tumors, osteosarcoma, thoracic oncology
MeSH terms: Case Reports

Background:
Phyllodes tumors (PTs), which account for less than 1% of mammary gland tumors, composed of both epithelial and stromal components. If a malignant heterologous component is encountered, PT is considered malignant. Malignant phyllodes tumors (MPTs) only account for 8% to 20% of PTs. We report a case of MPT with osteosarcoma and chondrosarcoma differentiation and review the literature to discuss the differential diagnosis and therapy.
Case presentation:
A 59-year-old Chinese woman come to our hospital because of a palpable mass she had had for 1 months in the left breast. Preoperative core needle biopsy (CNB) was performed on the left breast mass on January 11, 2023. Pathological diagnosis was malignant tumor, the specific type was not clear. Mastectomy and sentinel lymph node biopsy of the left breast was performed. No metastasis was found in 3 sentinel lymph nodes identified by carbon nanoparticles and methylene blue double staining. Heterologous osteosarcoma and chondrosarcomatous differentiation of phyllodes tumor were observed. Immunohistochemistry: spindle tumor cells ER(-), PR(-), HER-2(-), CK-pan(-), CK7(-), CK8(-), SOX10(-), S100(-), and MDM2(-), CK5/6(-), P63(-), P40(-) were all negative. CD34:(+), SATB2(+), P53(90% strong), CD68 (+), Ki-67(LI: about 60%). No ductal carcinoma in situ was found in the breast. Fluorescence in situ hybridization (FISH) indicated USP6 was negatively expressed on formalin-fixed, paraffin-embedded (FFPE) tissue sections.
Conclusion:
MPTs are rare, and heterologous differentiation in MPTs is exceedingly rare. It could be diagnosed by pathology when metaplastic carcinoma, primary osteosarcoma, or myositis ossificans were excluded. This case could help clinicians to improve the prognosis and treatment of this disease.