OpenMed/multicare-articles huggingface dataset:
Case report of severe constrictive perimyocarditis and ischemic hepatitis in a Crohn's disease patient upon infliximab-induced lupus-like syndrome
Authors: Simon Hirschmann, Sarah Fischer, Entcho Klenske, Katharina Dechant, Jörg H W Distler, Christoph Treutlein, Markus F Neurath, Raja Atreya
Journal: Therap Adv Gastroenterol (2021)
Keywords: crohn’s disease, anti-tnf antibody, case report, infliximab, lupus-like syndrome
MeSH terms: Case Reports

Anti-tumor necrosis factor (TNF) antibodies have become an indispensable part in the therapeutic landscape of treating inflammatory bowel disease (IBD) patients. Nevertheless, they can be associated with the occurrence of severe systemic side effects. Here, we report the case of a 23-year-old patient with ileocolonic Crohn's disease in endoscopic remission under ongoing anti-TNF infliximab therapy with occurrence of novel generalized arthralgia, pleuritic chest pain, and dyspnea. Clinical, laboratory, and imaging diagnostic workup in an extended clinical routine setting at the University Hospital of Erlangen, Germany, was used by a multidisciplinary team consisting of gastroenterologists, radiologists, cardiologists, and rheumatologists to investigate the underlying cause of the clinical symptoms in the patient. The results received using the aforementioned diagnostic setup led to the diagnosis of severe constrictive perimyocarditis due to infliximab-induced lupus-like syndrome with distinct ANA reactivity and elevated anti-dsDNA levels. Furthermore, pronounced ischemic hepatitis was diagnosed. Infliximab treatment was immediately stopped, and initiated corticosteroid pulse therapy only led to partial response as it had to be reduced due to pronounced psychiatric side effects. Persistent signs of pericarditis required additional ibuprofen therapy, which led to subsequent resolution of cardial symptoms. Formerly elevated liver enzymes returned to normal, and there were no clinical signs of recurrence of Crohn's disease activity over 18 months of follow-up. The patient was subsequently switched to ustekinumab therapy for further treatment of underlying Crohn's disease. This case report describes for the first time severe infliximab-induced lupus-like syndrome in an IBD patient, concurrently mimicking ST-elevation myocardial infarction with MRI visualization of pericarditis, occurrence of ischemic hepatitis, and pronounced signs of systemic inflammation.
Appendicitis in De Garengeot's Hernia Presenting as a Nontender Inguinal Mass: Case Report and Review of the Literature
Authors: K Ahmed, K Bashar, T J McHugh, S M McHugh, E Kavanagh
Journal: Case Rep Surg (2014)

Introduction. De Garengeot first described a femoral hernia containing the appendix in 1731. Appendicitis occurring in this rare clinical setting represents a challenge in diagnosis and management. Case Presentation. We present the case of a 71-year-old male with a three-day history of a nontender inguinal mass. Computed tomography (CT) suggested a femoral hernia. Intraoperatively, the hernia sac was noted to contain a perforated appendix. Discussion. This is an infrequently reported clinical scenario and only the 14th reported case in peer-reviewed literature which includes preoperative CT images. Our case adds to previously reported low sensitivity of CT for diagnosing De Garengeot's hernia. Furthermore, unlike our case the vast majority of previous reports noted a painful inguinal swelling. Conclusion. Perforated appendicitis in a femoral hernia is an extremely uncommon presentation. However, consideration should be given to De Garengeot's hernia in patients with a groin mass, even if nontender.
Novel TTN Mutation Causing Severe Congenital Myopathy and Uncertain Association with Infantile Hydrocephalus
Authors: Palanikumar Balasundaram, Indirapriya Darshini Avulakunta, Leslie Delfiner, Paul Levy, Katie R Forman
Journal: Case Rep Genet (2023)
MeSH terms: Case Reports

Arthrogryposis multiplex congenita (AMC) is characterized by nonprogressive symmetric contractures of multiple joints with normal intellect and normal systemic examination. AMC is often due to fetal akinesia, which has neurologic, muscular, and connective tissue etiologies. We present a case of AMC due to a variant in the titin (TTN) gene in a term neonate. The infant is homozygous for this variant, c.38442dup, which is predicted to result in a truncated protein (p.Pro12815Thr fs∗37, NM_001267550.2). A literature search (PubMed) failed to find reports of this TTN variant. The variant was classified as pathogenic and submitted to ClinVar. Titin is the body's largest protein, expressed in skeletal and cardiac muscles and encoded by the TTN gene. Due to its large size (364 exons), the TTN gene has been difficult to sequence; the number of variants in the TTN gene and the spectrum of titinopathies are probably underestimated.
Penile degloving injury in an adolescent with congenital hypothyroid
Authors: Chauniqua Kiffin, Matthew Porcelli, Oksana Prychyna, Byron Pazmino, Daniel Pust, Joseph Decostanza
Journal: Case Rep Med (2012)
MeSH terms: Case Reports

This case follows a 17-year-old boy with congenital hypothyroidism who sustained penile skin avulsion secondary to a dog bite. Initially, an attempt was made to repair the wound using the avulsed skin flap itself as coverage. The repair was done immediately upon presentation to enhance the chances of adequate flap viability; however, the dorsal portion of the reconstruction necrosed within the following week. Ultimately a full thickness surgical graft (FTSG) repair was performed to ensure the most desirable outcome.
Unusual case of cavitary lung metastasis from squamous cell carcinoma of the uterine cervix
Authors: Soundouss Raissouni, Rais Ghizlane, Houda Mouzount, Kharmoum Saoussane, Setti Khadija, Fouad Zouaidia, Rachida Latib, Hind Mrabti, Hassan Errihani
Journal: Pan Afr Med J (2013)
Keywords: cavitary lung metastasis, squamous cell carcinoma, uterine cervix
MeSH terms: Carcinoma, Squamous Cell / secondary, Lung Neoplasms / secondary, Uterine Neoplasms / pathology, Case Reports, Adult, Carcinoma, Squamous Cell / diagnosis, Female, Humans, Lung Neoplasms / diagnosis

Spontaneous excavation of primary lung cancer is common; however cavitation of metastatic lung lesions is rare and usually confused with benign lesions. In Moroccan context tuberculosis is the first suspected diagnosis of lung excavations. We report a rare case of cavitary lung metastasis of a uterine cervix cancer, treated initially as tuberculosis. A 40-year old non-smoking woman with a known history of squamous cell carcinoma of the uterine cervix since August 2005; presented on September 2008 with right chest pain without fever, hemoptysis or weight loss. CT scan showed a thin walled cavity. Empirical Antibiotic therapy was conducted 15 days with poor outcome. Then antibacillary treatment was started with no proof of mycobacterial infection. A month later, the patient presented with gynecological bleeding and a pneumothorax. Bronchoscopy with transbronchial biopsy of the cavitary mass was performed. Pathology demonstrated a metastatic squamous cell carcinoma. Pelvic examination and MRI showed a subsequent local cervix recurrence. Patient underwent 3 courses of systemic chemotherapy. She died on June 2009 due to progressive disease. Even cavitary lung metastases are rare and benign differential diagnosis are more common, clinician should be careful in neoplastic context and investigation should be done to eliminate a recurrence.
Association Between Late-Onset Leukoencephalopathy With Vanishing White Matter and Compound Heterozygous EIF2B5 Gene Mutations: A Case Report and Review of the Literature
Authors: Fanxin Kong, Haotao Zheng, Xuan Liu, Songjun Lin, Jianjun Wang, Zhouke Guo
Journal: Front Neurol (2022)
Keywords: eif2b5, adult-onset, compound mutation, leukoencephalopathy with vanishing white matter, ovarian failure
MeSH terms: Case Reports

Leukoencephalopathy with vanishing white matter (LVWM) is an autosomal recessive disease. Ovarioleukodystrophy is defined as LVWM in females showing signs or symptoms of gradual ovarian failure. We present a 38-year-old female with ovarioleukodystrophy who showed status epilepticus, gait instability, slurred speech, abdominal tendon hyperreflexia, and ovarian failure. Abnormal EEG, characteristic magnetic resonance, and unreported EIF2B5 compound heterozygous mutations [c.1016G>A (p.R339Q) and c.1157G>A (p.G386D)] were found. Furthermore, the present report summarizes 20 female patients with adult-onset ovarioleukodystrophy and EIF2B5 gene mutations. In conclusion, a new genetic locus for LVWM was discovered. Compared with previous cases, mutations at different EIF2B5 sites might have different clinical manifestations and obvious clinical heterogeneity.
Acute Cardiac Failure due to Intra-Atrial Mass Caused by Zygomycetes in an Immunocompromised Paediatric Patient
Authors: G J van de Glind, C E M Gidding, C M W Verlaat, K Duthoi, A P C Backx, P E Verweij, A Warris
Journal: Case Rep Med (2010)
MeSH terms: Case Reports

Cardiac zygomycosis can be a critical condition with sudden onset of severe congestive heart failure followed by severe hemodynamic deterioration. We report a fatal course of disseminated fungal infection with a massive intra-atrial thrombosis caused by a zygomycete, in a five year old boy treated for acute lymphoblastic leukaemia. In addition, we discuss the literature concerning infections caused by zygomycetes involving the heart. Prognosis is poor. A high index of suspicion and an aggressive diagnostic and therapeutic approach with the prompt start of preemptive antifungal therapy are key factors to improve outcome.
Unusual presentation of uncommon disease: anorexia nervosa presenting as wernicke-korsakoff syndrome-a case report from southeast Asia
Authors: Raheel Mushtaq, Sheikh Shoib, Tabindah Shah, Mudasir Bhat, Randhir Singh, Sahil Mushtaq
Journal: Case Rep Psychiatry (2014)

Anorexia nervosa presenting as Wernicke-Korsakoff syndrome is rare. The causes of Wernicke-Korsakoff syndrome are multiple like alcohol abuse, thyrotoxicosis, haemodialysis, severe malnutrition because of gastric carcinoma and pyloric obstruction, hyperemesis gravidarum, and prolonged parenteral feeding. We report a case of anorexia nervosa, who presented with Wernicke's encephalopathy and progressed to Korsakoff's syndrome. Knowledge, awareness, and early intervention of anorexia nervosa by mental health professionals can prevent development of Wernicke-Korsakoff syndrome.
Spontaneous celiacomesenteric trunk dissection: Case report
Authors: Mabrouka Boukoucha, Abdelwahed Yahmadi, Hakim Znaidi, Raoudha Ben Khelifa, Alifa Daghfous
Journal: Int J Surg Case Rep (2020)
Keywords: acute abdominal, celiacomesenteric trunk, dissection, mesenteric ischaemia
MeSH terms: Case Reports

Introduction:
The celiacomesenteric trunk (CMT) is one of the most striking among the different variations of the normal vascularisation of the gastro-intestinal tract. It is often accidentally discovered during autoptical dissections, angiography or abdominal computed tomography (CT).
Case presentation:
A 27-year-old man was admitted to emergency for an acute abdominal pain. For his critical condition, the patient was immediately brought to the operating room. A extensive intestinal necrosis was found. Post-operator CT discovered a common CMT that is complicated by extended thrombosis. Despite all resuscitation measures, the patient died of septic shock two days later. An autopsy was performed showed that the main cause of intestinal ischemia was related to dissection of a common CMT.
Discussion:
A CMT is a highly unusual variation in humans. It is usually asymptomatic and may be discovered incidentally during vascular surgery, radiologic imaging, or cadaver's dissection. Lesion of this entity can lead to serious gastrointestinal complications including necrosis. Different classifications are proposed in the literature.
Conclusion:
As it is associated with the risk of mesenteric ischaemia, CMT should be kept in mind as a differential diagnosis for cases of recurrent non-specific abdominal pain.
Pericapsular Nerve Group Block: An Excellent Option for Analgesia for Positional Pain in Hip Fractures
Authors: Utsav Acharya, Ritesh Lamsal
Journal: Case Rep Anesthesiol (2020)
MeSH terms: Case Reports

Fractures in and around the hip are common presentations in the emergency department. It is commonly seen in the elderly as a result of osteoporotic changes. However, younger age groups are also affected, especially as a result of high velocity trauma. Irrespective of age, hip fractures are extremely painful, and it is difficult to position the patients for anesthesia procedures. Most of these cases are performed under subarachnoid block (SAB) or combined spinal-epidural anesthesia (CSEA), which requires the patient to be in sitting or lateral position. Here, we report a series of ten cases where pericapsular nerve group (PENG) block was administered prior to positioning the patients for SAB or CSEA. This block is a recently described regional anesthesia technique that provides excellent analgesia for hip fractures. It also provides very good analgesia for patient positioning during procedures such as SAB or CSEA.
A case of double cystic esophageal duplication in VACTERL syndrome: the first case report and a review of the literature
Authors: G Vasta, S Tursini, E Rovero, R Angotti, F Molinaro, V Briganti
Journal: Front Pediatr (2023)
Keywords: vacterl, case report, esophageal duplication cyst, pediatric surgery, thoracoscopy
MeSH terms: Case Reports

Background:
An esophageal duplication cyst (EDC) is a rare malformation resulting from the embryonic foregut. VACTERL syndrome is a genetic disorder affecting many systems of the human body. We report the first case of VACTERL syndrome associated to asymptomatic double EDC.
Case report:
A girl with anorectal malformation and rectovestibular fistula, kidney malformation, and various vertebral defects came to our attention at the time of birth. VACTERL disease was diagnosed. She underwent Peña anoplasty at 4 months of life without complications. MRI was conducted at the age of 2. It accidentally showed a double esophageal duplication (12 mm × 35 mm × 10 mm) at the D7-D9 level. We planned a thoracoscopy; previous intraoperative esophagogastroduodenoscopy showed an external compression of the native esophagus. Two duplicated esophageal lesions were removed. The patient made an uneventful recovery and was completely asymptomatic at long-term follow-up.
Conclusions:
VACTERL syndrome is still a not well-defined disease. Based on the current literature, this is the first case of a double esophageal duplication in a patient affected by VACTERL syndrome. According to us, the thoracoscopic approach of esophageal duplications can be followed by experts. Complete surgical excision is possible even if the cyst shares a common muscular wall with the esophagus. For this reason, we suggest to close the muscular wall by a simple interrupted suture.
Case Report: Vitreous Amyloidosis Caused by a TTR Lys55Asn Mutation With Intraoperative Suprachoroidal Hemorrhage
Authors: Qing Xu, Xinting Wang, Zhengpei Zhang, Jie Li, Haiyang Liu, Sujuan Ji, Lei Qiao, Chaoju Gong, Ruifang Feng, Suyan Li
Journal: Front Med (Lausanne) (2022)
Keywords: lys55asn mutation, ttr, case report, suprachoroidal hemorrhage, vitrectomy, vitreous amyloidosis, vitreous opacity
MeSH terms: Case Reports

We report a vitreous amyloidosis patient with white vitreous opacities (footplates) adhering to the posterior lens capsule. A positive Congo-red stain and transthyretin (TTR) Lys55Asn mutation confirmed the diagnosis of vitreous amyloidosis. Optical coherence tomography (OCT) revealed fern-like material adhering to the the posterior pole retinal surface in both eyes. Visual acuity significantly improved after the first vitrectomy, but vitreous opacities recurred 4 years later. The patient appeared to have aggravated sensorimotor neuropathy and severe autonomic dysfunction at the same time. He developed intraoperative suprachoroidal hemorrhage during the second vitrectomy.
Complex Twisted Knots of Umbilical Cord in a Monochorionic-Diamniotic Twin Gestation: A Case Report
Authors: Behnaz Razavi, Maryam Kasraeian, Atefe Hashemi, Shaghayegh Moradi Alamdarloo, Fateme Sadat Najib
Journal: Galen Med J (2020)
Keywords: multiple entanglement, nuchal cord, twin pregnancy, umbilical cord entanglement
MeSH terms: Case Reports

Background:
True knots and tight loops of umbilical cord can cause serious fetal complications in monochorionic-monoamniotic twins but are usually unexpected in Monochorionic-diamniotic twins because of the presence of the intertwin membrane. This report presents a case of monochorionic-diamniotic twin gestation with a complex cord knots.
Case report:
A 31-year-old G2Ab1 with monochorionic-diamniotic twin pregnancy in the gestational age of 30 weeks presented with ruptured membrane since 3weeks before delivery. At the delivery time, multiple umbilical cord knots was found.
Conclusion:
Premature ruptured membrane can cause septostomy of the intertwin membrane, multiple umbilical cord knots and its complications. Therefore, these cases should be considered for evaluation of the presence of intertwin membrane and umbilical cord knots in each sonography examination.
Implementation of Electronic Adherence Monitors and Associated Interventions for Routine HIV Antiretroviral Therapy in Uganda: Promising Findings
Authors: Jessica E Haberer, Robert Baijuka, John Bosco Tumuhairwe, Edna B Tindimwebwa, James Tinkamanyire, Ellyk Tuhanamagyezi, Lawrence Musoke, Lindsey E Garrison, Marisa DelSignore, Nicholas Musinguzi, Stephen Asiimwe
Journal: Front Digit Health (2022)
Keywords: africa, hiv, sms intervention, electronic adherence monitoring, implementation science
MeSH terms: Case Reports

Background:
High, sustained adherence is critical for achieving the individual and public health benefits of HIV antiretroviral therapy (ART). Electronic monitors provide detailed adherence information and can enable real-time interventions; however, their use to date has largely been confined to research. This pilot study (NCT03825952) sought to understand feasibility and acceptability a relatively low-cost version of this technology and associated interventions for routine ART delivery in sub-Saharan Africa.
Methods:
We provided two ART clinics in rural, southwestern Uganda with electronic adherence monitors for data-informed counseling as well as optional SMS messages to clients and/or social supporters (daily or triggered by missed or delayed doses) and/or an alarm. Clinic and ART client experiences were observed for 3 months per client, including time and motion studies. Qualitative interviews among clients, clinicians, and healthcare administrators were informed by the Consolidated Framework for Implementation Research.
Results:
Fifty-one ART clients were enrolled; 57% were male and the median age was 34 years. Choice of associated intervention varied among participants. The median number of visits during follow-up was two per client. Counselors reviewed the adherence data with 90% of clients at least once; 67% reviewed data at all visits. Average adherence was 94%; four clients had adherence gaps >1 week. Acceptability was high; all but one client found the monitor "very useful" and all found SMS "very useful." Clinic visits among clients with the intervention lasted 4 min longer on average than those in standard care. The monitors and daily SMS generally functioned well, although excess SMS were triggered, primarily due to cellular network delays. Overall, participants felt the technology improved adherence, clinic experiences, and clinician-client relationships. Few worried about stigma and privacy. Cost was a concern for implementation, particularly at scale.
Conclusion:
We successfully implemented a relatively low-cost electronic ART adherence monitor and associated interventions for routine care in rural Uganda. Feasibility and acceptability were generally high, and individuals were identified who could benefit from adherence support. Future work should involve longitudinal follow-up of diverse populations, clinical outcomes, and detailed cost-effectiveness analysis to help drive policy decisions around the uptake of this technology for routine clinical care.
Clinical trial registration:
identifier: NCT03825952.
The Development of Instruments to Detect Indicators of Behavioral Changes in Therapeutic Communities: A Clinical Case Study
Authors: Stefania Cristofanelli, Agata Ando', Laura Ferro
Journal: Front Public Health (2019)
Keywords: adolescence, behavioral problems, psychological assessment, psychological functioning, therapeutic communities

Clinicians involving in the treatment of adolescent patients should use a valid and efficient psychological assessment. The evaluation of the efficiency in clinical interventions may provide helpful information in terms of cost-effectiveness and may contribute to increase the quality and efficacy of the public services. Despite the importance of clinical and therapeutic interventions, we may observe several aspects limiting the chance in using them. For example, the neuropsychiatry context due to heterogeneous users (such as children and adolescents) makes the replicability of clinical trials difficult in terms of results. Thus, efficient clinical programs and interventions-potentially able to identify specific and long-term effects-need to be defined. In clinical contexts (i.e., therapeutic communities). It should be a priority both to manage aspects of emergency/urgency we may observe in adolescents, and to focus on those aspects placed on a timing dimension. The current study reports a description of innovative measures developed specifically for assessing adolescent patients and for tracking psychological features and behavioral changes. Furthermore, a clinical case is examined by using a multimethod assessment including such innovative measures. Clinical implications are discussed. The development and sharing of "assessment cultures" among professionals should represent a priority in improving the effectiveness of therapeutic communities.
Suboptimal Response to Tenofovir Alafenamide in Two Patients With HBeAg-Positive Hepatitis B: A Case Report
Authors: Ruochan Chen, Siya Pei, Yayu Chen, Linxia Tan, Ying Xue, Shao Liu, Yan Huang, Xuegong Fan
Journal: Front Med (Lausanne) (2021)
Keywords: hbeag-positive chronic hepatitis b, case report, monotherapy, suboptimal response, tenofovir alafenamide
MeSH terms: Case Reports

Tenofovir alafenamide (TAF) is one of the most potent first-line nucleot(s)ide analogs for treating chronic hepatitis B virus (HBV) infections. To date, no cases of TAF drug resistance and/or suboptimal response have been reported. To our knowledge, this is the first report of two adult male patients presenting a suboptimal response response to TAF monotherapy. Our study indicates long-term observations and extensive data are needed to further evaluate the efficacy and safety of TAF, and highlights the need for the development of robust novel direct-acting antivirals and immune therapies for HBV.