OpenMed/multicare-articles huggingface dataset:
Case Report: Long-term observations from the tacrolimus weaning randomized clinical trial depicts the challenging aspects for determination of low-immunological risk patients
Authors: Christophe Masset, Jacques Dantal, Jean-Paul Soulillou, Alexandre Walencik, Florent Delbos, Sophie Brouard, Magali Giral, Nantes DIVAT Consortium, Gilles Blancho, Julien Branchereau, Diego Cantarovich, Anne Cesbron, Agn Amp Xe S Chapelet, Anne Devis, Cl Amp Xe Ment Deltombe, Lucile Figueres, Raphael Gaisne, Claire Garandeau, Caroline Gourraud-Vercel, Maryvonne Hourmant, Christine Kandel-Aznar, Georges Karam, Clarisse Kerleau, Delphine Kervella, Claire Leman, Alice Leclech, Aur Amp Xe Lie Meurette, Karine Renaudin, Simon Ville
Journal: Front Immunol (2022)
Keywords: allograft rejection, calcineurin inhibitor withdrawal, case report, donor specific antibodies, kidney transplantation
MeSH terms: Graft Rejection, Tacrolimus / adverse effects, Randomized Controlled Trial, Case Reports, Research Support, Non-U.S. Gov't, Calcineurin Inhibitors / adverse effects, Humans, Neoplasm Recurrence, Local / drug therapy, Retrospective Studies, Weaning, Tacrolimus, Calcineurin Inhibitors

Whilst calcineurin inhibitors (CNI) are the cornerstone of immunosuppressive maintenance therapy in kidney transplantation, several studies have investigated the safety of CNI withdrawal in order to avoid their numerous side effects. In this context, we performed several years ago a clinical randomized trial evaluating CNI weaning in stable kidney transplant recipients without anti-HLA immunization. The trial was interrupted prematurely due to a high number of de novo DSA (dnDSA) and biopsy proven acute rejection (BPAR) in patients who underwent tacrolimus weaning, resulting in treatment for rejection and resumption of tacrolimus. We report here the long-term outcomes of patients included in this clinical trial. Ten years after randomization, all patients are alive with a functional allograft. They all receive tacrolimus therapy except one with recurrent cutaneous neoplasia issues. Long-term eGFR was comparable between patients of the two randomized groups (46.4 ml/min vs 42.8 ml/min). All dnDSA that occurred during the study period became non-detectable and all rejections episodes were reversed. The retrospective assessment of HLA DQ single molecule epitope mismatching determined that a majority of patients who developed dnDSA after tacrolimus withdrawal would have been considered at high immunological risk. Minimization of immunosuppression remains a challenging objective, mainly because of the issues to properly select very low immunological risk patients. Valuable improvements have been made the last decade regarding evaluation of the allograft rejection notably through the determination of numerous at-risk biomarkers. However, even if the impact of such tools still need to be clarify in clinical routine, they may permit an improvement in patients' selection for immunosuppression minimization without increasing the risk of allograft rejection.
Osteogenesis Imperfecta with Celiac Disease and Type II Diabetes Mellitus Associated: Improvement with a Gluten-Free Diet
Authors: Luis Rodrigo, Isabel Pérez-Martinez
Journal: Case Rep Med (2012)
MeSH terms: Case Reports

Osteogenesis imperfecta (OI) is a genetic disease, with a connective tissue alteration, consisting in the presence of multiple spontaneous fractures or after minimal traumatism. Its association with other metabolic processes is rarely described. We present the clinical case of a female adult patient of 43 years. From her infancy, she has had multiple fractures, needing several surgical interventions, and she was diagnosed of OI type 2 at adolescence age. Due mainly to difficulties in walking remaining in wheel-chair in the last three years, she was overweight with morbid obesity (BMI = 45.4) and had a type-II DM associated. She suffered from recurrent abdominal pain and chronic diarrhea and was diagnosed of celiac disease (CD) with increased intraepithelial duodenal infiltration, being classified as lymphocytic enteritis, Marsh I type. She was put on a gluten-free diet (GFD), having lost 6 kg of weight after 6 months, with a good control of DM-II and presenting a significant clinical improvement. It is rewarding to search the presence of two coincidental metabolic diseases associated to OI, specially CD, because of the dramatic clinical benefit in the general found after putting on a GFD.
Hemi-Clamshell Approach for Fetal Lung Interstitial Tumor Resection in a Neonate: A Case Report
Authors: Yasuhiro Kuroda, Hiroaki Fukuzawa, Insu Kawahara, Keiichi Morita
Journal: European J Pediatr Surg Rep (2021)
Keywords: fetal lung interstitial tumor, hemi-clamshell approach, neonate tumors
MeSH terms: Case Reports

Fetal lung interstitial tumor (FLIT) is a rare primary lung mass in neonates. Classical incisions, such as posterolateral thoracotomy or median sternotomy, do not provide optimal exposure of the operative field for the resection of pediatric thoracic giant tumors. Herein, we report a rare case of a FLIT in a full-term male neonate, with complete resection achieved using a hemi-clamshell approach, which provided the required visualization of the operative field. The neonate was transferred to our hospital because of mild respiratory distress, which developed 18-hour after normal vaginal delivery. A mass in his right chest, without a midline shift, was observed on chest radiographs. Computed tomography showed a well-circumscribed solid anterior cervicothoracic mass, with a uniform density and no apparent cysts, diagnosed as a primary thoracic giant tumor. Once the patient was clinically stabilized, we proceeded with right upper lobectomy, using a hemi-clamshell approach, full sternotomy, and anterolateral thoracotomy, on postnatal day 22. Histopathologic examination revealed an 8.5 × 6.5 × 4.0 cm solid mass within the right upper lobe, which was diagnosed as a FLIT. His postoperative recovery was uneventful. The patient was followed up for 1 year, with no complaints or symptoms and no postoperative shoulder dysfunction. Gross total resection of primary thoracic giant tumors can be accomplished in neonates with optimal exposure of the chest cavity using a hemi-clamshell approach.
Bathing Suit Variant of Autosomal Recessive Congenital Ichthyosis (ARCI) in Two Indian Patients
Authors: Dharshini Sathishkumar, Dincy Peter, Susanne Pulimood, Henning Wiegmann, Frederic Valentin, Meera Thomas, Hans Christian Hennies, Vinzenz Oji
Journal: Case Rep Dermatol Med (2018)
MeSH terms: Case Reports

Bathing suit ichthyosis (BSI) is a rare variant of autosomal recessive congenital ichthyosis (ARCI) due to transglutaminase-1 gene (TGM1) mutations leading to a temperature sensitive phenotype. It is characterized by dark-grey or brownish scaling restricted to the "bathing suit" areas. We report two Indian girls with bathing suit ichthyosis and mutations in TGM1 (patient 1: homozygous for c.1147G>A; patient 2: compound heterozygous for c.832G>A, c.919C>G).
Myasthenia Gravis Coexisting With Primary Sjögren's Syndrome: Report of Three Cases and Literature Review
Authors: Xia Li, Yi Zhao, Qiuju Liao, Yuwei Da
Journal: Front Neurol (2020)
Keywords: autoimmune diseases, coexistence, myasthenia gravis, outcome, primary sjögren's syndrome

Objective: The coexistence of myasthenia gravis (MG) and primary Sjögren's syndrome (pSS) is rarely reported. This study aims to describe the clinical features, treatment and outcome of MG coexisting with pSS. Materials and Methods: Herein we reported three cases with the two coexisting diseases, and also searched the PubMed, Medline databases, and China Wanfang databases for the relevant case reports written in English, Chinese, or Japanese with detailed data. Results: We reviewed a total of 17 patients with both diseases. Fifteen patients were female. The median age at onset was 48 years (range 28-78 years). MG was the initial disease in nine of 17 cases. The median interval between the onsets of the two diseases was 30 months (range 7 months to 20 years). The symptoms of MG included fatigable ptosis (64.7%), bulbar symptoms (58.8%), muscle fatigability (64.7%), diplopia (64.7%), dyspnea (23.5%), and facial paralysis (5.9%). Anti-acetylcholine receptor antibody was positive in 70.6% patients. All the patients had sicca symptoms. Manifestations of pSS also included swollen exocrine glands (23.5%), joint pain (23.5%), hair loss (11.8%), leukopenia (11.8%), recurrent oral ulcers (5.9%), Raynaud phenomenon (5.9%), and fever (5.9%). ANA positivity was present in 70.6% patients, anti-SSA positivity in 47.1%, and double positivity of anti-SSA and anti-SSB in 17.6%. There were 12 patients (70.6%) with two autoimmune diseases (pSS and MG), and five patients with more than two autoimmune diseases. Cholinesterase inhibitors were the most commonly prescribed drugs (82.4%). Seven patients received thymectomy and one patient improved after the operation. Two patients were given intravenous methylprednisolone pulse therapy, and four patients oral steroids combined with immunosuppressants initially. Intravenous immunoglobulin and plasma exchange were used in two patients, respectively, for the respiratory failure. All the patients improved following treatment except one patient who died of MG crisis due to medication withdrawal. Conclusion: The coexistence of SS with MG is quite rare. The onset of MG may occur before or after the diagnosis of SS. Co-morbidity with MG does not seem to adversely affect the course of SS. Thus, controlling the progress of MG is the critical aspect of treatment.
The Important Role of Endoscopy in Management of Pediatric Pseudomembranous Necrotizing Tracheitis
Authors: Xiling Wu, Lei Wu, Zhimin Chen
Journal: Front Pediatr (2020)
Keywords: airway obstruction, endoscopy, mechanical debridement, necrotizing tracheitis, pediatric
MeSH terms: Case Reports

Pseudomembranous necrotizing tracheitis is a rare, but life-threatening cause of central airway obstruction. Here, we reported three cases of pediatric pseudomembranous necrotizing tracheitis. The infectious etiologies were Staphylococcus aureus secondary to influenza A virus and Aspergillus fumigatus. Endoscopy was used in diagnosis and management of all patients and two patients survived. The improvement in mortality rate of these diseases need early recognition and prompt treatment with mechanical debridement by endoscope and early initiation of broad spectrum antibiotics. Endoscopy is a promising tool to diagnose and remove the pseudomembrane, therefore relieving central airway obstruction.
Follicle-Stimulating Hormone-Secreting Pituitary Adenoma Inducing Spontaneous Ovarian Hyperstimulation Syndrome, Treatment Using In Vitro Fertilization and Embryo Transfer: A Case Report
Authors: Xiaofang Du, Wen Zhang, Xingling Wang, Xiaona Yu, Zhen Li, Yichun Guan
Journal: Front Endocrinol (Lausanne) (2021)
Keywords: fsh-secreting pituitary adenoma, ivf-et, endocrine disorders, spontaneous ovarian hyperstimulation syndrome, transsphenoidal surgery
MeSH terms: Adenoma / complications, Embryo Transfer, Fertilization in Vitro, Follicle Stimulating Hormone / metabolism, Ovarian Hyperstimulation Syndrome / therapy, Pituitary Neoplasms / complications, Case Reports, Adenoma / metabolism, Adenoma / pathology, Adult, Female, Humans, Ovarian Hyperstimulation Syndrome / etiology, Pituitary Neoplasms / metabolism, Pituitary Neoplasms / pathology, Treatment Outcome, Follicle Stimulating Hormone

Objective:
To describe the management of a patient with a pituitary adenoma secreting follicle-stimulating hormone (FSH) associated with spontaneous ovarian hyperstimulation syndrome (sOHSS) who was treated with in vitro fertilization and embryo transfer (IVF-ET).
Methods:
We report a clinical case of a woman of reproductive age with menstrual irregularity, infertility and ovarian hyperstimulation due to recurrent pituitary adenoma secreting FSH, which persisted after transsphenoidal surgery.She underwent the diagnosis by magnetic resonance imaging (MRI) and laboratory tests,and finally she was treated with IVF-ET.
Results:
The patient was plagued by a recurrent pituitary adenoma for many years and tried various treatments. After complete transsphenoidal surgery, sOHSS decreased, as shown by a reduction in oestradiol levels and an improvement in the ultrasonography parameters; however, secondary amenorrhea occurred. Finally, pregnancy was achieved through IVF-ET and the symptoms of ovarian hyperstimulation were relieved.
Conclusions:
IVF-ET was found to be effective for the treatment of recurrent pituitary adenoma, thus representing a therapeutic option that should be taken into consideration in such cases.
A novel surgical approach with peritonectomy to extranodal multisystemic histiocytic sarcoma: A case report and literature review
Authors: Giuseppe Barbato, Alessandro Tarantini, Francesco Serra, Francesca Cabry, Alberto Farinetti, Lorena Sorrentino, Anna Vittoria Mattioli, Roberta Gelmini
Journal: Int J Surg Case Rep (2019)
Keywords: histiocytic sarcoma, peritoneal metastases, peritonectomy

Introduction:
Histiocytic sarcoma (HS) is a very rare malignant histiocytic derivation cancer. The extranodal multisystemic HS has an aggressive clinical course and poor Disease-Free Survival (DFS) and Overall Survival (OS). There are no shared and effective therapeutic protocols; our approach aims to improve the prognosis for advanced diseases.
Presentation of the case:
53-year-old female patient admitted to hospital for intestinal obstruction in April 2016 with evidence of an ileal mass in the right iliac fossa and peritoneal metastases, undergoing urgent surgery of ileal resection, ileostomy and nodular peritoneal mass biopsy. Histological examination diagnosed HS. Staging exams excluded the involvement of other gastro-intestinal districts in the absence of concomitant lymphoproliferative disorders, and PET-CT revealed multiple abdominal and mediastinal nodes hyperplasia. The patient performed chemotherapy and, seeing the partial abdominal extranodal response, was performed cytoreductive surgery (CRS). Histological examination confirmed the diagnosis of HS with multiple peritoneal, ileal, colic and omental localisation. Adjuvant second-line chemotherapy treatment (4 DHAP cycles) was performed. Disease Free Survival at ten months from cytoreductive surgery, Overall Survival at 21 months from diagnosis.
Discussion:
Despite a multimodal therapy with surgery and chemotherapy, extranodal multisystemic HS has a poor prognosis. Up to now the role of surgery is limited to biopsies or complications treatment. Our results of DFS and OS show that cytoreductive surgery may be a valid therapeutic choice.
Conclusion:
The surgical approach with major cytoreductive purposes could improve the prognosis in cases with prevalent abdominal extranodal localisation.
Emergency primary repair of grade V bladder neck injury complicating pelvic fracture
Authors: Elroy P Weledji, Pius Fokam, Djatche Nzade, Divine Eyongeta
Journal: Ann Surg Innov Res (2014)
Keywords: bladder neck injury, extravasation, pelvic injury, primary repair, scrotal necrotizing fasciitis
MeSH terms: Case Reports

We report a case of a grade V bladder injury complicating an open-book pelvic fracture following a road traffic accident. The bladder neck injury was primarily repaired in the emergency setting of a poor-resourced area with successful outcome. The dangers of urinary extravasation are still to be considered of importance and we advocate and encourage immediate/emergency open intervention although it remains controversial to say the least in a lesser resourced healthcare set up.
Repetitive transcranial magnetic stimulation to maintain treatment response to electroconvulsive therapy in depression: a case series
Authors: Yoshihiro Noda, Zafiris J Daskalakis, Cinthia Ramos, Daniel M Blumberger
Journal: Front Psychiatry (2013)
Keywords: ect, bilateral rtms, maintenance rtms, major depression, relapse prevention

Electroconvulsive therapy (ECT) is the most effective treatment for a refractory major depression in the context of both unipolar and bipolar affective disorders. However, the relapse rate within the first 6 months after a successful course of ECT to treat a depressive episode can be as high 50%. Evidence-based strategies to prevent relapse have partial efficacy and are associated with problematic adverse effects limiting their use as long-term treatments. Repetitive transcranial magnetic stimulation (rTMS) has demonstrated efficacy in treatment-resistant depression with a favorable adverse effect profile. Herein, we describe six patients, four with unipolar and two with bipolar depression, where rTMS was used to maintain response after a successful course of acute and continuation ECT. rTMS was administered once or twice weekly, at 120% of the resting motor threshold. Patients received sequential bilateral rTMS (low frequency right: 600 pulses, then high frequency left: 3000 pulses). The site of stimulation was 6 cm anterior and 1 cm lateral from the site of maximum stimulation of the abductor pollicis brevis muscle. Depressive symptoms were monitored with the quick inventory of depressive symptoms-self rated. Five of the six patients were able to maintain their response status from 6 to 13 months at the time of last observation. The use of rTMS may be an important relapse prevention strategy following an acute course of ECT. Controlled studies comparing rTMS to current evidence-based relapse prevention strategies are warranted.
Gout in the Chest Misdiagnosed as Ankylosing Spondylitis
Authors: Wenjing Xue, Shengkai Zhang, Qinqin Wang, Wenzhong Que, Shanghua Xu
Journal: Front Med (Lausanne) (2020)
Keywords: ankylosing spondylitis, case report (source
MeSH terms: Case Reports

Gout is a crystal-related joint disease caused by single sodium urate deposition in the joints or in soft tissues. In recent years, the incidence of gout has increased, but cases of urate crystals deposited in the chest-ribs are rare. Here, we describe a 39-year-old man who complained of frequent pain and a feeling of tightness in chest-ribs and was misdiagnosed as ankylosing spondylitis. In addition, treatment with non-steroidal anti-inflammatory drugs (NSAIDs) and etanercept for 6 months showed no improvement, which confirmed the misdiagnosis. After physical examination, blood examination, and dual-energy CT examination, the patient was diagnosed with gout and received 50 mg benzbromarone once a day with treatment of low serum uric acid. In conclusion, gout in the chest and ribs is an unusual manifestation and has rarely been reported in the literature. This case highlights an important but overlooked history of hyperuricemia in the diagnosis, and dual-energy CT is the preferred method for differential diagnosis of chest-ribs gout.
Common Bile Duct Stone Formed around a Migrated Clip: An Unexpected Complication of Laparoscopic Cholecystectomy
Authors: Anas M Hussameddin, Iba Ibrahim AlFawaz, Reema Fahad AlOtaibi
Journal: Case Rep Gastrointest Med (2018)
MeSH terms: Case Reports

Surgical clip migration into the common bile duct with subsequent stone formation is a rare complication following laparoscopic cholecystectomy. Very few cases have been reported in the literature. We report a case of bile duct stone formation around a migrated surgical clip 16 years after laparoscopic cholecystectomy. The patient presented with right upper quadrant pain, fever, and chills for one week. Investigation with abdominal ultrasound showed dilatation of the common bile duct and moderate dilatation of the intrahepatic bile ducts. The diagnosis was confirmed by endoscopic retrograde cholangiopancreatography and the patient was managed successfully with sphincterotomy and stone extraction. The exact mechanism of clip migration is not fully understood. Presenting symptoms are similar to non-clip-induced choledocholithiasis. Time of presentation can vary significantly with an average of 26 months. Most cases reported in the literature required surgical intervention. Clip migration should be considered in the differential diagnosis of postcholecystectomy biliary colic and cholangitis. Management with endoscopic retrograde cholangiopancreatography is the treatment of choice.
The cultural context of teaching and learning sexual health care examinations in Japan: a mixed methods case study assessing the use of standardized patient instructors among Japanese family physician trainees of the Shizuoka Family Medicine Program
Authors: Cameron G Shultz, Michael S Chu, Ayaka Yajima, Eric P Skye, Kiyoshi Sano, Machiko Inoue, Tsukasa Tsuda, Michael D Fetters
Journal: Asia Pac Fam Med (2015)
Keywords: family medicine, japan, sexual health, standardized patient instructors

Background:
In contrast to many western nations where family medicine is a cornerstone of the primary care workforce, in Japan the specialty is still developing. A number of services within the bailiwick of family medicine have yet to be fully incorporated into Japanese family medicine training programs, especially those associated with sexual health. This gap constitutes a lost opportunity for addressing sexual health-related conditions, including cancer prevention, diagnosis, and treatment. In this mixed methods case study we investigated the perceived acceptability and impact of a standardized patient instructor (SPI) program that trained Japanese family medicine residents in female breast, pelvic, male genital, and prostate examinations.
Case description:
Building on an existing partnership between the University of Michigan, USA, and the Shizuoka Family Medicine Program, Japan, Japanese family medicine residents received SPI-based training in female breast, pelvic, male genital, and prostate examinations at the University of Michigan. A mixed methods case study targeting residents, trainers, and staff was employed using post-training feedback, semi-structured interviews, and web-based questionnaire.
Discussion and evaluation:
Residents' and SPIs' perceptions of the training were universally positive, with SPIs observing a positive effect on residents' knowledge, confidence, and skill. SPIs found specific instruction-related approaches to be particularly helpful, such as the positioning of the interpreter and the timing of interpreter use. SPIs provided an important opportunity for residents to learn about the patient's perspective and to practice newly learned skills. Respondents noted a general preference for gender concordance when providing gender-specific health care; also noted were too few opportunities to practice skills after returning to Japan. For cultural reasons, both residents and staff deemed it would be difficult to implement a similar SPI-based program within Japan.
Conclusions:
While the SPI program was perceived favorably, without sufficient practice and supervision the skills acquired by residents during the training may not be fully retained. Deep-rooted taboos surrounding gender-specific health care appear to be a significant barrier preventing experimentation with SPI-based sexual health training in Japan. The feasibility of implementing a similar training program within Japan remains uncertain. More research is needed to understand challenges and how they can be overcome.