Case Series Research Articles

Severe ovarian hyperstimulation syndrome following sole gonadotropin-releasing hormone (GnRH) agonist trigger: a case series and literature review

Abstract Objective The aim of this study was to report three cases of early severe ovarian hyperstimulation syndrome (OHSS) in patients undergoing a GnRH antagonist protocol triggered with GnRH agonist (GnRH-a), leading to hospitalization and the need for peritoneal drainage. Additionally, a review of the existing literature on this topic is provided. Design This is a retrospective case series and a literature review. Setting This study was conducted at obstetrics and gynecology department of tertiary academic referral hospitals, Israel. Participants This study included three patients presented with severe OHSS symptoms, including abdominal distension, ascites, and hemoconcentration. Main outcome measures The main focus of the treatment was to address the symptoms and prevent any further complications. The outcome was the complete recovery of the patients. Results The presented cases detail instances of severe OHSS following oocyte retrieval, utilizing GnRH-a for triggering. Case 1 involved a 33-year-old patient with a history of polycystic ovary syndrome (PCOS), Case 2 featured a 22-year-old patient with familial adenomatous polyposis (FAP), and Case 3 included a 41-year-old patient with a history of depressive disorder. All patients receiving supportive care, including infusions and medications, exhibited gradual improvement during hospitalization, with complete resolution observed during the 20-day post-hospitalization check-up. Conclusions These three cases highlight the occurrence of severe early OHSS following a GnRH antagonist protocol triggered with GnRH-a in the absence of human chorionic gonadotropin (hCG) administration for trigger or luteal-phase support. Clinicians must be aware that a GnRH-a trigger followed by a freeze-all approach does not guarantee the complete elimination of OHSS in all patients.

Steven-Johnson Syndrome/Toxic Epidermal Necrolysis is Associated with PD-1/PD-L1 Inhibitors Usage: A Case Series

Steven-Johnson Syndrome/Toxic Epidermal Necrolysis is Associated with PD-1/PD-L1 Inhibitors Usage: A Case Series

Bilateral triceps rupture: a review of the literature and case series

Bilateral triceps rupture: a review of the literature and case series

Case Report: Respiratory lesions successfully treated with intravenous plasminogen, human-tvmh, replacement therapy in four patients with plasminogen deficiency type 1

Plasminogen deficiency type 1 (PLGD-1, hypoplasminogenemia) is an ultra-rare, lifelong disease associated with development of fibrinous lesions in multiple organ systems. Depending on lesion location, clinical manifestations of PLGD-1 can result in acute and/or chronic respiratory airway disease which can compromise respiratory function leading to life-threatening events. Early recognition and effective treatment of airway obstruction caused by fibrinous lesions are critical to prevent morbidity due to respiratory compromise. However, physicians may not be familiar with the clinical presentation and management of PLGD-1, causing delays in diagnosis and treatment and potentially contributing to morbidity. Presented here is a case series of one adult and three pediatric patients with severe respiratory complications of PLGD-1 successfully managed by infusions of plasminogen, human-tvmh replacement therapy. Patients’ respiratory symptoms were resolved or greatly improved, and treatment was generally well tolerated. In all patients, baseline plasminogen activity was substantially increased with plasminogen replacement therapy administered initially every one to two days followed by extended interval dosing as symptoms were controlled or resolved. All four described cases support the clinical benefit of replacement therapy with plasminogen, human-tvmh in the resolution of life-threatening respiratory complications associated with PLGD-1. Clinical manifestations in addition to respiratory lesions were also improved or resolved with continued treatment.

Accuracy of robotic computer‐assisted implant surgery for immediate implant placement: A retrospective case series study

Accuracy of robotic computer‐assisted implant surgery for immediate implant placement: A retrospective case series study

This is the first in‐human trial and prospective case series of a novel single‐port robotic system for gynaecological surgery: An IDEAL stage 2a study

This is the first in‐human trial and prospective case series of a novel single‐port robotic system for gynaecological surgery: An IDEAL stage 2a study

Optical Coherence Tomography as a Diagnosis-Assisted Tool for Guiding the Treatment of Melasma: A Case Series Study

Background/Objectives: Multiple underlying pathomechanisms may lead to melasma, but there has been no report on the use of optical coherence tomography (OCT) to reveal specific pathomechanisms in individual patients and provide individualized treatments accordingly. Using real-time OCT images, we studied the pathomechanisms of melasma in 12 female patients and the effects of individualized treatments. Methods: Patients were divided into good and bad improved groups according to the improvement in hyperpigmentation at month 4. Results: In the bad improved group, all melanin or confetti melanin had significantly decreased at month 2 or month 4 while granular melanin ratio at month or month 4 significantly increased, the most parameters of dendritic-sharped cells (DCs) before and after treatment were not significantly different, the collagen area or collagen density at month 4 significantly decreased. In the good improved group, there was slightly low all melanin/confetti melanin at month 4 and high granular melanin at month 4 in comparison to the bad improved group. Moreover, most of the parameters in the DCs at month 4 significantly increased while most parameters in collagen at month 4 significantly decreased. Conclusions: OCT is useful in revealing the involved pathomechanisms of melasma in individualized patients. Positive treatment results can be achieved through individualized therapy regimen targeting the pathomechanisms.

Reconstructive surgical therapy of peri-implant defects with ribose cross-linked collagen matrix and crosslinked hyaluronic acid – a prospective case series

ObjectiveTo investigate the efficacy of ribose-crosslinked collagen (RCLC) matrices functionalized by crosslinked hyaluronic acid (xHya) for reconstructive treatment of class I and III (b-c) peri-implantitis lesions in a transmucosal healing mode.Materials and methodsThirteen patients presenting with 15 implants were included in this prospective case series. Upon flap reflection, the implants were thoroughly decontaminated employing glycine powder air polishing and adjunctive sodium hypochlorite. For defect augmentation, xHyA was administered to the bony defect walls, exposed implant surfaces, and the RCLC matrix before defect grafting. The full-thickness flap was readapted and sutured around the implant neck for transmucosal healing. Baseline and respective values at the 12 months post-op evaluation were recorded for the clinical parameters peri-implant probing depth (PPD), buccal soft tissue dehiscence (BSTD) and bleeding on probing (BoP). Furthermore, two independent investigators analyzed radiographic changes in the defect area. The mean changes for all variables were analyzed with a paired t-test.ResultsThe initial mean PPD was 7.2 ± 1.9 mm, and BoP was present in 63% of sites. After 12 months, PPD at the latest visit was 3.2 ± 0.66 mm, which amounted to a respective 3.9 ± 1.85 mm reduction, while the BoP frequency dropped to 10% at all sites. Radiographic bone fill was accomplished for 62.8% of the former defect area, accompanied by a mean MBL gain of 1.02 mm around the treated implants (all p < 0.001).ConclusionsWithin the limits of this case series, we conclude that the proposed treatment sequence substantially improved peri-implant defects and offered a simplified but predictive technique.Clinical relevanceReconstructive treatment approaches for peri-implantitis are effective but remain non-superior to open flap debridement. Further research on novel biomaterial combinations that may improve reconstructive treatment outcomes are warranted. Ribose-crosslinked collagen matrices biofunctionalized by hyaluronic acid used in this study yield improved clinical and radiographic peri-implant conditions after 12 months.Graphical

Real‐world effectiveness of eptacog beta in patients with haemophilia and inhibitors: A multi‐institutional case series

AbstractIntroductionThe management of bleeding events (BEs) in haemophilia A (HA) and B (HB) patients with inhibitors necessitates the use of bypassing agents. The recombinant factor VIIa bypassing agent eptacog beta has demonstrated efficacy at treating BEs and managing perioperative bleeding in adults in phase three clinical studies.AimTo provide real‐world descriptions of eptacog beta use for BE treatment in patients on emicizumab or eptacog beta prophylaxis.MethodsThis is a retrospective case series of 14 patients who received eptacog beta at seven haemophilia treatment centres, with HA (n = 11) or HB (n = 3) and inhibitors or anaphylaxis to factor replacement.ResultsTwenty‐four spontaneous and traumatic BEs are described (muscle hematomas, joint hemarthroses, port site, and epistaxis) involving 11 subjects. Eptacog beta was effective for acute bleed treatment as both first‐line therapy and for treatment of BEs refractory to eptacog alfa in 23/24 events. When eptacog beta was used for prophylaxis, 2/3 patients reported a decreased frequency of breakthrough BEs compared with prophylactic eptacog alfa and one patient experienced a similar frequency of breakthrough BEs compared with prophylactic activated prothrombin complex concentrate. Eptacog beta provided effective bleed control for three subjects who underwent minor surgical procedures. Treatment with eptacog beta was estimated to be 46%–72% more cost‐effective than eptacog alfa. No safety concerns or adverse events were reported.ConclusionsIn this case series, eptacog beta was safe, effective, and economical as first‐line therapy, treatment of refractory BEs, management of perioperative bleeding, or prophylaxis in haemophilia patients with inhibitors.

Fluocinolone acetonide 0.18-mg implant for treatment of recurrent inflammation due to non-infectious uveitis: a case series of 15 patients

IntroductionUncontrolled non-infectious uveitis affecting the posterior segment (NIU-PS) can lead to vision loss due to repeated bouts of inflammation and consequent tissue damage. Patients with chronic NIU-PS who experience recurrent uveitis after being treated with systemic and short-acting local corticosteroids may benefit from the sustained-release 0.18-mg fluocinolone acetonide implant (FAi).MethodsIn this case series, 18 eyes with chronic, recurrent NIU-PS and cystoid macular edema (CME) treated with the 0.18-mg FAi were analyzed retrospectively. Data on patient demographics, clinical history, previous and concomitant treatments for uveitis recurrence, time to and number of uveitis recurrences, intraocular pressure (IOP), central subfield thickness (CST), and visual acuity (VA) were collected and summarized.ResultsA majority of patients (14/15 [93%]) had a history of ocular surgery, largely cataract extraction, and all developed chronic and recurrent NIU-PS and CME. At baseline, patients had a mean age of 72 years (range: 46 to 93), were 53% male, and had a mean duration of NIU-PS of 3 years (range: 1 to 19). Patients were followed for an average of 16.5 months (range: 2 to 42.5 months) post FAi. Eleven of the 18 eyes (61%) had ≥ 5 recurrences of uveitis since diagnosis, with an average time to recurrence of approximately 12 weeks (range: 1 to 27). All eyes treated with the 0.18-mg FAi showed reduced NIU-PS recurrence and visual and anatomical improvement, as measured by VA and CST, respectively. Two eyes had an IOP elevation that was managed with topical therapy, and one eye was treated with topical prednisolone for additional inflammation management. Two eyes required adjunct therapy with short-acting intravitreal corticosteroids at 7 and 16 weeks for NIU-PS recurrence after 0.18-mg FAi insertion.ConclusionAfter receiving the 0.18-mg FAi, eyes with uncontrolled NIU-PS had sustained resolution of CME and inflammation with limited need for supplementary steroid drops or injections and minimal steroid class-specific adverse effects; none required incisional IOP-lowering surgery.

Silent Threats of the Heart: A Case Series and Narrative Review on Suicide Left Ventricle Post-Aortic Valve Replacement in Patients with Dynamic LVOT Obstruction and Aortic Stenosis

Aortic stenosis (AS) is the most prevalent valvular heart disease in Europe and North America, with transcatheter aortic valve implantation (TAVI) revolutionizing its management. Hypertrophic left ventricle (HLV) frequently coexists with AS, complicating treatment due to the associated risk of left ventricular outflow tract (LVOT) obstruction, heart failure, and sudden death. A rare but severe post-aortic valve replacement (AVR) complication, termed “suicide left ventricle” (SLV), has emerged, necessitating further study. This report synthesizes current literature on SLV, its pathophysiology, and management strategies, alongside four patient case studies. The patients aged 79–87 years, underwent AVR for symptomatic AS with HLV. Post-AVR, all experienced severe complications, including dynamicLVOT gradients, systolic anterior motion (SAM) of the mitral valve, and severe hypotension, leading to death in two cases. One patient survived following surgical aortic valve replacement (SAVR) with surgical myectomy. One patient survived after TAVI. These cases highlight the critical importance of multidisciplinary Heart Team evaluations and personalized treatment plans in managing SLV. Despite advancements in AVR, SLV remains a complex, life-threatening condition, requiring an exhaustive and multifaceted approach for optimal patient outcomes. This report offers valuable insights into SLV occurrence and management from a clinical perspective.

Clinico-genetic profile of seven patients with PARK-PINK1: A case series from a tertiary care centre from India and review of literature.

Recessive variants in the PINK1 gene is a known cause of early-onset Parkinson's disease (EOPD). To describe the clinical features and genetic profile of patients of PARK-PINK1. Retrospective chart review of demographic, clinical and genetic details of patients carrying biallelic PINK1 variants from our database. Seven cases were recruited with median age at onset 33 years (Range: 20-49). All had asymmetrical onset, tremor in four, abnormal posturing in two and slowness in one patient. Parkinsonism phenotype was noted in six patients (with dystonia in four) and isolated dystonia in one. Among 6 patients with parkinsonism, five had rest tremor, all had good levodopa-response, and four had motor-fluctuation with choreiform-dyskinesia. Exome-sequencing revealed bi-allelic pathogenic/likely pathogenic variants in all of which five were novel. PARK-PINK1 presents as an EOPD with tremor-predominant phenotype, good levodopa-responsiveness, early motor fluctuation and dyskinesia. We describe five novel variants in PINK1 gene.

Oral mucosal changes caused by nicotine pouches: case series

Oral nicotine pouches are the latest products in the tobacco industry. They are manufactured by large tobacco companies and entice tobacco or nicotine addicts, although the products are presented as a ‘harmless choice.’ Nevertheless, dentists and oral health specialists worry about oral mucosal changes due to product interactions with the oral mucosa. Unfortunately, there are no case reports of oral mucosal changes from nicotine pouches that are also investigated histopathologically. The aim of the present study was to visually and histopathologically investigate oral mucosal changes in nicotine pouch users. An online retrospective survey regarding medical and dental health, dietary habits, and tobacco consumption habits was conducted (n = 50). Respondents were selected for further intraoral and histopathological investigation based on the inclusion criteria. All five respondents had oral lesions that were histopathologically analyzed. Visually, the lesions varied in form and intensity, but all appeared white at the location where the pouches were placed. Histopathological analyses revealed parakeratosis with acanthotic epithelium, intraepithelial and connective tissue oedema, and chronic inflammatory infiltration with lymphocytes and macrophages. Participants received information about nicotine cessation and oral health recommendations. In conclusion, nicotine pouches significantly impacted oral mucosa with white lesions that revealed important changes at the cellular level.

Improvement of Functional Capacity and Quality of Life by Cardiac Rehabilitation: Case Series

Cardiac rehabilitation (CR) is a long-term program involving prescribed exercise, education, and counseling to mitigate the physiological and psychological impacts of cardiac disease. Despite evidence supporting its effectiveness in reducing mortality rates, CR remains underutilized, particularly among patients with heart failure. This research aims to highlight the importance of CR in improving the quality of life and functional capacity for post-cardiac surgery and heart failure patients. The study involved a series of case illustrations, including patients undergoing aortic valve replacement, coronary artery bypass graft surgery, and congenital heart disease repair. Each patient completed a Phase II rehabilitation program consisting of 12 exercise sessions, with functional capacity measured using the six-minute walk test (6MWT). The results indicated significant improvements in both functional capacity and quality of life, including reductions in depression levels. The findings suggest that CR is highly beneficial in enhancing patients' overall recovery and quality of life, particularly when integrated into standard postoperative care. Given these results, healthcare systems should promote broader access to CR programs to maximize patient outcomes. Additionally, future research should explore long-term benefits and develop personalized CR programs to cater to diverse patient needs.

Monostotic fibrous dysplasia of jaw bones: a case series

Abstract Background Fibrous dysplasia (FD) is a benign fibro-osseous lesion, a skeletal developmental anomaly of the bone-forming mesenchyme. The diagnosis of fibro-osseous lesions, particularly those of the jaw bones, poses significant challenges to clinicians and pathologists since it requires a correlation of clinical, radiological, histological, and surgical findings. Accurate and specific diagnosis is crucial as treatment modalities differ with different fibro-osseous lesions. Methods This retrospective analysis presents a case series of a rare condition of monostotic FD in the maxillofacial region affecting jaw bones diagnosed and/or treated over period of 10 years. Results Five cases of monostotic FD were diagnosed and treated between a period of 2013 and 2023. The cases from the 2nd to 8th decade were included in the analysis with equal involvement of males and females. Out of five cases, four cases were involving maxilla and 1 showed involvement of mandible. Conclusion FD is a rare entity affecting the jaw bones which often lead to disfigurement of face. Early detection is warranted to decrease potential complications. In addition, genetic analysis could help in understanding the occurrence in certain population.

Rigid iatrogenic hallux varus: a decades’ worth experience with arthrodesis of the metatarsophalangeal joint

Abstract Purpose Arthrodesis of the first ray metatarsophalangeal joint (MPJ) is the gold standard in iatrogenic hallux varus (IHV) in the presence of stiffness and osteoarthritis. The purpose of this study is to collect clinical and radiographic results and complications of MPJ arthrodesis in rigid iatrogenic HV. Methods A retrospective evaluation of rigid iatrogenic HV undergoing arthrodesis with a minimum follow-up (FU) of two years was performed. The clinical parameters assessed were visual analog scale (VAS), the AOFAS Hallux Metatarsophalangeal-Interphalangeal Scale score and the satisfaction scale. The radiological parameters evaluated the first to second metatarsal angle (IMA) and the angle of hallux valgus (HVA). Complications were also analysed. Result A total of 18 patients (19 procedures) with a mean FU of 5.5 ± 2.5 years were included. The mean VAS improved from 7.3 ± 1.6 to 1.3 ± 1.2 (p &lt; 0.05) at the last FU. Similarly, the AOFAS Hallux Metatarsophalangeal-Interphalangeal scale score significantly improved to 82 ± 9.2 (p &lt; 0.05). Radiological evaluation demonstrated a 1–2 IMA improvement from 4.4 ± 2.2° preoperatively to 8.9 ± 2.4° at 3 months post-operatively. Similarly, there was a significant (p &lt; 0.05) improvement of the HVA from − 22.7 ± 4.1° to 13.1 ± 4.1° at three months post-operative (p &lt; 0.05). No signification loss of correction was noted at the last follow-up (p &gt; 0.05). In one case, a delayed fusion at the arthrodesis site required surgical revision to promote fusion. No patient experienced pain with stress from the first MTP joint arthrodesis site or identified the arthrodesis site as a source of pain. No patient required implant removal. Re-operation and revision rates were 5.3%. The overall complications rate was 15.8%. Conclusions MPJ fusion effectively corrects Iatrogenic Hallux Varus in cases of rigid and fixed deformities in the medium- to long-term follow-up, with lasting improvements in AOFAS and VAS scores. The procedure is characterised by a non-negligible risk of complications, reoperations and revisions. Level of evidence Level IV, case series.

Case Series of Abdominal Actinomycosis: An Old Diagnostic Conundrum

Case Series of Abdominal Actinomycosis: An Old Diagnostic Conundrum

Clinical presentation, imaging characteristics, and short‐term outcome of eight cats presented with suspected traumatic atlantoaxial hyperflexion

AbstractTraumatic atlantoaxial hyperflexion is considered rare in cats, and only a few case reports have been published. There are other conditions that can result in a peracute onset of neurological signs localized to the cervical spinal cord segments, including acute noncompressive nucleus pulposus extrusion, ischemic myelopathy, or vertebral fractures. Appropriate treatment for these conditions can only be initiated after an accurate diagnosis has been obtained. The aim of this observational, retrospective, single‐center, descriptive case series study was to describe the clinical presentation, imaging characteristics, and short‐term outcome of eight cats presented with suspected traumatic atlantoaxial hyperflexion. Young male healthy domestic shorthair cats were overrepresented (7/8) and typically presented with a peracute, nonprogressive, nonpainful, nonlateralizing C1–C5 myelopathy (tetraplegia or nonambulatory tetraparesis) following a road traffic accident or head trauma. All MRI studies demonstrated a solitary, focal, ill‐defined intramedullary lesion immediately dorsal to the dens of the axis, affecting both grey and white matter. All cats were treated medically. In 50% of the cats, the neurological grade improved at discharge or short‐term follow‐up, 25% of the cats recovered completely, one cat was static at short‐term follow‐up, and one cat was euthanized due to persistent forebrain signs and lack of neurological improvement.

Survivorship and Functional Outcomes After Complex Primary Total Knee Arthroplasty With Contemporary Rotating-Hinge Implants

Background: Initial fixed-bearing hinge designs for primary total knee arthroplasty (TKA) had high rates of aseptic loosening. There are limited data on contemporary rotating-hinge implants. Purpose: We sought to determine survivorship and functional outcomes of contemporary rotating-hinge implants used in primary TKA. Methods: Retrospective review identified 54 primary rotating-hinge TKAs implanted in 49 patients from 2014 to 2018 at a single institution. Patients identified were 76% women, the mean body mass index was 29 kg/m2, the mean age was 65 years, and the mean follow-up was 3 years. The primary diagnosis for TKA in all cases was severe instability and ligamentous compromise. Secondary diagnoses included post-traumatic osteoarthritis (11, 20%), neurologic disease (10, 19%), inflammatory arthritis (10, 19%), connective tissue disease (3, 6%), valgus deformity (16, 30%), varus deformity (2, 4%), and recurvatum (2, 4%). Preoperative, postoperative (within 6 weeks), and most recent radiographs were reviewed. In this study, we collected preoperative, 1-year, and 2-year patient-reported outcome measures (PROMs) for patients with primary rotating-hinge TKA. Patient-reported outcome measures were prospectively collected, including the Knee Injury and Osteoarthritis Outcome Survey for Joint Replacement (KOOS JR) scores and the Mental (MCS) and Physical Component Scores (PCS) of the Veterans RAND 12-Item Health Survey (VR-12). Kaplan-Meier analysis was used to determine implant survivorship. Results: Reoperation was required in 6% (3/54); indications included periprosthetic joint infection (1), peripatellar fibrosis (1), and periprosthetic femur fracture (1). At both 2 and 5 years, survivorship free from all-cause reoperation was 95% and from revision for aseptic loosening was 100%. Mean KOOS JR scores increased from 47 preoperatively to 65 at 2 years postoperatively. On radiographic review, there were no progressive radiolucent lines consistent with aseptic loosening at final follow-up. Conclusion: The findings of this single-center, multi-surgeon retrospective case series on the use of rotating-hinge implants for primary TKA suggest excellent 2-year survivorship free from reoperation and no revisions for aseptic loosening. We report modest improvement in a variety of PROMs at 1-year and 2-year follow-up. Despite improvement, clinical outcomes were poor for a primary implant. Longer-term follow-up is required to monitor the durability of primary hinges.

Retinal displacement following direct versus indirect fluid exchange in pars Plana vitrectomy for rhegmatogenous retinal detachment.

To compare the occurrence and characteristics of retinal displacement following direct perfluorocarbon liquid (PFCL)-silicone oil (SO) exchange versus indirect PFCL for air and air for SO exchange methods during pars plana vitrectomy (PPV) in patients with rhegmatogenous retinal detachment (RRD). A comparative case series study was conducted on 58 eyes with recent RRD, undergoing standard three-port PPV with SO tamponade. Fluid exchange was performed using either direct or indirect methods. Postoperatively, a comprehensive ophthalmic examination and fundus autofluorescence (FAF) imaging were conducted at one month. The presence, amount, and direction of retinal displacement were assessed based on FAF imaging and compared between the direct and indirect fluid exchange groups. FAF imaging at one month revealed retinal displacement in 41.4% of eyes in the direct group and 62.1% in the indirect group, with no statistical difference between them (P = 0.537). However, the mean displacement was significantly higher in the indirect group (282.61 ± 110.83 µm) compared to the direct group (220.33 ± 39.67 µm, P = 0.04). The direction of displacement (downward or upward) did not differ significantly between the groups (P = 0.093). While the occurrence and location of postoperative retinal displacement did not significantly differ between direct and indirect fluid exchange methods during PPV for RRD, eyes treated with the direct method exhibited lower mean displacement compared to the indirect method. These findings suggest potential benefits of the direct exchange approach in minimizing retinal displacement following surgery.