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10 pages, 6574 KB  
Case Report
Structured Differential Diagnosis of Orofacial Pain Associated with an Enamel Crack Using ICOP and ICHD-3: A Case Report
by Kohei Shimizu, Takuya Yasukawa, Masayuki Okano, Aki Kawamoto, Noboru Noma, Makoto Hayashi and Osamu Takeichi
Healthcare 2026, 14(13), 2001; https://doi.org/10.3390/healthcare14132001 (registering DOI) - 6 Jul 2026
Abstract
Background: Cracked teeth may present with variable and atypical symptoms, sometimes mimicking non-odontogenic orofacial pain conditions, making diagnosis challenging, particularly when cracks appear limited to enamel. Case presentation: A 36-year-old woman presented with intermittent pain in a mandibular molar radiating to the ipsilateral [...] Read more.
Background: Cracked teeth may present with variable and atypical symptoms, sometimes mimicking non-odontogenic orofacial pain conditions, making diagnosis challenging, particularly when cracks appear limited to enamel. Case presentation: A 36-year-old woman presented with intermittent pain in a mandibular molar radiating to the ipsilateral temporal region. Clinical examination identified a crack line on the lingual surface of the mandibular first molar. Pulp sensibility testing (cold test and electric pulp test), occlusal loading tests, and cone-beam computed tomography (CBCT) were performed. CBCT was used primarily to exclude vertical root fracture and periapical pathology, and no radiographic abnormalities were identified. Differential diagnosis was conducted using structured diagnostic frameworks, including the International Classification of Orofacial Pain and the International Classification of Headache Disorders (3rd edition). Diagnostic local anaesthesia eliminated both biting pain and referred pain, supporting an odontogenic source. Collectively, the findings suggested that the enamel crack was the most likely source of odontogenic pain, although a definitive causal relationship could not be established. Because pulp sensibility remained normal, conservative management was selected. Crack sealing with a methyl methacrylate-based adhesive resin resulted in complete symptom resolution that was maintained throughout a 3-year follow-up period without the need for root canal treatment. Conclusions: Although the diagnosis remained probabilistic, the structured diagnostic approach, together with the favourable clinical response after crack sealing, supported the enamel crack as the most likely source of odontogenic pain. Full article
(This article belongs to the Special Issue Contemporary Clinical Advances in Endodontics)
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19 pages, 524 KB  
Systematic Review
Nutritional Practices During the Transition to Motherhood: A Systematic Qualitative Review
by Artemisia Kokkinari, Maria Dagla, Kleanthi Gourounti, Evangelia Antoniou and Georgios Iatrakis
Nurs. Rep. 2026, 16(7), 234; https://doi.org/10.3390/nursrep16070234 (registering DOI) - 6 Jul 2026
Abstract
Background: The transition to motherhood represents a critical life phase marked by profound biological, psychological and social changes. During this period, women’s nutritional practices are shaped not only by physiological needs but also by shifting identities, caregiving responsibilities and social expectations. Although nutrition [...] Read more.
Background: The transition to motherhood represents a critical life phase marked by profound biological, psychological and social changes. During this period, women’s nutritional practices are shaped not only by physiological needs but also by shifting identities, caregiving responsibilities and social expectations. Although nutrition during pregnancy and the postpartum period has been widely studied from a biomedical perspective, less attention has been paid to how women experience, negotiate and attribute meaning to food during the transition to motherhood. Objective: This systematic qualitative review aimed to synthesise existing qualitative evidence on women’s experiences of nutritional practices during the transition to motherhood, with particular attention to food as self-care, control, autonomy, identity formation and mental well-being. Methods: A systematic search of electronic databases was conducted to identify qualitative studies exploring women’s experiences of nutrition during pregnancy and early motherhood. Eligible studies employed qualitative methodologies such as interviews, focus groups or ethnographic approaches. Study selection followed PRISMA guidelines. Methodological quality was appraised using established qualitative appraisal tools. A thematic synthesis approach was used to integrate findings across studies. Results: The synthesis identified several interrelated themes: nutrition as a form of self-care and emotional regulation; loss of autonomy and heightened moral surveillance around food choices; food practices as a means of performing and negotiating “good motherhood”; and the emotional burden of dietary expectations in relation to mental health and identity. Women described navigating competing demands between their own nutritional needs and those of their infants, often within contexts of social judgement and limited support. Conclusions: Nutritional practices during the transition to motherhood extend beyond health behaviours and are deeply embedded in issues of identity, autonomy and care. Recognising the social and emotional dimensions of maternal nutrition may inform more holistic, woman-centred approaches to nutritional guidance and maternal health support. Full article
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16 pages, 2055 KB  
Case Report
MEIS1::NCOA2 Fusion Sarcoma of the Bartholin Gland: A Case Report and Review of the Literature
by Pauline Dumonceaux, Loréane Sims, Aline Francois, Sabrina Croce, Latifa Fellah, Sophie Cvilic, Charlotte Maillard and Pascale Jadoul
Diagnostics 2026, 16(13), 2098; https://doi.org/10.3390/diagnostics16132098 - 3 Jul 2026
Viewed by 146
Abstract
Background: MEIS1::NCOA1/2 fusion sarcomas are a recently described molecular entity arising predominantly in the genitourinary and gynecologic tracts. Their clinical presentation is often misleading, and no standardized treatment guidelines currently exist. Methods: A literature review was conducted using PubMed to identify all reported [...] Read more.
Background: MEIS1::NCOA1/2 fusion sarcomas are a recently described molecular entity arising predominantly in the genitourinary and gynecologic tracts. Their clinical presentation is often misleading, and no standardized treatment guidelines currently exist. Methods: A literature review was conducted using PubMed to identify all reported cases of molecularly confirmed MEIS1::NCOA1/2 fusion sarcomas. Clinicopathological, molecular, treatment, and outcome data were extracted for comparative analysis. Case: We report the case of a 38-year-old nulliparous woman who presented with a right vulvar induration clinically consistent with a Bartholin gland cyst. Surgical excision revealed a spindle cell mesenchymal tumor harboring a MEIS1::NCOA2 fusion transcript and a CTNNB1 exon 3 mutation, with probable incomplete resection margins. A local recurrence was documented by MRI and PET–CT at eight months. Surgical re-excision revealed diffuse involvement and complete excision was considered uncertain. Adjuvant external beam radiotherapy followed by an MR-Linac boost was administered. Discussion: This case highlights the diagnostic challenge of Bartholin gland masses. We provide a review of the literature on MEIS1::NCOA1/2 fusion sarcomas and examine the potential aggressiveness of tumors that additionally harbor a CTNNB1 mutation. Given the nonspecific immunophenotype of this entity, this case underscores the indispensable role of RNA-based molecular sequencing in the diagnosis of low-grade spindle cell tumors when immunohistochemistry proves inconclusive. We further discuss the surgical challenges inherent to this anatomical region and explore intention-to-treat radiotherapy as a potential therapeutic option. Conclusions: We report a rare case of a MEIS1::NCOA2 fusion-positive sarcoma arising in the Bartholin gland region, and, to our knowledge, the first case in which radiotherapy with curative intent has been explored for this entity. This observation expands the limited literature on this emerging clinicopathological entity. Full article
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9 pages, 1077 KB  
Case Report
Complete Imaging Resolution of Ductal Carcinoma In Situ During Osimertinib Therapy for Synchronous EGFR Exon 19-Mutant Non-Small Cell Lung Cancer: A Case Report
by Leticia Assad Maia Sandoval, Richard E. Sharpe, Austin J. Fullenkamp, Erinn Downs and Lida Mina
Int. J. Mol. Sci. 2026, 27(13), 5995; https://doi.org/10.3390/ijms27135995 - 3 Jul 2026
Viewed by 73
Abstract
A 64-year-old Asian woman diagnosed with synchronous breast ductal carcinoma in situ (DCIS) and stage IV EGFR-mutated non-small cell carcinoma of the lung (NSCLC). A decision was made to defer management of the DCIS and initiate Osimertinib for lung cancer treatment, since this [...] Read more.
A 64-year-old Asian woman diagnosed with synchronous breast ductal carcinoma in situ (DCIS) and stage IV EGFR-mutated non-small cell carcinoma of the lung (NSCLC). A decision was made to defer management of the DCIS and initiate Osimertinib for lung cancer treatment, since this was a life-limiting diagnosis. At 9 months, restaging FDG-PET CT showed an interval response in the NSCLC and complete loss of FDG avidity at the biopsy-proven DCIS site. Breast MRI confirmed complete imaging resolution of the DCIS. The clinical resolution of breast DCIS during third-generation EGFR inhibitor therapy has not been previously reported in humans and highlights a potential role for the EGFR/HER2 (ERBB) pathway in pre-invasive breast cancer. Full article
(This article belongs to the Special Issue Breast Cancer: From Pathophysiology to Novel Therapies, 2nd Edition)
12 pages, 1081 KB  
Case Report
Successful Dialysis Weaning in Refractory Membranous Nephropathy Through Long-Term Multi-Disciplinary Management: A Case Report
by Reina Suetsugu-Ishizawa, Megumi Matsumoto, Hirofumi Sakuma, Motoki Matsuki, Mitsuru Yanai, Yayoi Ogawa and Naoki Nakagawa
Kidney Dial. 2026, 6(3), 46; https://doi.org/10.3390/kidneydial6030046 - 3 Jul 2026
Viewed by 59
Abstract
Membranous nephropathy (MN) is a leading cause of nephrotic syndrome (NS). The remission rate of MN remains limited, and effective strategies for refractory MN are not established. We present the case of a 49-year-old Japanese woman with severe NS caused by MN. Kidney [...] Read more.
Membranous nephropathy (MN) is a leading cause of nephrotic syndrome (NS). The remission rate of MN remains limited, and effective strategies for refractory MN are not established. We present the case of a 49-year-old Japanese woman with severe NS caused by MN. Kidney biopsy revealed glomerular basement membrane thickening with granular deposition of immunoglobulin G (IgG) and complement component 3. IgG subclass analysis showed predominant IgG1 deposition, with weak IgG2 and IgG3 deposition. Phospholipase A2 receptor (PLA2R) deposition was equivocal in the first kidney biopsy and negative in the second. Serum anti-PLA2R antibody was not detected. Electron microscopy revealed subepithelial, subendothelial, and mesangial electron-dense deposits. Detailed screening revealed no significant abnormalities other than appendiceal findings, suggesting secondary MN associated with appendiceal infection. Although combined therapy with prednisolone, cyclosporine, rituximab, and low-density lipoprotein apheresis was administered during the first 6 months, remission of MN was not achieved. During dialysis, initiated because of kidney failure, long-term multidisciplinary management, including control of appendiceal infection and inflammation and initiation of angiotensin II receptor blocker therapy, ultimately led to remission of MN and discontinuation of dialysis. Overall, even refractory MN requiring dialysis may have a reversible clinical course with careful conservative management and long-term follow-up. Full article
13 pages, 6354 KB  
Case Report
Hydroxychloroquine-Induced AGEP with Positive Rechallenge: A Case Report and Mini Review of the Literature
by Kristijan Jovanović, Tamara Umeljic Sočević, Milos Stepovic, Jovana Milosavljević, Jovica Tomović, Miroslav M. Sovrlić, Marko Folić, Miloš N. Milosavljević, Dalibor Jovanović and Nevena Folić
Dermatopathology 2026, 13(3), 30; https://doi.org/10.3390/dermatopathology13030030 - 3 Jul 2026
Viewed by 113
Abstract
Background/Objectives: Hydroxychloroquine is widely used in the treatment of autoimmune and dermatologic diseases; however, it may rarely induce severe cutaneous adverse reactions. Acute Generalized Exanthematous Pustulosis is an uncommon, acute pustular eruption most frequently associated with antibiotics. Hydroxychloroquine-induced AGEP remains relatively rare and [...] Read more.
Background/Objectives: Hydroxychloroquine is widely used in the treatment of autoimmune and dermatologic diseases; however, it may rarely induce severe cutaneous adverse reactions. Acute Generalized Exanthematous Pustulosis is an uncommon, acute pustular eruption most frequently associated with antibiotics. Hydroxychloroquine-induced AGEP remains relatively rare and diagnostically challenging due to its atypical and prolonged clinical course. Case presentation: We report the case of a 45-year-old woman with rheumatoid arthritis and a complex medical history who developed generalized urticarial and pustular dermatosis following re-exposure to hydroxychloroquine. Notably, the patient had experienced a similar cutaneous reaction after previous exposure to the same medication several years earlier. Ten days after completing a treatment course of hydroxychloroquine, she developed rapidly progressive pruritic erythematous and urticarial plaques that evolved into generalized annular lesions with peripheral scaling and grouped sterile pustules. Laboratory evaluation demonstrated leukocytosis, intermittent eosinophilia, and elevated IgE levels, while the infectious workup was negative. Histopathological examination revealed subcorneal pustules with neutrophilic infiltration, mild spongiosis, and scattered individual eosinophils, perivascular inflammatory infiltrates, findings consistent with AGEP. Retrospective assessment using the EuroSCAR scoring system classified the reaction as probable AGEP, while the Naranjo adverse drug reaction scale supported a probable causal relationship with hydroxychloroquine. Clinical improvement was achieved after withdrawal of the drug and treatment with systemic corticosteroids and supportive therapy. Conclusions: This case highlights the importance of recognizing atypical presentations compatible with hydroxychloroquine-induced probable AGEP and emphasizes the diagnostic value of a positive rechallenge as supportive evidence of drug causality. Early recognition and prompt discontinuation of the offending agent are essential to prevent severe complications and recurrence. Full article
(This article belongs to the Section Clinico-Pathological Correlation in Dermatopathology)
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21 pages, 3693 KB  
Article
Evaluating the Feasibility and Acceptability of a Culturally Adapted Intervention to Promote Resistance Exercise in Young Black Women: A Randomized Controlled Trial
by Chloe S. Jones, Katherine E. Spring and Danielle D. Wadsworth
Int. J. Environ. Res. Public Health 2026, 23(7), 867; https://doi.org/10.3390/ijerph23070867 - 3 Jul 2026
Viewed by 176
Abstract
Young Black women face barriers to exercise and elevated cardiometabolic risk, yet resistance exercise (RE) remains underutilized despite its benefits. We evaluated the feasibility and acceptability of a 24-week culturally adapted RE intervention + text messages in young Black women. Participants were randomized [...] Read more.
Young Black women face barriers to exercise and elevated cardiometabolic risk, yet resistance exercise (RE) remains underutilized despite its benefits. We evaluated the feasibility and acceptability of a 24-week culturally adapted RE intervention + text messages in young Black women. Participants were randomized to the motivational exercise group (MEG; n = 14) or the standard exercise group (SEG; n = 13). Both groups received 10 and 11 weeks of supervised (by a Black woman) and unsupervised RE. MEG received additional cultural adaptations and weekly discussions to build competence, autonomy, and self-regulation strategies + mobile support. Feasibility and acceptability were assessed via recruitment, consent, and retention rates, adherence, and thematic analysis of semi-structured interviews. Recruitment and consent rates were 97.2% and 100.0%, respectively. Retention rates were 93.3% (MEG) and 86.7% (SEG) at 12 weeks, and 93.3% and 80.0% at 24 weeks, respectively. Supervised adherence was 93.9% and 88.8% in MEG and SEG, and 14.3% and 15.4%, respectively, during unsupervised RE. Participants desired continued support and a more tailored mobile experience during unsupervised RE. Supervised RE with ethnically matched trainers was feasible and acceptable. Future interventions should incorporate mobile tools with tailored feedback and accountability strategies to sustain long-term RE to improve health outcomes in this population. Full article
(This article belongs to the Special Issue Adherence to Physical Activity and Its Role in Health Promotion)
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16 pages, 304 KB  
Article
Factors Associated with Perceptions of Maternal Control and Support During Childbirth and Relationship with Childbirth Satisfaction Among Women in Spain
by Sergio Martínez-Vázquez, Rocío Adriana Peinado-Molina, Leticia Molina-García and Antonio Hernández-Martínez
Medicina 2026, 62(7), 1281; https://doi.org/10.3390/medicina62071281 - 3 Jul 2026
Viewed by 145
Abstract
Background and Objectives: The World Health Organisation (WHO) promotes respectful, woman-centred care during childbirth, aimed at preserving women’s autonomy, their sense of control and the fulfilment of their expectations. Loss of control during childbirth has been linked to a poorer birth experience [...] Read more.
Background and Objectives: The World Health Organisation (WHO) promotes respectful, woman-centred care during childbirth, aimed at preserving women’s autonomy, their sense of control and the fulfilment of their expectations. Loss of control during childbirth has been linked to a poorer birth experience and adverse emotional consequences in the postpartum period. To analyse the maternal and perinatal factors associated with perceived control and support during childbirth, and their relationship with childbirth satisfaction in women in Spain. Materials and Methods: A cross-sectional study was conducted with postpartum women who had given birth within the previous six months in Spain. Validated instruments were used, including the “Support and Control in Birth” (SCIB) scale and the “Birth Satisfaction Scale-Revised” (BSS-R). Sociodemographic, obstetric and neonatal variables were also collected. Associations were assessed using multiple linear regression models. Results: A total of 302 postpartum women participated, with a mean age of 35.2 years (SD = 4.18), of whom 70.2% (n = 212) were primiparous. Satisfaction with childbirth (BSS-R) was the strongest positive association with all dimensions of the SCIB: internal control (adjusted mean difference [aMD] = 0.56; 95% CI: 0.48, 0.64), external control (aMD = 0.75; 95% CI: 0.61, 0.89), support (aMD = 0.89; 95% CI: 0.77, 1.01) and overall score (aMD = 2.34; 95% CI: 2.13, 2.56). Greater pain intensity was associated with a lower perception of internal control (aMD = −1.00; 95% CI: −1.31, −0.70). In comparison with adherence to the birth plan, non-adherence showed a negative association with the support dimension (aMD = −7.71; 95% CI: −10.72, −4.71) and with the overall SCIB score (aMD = −10.21; 95% CI: −16.01, −4.42). Women’s active participation in decision-making was positively associated with perceived control and support (aMD = 7.15; 95% CI: 2.21, 12.09). Conclusion: A greater perception of support and control during labour is associated with a more positive birth experience. Deviation from the birth plan and greater pain intensity are associated with a poorer perception of control, whilst women’s active participation was associated with higher perceived control. These findings underscore the importance of personalised and respectful care to improve women’s childbirth experience, particularly perceived support, control and satisfaction. Full article
3 pages, 819 KB  
Interesting Images
Retrospective Diagnosis of Recurrent Takotsubo Syndrome Episodes in a 40-Year-Old Woman
by Malgorzata Zalewska-Adamiec, Hanna Bachorzewska-Gajewska and Slawomir Dobrzycki
Diagnostics 2026, 16(13), 2083; https://doi.org/10.3390/diagnostics16132083 - 3 Jul 2026
Viewed by 108
Abstract
Recurrence of Takotsubo syndrome (TS) is the most significant challenge in the long-term follow-up of patients with TS. We present the case of a 40-year-old woman with a history of two acute coronary syndromes and myocarditis who was hospitalized in our clinic due [...] Read more.
Recurrence of Takotsubo syndrome (TS) is the most significant challenge in the long-term follow-up of patients with TS. We present the case of a 40-year-old woman with a history of two acute coronary syndromes and myocarditis who was hospitalized in our clinic due to Takotsubo syndrome. Left ventriculography demonstrated the rare mid-ventricular variant of Takotsubo syndrome. Based on a carefully obtained medical history, it was established that all previous coronary events, as well as the current TS episode, had been preceded by psychological stress. Following an additional review of the patient’s medical records and application of the InterTAK score, we concluded that the previous incidents may have been episodes of TS and that the patient had now experienced her fourth episode of Takotsubo syndrome. Full article
(This article belongs to the Special Issue Insights into Imaging Diagnosis of Heart Disease)
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6 pages, 1406 KB  
Case Report
Fluoroscopy-Guided Injection of Autologous Mechanically Filtered Adipose Tissue for Chronic Sacroiliac Joint Pain: A Case Report
by Bruno De Meo, Alfonso Maria Forte, Elisa Palombo, Hassan Zmerly and Luigi Di Lorenzo
Surgeries 2026, 7(3), 80; https://doi.org/10.3390/surgeries7030080 - 2 Jul 2026
Viewed by 101
Abstract
Introduction: Chronic sacroiliac joint (SIJ) pain is a frequent cause of low-back pain and remains challenging to diagnose and treat due to complex anatomy, overlapping clinical features, and limited long-term efficacy of conventional therapies. Case Presentation: We report the case of a 74-year-old [...] Read more.
Introduction: Chronic sacroiliac joint (SIJ) pain is a frequent cause of low-back pain and remains challenging to diagnose and treat due to complex anatomy, overlapping clinical features, and limited long-term efficacy of conventional therapies. Case Presentation: We report the case of a 74-year-old woman with chronic right-sided sacroiliitis associated with spondyloarthritis, presenting with persistent gluteal pain refractory to nonsteroidal anti-inflammatory drugs, physiotherapy, and steroid injections. The patient underwent a dual imaging-guided intra-articular injection, consisting of pre-procedural ultrasound assessment and fluoroscopy-guided needle placement to confirm intra-articular access prior to injection. Autologous adipose tissue purified through mechanical filtration, without enzymatic manipulation, was administered. Pain intensity decreased from a visual analog scale (VAS) score of 8/10 at baseline to 2/10 at three months, with sustained improvement up to 12 months, functional recovery, and discontinuation of analgesic therapy, without procedure-related complications. Conclusions: This case suggests that dual imaging-guided intra-articular injection of mechanically filtered autologous adipose tissue may be feasible in selected patients with refractory SIJ pain. No causal inference can be drawn from a single case, and further studies are required to evaluate safety and clinical effectiveness. Full article
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17 pages, 720 KB  
Systematic Review
Psychological Interventions Targeting Maternal Role Development and Identity in Perinatal Mental Health: A Systematic Review with Qualitative Synthesis
by Lorena Gutiérrez Hermoso, Cecilia Peñacoba Puente, Carmen Écija Gallardo, Livia Gomes Viana Meireles and Patricia Catalá Mesón
Healthcare 2026, 14(13), 1958; https://doi.org/10.3390/healthcare14131958 - 2 Jul 2026
Viewed by 154
Abstract
Background: Maternal identity is the perception and recognition of a woman as a mother. Within this emerging identity, the maternal role takes on special importance as a manifestation of the set of responsibilities that a woman assumes in the care and upbringing [...] Read more.
Background: Maternal identity is the perception and recognition of a woman as a mother. Within this emerging identity, the maternal role takes on special importance as a manifestation of the set of responsibilities that a woman assumes in the care and upbringing of her baby. Respectful professional accompaniment during the period of maternal role acquisition is key to perinatal mental health and secure bonding with the baby. The main objective of this systematic review with narrative synthesis was to analyze the effects of psychological support programs aimed at maternal role acquisition during the transition to motherhood. Methods: Studies with experimental and quasi-experimental designs addressing maternal role acquisition in pregnant or postpartum women were included. A systematic search was conducted in PsycINFO, MEDLINE, PubMed and SCOPUS from inception to March 2025 following PRISMA recommendations. Due to the heterogeneity in study designs, interventions and outcome measures, a narrative synthesis was performed instead of a meta-analysis. Results: A total of 11 studies were extracted with a total sample of 1244 women, including five randomized controlled trials and six quasi-experimental studies. Psychological support programs focusing on maternal role acquisition generally showed improvements in maternal identity construction, self-efficacy and maternal competence, although not all findings reached statistical significance. In addition, several studies reported reductions in postnatal depressive symptoms, as well as improvements in subjective well-being and maternal role perception. Conclusions: results suggest that psychological support programs targeting maternal role acquisition may represent a promising approach for supporting perinatal mental health. However, the evidence should be interpreted with caution due to methodological limitations and heterogeneity across studies. In fact, most included studies were conducted in Eastern cultural contexts (Iran, China), limiting generalizability to Western populations without further adaptation and validation. Additionally, incomplete reporting of standardized effect sizes and precision measures across studies limits the quantitative interpretation of the findings. This review was not prospectively registered, and title/abstract screening was conducted by a single reviewer, increasing the risk of selection bias. Further research using rigorous and standardized designs is needed to clarify the effectiveness and generalizability of these interventions. Full article
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14 pages, 27965 KB  
Case Report
An Autopsy Report of Beta-Propeller Protein-Associated Neurodegeneration with 68-Year Survival, Focusing on Isoform-Specific Distribution of Hyperphosphorylated Tau
by Tomonori Kai, Keiko Tominaga, Atsumi Matsunaga, Hiroshi Shimizu, Kazuhiro Iwama and Keisuke Ishizawa
Reports 2026, 9(3), 209; https://doi.org/10.3390/reports9030209 - 1 Jul 2026
Viewed by 138
Abstract
Background and Clinical Significance: Beta-propeller protein–associated neurodegeneration (BPAN), also known as static encephalopathy of childhood with neurodegeneration in adulthood (SENDA), is a subtype of neurodegeneration with brain iron accumulation caused by pathogenic variants in WDR45. Although its clinical course and neuroimaging [...] Read more.
Background and Clinical Significance: Beta-propeller protein–associated neurodegeneration (BPAN), also known as static encephalopathy of childhood with neurodegeneration in adulthood (SENDA), is a subtype of neurodegeneration with brain iron accumulation caused by pathogenic variants in WDR45. Although its clinical course and neuroimaging features are increasingly recognized, detailed neuropathological characterization, especially at its terminal stage, remains limited. Case presentation: We report a 68-year-old woman with a heterozygous WDR45 splice-site variant (NM_007075.4:c.830+1G>A), representing the longest-surviving case of SENDA/BPAN described to date. After static developmental delay in childhood, she rapidly developed progressive parkinsonism, dystonia, and cognitive decline in early adulthood, ultimately becoming bedridden with profound motor and autonomic dysfunction. Serial MRI demonstrated progressive cerebral and cerebellar atrophy with iron-related signal changes in the globus pallidus and substantia nigra. She died of sepsis at the age of 68 and was subjected to an autopsy including the brain. Neuropathological findings: Autopsy revealed severe, diffuse neuronal loss and gliosis throughout the central nervous system, with marked iron deposition and complete neuronal loss in the globus pallidus and substantia nigra. Immunohistochemistry demonstrated widespread tau pathology. Notably, neuronal tau inclusions contained both four-repeat (4R) and three-repeat (3R) isoforms, whereas glial tau was predominantly 4R-positive, indicating a mixed neuronal 4R/3R and glial 4R-dominant tauopathy. Perivascular and subpial 4R-tau–dominant deposits consistent with aging-related tau astrogliopathy were also present. LC3-positive and ferritin-positive cells suggested impaired autophagic flux, supporting the proposed autophagy-related pathogenesis of SENDA/BPAN. Conclusions: This case provides comprehensive clinicopathological insight into end-stage SENDA/BPAN, highlighting distinctive tau isoform patterns in neurons versus glia and pathological evidence of autophagy dysfunction. These findings expand the neuropathological spectrum of SENDA/BPAN and may inform future mechanistic and therapeutic research. Full article
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10 pages, 1110 KB  
Case Report
Lightning Strike-Induced Cutaneous Burns and Airway Injury: A Case Report Highlighting Recurrent Extubation Failure in a Critically Ill Patient
by Robert Canelli, Dhanesh D. Binda, Maxwell B. Baker, Christa M. Lam and Ala Nozari
Eur. Burn J. 2026, 7(3), 36; https://doi.org/10.3390/ebj7030036 - 1 Jul 2026
Viewed by 89
Abstract
Introduction: Lightning strikes cause a unique spectrum of cutaneous burn injuries that differ substantially from conventional thermal or high-voltage electrical burns. Despite the well-documented systemic lethality of lightning injuries, the burn-specific sequelae—including airway involvement and its contribution to ventilatory failure—remain underreported in the [...] Read more.
Introduction: Lightning strikes cause a unique spectrum of cutaneous burn injuries that differ substantially from conventional thermal or high-voltage electrical burns. Despite the well-documented systemic lethality of lightning injuries, the burn-specific sequelae—including airway involvement and its contribution to ventilatory failure—remain underreported in the burn literature. Case Report: We present a 31-year-old woman who sustained multiple cutaneous burns following a lightning strike, including a circumferential necklace-pattern burn to the neck, a large midline torso burn, and burns to the right lower extremity and foot. Following resuscitation from ventricular fibrillation cardiac arrest, she required mechanical ventilation and experienced three sequential extubation failures before ultimately requiring tracheostomy. We propose that direct supraglottic and glottic injury, together with airway mucosal edema from localized thermal and electrical injury of the neck, compounded further by systemic inflammatory and infectious complications, contributed to her inability to sustain independent ventilation. Conclusions: In lightning strike victims with burns involving the neck or thorax, direct upper airway injury should be actively considered, excluded when possible, and closely monitored as a potential cause of extubation failure. A low threshold for early bronchoscopic airway assessment and conservative extubation protocols is warranted. Full article
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9 pages, 655 KB  
Case Report
Polyclonal Hyperviscosity Crisis and Severe Depletion Coagulopathy Induced by Therapeutic Plasma Exchange in Sjögren’s Syndrome: A Case Report and Therapeutic Dilemma
by Gabriela Rybka, Andrzej Boryczko, Radosław Dziedzic, Łukasz Chmura and Joanna Kosałka-Węgiel
Reports 2026, 9(3), 207; https://doi.org/10.3390/reports9030207 - 1 Jul 2026
Viewed by 140
Abstract
Background and Clinical Significance: Hyperviscosity syndrome (HVS) is a rare complication of primary Sjögren’s syndrome (pSS). While therapeutic plasma exchange (TPE) is the standard treatment to clear pathogenic immunoglobulins, its execution can trigger severe, atypical systemic risks. Case Presentation: A 60-year-old woman with [...] Read more.
Background and Clinical Significance: Hyperviscosity syndrome (HVS) is a rare complication of primary Sjögren’s syndrome (pSS). While therapeutic plasma exchange (TPE) is the standard treatment to clear pathogenic immunoglobulins, its execution can trigger severe, atypical systemic risks. Case Presentation: A 60-year-old woman with pSS and extreme polyclonal hypergammaglobulinemia (total protein 100 g/L, IgM 41 g/L) presented with an acute hyperviscosity crisis, causing retinopathy, neurological deficits, and skin ischemia. Emergency TPE with 5% albumin replacement successfully reduced IgM by ~90% (to 6.39 g/L), resolving HVS symptoms. However, 20 min post-procedure, the patient suffered sudden hemodynamic collapse (BP 50/30 mmHg) and developed multiple massive, expanding soft-tissue hematomas. Laboratory tests revealed a coagulopathy consistent with plasma protein depletion following therapeutic plasma exchange, characterized by severe hypofibrinogenemia (1.35 g/L) and a 50% reduction in total serum protein. TPE was permanently discontinued. The patient was successfully stabilized using aggressive fluid resuscitation, vasopressors, and fresh frozen plasma (FFP) transfusions, followed by maintenance therapy with rituximab. Conclusions: In conclusion, clinicians should remain vigilant that severe hyperviscosity syndrome can be driven by a polyclonal increase in immunoglobulins rather than just monoclonal entities; furthermore, managing this condition requires careful balancing of TPE efficacy against its potential to trigger profound depletion coagulopathy. Full article
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Article
‘I Hope Now You Will Do Something for Her, as She Is a Great Burden to Me’: The Impact of Economic Dependency on the Afterlives of Cork Single Women Active in the Irish Revolutionary Period
by Leeann Lane
Humanities 2026, 15(7), 86; https://doi.org/10.3390/h15070086 - 30 Jun 2026
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Abstract
This article examines the later life experiences of women who participated in the revolutionary period in Cork city and county who did not go on to marry. Many within this cohort of Cork female activists experienced poverty, ill health, economic dependency and loneliness [...] Read more.
This article examines the later life experiences of women who participated in the revolutionary period in Cork city and county who did not go on to marry. Many within this cohort of Cork female activists experienced poverty, ill health, economic dependency and loneliness in the aftermath of the revolutionary period. Their unmarried status left these women particularly vulnerable in a patriarchal state structured—legally, economically and culturally—on an essentialist view of the married, childbearing woman. The invisibility of adult dependency in Ireland was summed up in the 1926 Census of Population report: ‘no account is taken in this inquiry of adults who may be supported out of the household income’. Mary Daly notes the ‘high proportion of the population working within a family economy which had little reliance on money incomes and waged employment’. This may, as she states, ‘have reduced the numbers exposed to the blunt instrument of unemployment’ in the context of the 1930s depression. However, the emotional and psychological effects of economic dependency have not been considered in any depth. The lack of statutory benefits for lower-class single women who were unable to access paid employment and who did not have the requisite social insurance stamps meant that they faced a vista of poverty and dependency on charity and family members. The latter were often begrudging and indisposed to assume the financial burdens of those who they often deemed economically ‘unproductive’. This article argues that, in so many cases, financial dependency resulted in the loss of agency and autonomy for unmarried women. Full article
(This article belongs to the Special Issue Celibacy in Irish Women’s Writing)
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