Reports, Volume 7, Issue 4 (December 2024) – 25 articles

Background and Clinical Significance: Voriconazole is a commonly prescribed second-generation azole used for the prevention and treatment of fungal infections. This report seeks to elucidate the relationship between certain intravenous infusion parameters and the causality and severity of potential visual adverse events associated with voriconazole administration, despite existing reports of visual disturbances such as hallucinations and altered visual perception, the underlying causes of which remain inadequately understood. Case Presentation: This case report describes a 32-year-old female patient who experienced sudden hallucinations and visual impairments after receiving an initial dose of intravenous voriconazole for the treatment of recurrent severe vulvovaginitis caused by Candida glabrata. The symptoms quickly disappeared when the dosage and infusion rate were reduced as per the recommendations of the clinical pharmacist. Conclusions: This example emphasizes the possible negative drug responses linked to voriconazole, especially those provoked by its irrational use described as an inappropriate infusion rate, and the crucial role of clinical pharmacists in recognizing and handling these reactions. Full article

Background: Persistent alterations in taste and smell affect a significant proportion of individuals following COVID-19, representing a component of post-acute COVID-19 syndrome, commonly referred to as long COVID. The degradation of sphingomyelin by acid sphingomyelinase is regarded as a biomarker for acquired demyelinating neuropathies. Objectives: This study was aimed to enroll women who contracted COVID-19 during pregnancy and experienced persistent alterations in taste and/or smell for more than 1 year post-infection, in comparison to pregnant women without any disturbances in these senses. Methods: The patients were subjected to a questionnaire investigating smell and taste disorders more than 1 year after the infection. Then, the levels of acid sphingomyelinase in the plasma of the participants were assessed. Results: The results showed that in women who had been pregnant and who had been infected with SARS Cov-2 during the COVID period and who still had taste and smell disorders 1 year later, plasma acid sphingomyelinase levels were double that of pregnant women who had contracted the infection during the COVID period but had not reported taste and smell disorders and that of pregnant women analyzed after the COVID period. Conclusions: The results suggest a hypothesis that the persistence of sensory disturbances in long COVID was probably due to a failure to utilize brain circuitry with demyelination resulting from chemosensory dysfunction of the olfactory epithelium. Full article

Background and Clinical Significance: Abnormal uterine bleeding during the postmenopausal years is a pathological sign that may be due to simultaneous intrauterine and ovarian pathology. Granulosa cell tumours of the ovary are malignant neoplasms producing oestradiol, which leads to the abnormal proliferation of the endometrium, precancerous lesions, and endometrial carcinoma type I. Case Presentation: The authors present a clinical case of a 67-year-old woman with postmenopausal bleeding who underwent a total abdominal hysterectomy with bilateral adnexectomy, pelvic lymphadenectomy, and partial omentectomy. The histopathological examination showed a granulosa cell adult-type ovarian tumour and a malignant endometrial polyp with atypical hyperplasia of the endometrium. Conclusions: The immunohistochemical analysis of the malignant endometrial polyp confirmed the expression of oestrogen, progesterone receptors, and the biomarker Ki-67. Full article

Background and Clinical Significance: Lophomonas is a multiflagellate anaerobic protozoan that usually inhabits the intestines of insects, mainly cockroaches. However, bronchopulmonary infections caused by this parasite have been increasingly reported worldwide in recent decades. We provide important information for the diagnosis of this disease, which often goes undetected and frequently leads to misdiagnosis and inadequate treatment. It is noteworthy that this is the third case reported in Mexico. Case Presentation: A 37-year-old male patient was hospitalized several times for pneumonia with a persistent granuloma in the right bronchial lobe. After extensive testing, the patient was diagnosed with bronchopulmonary lophomoniasis and successfully treated with metronidazole. Conclusions: Clinicians worldwide should be aware of the existence of lophomoniasis, especially in low-income regions with poor sanitation and high insect exposure. This parasitic infection, although rare, may be underestimated due to its nonspecific respiratory symptoms, which may mimic other infections. Full article

Background and Clinical Significance: Guyon’s canal syndrome is a pathological condition caused by compression of the ulnar nerve at the level of the wrist. It is less frequent than other compression syndromes of the upper limb (cubital and carpal tunnel), and different causative agents, including vascular lesions, are described. Among these, aneurysm of the ulnar artery is described in the literature as an infrequent aetiology. Case Presentation: We report the case of a 25-year-old young man with Guyon’s canal syndrome caused by an aneurysm of the ulnar artery, who underwent surgical decompression of the Guyon’s canal without intervening on the aneurysm. The postoperative course was free of complications, and the patient reported satisfaction, with reduced symptoms. Clinical examination and ultrasound imaging showed mass reduction of the aneurysm in the postoperative period, which appears to be an evolution hitherto undocumented in the literature. Conclusions: Many treatments are available for Guyon’s canal syndrome. Past medical and surgical treatments, duration and severity of symptoms, causes, and pathogenesis are important for therapeutic choice. Surgical treatment based on ligament section and lysis of the Guyon’s canal downstream, without any action on the aneurysm and with ulnar artery preservation, determined a reduction in terms of volume, relief of the symptoms, and patient satisfaction. With this case we describe a surgical therapeutic option for the treatment of Guyon’s canal syndrome caused by an aneurysm of the ulnar artery, in which surgery is limited to canal decompression and consequential aneurysm mass reduction with concomitant relief of symptoms. Full article

Background and Clinical Significance: Fever in the returning traveler is a medical emergency warranting prompt exclusion of potentially life-threatening infections such as malaria. Case Presentation: We describe a case of a febrile returned traveler to South Africa whose prompt initial diagnostic work-up was notable for a false-positive malaria rapid diagnostic test (RDT), and who nevertheless responded quickly to oral atovaquone-proguanil, despite an ultimate diagnosis of African tick bite fever. Subsequent RDT and malaria thick- and thin-film blood examination failed to corroborate a diagnosis of malaria and all other microbiological testing other than rickettsial serology remained non-contributory. Conclusions: The case presented highlights important points regarding diagnostic test performance characteristics and premature diagnostic closure. Full article

Background and Clinical Significance: Epithelial–myoepithelial carcinoma (EMC) is a rare, low-grade malignant tumor of the salivary glands. It is characterized by a low malignancy potential, as indicated by its low rate of lymph node involvement and distant metastasis, and has a local recurrence rate of approximately 50%. Due to the rarity of EMC and the limited data available in the literature, there are no established treatment or follow-up guidelines. Case Presentation: We report the case of an 83-year-old man who presented with swelling in the left submandibular region, occurring 9 years following an initial diagnosis of EMC in the ipsilateral parotid gland. After radiological examinations and an ultrasound-guided fine-needle aspiration biopsy, the patient underwent surgical excision of the lesion with a histological diagnosis of recurrence/metastasis of low-grade epithelial–myoepithelial carcinoma. Conclusions: This is the first documented case of loco-regional lymph node metastasis 9 years after an initial diagnosis of EMC of the parotid gland. Based on our experience, EMC of the parotid gland, even when diagnosed at an early stage, seems to require a long follow-up period. Full article

Background and Clinical Significance: Kawasaki disease (KD) is an acute febrile vasculitis that primarily affects children and is associated with systemic inflammation, particularly in the coronary arteries. Coxsackievirus A6 (CVA6) has emerged as a significant agent in atypical presentations of hand, foot, and mouth disease (HFMD), raising the possibility of its involvement in KD. Case Presentation: This report presents the case of an 18-month-old Thai boy admitted with symptoms of high fever, sore throat, and ulcerative lesions, initially diagnosed with herpangina. As his condition progressed, additional KD symptoms developed, including conjunctival injection, rash, and elevated inflammatory markers, fulfilling the diagnostic criteria for KD. Notably, throat swab analysis confirmed CVA6 as the causative agent. Phylogenetic analysis revealed that the CVA6 strain closely aligned with Chinese strains from 2023, showing a high nucleotide sequence homology of 98.4%. Conclusions: In conclusion, this case highlights a possible association between CVA6-associated herpangina and KD, suggesting that CVA6 infection may act as a trigger for KD in genetically susceptible children. These findings highlight the need for increased awareness among healthcare providers to promptly identify and manage Kawasaki Disease during peak enterovirus seasons, reducing its impact on children. Full article

Background and Clinical Significance: Veno-venous extracorporeal membrane oxygenation (VV ECMO) has become a widely accepted supportive treatment for severe acute respiratory distress syndrome (ARDS) in intensive care units (ICUs). Although it has gained popularity, some of its aspects, including optimal anticoagulation management and the best means of monitoring hemostasis, remain unresolved. Thrombosis and bleeding are still important complications of ECMO. Case Presentation: A 44-year-old male patient, with no underlying conditions, was diagnosed with severe acute respiratory distress syndrome (ARDS) due to AH1N1 influenza. He presented severe hypoxemia despite the use of mechanical ventilation, neuromuscular blocking agent infusion and prone position. VV ECMO was used, and coagulation was stopped on ECLS day 6 due to severe pulmonary hemorrhage. The systemic hemostatic disorders found in this patient were difficult to differentiate from disseminated intravascular coagulation (DIC) or sepsis-induced coagulopathy (SIC), improved transiently after circuit exchange, and resolved only after discontinuation of ECMO. The patient was discharged fully conscious and cooperative, with no apparent neurological deficit. Conclusions: Systemic hemostatic abnormalities may precede oxygenator failure and mimic DIC or SIC. Timely oxygenator exchange may therefore be considered. However, it is a high-risk procedure, especially in fully ECLS-dependent patients. Full article

Background and Clinical Significance: Denture stomatitis is a clinical manifestation of oral candidiasis, often seen in individuals wearing removable dentures that lead to the formation of sub-prosthetic stomatitis. This is particularly common in maladjusted appliances that have been in use for many years. Studies have shown that patients with systemic diseases such as diabetes, or other medical complexities, have a higher likelihood of developing denture stomatitis. To address this problem, the use of implant-retained dentures with different types of attachments has been introduced, providing increased comfort and hygiene for edentulous patients. However, this solution is not without its own set of challenges, being that the prolonged contact with mucosal surfaces can lead to challenges in cleaning and managing plaque depending on the attachment. Case Presentation: In this clinical case report, we present a female patient who developed bar-retained prosthetic stomatitis induced by oral candidiasis a few months after receiving her provisional prosthesis. Conclusions: In conclusion, this case report emphasizes the need to consider both systemic and local factors when preventing and treating denture stomatitis. By understanding the risk factors involved, healthcare professionals can provide their patients with the best possible care, helping to reduce the prevalence of oral candidiasis in individuals who use implant-retained dentures. Full article

Background and Clinical Significance: Biliary tract injuries are a recognized complication of laparoscopic cholecystectomy. Early diagnosis and prompt management are crucial to minimize complications such as bile leaks, strictures, and fistula formation. This case report highlights the use of a biodegradable biliary stent in managing a complex biliary injury and discusses the impact of delayed diagnosis on treatment outcomes. Case Presentation: We present the case of a 30-year-old male who sustained a Strasberg E2 biliary tract injury during a laparoscopic cholecystectomy. Initially misdiagnosed, the injury was only recognized on the fourth postoperative day. The patient underwent a Roux-en-Y hepaticojejunostomy and subsequently developed a postoperative biliary fistula, which was managed with percutaneous drainage. A biodegradable biliary stent was later placed to address a stricture and minimize the need for future interventions. One year later, the patient presented with symptoms of cholangitis, and radiological findings revealed a narrowing of the biliary lumen. The stricture was resolved and an endoscopic gastrojejunal shunt was placed to prevent further complications. The patient is currently in good condition with no signs of further complications. Conclusions: This case emphasizes the importance of early diagnosis in managing biliary tract injuries and highlights the potential of biodegradable stents to reduce the need for repeat interventions. Despite a delayed diagnosis necessitating complex surgical procedures, the use of a biodegradable stent proved effective in managing postoperative complications. Further studies are needed to evaluate the long-term efficacy of biodegradable stents in similar clinical scenarios. Full article

Background and Clinical Significance: Heart failure and kidney diseases often coexist and are difficult to clinically manage. Dysfunction in either organ exacerbates dysfunction in the other, potentially leading to cardiorenal syndrome (CRS). CRS has five different subtypes, with CRS type 5 being the most problematic given that it consists of an acute insult superimposed upon chronic CRS. Additionally, type 5 CRS can be complicated by heart failure-related cardiogenic shock (HF-CS), which is associated with increased hospitalizations and has a high 1-year mortality rate. The standard treatment for patients with HF-CS consists of guideline-directed medical therapy for heart failure with reduced ejection fraction (HFrEF) as tolerated, along with inotropic therapies and surgical mechanical left ventricular (LV) support, guided by invasive hemodynamic monitoring. Case Presentation: This case study reports the presentation of a 57-year-old man who presented with type 5 CRS who rapidly decompensated to stage E HF-CS and was effectively and safely treated with aggressive intravenous hydration, a defined plant-based diet (DPBD), and reduction of guideline-directed prescription medications without invasive hemodynamic monitoring. Conclusions: Hydration, a DPBD, and a reduction in medication burden may be effective in CRS. Pilot studies are warranted to evaluate the efficacy of this intervention in CRS in a larger cohort. Full article

Background and Clinical Significance: Among the odontogenic tumors, ameloblastoma is one of the most notorious, although it remains relatively rare, accounting for approximately one percent of all oral tumors. This neoplasm, derived from odontogenic epithelium, may arise from the developing enamel organ, epithelial cell rests of dental lamina, epithelial lining of odontogenic cysts, and basal cells of oral epithelium. This is a case presentation of a mural unicystic ameloblastoma, the most aggressive subtype and the one with the highest chance of recurrence. Case Presentation: A patient was referred by his dentist for root canal treatment at the Emergency Dental Clinic of Boston University. The patient complained of mandibular numbness. A panoramic radiograph was acquired, revealing a radiolucent lesion in the right mandible. Clinical examination detected a soft swelling perforating the buccal cortex in the area of #27–#30. A Cone-Beam CT (CBCT) was acquired in the Oral and Maxillofacial Radiology Clinic revealing a well-defined, partially corticated entity in the periapical area of teeth #27 through #30, with evidence of scalloping borders. The internal structure was unilocular and uniformly low-density. The entity caused interruption of the lamina dura of the associated teeth and inferior displacement of the inferior alveolar canal. Differential diagnoses included unicystic ameloblastoma (UA) and central giant cell granuloma as a second less likely diagnosis. An incisional biopsy was performed for further evaluation. Biopsy confirmed UA with mural involvement. Conclusions: UAs typically exhibit less aggressive behavior. However, cases like this one, where mural involvement is noted and no associated impaction is detected, underline the possibility of variable radiographic presentation and the significance of a multidisciplinary approach in correct diagnosis and treatment. Histological subtyping is crucial for guiding treatment. Full article

A healthy 54-year-old man previously presented to vascular surgeons with a 4-year history of swelling of the penis and scrotum was scheduled for ultrasound evaluation in the angiology office in our department. At presentation, there was a giant enlargement of the penis and scrotum, without swelling of the legs. Ultrasound evaluation was negative for vascular abnormalities. A diagnosis of chronic lymphatic disease was suspected; thus, a lymphoscintigraphy was performed. This test was normal showing, a good visualization of major lymphatics. The patients had a history of psoriasis with a documented previous event of flexural psoriasis involving his genitals with secondary infection 4 years before. Since that infection, his genitals progressively increased in size, and despite medical treatment and different surgical evaluations, the patient’s symptoms have not resolved, with marked disability associated with walking and sexual activity. Full article

Background and Clinical Significance: Radiation recall dermatitis (RRD) following immune checkpoint inhibitor (ICI) therapy has been infrequently reported. Case Presentation: We present a 47-year-old female patient who developed RRD of the breast following three doses of pembrolizumab administered as an adjuvant treatment post-nephrectomy for Stage III renal cell carcinoma (RCC). Notably, the affected breast had previously undergone external beam radiotherapy 247 weeks earlier for Stage IA invasive ductal carcinoma. She had received no prior chemotherapy at any point. RRD manifested as breast induration, erythema, and peau d’orange, and contraction of breast volume was noted following three cycles of pembrolizumab on week 17 (400 mg dose every 6 weeks). The dermatitis responded rapidly to systemic corticosteroids and no treatment interruption was needed. Conclusions: To date, this is the longest reported interval from completion of radiotherapy to RRD. A literature search underscores the variability in presentation and management of ICI-associated RRD. Full article

Background and Clinical Significance: Chryseobacterium gleum is a Gram-negative opportunistic and emerging pathogen able to induce systemic manifestations (e.g., peritonitis, pneumonia, urinary tract infections, meningitis) in immunocompromised patients. No data on children have been published. Case Presentation: A 2-year-old child presented in the pediatric ambulatory room with recurrent fever, submandibular lymphadenopathy, and skin rash. Laboratory findings revealed the presence of microcytic anemia with an increase in c-reactive protein. Chest X-ray reported mild accentuation of the bronchial structure, especially on the right side and middle–lower zone. In the peripheral blood smear, anisopoikilocytosis and elliptical red cells were evident. Clinical evaluation revealed the presence of conjunctivitis and polymorphic erythema, hyperemic pharynx and tonsils, SPO2 99%, auscultation of the chest, harsh vesicular murmur all over the area, and some wheezing. Microbiological analysis of sputum and throat swabs revealed the presence of numerous colonies of Chryseobacterium gleum confirmed using matrix-assisted laser desorption/ionization time-of-flight mass spectrometry (MALDI-TOF MS score > 2.2). Conclusions: This is the first case of Chryseobacterium gleum post-COVID in a child. We suggest that a quick identification and an appropriate treatment represent the critical factors able to prevent the adverse outcomes related to C. gleum infection. Full article

Background: The systemic stress response to surgery is a complex physiological process characterized by neuroendocrine, sympathetic, and inflammatory activation. While necessary for survival, this response can lead to adverse outcomes such as hyperglycemia, immune suppression, cardiovascular complications, and delayed recovery. Regional anesthesia (RA) has been shown to modulate this stress response more effectively than general anesthesia (GA) by blocking nociceptive signaling and attenuating the release of stress mediators. Objectives: This review aims to elucidate how RA influences the systemic stress response, highlighting its clinical benefits in reducing postoperative pain, improving hemodynamic stability, minimizing inflammatory responses, and preserving immune function. Additionally, this review examines evidence from clinical trials supporting using RA to improve surgical outcomes, particularly in high-risk populations. Methods: A comprehensive narrative review of the literature was conducted to explore the physiological impact of RA on the systemic stress response and its associated clinical outcomes. Studies comparing RA to GA across various surgical procedures were evaluated, focusing on neuroendocrine modulation, sympathetic inhibition, inflammatory attenuation, and the implications for pain management, cardiovascular and pulmonary function, and immune preservation. Results: RA significantly attenuates the neuroendocrine response by reducing the release of cortisol and catecholamines, thereby improving hemodynamic stability and reducing myocardial oxygen consumption. RA also inhibits the sympathetic nervous system, leading to improved cardiovascular outcomes. Furthermore, RA mitigates the inflammatory response by reducing pro-inflammatory cytokine levels, reducing the risk of systemic inflammatory response syndrome (SIRS), sepsis, and pulmonary complications. Clinical studies and meta-analyses consistently demonstrate that RA reduces postoperative pain, opioid consumption, and the incidence of cardiovascular and pulmonary complications, particularly in elderly and high-risk patients. Conclusions: RA offers a significant advantage in modulating the systemic stress response to surgery, improving postoperative outcomes by reducing pain, enhancing cardiovascular stability, and preserving immune function. Its benefits are particularly pronounced in high-risk populations such as the elderly or those with pre-existing comorbidities. Given the growing evidence supporting its efficacy, RA should be considered a critical component of multimodal perioperative care strategies aimed at minimizing the systemic stress response and improving recovery. Future research should optimize RA techniques and identify patient-specific factors to enhance therapeutic benefits. Full article

Objective: The present study investigates the incidence of postoperative acute kidney injury (AKI) and related risk factors in patients undergoing total joint arthroplasty. Methods: Included in the study were patients undergoing joint arthroplasty in 2015–2020. The patients with acute or chronic renal failure were excluded. The participants’ demographical data, American Society of Anesthesiologist (ASA) score, Charlson Comorbidity Index (CCI), type of operation, duration of surgery, presence of comorbidities, preoperative anemia, preoperative albumin levels, use of nephrotoxic agents, number of transfusions during perioperative period, presence of AKI according to Kidney Disease Improving Global Outcome (KDIGO) scores, and length of hospital and intensive care unit stay were evaluated. Results: The study was initiated with 1780 patients: 113 patients were excluded due to chronic kidney failure, 108 patients were excluded due to acute kidney failure, 648 patients were excluded because their data could not be reached, and finally, 911 patients were included in the study. AKI was detected in 134 patients (14.7%), and the number of patients in the KDIGO1 and KDIGO2 groups were 120 and 14, respectively. When evaluated according to the variable significance test result and clinical significance, the model consists of variables such as ASA, CCI, hypertension, nonsteroidal anti-inflammatory drugs (NSAIDs), vancomycin, beta lactam, contrast material and preoperative anemia, operation type, and anesthesia management. Machine learning analyses were performed using 11 variables (10 independent and 1 dependent variable). Logistic regression, naive Bayes, multilayer perceptron, bagging, and random forrest approaches were used for evaluation of the predictive performance. In an evaluation of the true classification ratio, the best result was obtained with the logistic regression method at 85.2%. Conclusions: The study revealed advanced age, high ASA and CCI, presence of diabetes and hypertension, NSAID, vancomycin and contrast material, and the presence of preoperative anemia to be independent risk factors for AKI. Full article

Background and Clinical Significance: The plantar plate (PP) tear of the second metatarsophalangeal joint (MTPJ) is a common cause of forefoot pain in clinical practice. The PP is the main stabilizing structure of the joint and, together with the collateral ligaments, is the key to maintaining the stability of the MTPJ. Many surgical procedures have been described to repair PP tears. Currently, there is still controversy regarding which is the surgical superior option (direct versus indirect PP repair techniques). Transfer of the flexor digitorum longus tendon to the dorsum of the proximal phalanx is one of the surgical techniques described to treat PP tears associated with MTPJ instability. Case Presentation: We present two cases that developed instability of the second MTPJ secondary to chronic PP tear with symptoms resolved after transfer of the flexor digitorum longus (FDL). Conclusions: Currently, the literature review shows that the procedure seems to be the most consistent surgical option in chronic cases of PP tears. Full article

Background: Multinodular Vacuolating Neuronal Tumors (MVNTs) are mixed glial–neuronal brain lesions classified as World Health Organization (WHO) CNS grade 1 tumors, often associated with long-term epilepsy. First described by Huse et al. in 2013 and included in the WHO CNS classification in 2016, MVNTs present a range of clinical manifestations, from symptomatic to asymptomatic. They typically affect young to middle-aged adults and exhibit diverse presentations. Radiologically, MVNTs are usually supratentorial, frequently located in the temporal lobe but also observed in the frontal and parietal lobes. MRI is essential for diagnosis, revealing multiple coalescing subcortical or cortical nodules with hyperintense signals on T2-weighted/FLAIR sequences, often without peripheral edema or mass effects. Case Reports: This paper presents two cases: one symptomatic MVNT with significant clinical manifestations, and the other documenting an incidental finding of MVNT in an asymptomatic patient. One case shows typical temporal lobe localization, while the other highlights a rare frontal lobe localization, with clear radiological findings on T2/FLAIR sequences. Conclusions: These cases illustrate the varied clinical presentations of MVNTs and emphasize MRI’s critical role in diagnosis and management. Asymptomatic cases often require conservative management, stressing the avoidance of unnecessary invasive procedures and the importance of regular monitoring. Full article

Background: Licorice (glycyrrhiza glabra) cough syrup intoxication is manifested with refractory hypokalemia, hypertension, and metabolic alkalosis. The transformation of glycyrrhiza glabra metabolic into glycyrrhetic acid after ingestion further inhibits the 11-β-hydroxysteroid dehydrogenase-2 enzyme, impeding the conversion of cortisol into cortisone. The accumulation of cortisol can also stimulate mineralocorticoid receptors, which leads to a pseudo-hyperaldosteronism-like effect. Case Presentation: We report a 60-year-old male patient with licorice intoxication due to the chronic consumption of licorice cough syrup. He exhibited a transient seizure lasting approximately one minute. Initially, hypokalemia (potassium level was 2.0 mmol/L), metabolic alkalosis, and QT interval prolongation with premature ventricular complexes were demonstrated on his electrocardiogram. Despite the administration of both intravenous and oral potassium supplements over two days, there was no significant improvement in hypokalemia. Spironolactone, an aldosterone receptor antagonist, was administered in addition to ongoing potassium supplementation from the 3rd day. This intervention led to a rapid normalization of hypokalemia in one day. The patient was ultimately discharged on the 6th day without any subsequent complications. Conclusions: The licorice-induced chronic intoxication, which led to pseudo-hyperaldosteronism and refractory hypokalemia, was successfully managed with aggressive potassium supplementation and spironolactone treatment. Full article

Background and Clinical Significance: Immune checkpoint inhibitors (ICIs) have revolutionized cancer treatment but may underlie diverse and potentially life-threatening immune-related adverse events (irAEs). They may cause various conditions leading to respiratory failure, including myasthenic syndromes and myositis. However, diaphragmatic paralysis (DP) has rarely been reported. To describe patients with diaphragmatic paralysis in a pharmacovigilance registry, we searched the prospective REISAMIC registry at the Gustave Roussy Cancer Center (Villejuif, France) for cases of diaphragmatic palsy (DP) occurring from September 2014 to December 2021. Case Presentation: We identified three patients, in whom DP was confirmed by diaphragmatic ultrasonography, pulmonary function tests, and/or diaphragmatic electroneuromyogram. Diaphragmatic palsy was life-threatening in all patients, as it caused respiratory failure requiring mechanical ventilation. In all cases, a pre-existing subclinical paraneoplastic syndrome was detected. Onconeural antibodies (anti-titin and anti-VGCC) were detected in these patients before and after the initiation of ICI therapy, suggesting a mixed paraneoplastic syndrome with features overlapping those of myasthenic syndrome (myasthenia gravis in one patient and Lambert–Eaton syndrome in another) and myositis. Conclusions: Diaphragmatic palsy is a severe irAE potentially resulting from different mechanisms, including myositis and neuromuscular junction involvement (myasthenia gravis, Lambert–Eaton). Antineuronal antibodies associated with such conditions were already present in our patients prior to immunotherapy initiation, suggesting ICIs could trigger flare-ups of pre-existing silent paraneoplastic autoimmune conditions. Full article

Background and Clinical Significance: The recombinant zoster vaccine (Shingrix) helps prevent shingles and its complications in adults 50 and older. While minor side effects are common, severe adverse reactions are thought to be rare, and long-term side effects are not well documented. Case Presentation: A 50-year-old woman with Crohn’s disease developed joint pain, effusion, and neurological symptoms such as numbness and tingling shortly after receiving the first dose of the recombinant zoster vaccine. Symptoms waxed and waned but persisted for over a year despite anti-inflammatories and specialist referrals. Diagnostic imaging and labs were unrevealing. Conclusions: This case of prolonged somatic and neurological symptoms associated temporally with Recombinant zoster vaccine administration reinforces the critical need for thorough pharmacovigilance and investigation of possible long-term adverse vaccine reactions. Provider documentation and reporting of individual experiences can help improve the detection of rare vaccine-related risks, elucidate potential risk factors, and refine safety screening. Diligent monitoring and research into longitudinal vaccine outcomes remain paramount, especially following recent emergency authorizations. Full article

Background and Clinical Significance: Tuberculosis (TB) poses a significant global health challenge; although low–middle income countries carry the heaviest burden, its diagnosis and treatment can be challenging in any country. The clinical picture can be complex and vary from person to person, with autoimmune complications that can hinder TB diagnosis and treatment. Case Presentation: We report the case of a 38-year-old man from Bangladesh who had recently arrived in Italy through the Balkan route. He presented with TB in the cervical lymph nodes and long-standing chronic myalgias. While a wide range of TB-triggered autoimmune entities can be found in the literature, this case is the first to describe immune-mediated necrotizing myopathy (IMNM) triggered by active TB. Conclusions: IMNM has been previously associated only with other infections like SARS-CoV-2 and Dengue. The successful diagnosis and management of TB-induced IMNM was achieved through a collaborative, multidisciplinary approach involving rheumatologists, immunologists, and infectious diseases specialists, showcasing an innovative treatment strategy and adding new insights into the complexities of TB and IMNM. Full article

Background and Clinical Significance: The treatment for pediatric Crohn’s disease (CD) has shifted over the years from steroids and immunomodulators to biologics with the goal of histological and clinical remission. Endoscopic balloon dilation (EBD) has been utilized for stricturing disease, even in the pediatric population. EBD has been shown to be effective and minimally invasive, though historically, has been performed on patients with persistent mucosal inflammation. As such, intralesional corticosteroid (ILC) injections have been traditionally utilized during EBD. However, intralesional corticosteroid efficacy among pediatrics patients in deep endoscopic remission is unknown. Case Presentation: We report four patients that demonstrated at least initial successful dilations without intralesional steroid injections. Conclusions: The use of ILC injections during routine EBDs in pediatric patients should be further explored in randomized control trials. Full article