Unusual Case Research Articles

An unusual case of upper airways obstruction

Tracheal stenosis poses a diagnostic challenge that can mimic other respiratory pathologies, particularly bronchial asthma. It is characterized by a narrowing of the tracheal caliber, leading to respiratory symptoms such as dyspnea, cough, and wheezing. However, these manifestations can overlap with those of bronchial asthma, making the differential diagnosis a challenging yet crucial task for an appropriate therapeutic approach. This study aims to underscore the importance of a comprehensive clinical approach, utilizing bronchoscopy and global spirometry with bronchodilator reversibility testing, to differentiate tracheal stenosis from bronchial asthma, thereby contributing to accurate and timely surgical treatment. Bronchoscopy stands as a cornerstone in investigating tracheal stenosis, allowing for a direct assessment of the site, extent, and nature of the stenosis. Endoscopic observation of the morphological characteristics of the stenosis, such as concentric narrowing or mucosal irregularities, can guide towards a more precise diagnosis. Asthma, characterized by a reversible bronchial obstruction, responds positively to bronchodilators, whereas tracheal stenosis shows a limited or absent response.

An unusual case of primary splenic soft part alveolar sarcoma: case report and review of the literature with emphasis on the spectrum of TFE3-associated neoplasms

BackgroundAlveolar soft part sarcoma is a rare tumour of soft tissues, mostly localized in muscles or deep soft tissues of the extremities. In rare occasions, this tumour develops in deep tissues of the abdomen or pelvis.Case presentationIn this case report, we described the case of a 46 year old man who developed a primary splenic alveolar soft part sarcoma. The tumour displayed typical morphological alveolar aspect, as well as immunohistochemical profile notably TFE3 nuclear staining. Detection of ASPSCR1 Exon 7::TFE3 Exon 6 fusion transcript in molecular biology and TFE3 rearrangement in FISH confirmed the diagnosis.ConclusionWe described the first case of primary splenic alveolar soft part sarcoma, which questions once again the cell of origin of this rare tumour.

Giant unruptured sinus of Valsalva aneurysms causing angina pectoris

BackgroundSinus of Valsalva aneurysm (SVA) is a rare cardiac anomaly which can be congenital or acquired. Patients with SVA are commonly asymptomatic when the occupying effect of SVA is insignificant, while ruptured SVA usually causes severe symptoms including heart failure and myocardial ischemia.Case presentationWe present an unusual case of a 64-year-old female manifesting with exertional dyspnea as well as angina pectoris for three months. Echocardiography and cardiac computed tomographic angiography confirmed unruptured left-coronary and non-coronary SVAs. The left anterior descending artery and left circumflex artery were stretched and compressed by the SVA which causing myocardial ischemia. The patient finally received aortic root replacement (Bentall procedure) and got symptom relieved.ConclusionsGiant unruptured SVA originating from left coronary sinus is extremely rare. Our case highlights that giant SVA should be considered in cases with angina pectoris. Echocardiography and coronary computed tomographic angiography are useful and important for diagnosis. Surgery is highly recommended in patients with SVA.

Aberrant sparganosis in cat caused by Spirometra mansoni (Cestoda: Diphyllobothriidae): a case report

BackgroundSparganosis is a rare zoonotic disease caused by plerocercoid larvae of the genera Spirometra or Sparganum (Cestoda: Diphyllobothriidae). The larvae of Spirometra generally do not undergo asexual reproduction, whereas those of Sparganum can induce proliferative lesions in infected tissues. This paper presents an unusual case of proliferative sparganosis due to infection with Spirometra mansoni in a cat, normally considered a definitive host of the species.Case presentationA 9-year-old male domestic cat was presented with a mass on the right side of the face that underwent progressive enlargement for 1 month. The morphological and histopathological examinations revealed multiple asexual proliferative cestode larvae in the lesions, suggestive of proliferative sparganosis. Next-generation sequencing analysis of formalin-fixed and paraffin-embedded specimens of surgically excised tissue indicated that the worm was Spirometra mansoni.ConclusionAlthough S. mansoni a common tapeworm species found in the small intestine of domestic cats and dogs in Japan, proliferative sparganosis is extremely rare. This is the first confirmed case of proliferative sparganosis due to infection with S. mansoni in cat.

Successful management of pulmonary edema secondary to accidental electrocution in a young dog

BackgroundHuman records describe pulmonary edema as a life-threatening complication of electric shock. Successful management requires prompt recognition and intensive care. However, in companion animals, electrocutions are rarely reported, even though domestic environments are full of electrical devices and there is always the possibility of accidental injury. Therefore, it is important for veterinarians to know more about this condition in order to achieve successful patient outcomes.Case presentationA 3-month-old male Labrador Retriever was presented with a history of transient loss of consciousness after chewing on a household electrical cord. On admission, the puppy showed an orthopneic position with moderate respiratory distress. Supplemental oxygen via nasal catheter was provided, but the patient showed marked worsening of respiratory status. Point-of-care ultrasound exams suggested neurogenic pulmonary edema due to electrical shock close to the central nervous system and increased B-lines without evidence of cardiac abnormalities. Mechanical ventilation of the patient was initiated using volume-controlled mode with a tidal volume of 9 to 15 ml/kg until reaching an end-tidal carbon dioxide ≤ 40 mm Hg, followed by a stepwise lung-recruitment maneuver in pressure-controlled mode with increases of the peak inspiratory pressure (15 to 20 cm H2O) and positive end-expiratory pressure (3 to 10 cm H2O) for 30 min, and return to volume-controlled mode with a tidal volume of 15 ml/kg until reaching a peripheral oxygen saturation ≥ 96%. Weaning from the ventilator was achieved in six hours, and the patient was discharged two days after admission without neurological or respiratory deficits.ConclusionsWe present a rather unusual case of a neurogenic pulmonary edema subsequent to accidental electrocution in a dog. Timely diagnosis by ultrasound and mechanical ventilation settings are described. Our case highlights that pulmonary edema should be considered a potentially life-threatening complication of electrical shock in small animal emergency and critical care medicine.

Profound hypoxemia and hypotension during posterior spinal fusion in a spinal muscular atrophy child with severe scoliosis: a case report

BackgroundAnesthesia for spinal muscular atrophy (SMA) patients undergoing spinal deformity surgery is challenging. We report an unusual case of an SMA girl who developed severe intraoperative hypoxemia and hypotension during posterior spinal fusion related with surgical positioning.Case presentationA 13-yr-old girl diagnosed with SMA type 2, severe kyphoscoliosis and thoracic deformity was scheduled for elective posterior spinal fusion. She developed severe hypoxemia and profound hypotension intraoperatively in the prone position with surgical table tilted 45° to the right. Though transesophageal echocardiography (TEE) could not be performed due to limited mouth opening, her preoperative computed tomography revealed a severely distorted thoracic cavity with much reduced volume of the right side. A reasonable explanation was when the surgeons performed surgical procedure with the tilted surgical table, the pressure was directly put on the shortest diameter of the significantly deformed thoracic cavity, causing severe compression of the pulmonary artery, resulting in both hypoxemia and hypotension. The patient stabilized when the surgical table was tilted back and successfully went through the surgery in the leveled prone position.ConclusionsSpinal fusion surgery is beneficial for SMA patients in preventing scoliosis progression and improving ventilation. However, severe scoliosis and thoracic deformities put them at risk of both hemodynamic and respiratory instability during surgical positioning. When advanced monitoring like TEE is not practical intraoperatively, preoperative imaging may help with differential diagnosis, and guide the surgical positioning to minimize mechanical compression of the thoracic cavity, thereby helping the patient complete the surgery safely.

Type 1 secretion necessitates a tight interplay between all domains of the ABC transporter

Type I secretion systems (T1SS) facilitate the secretion of substrates in one step across both membranes of Gram-negative bacteria. A prime example is the hemolysin T1SS which secretes the toxin HlyA. Secretion is energized by the ABC transporter HlyB, which forms a complex together with the membrane fusion protein HlyD and the outer membrane protein TolC. HlyB features three domains: an N-terminal C39 peptidase-like domain (CLD), a transmembrane domain (TMD) and a C-terminal nucleotide binding domain (NBD). Here, we created chimeric transporters by swapping one or more domains of HlyB with the respective domain(s) of RtxB, a HlyB homolog from Kingella kingae. We tested all chimeric transporters for their ability to secrete pro-HlyA when co-expressed with HlyD. The CLD proved to be most critical, as a substitution abolished secretion. Swapping only the TMD or NBD reduced the secretion efficiency, while a simultaneous exchange abolished secretion. These results indicate that the CLD is the most critical secretion determinant, while TMD and NBD might possess additional recognition or interaction sites. This mode of recognition represents a hierarchical and extreme unusual case of substrate recognition for ABC transporters and optimal secretion requires a tight interplay between all domains.

Hematuria Due to Possible Histoplasma-Associated Urinary Bladder Pseudotumor With Negative Serologic and Urine Antigen Testing

Abstract Histoplasma capsulatum is a ubiquitous dimorphic fungus causing multiple infectious syndromes, ranging from subclinical to severe disseminated disease. We present an unusual case of hematuria due to pedunculated urinary bladder mass in an immunocompetent host. Although the gold standard for diagnosis of histoplasmosis is through demonstration of characteristic yeast forms on histopathologic examination of infected tissue, or observation of typical mycelial growth in culture of clinical specimens, investigational multiplex polymerase chain reaction of formalinized tissue was helpful in this case due to conflicting serologic testing, equivocal morphologic findings on histopathologic examination and, surprisingly, a negative urine Histoplasma antigen despite anatomically proximal location within the urinary bladder. Although antigen immunoassay and serology are commonly used proxy diagnostics in Histoplasma-associated disease, varying performance characteristics in certain disease states, such as cases of locally proliferative infection mimicking neoplastic growth similar to this report, may lead to elusive diagnosis.

Enterovirus Infection in Preterm Quadruplets with Clinical Presentation of Neonatal Late-Onset Sepsis: a Case Report.

This is the unusual case of concurrent late-onset sepsis caused by enteroviruses in preterm quadruplets in a neonatal ward. While viral infections are an important differential diagnosis of bacterial sepsis in the outpatient setting, the occurrence in a neonatal ward with strict prophylactic hygiene measures is unusual. In this case, transmission of enteroviruses via breast milk appears possible.

Unusual case of infertility reversal in a woman with atypical undiagnosed coeliac disease

Unusual case of infertility reversal in a woman with atypical undiagnosed coeliac disease

Fear, Dependency and Complicity in Late Eighteenth-Century Grenada, 1784–1796

ABSTRACT This article uses an unusual case of murder in the British, formally French, colony of Grenada in 1784 as a useful window to explore the themes of fear and dependency among planters, and complicity among enslaved and free Africans in a Caribbean slave colony at the end of the eighteenth century. The story of Korey, the enslaved nightwatchman, reprieved from the death penalty because of his worth to the plantocracy, highlights a process in the developing colony that saw large numbers of Africans in the colony employed outside of the planting regime throughout the 1780s and 1790s. By the end of the Fedon rebellion in 1795, armed Africans, some free some still enslaved, dominated the free spaces of the colony. The timing and unique circumstances of the rebellion and crucially the build up to it saw through a process in the colony that directly led to the formation of the West Indian Regiments. Grenada’s essential role in this process has been recognized but by marking the specific developments across the 1780s and 90s, we can see with more definition the interplay between planters and their fears, the military, the government, and Africans in this moment of genesis.

Severe hypocalcemia, an atypical presentation after thyroid cancer surgery, about a case

Hypoparathyroidism, secondary to postoperative thyroid cancer, is characterized by hypocalcemia, hyperphosphatemia, and hypercalciuria due to low concentrations of parathyroid hormone. The prevalence in the United States is 23 to 37 cases/100,000 people/year. Complications develop in 78 % of cases after cervical surgery; 75 % resolve spontaneously in the first 6 months, while the remaining 25 % resolve permanently; no incidence data were obtained in Bolivia. Treatment requires long-term oral calcium with citrate, calcium carbonate, and vitamin D analogs (calcitriol and alfacalcidol); in complex cases, calcium can be used intravenously (calcium gluconate); Some patients do not respond to standard treatment. In this way, the unusual clinical case of a 38-year-old male patient with a history of papillary thyroid carcinoma, post-total thyroidectomy, with a clinical picture of 48 hours of evolution characterized by pain at the cervical level, associated with a Trosseau sign, is presented. (+) and frank signs of respiratory distress with laryngeal spasm, which required management in the intensive care unit (ICU), correction of severe hypocalcemia, with calcium gluconate IV and calcium plus vitamin D PO; presenting an optimal evolution, graduating on the fourth day; He was subsequently discharged from the hospital two weeks later in good general condition.

Unusual Case of Recurrent Falls Secondary to Tumefactive Perivascular Spaces (P9-4.006)

Unusual Case of Recurrent Falls Secondary to Tumefactive Perivascular Spaces (P9-4.006)

Unusual Case of Plesiomonas Shigelloides Sepsis and Meningitis in a Neonate With Targetoid Vasculitic Rash.

Unusual Case of Plesiomonas Shigelloides Sepsis and Meningitis in a Neonate With Targetoid Vasculitic Rash.

Chronic stridor in a toddler after ingestion of a discharged button battery: a case report

BackgroundButton battery (BB) ingestions (BBI) are increasingly prevalent in children and constitute a significant, potentially life-threatening health hazard, and thus a pediatric emergency. Ingested BBs are usually charged and can cause severe symptom within 2 h. Discharged BBs ingestion is very rare and protracted symptom trajectories complicate diagnosis. Timely imaging is all the more important. Discharged BBs pose specific hazards, such as impaction, and necessitate additional interventions.Case presentationWe present the case of a previously healthy 19-month-old girl who was admitted to our pediatric university clinic in Germany for assessment of a three-month history of intermittent, mainly inspiratory stridor, snoring and feeding problems (swallowing, crying at the sight of food). The child’s physical examination and vital signs were normal. Common infectious causes, such as bronchitis, were ruled out by normal lab results including normal infection parameters, negative serology for common respiratory viruses, and normal blood gas analysis, the absence of fever or pathological auscultation findings. The patient’s history contained no evidence of an ingestion or aspiration event, no other red flags (e.g., traveling, contact to TBC). Considering this and with bronchoscopy being the gold standard for foreign body (FB) detection, an x-ray was initially deferred. A diagnostic bronchoscopy, performed to check for airway pathologies, revealed normal mucosal and anatomic findings, but a non-pulsatile bulge in the trachea. Subsequent esophagoscopy showed an undefined FB, lodged in the upper third of the otherwise intact esophagus. The FB was identified as a BB by a chest X-ray. Retrieval of the battery proved extremely difficult due to its wedged position and prolonged ingestion and required a two-stage procedure with consultation of Ear Nose Throat colleagues. Recurring stenosis and regurgitation required one-time esophageal bougienage during follow-up examinations. Since then, the child has been asymptomatic in the biannual endoscopic controls and is thriving satisfactorily.ConclusionThis case describes the rare and unusual case of a long-term ingested, discharged BB. It underscores the need for heightened vigilance among healthcare providers regarding the potential hazards posed by discharged BBIs in otherwise healthy children with newly, unexplained stridor and feeding problems. This case emphasizes the critical role of early diagnostic imaging and interdisciplinary interventions in ensuring timely management and preventing long-term complications associated even to discharged BBs.

Sodium-glucose co-transporter-2 inhibitor-associated non-diabetic ketoacidosis: a case report

Ketoacidosis is considered an emergency metabolic disorder that can be triggered by starvation and alcohol consumption in addition to diabetes. We described an unusual case of ketoacidosis in a non-diabetic young man who presented to our hospital with a complaint of rapid and shallow breathing, weakness and nausea. Fourteen days back, due to heart failure, he started sodiumglucose co-transporter 2 inhibitors (SGLT2-i) in addition to fumarate bisoprolol, sacubitril/valsartan and eplerenone. Based on clinical examination, ketoacidosis was suspected, although his glucose and glycosylated hemoglobin levels were in range. Overall, based on the clinical and laboratory findings, the diagnosis of euglycemic non-diabetic ketoacidosis due to the use of the SGLT2- i was made and SGLT2-i has been discontinued. Although SGLT2-i are effective and safe drugs, it is advisable to carefully evaluate their use also in non-diabetic patients especially when hepatic fibrosis or incorrect eating habits are present.

An unusual case of gallbladder ascariasis in pregnancy.

SynopsisGallbladder ascariasis in pregnancy presents with protracted nausea and vomiting.

Odontogenic Infection of the Infratemporal Fossa: A Diagnostic Challenge

The infratemporal fossa (IF) is an important anatomical space that carries vital neurovascular structures within it. The formation of an abscess in the IF, particularly of odontogenic aetiology, is rare. It can pose a diagnostic challenge due its uncommon presentation and can be life-threatening with delayed treatment. We present an unusual case of an IF abscess in an otherwise healthy female patient. Due to the rising difficulty accessing emergency NHS dental services, the untimely diagnosis and management of odontogenic infections may lead to similar scenarios. CPD/Clinical Relevance: It is important that clinicians have good knowledge of the anatomy of fascial spaces and be aware of signs and symptoms of spreading infections.

A Rare Case of Lemierre’s Syndrome due to Veillonella Parvula: A Dangerous and Forgotten Complication of a Septic Condition

AbstractThis clinical case presents an unusual case of Lemierre’s syndrome (LS) in a young woman of 38-year-old. She arrived in the Emergency Department with a high fever and pharyngology resistant to antibiotic therapy with clarithromycin, ceftriaxone, and cortisone for two weeks. At the blood sampling, there is a marked leucocytosis, and the advice of the otolaryngologist is required given the strong pain in the throat. Due to the tonsillar abscess, a neck CT with a contrast medium is necessary for the otolaryngologist’s opinion. The CT shows thrombosis of the jugular vein and left subclavian, with thickening of soft perivascular tissues; these findings suggest Lemierre’s syndrome: a septic thrombophlebitis of the jugular vein that occurs as a complication of a peritonsillar abscess. The diagnostic process is then completed with a chest HR-CT, which reveals lung density and excavation areas suggesting tuberculosis. Blood culture reveals the presence of Veillonella Parvula (an anaerobic gram-negative coccus), sputum culture reveals the presence of some colonies of Enterobacter cloacae complex, real-time PCR examination on sputum reveals the presence of Streptococcus Pneumoniae and the borderline presence of rhinovirus. Microbiologists, after these results and neck and chest CT with a contrast agent, agree with the diagnosis of suspected LS at an early stage: a septic dissemination fortunately limited only to the neck and lungs region.

Aesthetic and Functional Outcomes of Upper Blepharoplasty Combined with Dacryoadenopexy in Lacrimal Gland Prolapse.

Lacrimal gland prolapse (LGP) is a term used to describe a benign, anterior displacement of the lacrimal gland. If this condition is not properly addressed during upper blepharoplasty, the aesthetic and functional results are less than optimal. This study aimed to report the surgical outcomes of upper blepharoplasty combined with dacryoadenopexy in patients with LGP. We reported an unusual case of severe LGP in a young patient. In addition, we performed a systematic review of the English literature on surgical cases of LGP published between 1973 and 2023. Eligible articles were analyzed for individual patient data (IPD) and aggregate patient data (APD). The primary predictor variable was the surgical technique used for lacrimal gland repositioning/suspension. The primary outcome variable was relapse rate. The bibliographic search retrieved 488 surgical cases of LGP: 12 in IPD, and 476 in APD datasets. The relapse rates in the IPD and APD cohorts were 8.3% (1/12) and 1.2% (6/476), respectively. Within the APD dataset, no significant difference in the relapse rates between dacryoadenopexy via suture suspension and Whitnall's ligament suspension (5/409 and 1/20, respectively; P > 0.05) was observed. Light cauterization of the lacrimal gland capsule and surrounding soft tissues was performed in mild LGP cases (< 4mm prolapse), with a relapse rate of 0% (0/47). Upper blepharoplasty combined with dacryoadenopexy proved to be a safe surgical procedure with very satisfactory aesthetic outcome and minimal relapse rate. This journal requires that authors assign a level of evidence to each article. For a full description of these Evidence-Based Medicine ratings, please refer to the Table of Contents or the online Instructions to Authors www.springer.com/00266 .