Partial facial paralysis induced by sialolithiasis of the parotid gland: a case report.

BACKGROUND: Facial paralysis due to parotid sialolithiasis-induced parotitis is a unusual clinical phenomenon that has not been reported in prior literature. This scenario can present a diagnostic challenge due to its rarity and complex symptomatology, particularly if a patient has other potential contributing factors such as facial trauma or bilateral forehead botox injections as in this patient. This case report elucidates such a complex presentation, aiming to increase awareness and promote timely recognition among clinicians. CASE PRESENTATION: A 56-year-old male, with a medical history significant for hyperlipidemia, recurrent parotitis secondary to parotid sialolithiasis, and recent bilateral forehead cosmetic Botox injections presented to the emergency department with right lower facial drooping. This onset was about an hour after waking up and was of 4 h duration. The patient also had a history of a recent ground level fall four days prior that resulted in facial trauma to his right eyebrow without any evident neurological deficits in the region of the injury. A thorough neurological exam revealed sensory and motor deficits across the entirety of the right face, indicating a potential lesion affecting the buccal and marginal mandibular branches of the facial nerve (CN VII). Several differential diagnoses were considered for the lower motor neuron lesion, including soft tissue trauma or swelling from the recent fall, compression due to the known parotid stone, stroke, and complex migraines. An MRI of the brain was conducted to rule out a stroke, with no significant findings. A subsequent CT scan of the neck revealed an obstructed and dilated right Stensen's duct with a noticeably larger and anteriorly displaced sialolith and evidence of parotid gland inflammation. A final diagnosis of facial palsy due to parotitis secondary to sialolithiasis was made. The patient was discharged and later scheduled for a procedure to remove the sialolith which resolved his facial paralysis. CONCLUSIONS: This case emphasizes the need for a comprehensive approach to the differential diagnosis in presentations of facial palsy. It underscores the potential involvement of parotid sialolithiasis, particularly in patients with a history of recurrent parotitis or facial trauma. Prompt recognition of such uncommon presentations can prevent undue interventions, aid in timely appropriate management, and significantly contribute to the patient's recovery and prevention of long-term complications.

Is seminal quality worsening? A 20-year experience in Córdoba, Argentina.

PURPOSE: To assess the possible variations in semen quality during the last 20 years in Córdoba, Argentina, and to identify possible causal lifestyle or genitourinary factors. METHODS: Retrospective study of 23,130 patients attending an andrology laboratory. The 20-year period (2001-2020) was divided into four quinquenniums. Seminal parameters (sperm concentration, motility, morphology, viability, and membrane functional integrity) were classified as normal or abnormal according to WHO, and results were expressed as percentage of patients abnormal for each parameter per quinquennium. In addition, the percentage of patients per quinquennium exposed to the different risk factors (daily alcohol and/or tobacco consumption; occupational exposure to heat or toxics; history of parotitis or varicocele; and high body mass index, BMI) was reported. RESULTS: Patients included in our study did not show impairment in seminal quality over time. Beyond a transient decrease in normozoospermia in the second and third quinquennium, possibly explained by a parallel increase in teratozoospermia, other important parameters of the spermogram did not change. In fact, abnormalities in sperm concentration (oligozoospermia), total sperm count, viability and response to hypoosmotic test showed a decreasing trend over time. On the other hand, parotitis, varicocele, morbid obesity and regular exposition to heat/toxics were the factors more frequently associated with semen abnormalities; the last two increased their frequency over the study period. CONCLUSION: The population included in this study did not show a clear impairment in semen quality during the last 20 years. The decreasing patterns found were associated with high BMI and exposure to heat/toxics.

The effect of point-of-care ultrasound on length of stay in the emergency department in children with neck swelling.

BACKGROUND: Ultrasound is the imaging modality of choice in children presenting to the emergency department (ED) with soft tissue neck swelling. Point of care ultrasound (POCUS) has good accuracy when compared to comprehensive radiology department ultrasound (RADUS). POCUS could potentially improve ED length of stay (LOS) by improving efficiency. We aimed to evaluate the LOS of pediatric patients seen in ED with soft tissue neck swelling who received POCUS compared to RADUS. We determined unscheduled 30-day return visit rates in both groups as a balancing measure. METHODS: We performed a retrospective review of the electronic medical record for our cross-sectional study of discharged patients ≤21 years of age who had a neck ultrasound performed by a credentialed POCUS physician or by the radiology department between July 2014 and January 2020. We included patients who had both POCUS and RADUS in the POCUS group. We compared median ED LOS in both groups using the Mann Whitney U test and proportion of unscheduled return visits to the ED in both groups using odds ratio and 95% CI. RESULTS: There were 925 patients: 76 with only POCUS, 6 with POCUS and RADUS, and 843 with only RADUS performed. Median LOS in the POCUS group was 68.5 min (IQR 38.3120.3) versus 154.0 min (IQR 111.0, 211.0) in the RADUS group (p < 0.001). Return visit overall was 7.6%: 13.2% in the POCUS group versus 7.1% in the RADUS group (p = 0.07). CONCLUSION: Pediatric patients evaluated in the ED for soft tissue neck swelling had a shorter LOS with POCUS than with RADUS without a statistically significant increase in 30-day return visits. We suggest a "POCUS First" approach to the care of these patients.

Parotitis on Ultrasound: The Pomegranate Sign.

The differential diagnosis for facial swelling is broad and can be a diagnostic challenge in the pediatric emergency department. We describe the first pediatric case of acute parotitis with sialolithiasis where the diagnosis was facilitated by point-of-care ultrasound.

Coincidence of granulomatosis and polyangiitis with atypical clinical manifestation and antiphospholipid syndrome.

Granulomatosis and angiitis (GPA) is a multisystemic disease characterized by a granulomatous inflammation, tissue necrosis, and vasculitis of small and medium-sized blood vessels. Although the disease has a predilection for the upper respiratory tract, lungs, and kidneys, any organ system may be affected. Here, we present a case of generalized GPA manifested initially by necrotizing isolated parotitis and later by pulmonary-renal syndrome. Simultaneously with pulmonary hemorrhage, our patient developed an antiphospholipid syndrome (APS) presenting with deep vein thrombosis and strongly positive lupus anticoagulant. To the best of our knowledge the coincidence of parotitis and pulmonary-renal syndrome due to GPA and APS has never been reported previously. Concomitant venous thromboembolism may be life-threatening in a patient with GPA. Early diagnosis and institution of the proper therapy are critical in order to prevent organ damage.

Idiopathic recurrent pneumoparotitis.

Parotid swelling in a child can represent any of a variety of pathologies, including lymphadenitis, autoimmune disorders, other inflammatory conditions, vascular malformations, or neoplasms. Parotitis is usually infectious in etiology and is generally treated with antibiotics and supportive management. We report a case of a child with recurrent idiopathic pneumoparotitis, and a review of literature and the disease process is provided.

Salivary gland ultrasound in clinical practice: What is its real usefulness?

BACKGROUND: Salivary gland ultrasound (SGU) provides information about structural gland abnormalities that can be graded and used for primary Sjögren's syndrome (pSS) diagnosis. Its potential role as a prognostic marker for detecting patients at high risk of lymphoma and extra-glandular manifestations is still under evaluation. We aim to assess the usefulness of SGU for SS diagnosis in routine clinical practice and its relationship with extra-glandular involvement and lymphoma risk in pSS patients. METHODS: We designed a retrospective observational single-center study. Data was collected using the electronic health records of patients referred to an ultrasound outpatient clinic for evaluation over a 4-year period. Data extraction included demographics, comorbidities, clinical data, laboratory tests, SGU results, salivary gland (SG) biopsy, and scintigraphy results. Comparisons were made between patients with and without pathological SGU. The external criterion for comparison was the fulfillment of the 2016 ACR/EULAR pSS criteria. RESULTS: A total of 179 SGU assessments were included from this 4-year period. Twenty-four cases (13.4%) were pathological. The most frequently diagnosed conditions prior to SGU-detected pathologies were pSS (9.7%), rheumatoid arthritis (RA) (13.1%), and systemic lupus (4.6%). One hundred and two patients (57%) had no previous diagnosis (sicca syndrome work-up); of these, 47 patients (46.1%) were ANA positive and 25 (24.5%) anti-SSA positive. In this study, the sensitivity and specificity of SGU for SS diagnosis were 48% and 98% respectively, with a positive predictive value of 95%. There were statistically significant relationships between a pathological SGU and the presence of recurrent parotitis (p=.0083), positive anti-SSB antibodies (p=.0083), and a positive sialography (p=.0351). CONCLUSIONS: SGU shows high global specificity but low sensitivity for pSS diagnosis in routine care. Pathological SGU findings are associated with positive autoantibodies (ANA and anti-SSB) and recurrent parotitis.

Recurrent inflammation of accessory parotid tissue associated with unilateral parotid gland aplasia: diagnostic and therapeutic implications.

Aplasia of the major salivary glands is a rare condition due to an alteration in the development of the ectodermal tissue of the oral cavity often related to other craniofacial abnormalities or alteration of structures deriving from the first or second archial branch, in particular the lacrimal glands; it can be total or partial and determine clinical states ranging from an asymptomatic condition to a severe xerostomia. The accessory parotid tissue is similar to normal parotid tissue, completely independent from the main gland and susceptible to the same pathological disorders. We describe a very unusual case of an inflammatory disorder of accessory parotid tissue in a 44-year-old male patient with concomitant, and previously unknown, aplasia of the main ipsilateral parotid gland. We also discuss the role of imaging and conservative therapeutic modalities such as botulinum toxin therapy and, in the future, minimally invasive endoscopic-assisted resection in the management of such salivary disorder.

Pulmonary manifestations of childhood-onset primary Sjogren's syndrome (SS) masquerading as reactive airways disease in a male patient and review of interstitial lung disease associated with SS.

BACKGROUND: Sjogren's syndrome (SS) is a rare chronic autoimmune disease involving exocrine glands presenting with sicca syndrome, recurrent parotitis and other extraglandular stigmata. SS is well characterized in the adult population with classification criteria; however, primary SS presenting in childhood is poorly defined and rare in males. Recurrent parotitis is the most common presenting symptom in children with primary SS; however, clinical phenotype in children appears more variable than in adults. The lungs are a common extraglandular location for manifestations of primary SS. However, interstitial lung disease (ILD) is rare in children with primary SS. There are only four published reports of ILD associated with primary SS in female children. Here, we present a very rare case of primary SS in a pediatric male with pulmonary manifestations and review of the literature on ILD in childhood-onset primary SS. CASE PRESENTATION: A 14-year-old White male with a history of chronic severe asthma, recurrent parotitis and idiopathic intracranial hypertension was referred to pediatric rheumatology for evaluation of a positive ANA. In early childhood, he was diagnosed with persistent asthma recalcitrant to therapy. At age 8, he developed recurrent episodes of bilateral parotitis despite multiple treatments with sialoendoscopy. At age 14, respiratory symptoms significantly worsened prompting reevaluation. Lab workup was notable for positive ANA and Sjogren's Syndrome A and B antibodies. Pulmonary function tests showed only a mild obstructive process. Computed tomography of chest was significant for small airway disease, and lung biopsy was positive for mild interstitial lymphocytic inflammation presenting a conflicting picture for ILD. The constellation of findings led to the diagnosis of primary SS with associated pulmonary manifestations. He was treated with hydroxychloroquine, mycophenolate mofetil and oral corticosteroids with resolution of symptoms. CONCLUSIONS: Primary SS is a rare disease in the pediatric population that is poorly characterized. This case is the very rare presentation of childhood-onset primary SS with pulmonary manifestations in a male patient. ILD associated with primary SS is also very rare with only four pediatric patients reported in the literature. Collaborative effort is needed to develop pediatric specific diagnostic and treatment guidelines in this rare condition.

Giant tumorous lesions (correction of legions) surrounding the right coronary artery associated with immunoglobulin-G4-related systemic disease.

Immunoglobulin G4 (IgG4)-related systemic disease was first recognized as a clinicopathological entity about 10 years ago, and since then, it has attracted growing attention. It is an autoimmune disease which affects multiple organs including the pancreas, bile duct, salivary glands and retroperitoneum. Further, it was recently reported that it can be manifested as periarteritis, often as inflammatory abdominal aortic aneurysm. We describe the case of a 75-year-old man with autoimmune pancreatitis and parotitis who presented with angina. The serum concentration of IgG4 was significantly increased at 2,510 mg/dl. Coronary angiography showed multiple stenotic lesions and pronounced dilatation of the right coronary artery. Cardiac computed tomography disclosed increased wall thickness of the coronary arteries and focal tumorous lesions surrounding the right coronary artery. Treatment with steroids proved only marginally effective and he underwent surgical resection of the aneurysm and coronary artery bypass grafting. The diagnosis of IgG4-related systemic disease was confirmed by histological examination of the resected mass, which showed a massive infiltration of IgG4-positive plasma cells. This case emphasizes the importance of considering the diagnosis in any patient with abnormally increased wall thickness or ectatic lesions in the coronary arteries.

Parotid abscess: an unusual cause of facial nerve palsy.

Facial nerve palsy with a parotid mass is usually associated with malignant neoplasm of parotid gland. Its occurrence as a complication of parotid abscess is extremely rare. A literature review revealed only 16 cases of facial nerve palsy associated with suppurative parotitis or parotid abscess were reported. We present a case of deep parotid abscess which is complicated by facial nerve dysfunction. Underlying neoplasia was excluded.

A case of Kawasaki disease presenting with parotitis: A case report and literature review.

RATIONALE: Kawasaki disease affects multiple organ systems. Its typical symptoms include fever, rash, oropharyngeal mucosal erythema, bilateral non-exudative conjunctivitis, cervical lymphadenopathy, extremity changes, and membranous desquamation of the fingers and toes. In severe cases, cardiovascular, respiratory, musculoskeletal, gastrointestinal, neurological, and genitourinary complications may occur. In the early stage, Kawasaki disease is often manifested by uncommon symptoms, such as pyuria, meningitis, shock, and retropharyngeal or parapharyngeal abscess, which may delay diagnosis and treatment. We have reported a case of Kawasaki disease presenting with mumps and reviewed the clinical features of 14 other similar cases, in order to facilitate the early diagnosis and treatment of this unusual presentation of Kawasaki disease. PATIENT CONCERNS: A 10-year-old boy presented with persistent fever and parotitis and was diagnosed with suppurative parotitis. After antibiotic therapy, the parotid swelling reduced, but the fever persisted and other typical symptoms of Kawasaki disease appeared, including bilateral conjunctival hyperaemia, cervical lymphadenopathy, oropharyngeal mucosal erythema, membranous desquamation of the fingers, and left coronary artery widening. DIAGNOSES: The patient was diagnosed with Kawasaki disease 12 days after the onset of fever. INTERVENTIONS: The patient was administered γ-globulin 1.0 g/kg·d for 2 consecutive days and oral aspirin 5 mg/kg·d. OUTCOMES: The left coronary artery returned to a width of 3.8 mm after 1 month and of 3.1 mm after 3 months. The dose of aspirin was reduced to 3 mg/kg·d after 2 months and to 1.5 mg/kg·d after 3 months. LESSONS: Physicians should be aware that Kawasaki disease may develop after parotitis.

Simultaneous parotitis and ipsilateral herpes zoster ophthalmicus: coincidence?

A 43-year-old previously healthy man presented to the primary care clinic with concurrent ipsilateral viral parotitis and herpes zoster ophthalmicus. The patient experienced painful swelling below the right ear as well as painful vesicles on the right forehead, eyelid and cheek in the V1 dermatomal region. There were no lesions in the oral cavity or nose. Antibody titres confirmed active varicella zoster virus in the absence of mumps or herpes simplex virus 1 and 2 and unilateral parotitis were confirmed to be non-suppurative by the patient's primary care physician and the neurologist. Both conditions resolved within 3 weeks with appropriate treatment.

Necrotizing fasciitis of eyelid secondary to parotitis.

PURPOSE: Evaluation of a patient with necrotizing fasciitis of the eyelid spreading from parotid gland. METHODS: Interventional case report. The patient admitted with rapidly progressive swelling, redness, and pain of four eyelids, and whole face. Pathogenic microorganism was searched, laboratory tests were evaluated. Intensive medical treatment and necrotic wound debridement was performed. RESULTS: The patient had been receiving oral cephalosporin at the time she was referred to our hospital, but her infection had spread rapidly under this treatment. In addition to her eyelid necrosis, septic shock developed within 24 hours. No pathogenic microorganism was detected in cultures. The patient responded to intravenous sulbactam ampicillin, surgical debridement of necrotic wound and supportive medical care. Moderate ptosis was the only complication observed in this patient. Reconstructive surgery was not needed. CONCLUSIONS: With prompt antibiotic treatment and surgical debridement, cosmetic and functional success may be obtained in necrotizing fasciitis cases rarely seen with parotid gland origin.

Facial paralysis due to an occult parotid abscess.

Facial paralysis associated with benign diseases of the parotid gland is very rare. It has been reported in approximately 16 cases of acute suppurative parotitis or parotid abscess. We presented a 45-year-old woman who developed facial paralysis secondary to an occult parotid abscess. Initially, there was no facial paralysis and the signs and symptoms were suggestive of acute parotitis, for which medical treatment was initiated. Three days later, left-sided facial palsy of HB (House-Brackmann) grade 5 developed. Ultrasonography revealed a pretragal, hypoechoic mass, 10x8 mm in size, causing inflammation in the surrounding tissue. Fine needle aspiration biopsy obtained from the mass revealed polymorphonuclear leukocytes and lymphocytes. No malignant cells were observed. The lesion was diagnosed as an occult parotid abscess. After a week, the mass disappeared and facial paralysis improved to HB grade 4. At the end of the first month, facial paralysis improved to HB grade 1. At three months, facial nerve function was nearly normal.

[Parotid pneumocele in Down's syndrome]. / Neumocele parotídeo en el síndrome de Down.

Pneumoparotiditis is related to the cause of an infrequent increase in the size of the parotid gland due to the entrance of air through the Stenon duct following an increase in the pressure in the oral cavity. It is observed in musicians who play wind instruments, glass blowers, patients undergoing diagnostic or therapeutic techniques, in adolescents that provoke it intentionally, and in mentally handicapped people as found in the clinical case reported here. The symptoms are usually non-specific and the diagnosis is confirmed with an axial tomography. The interest in this clinical case lies in the peculiar form of presentation and its diagnostic confirmation through the use of CT imaging.