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.

Juvenile idiopathic recurrent parotitis (JIRP) treated with short course steroids, a case series study and one decade follow up for potential autoimmune disorder.

BACKGROUND: Juvenile idiopathic recurrent parotitis (JIRP) in children is a condition characterized with recurrent episodes of idiopathic parotid gland inflammation. Since there are no definitive guidelines for diagnosis and management of this condition, we present a consecutive case series of patients with more than one decade follow up and their dramatic response to short course treatment by prednisolone. METHODS: We conducted this study by retrospectively reviewed medical charts of children who were diagnosed with JIRP, from 1 January 2002 to 29 February 2023. We performed usual serological tests to exclude some possible background. We administered short course prednisolone on first day of episode as divided dosage (0.5 mg /kg). RESULTS: In this case series of 10 patients (70%) were male, median age of onset was 5 years, duration of episodes 5 days, and the mean course of disease were 3.8 years. The average follows up of patients was near 10 years. In comparison with their natural course of disease all patients showed a dramatic response to treatment on the first day of administration of prednisolone (P Value 0.005). For ten years follow up there was not any additional accompanying autoimmune disorder. CONCLUSION: Short course prednisolone on first day of each episode and its dramatic and meaningful response in our patients, introduce a new, effective, fast, and inexpensive regimen of therapy in patients with JIRP.

Sialendoscopy approach in treating juvenile recurrent parotitis: a systematic review.

BACKGROUND: Juvenile recurrent parotitis (JRP) is characterized by recurrent episodes of painful parotid swelling in children. The purpose of this systematic review was to determine the diagnostic and therapeutic effectiveness of sialendoscopy in children affected by JRP. METHODS: A systematic literature search was performed in PubMed, EMBASE, Scopus and the Cochrane Library until April 2022, without language restrictions or specified start date. Quality assessment was performed using the Newcastle-Ottawa Scale (NOS). RESULTS: Our review included 524 patients and 646 sialendoscopies. The sample sizes of the different studies ranged from 3 to 77 subjects. Most authors performed sialendoscopy under general anesthesia. The mean percentage of recurrences observed was 25.1% (95% confidence intervals) (CI 23.6-26.6). There was a statistically significant relationship between the number of attacks/year and recurrences (p < 0.05). The percentage of recurrences according to the type of irrigation/flushing used ranged from 22.2% to 25.2%, with no significant differences between the use of corticosteroids alone (25.2% of recurrences), corticosteroids plus antibiotics (25% of recurrences) or saline alone (22.2% of recurrences). Sialoendoscopy has proved in all cases to be a valid method for the diagnosis of JRP, but it does not allow a reliable differential diagnosis with other autoimmune parotitis such as Sjögren's syndrome. CONCLUSION: According to our results, parotid sialoendoscopy was 74.9% effective as a primary treatment in the prevention of recurrent symptoms in JRP. The type of ductal irrigation used did not significantly influence the prognostic outcome.

Is Reflex Parotitis a Forgotten or an Unknown Pathology?

Reflex parotitis is a recurrent parotid inflammation caused by an imbalance in the parasympathetic innervation of the salivary gland. However, due to a poor understanding of its pathophysiology, it can go unnoticed. Causes of ignored norms can trigger these repetitive episodes of glandular disease with a difficult diagnosis and inadequate treatment.

Fungal abscess of the parotid gland - the value of microbiological assessment.

SUMMARY: Fungal parotitis is rare and the sequela parotid abscess exceedingly so. We report our experience with Candida glabrata and Candida albicans parotid gland abscesses in critically ill HIV-positive patients and highlight the value of microbiological assessment to tailor management.

Neonatal Parotitis: A Case Report and Review of the Literature.

BACKGROUND: Neonatal parotitis is a rare disease. Between 1970 and 2011 only 44 cases have been reported in the English literature. METHODS: In this case report, we describe a case of neonatal parotitis caused by Group B streptococcus (GBS). Additionally, we performed a review of the recent literature. We found 18 new cases published between 2011 and 2020. These cases were analyzed together with the 44 cases published before 2011. RESULTS: All patients presented with swelling over the parotid area, with varying degrees of local inflammation and general symptoms. Purulent discharge from the Stensen's duct was present in 85% of the patients. The swelling was usually unilateral (84%). In total 70% of the patients were male. Prematurity was reported in 29% of the cases. The most common isolated pathogen was Staphylococcus aureus (68%). Only 5 cases were found describing GBS as the causative pathogen in neonatal parotitis. In most of the cases treatment with intravenous antibiotics was successful, 27% of the patients needed surgical drainage. The reported outcomes were good. CONCLUSIONS: When comparing GBS cases and non-GBS cases there seems to be a difference in presenting symptoms and pathophysiology, with GBS patients presenting without purulent discharge form the Stensen's duct and with more severe generalized symptoms. Additionally, all GBS patients had a positive blood culture, compared to 27% of the non-GBS patients, which indicates that in GBS cases the major route of parotid infection is hematogenous, compared to a retrograde flow from the oral cavity to the parotid gland in non-GBS cases.

Bilateral Candidal abscess of the parotid gland: A case report and literature review.

We report the case of a 75-year-old diabetic man who developed a bilateral abscess of the parotid gland. Although a bacterial parotitis was first suspected, cultures of fine-needle aspiration identified Candida albicans as the causal agent. Failure of medical treatment led us to perform a surgical drainage of the right abscess. The patient was then treated with oral fluconazole for 6 weeks, with complete recovery during follow-up. Of interest, the patient had a history of right superficial parotidectomy for a benign tumor 30 years ago. Despite the high prevalence of oral carriage, fungal abscesses of the parotid gland are extremely rare and have only been reported in a few cases. This might be due to the fungal toxicity of the salivary proteins, like histatins. To our knowledge, this is the first report of a Candida albicans abscess of the parotid gland developed bilaterally.

COVID-19-associated parotitis and sublingual gland sialadenitis.

The COVID-19 mainly affects the respiratory system; however, a variety of atypical manifestations of this disease have been also reported. Herein, we report a case of a man in his late 50s with severe COVID-19 presenting with parotitis and sublingual gland sialadenitis. Six days after admission, swelling in the bilateral preauricular and lower jaw areas developed. CT demonstrated parotid and sublingual gland enlargement with surrounding fat stranding, indicating sialadenitis. This case suggests that, in the present COVID-19 pandemic, COVID-19 should be included in the differential diagnosis of sialadenitis and prompt isolation should be considered to reduce the spread of infection.

Bilateral parotid gland swelling: An unusual presentation of B cell lymphoma.

ABSTRACT: This case report describes a 45-year-old woman with acute, bilateral parotid gland swelling caused by B-cell lymphoma. Enlarged, tender salivary glands are more commonly caused by infection, inflammation, or obstruction from salivary gland stones or tumors. This patient initially was treated for common causes of parotitis; however, her condition did not respond to the standard management and she ultimately was diagnosed with B-cell lymphoma.

Pediatric Sjögren Syndrome: Case Report On A Rare Entity.

Pediatric Sjögren's Syndrome (SS) is an auto-immune disorder of unknown prevalence with significant risk of comorbidity. In contrast to the classical dyad of xerostomia and xeropthalmia frequently seen in adults, in children and adolescents, recurrent parotiditis and sialadenitis are more often the presenting symptoms. We describe the case of a previously healthy 16-year-old girl with recurrent cervical lymphadenopathy and parotid swelling. Over the course of nine months, extensive investigation established chronic bilateral recurrent sialadenitis of unknown cause. The patient's clinic and complementary exams favor a primary SS diagnosis; however, she later meets classification criteria for Systemic Lupus Erythematosus. Although currently clinically stable under hydroxychloroquine with minor parotid swelling and eye redness, long term multidisciplinary follow-up will be needed to manage the patient's disease. This report aims to bring awareness to this diagnostic challenge and to the need for pediatric criteria for SS.

Polymicrobial Acute Suppurative Parotitis in a 33-Day-Old Infant: A Case Report and Review of the Literature.

BACKGROUND: Acute suppurative parotitis (ASP) of neonates is a rare condition characterized by irritability, erythema, and tenderness of the affected gland. METHODS/RESULTS: Only few cases have been reported in Engilsh literature, mostly in male neonates, in a unilateral fashion. In our case, a polymicrobial etiology (Klebsiella pneumoniae, Staphylococcus aureus, Acinetobacter ursingii, and Acinetobacter junii) was found. Based on the review of the microbiological findings of cases of ASP in English literature for the years 1970 to 2020, S. aureus is the most commonly isolated microorganism (47% of the total 65 patients). Our patient was born with a C-section procedure and was not breast-fed, making dysbiosis along with the usage of the feeding bottle, possible risk factors for the development of ASP. CONCLUSIONS: ASP may be due to polymicrobial etiology. Initial presentation in neonates may not include typical signs and symptoms, like fever. Aseptic technique of oral procedures is of utmost importance also in immune-competent neonates.

Updates in childhood Sjogren's syndrome.

PURPOSE OF REVIEW: Childhood Sjogren's syndrome (cSS) is a rare, chronic autoimmune disease characterized by inflammation of the exocrine glands. cSS is underrecognized because of differences in clinical presentation compared with adults. Until recently, publications describing clinical manifestations in cSS were limited to case reports and case series with small numbers of patients. Diagnostic studies to assess glandular symptoms in adults, are less commonly obtained in children. RECENT FINDINGS: Recent cohort studies describe presenting diagnostic clinical features in large populations of cSS and demonstrate how current classification criteria, used in adults, are not applicable to children. Recurrent parotitis is the consistent predominant manifestation that is inversely correlated with age. Novel salivary biomarkers and salivary gland ultrasonography are important objective measure, which may improve diagnosis and disease monitoring. Standardized treatment recommendations are needed. SUMMARY: Findings from large cohort studies provide a framework for the future development of diagnostic criteria for cSS. Such criteria should incorporate objective measures that are easily obtained in children. Future research to improve understanding of the application of novel biomarkers and imaging and developing consensus on treatment recommendations is needed.

Neonatal suppurative parotitis: Case reports and literature review.

BACKGROUND: Neonatal suppurative parotitis is a rare disease, characterized mainly by unilateral parotid swelling with erythema and tenderness, and often purulent discharge from the Stensen's duct into the oral cavity. Only 44 cases were reported in the English literature between 1970 and 2013. METHODS: A MEDLINE search was conducted using the terms acute, neonatal, newborn, suppurative, bacterial, purulent, parotitis, parotid swelling, and parotid abscess, limited to the English-language literature starting from 2011. We reviewed all reported cases, together with two more managed cases in our hospital. We also describe the magnetic resonance imaging findings of the early stage of this disease. RESULTS: We identified 26 new cases since 2011. The total number of patients reviewed was 72, including our patients. The infection was unilateral in 83% of patients, and 67% of the affected patients were males. The serum amylase levels were generally not elevated despite marked parotid swelling. Of the causative agents of this disease, 65% were Staphylococcus aureus, of which 19% were methicillin-resistant S. aureus. As the rate of cesarean section was high in patients with this disease, it was considered a risk factor. The diffusion-weighted magnetic resonance images showed multiple punctate hyperintensity regions with reduced apparent diffusion coefficient, suggesting microabscess formation in the affected gland. CONCLUSIONS: Acute suppurative parotitis should be considered in cases of swelling and tenderness in the parotid gland during the neonatal period. Multiple punctate hyperintensities in the parotid gland on the diffusion-weighted images may indicate a retrograde bacterial infection from the Stensen's duct.

Juvenile recurrent parotitis in a 4-year-old patient: a case report.

Juvenile recurrent parotitis (JRP) is a rare disease. It is most commonly occurring between the ages of 3 and 5 years, that classically resolves at adolescence. It is characterized by recurrent non-suppurative parotitis, with several acute inflammatory episodes per year. The parotid´s swelling tends to be unilateral, but it can occur bilaterally, with a more predominant side. The aim of this work was to present a case report that highlights signs and symptoms of this unusual condition and to stress on the value of ultrasonography as an aid to diagnosis.

IgG4-related chronic sclerosing sialadenitis in a child with recurrent parotitis: a case report.

BACKGROUND: IgG4-related disease (IgG4-RD) includes a group of immune-mediated diseases histologically characterized by lymphoplasmacytic infiltrate with a prevalence of IgG4-positive plasma cells, storiform fibrosis and obliterative phlebitis. Autoimmune pancreatitis, sialadenitis, dacryoadenitis and retroperitoneal fibrosis are the most frequent manifestations. IgG4-related sialadenitis usually affects submandibular glands and is very rare in children. Here we report the case of IgG4-related sialadenitis in a six-year-old patient previously diagnosed as juvenile recurrent parotitis. CASE PRESENTATION: A six-year-old patient was referred to our Centre for left parotid swelling of 4 × 3 cm, that was tender, soft in consistency, with overlying red and warm skin. His general condition was good but he was subfebrile; general examination revealed mild enlargement of left cervical lymph nodes. In the last 2 years he had had five episodes of parotitis, diagnosed by another pediatric Center as juvenile recurrent parotitis. On ultrasound examination the left parotid gland appeared enlarged, inhomogeneous, with a colliquative intraparotid lymph node and no evidence of sialolithiasis. Laboratory tests showed an increase of white blood cells and anti-VCA IgM and IgG positivity, with anti-EBNA e anti-EA I negativity. The patient was initially treated with oral antibiotics, but after 10 days the parotid became fluctuating, requiring surgical biopsy and drainage. Postoperative course was regular, with complete remission under oral antibiotic and steroid therapy. Microbiological tests, including cultures for aerobic and anaerobic bacteria, mycobacteria and Bartonella, were negative. Surprisingly, histology showed marked fibrosis and histiocytic and lymphoplasmacellular infiltrate with polyclonal plasma cells mostly expressing IgG4 immunoglobulins. Thus, the diagnosis of IgG4 related chronic sialadenitis in recurrent parotitis and recent EBV infection was made. CONCLUSIONS: IgG4-related sialadenitis is very unusual in children. Histology plays a key role in diagnosis, considering that up to 30% of patients have normal serum IgG4 levels, as shown in our case. The lack of previous histological data makes it impossible to attribute our patient's previous episodes of parotitis to IgG4-RD, though it is a very consistent possibility.

Immune disorders associated with juvenile recurrent chronic parotitis.

INTRODUCTION: Juvenile recurrent chronic parotitis (JRCP) is a rare disease of unknown cause. There is a growing interest in its autoimmune aetiology and its relationship with dysfunctions of cellular and humoral immunity, although there is no agreed protocol for complementary investigations for its study. A consecutive series of cases is presented where the immune alterations and associated autoimmune disorders are investigated, proposing a study algorithm. PATIENTS AND METHODS: A retrospective study was carried out on patients who had JRCP during the period from 2013 to 2016 and a follow-up of at least 2 years. After its clinical and ultrasound diagnosis, complementary examinations were systematically carried out to investigate infectious, immune, and autoimmune diseases. RESULTS: Of a total of 36 patients with inclusion criteria, 16 (44%) were found with some analytical alteration of a non-specific immunological nature (positive ANA, high IgG, low complement factor 4), or associated with a specific diagnosis, as occurred in 11 patients: Selective IgA deficiency (2), Sjögren's syndrome associated or not with systemic lupus erythematosus (3), coeliac disease associated or not with diabetes mellitus (4), Hashimoto's thyroiditis (1), and acquired immunodeficiency syndrome (1). CONCLUSION: Juvenile recurrent chronic parotitis can be considered a sentinel sign of other diseases of immunological/autoimmune aetiology for which the diagnosis, monitoring and early treatment can improve its prognosis. Viral infectious aetiology, with the exception of HIV, is not a priority in the study of recurrences.