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.

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.

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.

Parotitis caused by Mycobacteroides abscessus subspecies abscessus.

Rapidly growing mycobacteria rarely causes parotitis. We report a rare case of Mycobacteroides abscessus subspecies abscessus (MAB) parotitis in a previously healthy 26-year-old woman. She presented to the previous hospital with a swelling over the right parotid region, and a computed tomography scan revealed multiple abscesses in the swollen parotid gland. Histopathology showed granulomatous inflammation with acid-fast bacilli; however, a subsequent culture failed to isolate mycobacterium. Despite repeated antibiotic therapy and multiple surgical interventions including partial incision and drainage of the abscesses, the parotitis did not resolved. At six months after presentation, she was referred to our institute. We performed enlarged resection of the necrotic tissue and abscesses, and the sample cultivated after homogenization was positive for mycobacterium. The isolate was finally identified as MAB. She underwent long-term postoperative antibiotic therapy for MAB, with a favorable outcome. To the best of our knowledge, this is the first case of MAB parotitis where the subspecies has been identified. MAB is much more intractable than the other subspecies. We highlight the importance of the correct identification of MAB, which leads to the appropriate treatment.

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.

Neonatal Parotitis: A case report.

The following is a case report of a 17-day-old female baby, born at 35 weeks' gestation, weighing 2.6 kg. She was brought to us with reluctance to feed, swelling over the left side of her face and a fever documented at 102oF, along with an erythematous, tender, localised swelling over the left pre-auricular region that measured 2 x 1.5 cm in size. Diagnostic workup and ultrasound findings were consistent with parotitis; however, her blood culture was negative. The patient was managed on antibiotics but subsequently, developed a nosocomial infection while she was admitted in the hospital, which prolonged her hospital stay to a total of 16 days. Nevertheless, she had complete resolution of the signs and symptoms on her follow-up visit. Acute parotitis should be considered in the differential diagnosis of a neonate presenting with facial swelling, reluctance to feed or incessant crying. Timely and appropriate management can result in good recovery and minimising the potential for complications.

Conservative Management of Chronic Suppurative Parotitis in Patients with Sjögren Syndrome: A Case Series.

BACKGROUND Parotitis is an inflammation of the parotid gland, which can be caused by factors including infection, radiation, and hyposalivation secondary to systemic conditions, such as Sjögren syndrome, rheumatoid arthritis, or medication. Bacterial parotitis is a rare complication that can be observed in patients with hyposalivation. However, it is also observed in elderly and immunocompromised patients. Lack of continuous flushing of salivary glands and their ducts due to decreased salivary flow renders the glands prone to retrograde colonization with oral microflora. Several microorganisms have been associated with bacterial infections of the parotid glands; Staphylococcus aureus is the most common, accounting for 80% of cases, followed by mixed bacterial communities, including streptococci, anaerobes, and gram-negative bacilli. Bacterial parotitis presents as tenderness, swelling, and purulent sialorrhea from the salivary gland's duct. Immediate administration of broad-spectrum antibiotics, based on the results of the patient's culture and sensitivity test, has shown success in treating these cases. CASE REPORT We report 3 cases of chronic suppurative parotitis secondary to dry mouth and due to Sjögren syndrome that did not respond to oral or intravenous antibiotics and was successfully managed using conservative methods, such as the local application of superficial moist heat and periodic pus drainage by manipulating the parotid glands at dental clinics. CONCLUSIONS We concluded that conservative approaches, such as massaging the glands, local application of superficial moist heat, and periodic pus drainage without using antibiotics, should be considered as the first-line management of bacterial infection of the parotid glands.

Lactant de 6 setmanes amb tumefacció preauricular I febre d'aparició sobtada / No disponible

No disponible

Positive Effects of Local Botulinum Toxin Injection and Colchicine for Complicated Chronic Recurrent Parotitis: A Case Report.

Chronic recurrent parotitis (CRP) is a rare affliction of unknown cause characterized by recurrent episodes of unilateral or bilateral nonobstructive and nonsuppurative inflammation of the parotid glands. Management of CRP is not standardized, and attempts at treatment often fail. We report a case of a 29-year-old female patient with CRP for 13 years, with recurrent acute episodes, complicated with a collection and cutaneous fistula, refractory to repeated courses of corticosteroids and antibiotics. Injections of botulinum toxin in the parotid gland and maintenance treatment with oral colchicine lead to a rapid and sustained improvement. Local botulinum toxin injections associated with colchicine might represent a safe and noninvasive treatment of CRP. The possible beneficial effect of colchicine could be an argument for auto-inflammatory participation in the pathogenesis of CRP.

Botulinum Toxin Injection for Chronic Parotitis: A Multi-Center and Prospective Trial.

OBJECTIVE: When conservative therapy fails for chronic parotitis, sialendoscopic treatment or surgical excision can be considered. However, these are financially costly or invasive. Thus, this study aimed to evaluate the clinical efficacy and safety of botulinum toxin (BTX) injection and to further analyze its effect on parotid gland function and volume using salivary single-photon emission computed tomography (SPECT)-computed tomography (CT). METHODS: This clinical trial has been registered in the Clinical Research Information Service, Republic of Korea. Fourteen patients with chronic parotitis received BTX injections into the parotid glands. Pain, postprandial pain, swelling, aesthetic discomfort, and salivary flow rate were assessed before injection, at 2 weeks, and at 1, 3, and 6 months. Salivary SPECT-CT was performed before injection and again 3 and 6 months after to assess the volume and uptake changes. RESULTS: All subjective symptoms decreased significantly until 1 month and then increased. However, at 6 months, all subjective symptoms were determined to be better than before injection. The unstimulated and stimulated salivary flow rate did not show a significant difference over time. No significant difference was noted in parotid gland volume or uptake on salivary SPECT-CT over time. CONCLUSION: BTX injection can be an alternative treatment option for chronic parotitis. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E1903-E1909, 2021.

Parotitis due to Burkholderia cepacia infection after autologous hematopoietic stem cell transplantation.

Burkholderia cepacia predominantly causes opportunistic infections in hospitalized and immunocompromised patients such as patients with cystic fibrosis, cancer, or human immunodeficiency virus (HIV). Nonetheless, Burkholderia cepacia is infrequently reported to cause infection in hematopoietic stem cell transplantation (HSCT) recipients. Herein, we report a rare case of suppurative parotitis in a 31-year-old patient with T-cell lymphoblastic lymphoma (T-LBL) who underwent auto-HSCT. The secretion from the Stensen duct was collected, and Burkholderia cepacia was detected using the VITEK-2 identification system. Additionally, sensitive antibiotic therapy against this bacterium was also effective. This is the first case of parotitis triggered by Burkholderia cepacia after auto-HSCT, and it is also the first reported domestic case. This case emphasizes the importance of considering bacterial infections in general and Burkholderia cepacia specifically in HSCT patients with post-transplant parotitis.

Diagnostic Challenge and Clinical Management of Juvenile Mandibular Chronic Osteomyelitis.

Juvenile mandibular chronic osteomyelitis (JMCO) is a rare, idiopathic disease of chronic bone inflammation without suppuration, sinus tract formation, or sequestration. As the name suggests, this condition predominately affects children. The few cases of JMCO reported in the literature describe different treatments, and thus a standard therapy protocol has not yet been established. The aim of this paper is to report a clinical case in a 9-year-old girl that was misdiagnosed and unsuccessfully treated for 1 year. After experiencing persistent symptoms, a correct diagnosis was subsequently rendered based on the physical and radiographic examination as well as successful treatment with non-steroidal anti-inflammatory drugs (NSAIDs). The patient received drug therapy followed by periods of remission over a 4 year follow-up period. Diagnosis and treatment of JMCO is a challenge given the rarity and nonspecific signs and symptoms of this condition. Treatment with NSAIDs and regular follow-up is a conservative option for these patients.

[Neonatal suppurative parotitis]. / Neonatal purulent parotit ovanlig men potentiellt allvarlig infektion - God prognos med intravenös antibiotikabehandling tidigt.

Neonatal suppurative parotitis is a rare condition characterized by swelling, pain and erythema over the parotid gland. There may be a purulent exsudate from the Stensen duct. The predominant etiology is Staphylococcus aureus but cases with gram negative bacteria and streptococci have been reported. Most cases are managed conservatively with intravenous antibiotic therapy, and early treatment reduces risks of complications like sepsis and intraglandular abscess. We report two cases of neonatal suppurative parotitis; two 14-days-old males, both with one day history of parotid swelling and erythema. In one of them purulent exudate could be extracted from the Stensen duct. One of them had positive blood culture with Staphylococcus aureus. Ultrasound examination showed an enlarged parotid gland but no abscess. Growth of Staphylococcus aureus was found in both cases and the patients responded well to intravenous antibiotic therapy.