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.

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.

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.

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.

Facial and limb angioedema with parotitis and Kikuchi-like necrotizing lymphadenitis preceding neuropsychiatric systemic lupus erythematosus in a young African American male.

Angioedema has been observed in a few cases secondary to systemic lupus erythematosus (SLE). Herein, we report a rare case where a young healthy male initially presented with angioedema, lymphadenopathy and parotitis and later on developed neuropsychiatric manifestations at the very onset of his SLE disease. This case illustrates the importance of prompt clinical consideration of lupus with unusual and atypical preceding manifestations.

An unusual case of acute parotitis in a young adult.

Acute bacterial parotitis is uncommon in young adults. Infection with Serratia marcescens is even rarer and usually found in hospitalized patients. This case report focuses on a young woman with acute bacterial parotitis caused by S. marcescens that required a longer-than-normal course of antibiotics.

Botulinum Toxin for the Management of Sjögren Syndrome-Associated Recurrent Parotitis.

Recurrent parotitis is a rare manifestation of Sjögren syndrome. The management of recurrent parotitis is challenging because conservative methods may be of limited efficacy and invasive approaches carry the risk of complications. Botulinum toxin has been shown to reduce salivary flow, and consequently, the results of its use in the management of recurrent parotitis have been encouraging. A 65-year-old female patient with recurrent parotitis due to Sjögren syndrome was referred to us, complaining of weekly bouts of inflammation. She required a course of antibiotics monthly to control bacterial superinfections. We treated her with onabotulinumtoxinA injections into both parotid glands at regular intervals. After her second injection cycle, she denied further inflammatory bouts, has not required antibiotics in more than 36 months, and denied any side effects. Botulinum toxin may be a safe and effective method of treating Sjögren syndrome-associated recurrent parotitis.

The diffuse infiltrative lymphocytosis syndrome (DILS). A comprehensive review.

The Diffuse Infiltrative Lymphocytosis Syndrome (DILS) is a rare multisystemic syndrome described in HIV-infected patients. It is characterised by CD8(+) T-cell lymphocytosis associated with a CD8(+) T-cell infiltration of multiple organs. DILS is usually seen in uncontrolled or untreated HIV infection but can also manifest itself independently of CD4(+) T-cell counts. The syndrome may present as a Sjögren-like disease that generally associates sicca signs with bilateral parotiditis, lymphadenopathy, and extraglandular organ involvement. The latter may affect the lungs, nervous system, liver, kidneys, and digestive tract. Anomalies of the respiratory system are often identified as lymphocytic interstitial pneumonia. Facial nerve palsy, aseptic meningitis or polyneuropathy are among the more frequent neurological features. Hepatic lymphocytic infiltration, lymphocytic interstitial nephropathy and digestive tract lymphocytic infiltration account for more rarely noted complications. Sicca syndrome, organomegaly and/or organ dysfunction associated with polyclonal CD8(+) T-cell organ-infiltration are greatly suggestive of DILS in people living with HIV. Labial salivary gland biopsy is therefore helpful when the focus score is equal or greater than 1 (or Chisholm Score ≥ 3). Primary Sjögren syndrome, chronic HCV or HTLV1 infection, graft versus host disease, IgG4-related disease, and immune reconstitution inflammatory syndrome are among the differential diagnoses that need to be considered. Treatment consists in highly active anti-retroviral therapy (HAART), which is usually effective in resolving clinical signs and symptoms. Steroids, however, may also be occasionally required when organ infiltration does not respond to HAART. This review should provide an insight into this rare entity complicating the course of HIV infection.

Acute suppurative parotitis in a 33-day-old patient.

Acute suppurative parotitis is a rare disease in childhood. Its incidence is higher in premature newborns. Parotid swelling and pus drainage from Stenson's duct is pathognomonic, and Staphylococcus aureus is the causative agent in most cases. Here, a 33-day-old patient with acute suppurative parotitis is presented.

Beware of parotitis induced by iodine-containing contrast media.

Carotid stenting is being increasingly used for revascularization of the moderate to severe carotid stenosis and thus its complications are increasingly being recognized. We report a rare complication of induced by iodine contrast in a patient undergoing carotid stenting. s. A 51 year old man after the second stenting developed multiple small infarcts in spite of the distal device. He also had painful parotid swelling which improved within a week. One should be aware of iodine parotitis s in the patients undergoing iodinated contrast study.

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.

Bi-institutional analysis of microbiological spectrum and therapeutic management of parotid abscesses.

BACKGROUND: A parotid abscess (PA) is a complication of an acute bacterial parotitis with a potentially life-threatening course. To date, data on the diagnosis and therapy of PA is sparse and mostly consists of case reports or case series. Therefore, this study aimed at comprehensively analyzing the microbiological spectrum and the therapeutic management in a bi-institutional setting. METHODS: A retrospective clinical chart review was performed to identify all patients surgically treated for PA at two tertiary care centers in Germany. Data on demographics, clinical management and microbiological data including species identification, pathogenicity, type of antibiotic therapy, adjustment of antibiotics, antibiotic sensitivity testing, and smear test results were extracted. Intervention-related variables and etiology were analyzed for their statistical association with outcome variables. RESULTS: Overall, 85 patients were included. Most patients (92.9%) underwent surgical incision. Around half of the patients (45.9%) were treated under local anesthesia. No facial nerve palsy was observed. The most frequently detected pathogens were Streptococci (n = 23), followed by Staphylococcus aureus (n = 6) including one case of methicillin-resistant Staphylococcus aureus. Most patients (68.2%) received an aminopenicillin ± beta-lactamase inhibitor as empiric antibiotic therapy. In 6 cases the antibiotic therapy was modified after receiving the antibiogram. Four patients (5.2%) presented with recurrent PA. Etiology was idiopathic (42.4%), followed by tumorous (12.9%), obstructive, and immunosuppressive (each 11.8%). Patients with a dental focus (p = 0.007) had a longer duration of hospitalization. CONCLUSION: The results show that the surgical therapy of PA under local anesthesia is safe. A dental examination should routinely be performed to rule out a dental focus. Obtaining a microbiological specimen in order to modify antibiotic therapy if necessary and a histopathological specimen to rule out a tumorous etiology is obligate.

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.

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.

[A parotid gland lymphoma presenting with steroid-responsive recurrent sialadenitis: a case report]. / Steroide yanit veren reküren siyaladenit seklinde ortaya çikan atipik seyirli parotis bezi lenfomasi: Olgu sunumu.

Parotid gland located lymphomas are rarely seen. The most common lymphomas involving salivary gland include B-cell MALT (mucosa-associated lymphoid tissue) lymphomas. Of them, extranodal marginal zone B-cell lymphomas are the most frequently seen pathologies. They usually present with painless swelling of the parotid gland. The diagnosis is often based on parotidectomy. In this article, we report a 46-year-old male patient who was admitted with recurrent sialadenitis attacks which responded well to steroid treatment for three years, but recur after the cessation of the therapy. With the initial diagnosis of recurrent parotitis, the patient was scheduled for total parotidectomy for the definitive diagnosis and treatment. When the frozen-section of specimens during superficial parotidectomy revealed lymphoma, the operation was terminated.

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.