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.

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.

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.

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.

Absceso parotídeo y mastitis por Staphylococcus aureus resistente a Meticillina en un neonato / Parotid abscess and mastitis by Methicillin- resistant Staphylococcus aureus in a newborn

Introducción: La parotiditis supurativa aguda se presenta con poca frecuencia en el período neonatal. Objetivo: Aportar un nuevo caso de parotiditis supurativa aguda por Staphylococcus aureus resistente a la Meticillina. Presentación del caso: El presente caso tiene la particularidad de que presentó foco de infección inicial (impétigo y conjuntamente mastitis bilateral), en el que se demostró el mismo microorganismo causal de la infección: Staphylococcus aureus resistente a la Meticillina. La mastitis bilateral evolucionó hacia la formación de absceso. En la literatura revisada solo se encontraron tres publicaciones que tratan de neonatos con un foco inicial de infección en sitios diferentes de la parotiditis. Estos aspectos fueron los que motivaron la presentación del caso. Conclusiones: Staphylococcus aureus resistente a la Meticillina ha emergido en los últimos años como agente causal de parotiditis supurativa aguda, que puede diseminarse hacia otro foco infeccioso, habitualmente se logra la curación con tratamiento antibiótico ajustado al agente causal, concretamente con Vancomicina, aunque puede requerir también tratamiento quirúrgico si ocurre abscedación(AU)

Introduction: Acute suppurative parotitis occurs infrequently in the neonatal period. Objective: To provide information of a new case of acute suppurative parotitis caused by Methicillin- resistant Staphylococcus aureus. Case presentation: The present case has the particularity that the patient presented a source of initial infection (impetigo and jointly bilateral mastitis), in which the same causal microorganism of the infection was found: Methicillin resistant Staphylococcus aureus. Bilateral mastitis evolved to the formation of abscess. In the literature reviewed, there were only 3 publications on neonates who presented an initial source of infection in sites different from parotitis. These aspects are those that motivated the presentation of this case. Conclusions: Methicillin resistant Staphylococcus aureus has emerged in the last years as a causal agent of acute suppurative parotitis that can lead to dissemination of another source of infection. Normally, the cure is achieved with antibiotic treatment adjusted to the causal microorganism, specifically with Vancomycin; although it can require surgical treatment if abscesses occurs(AU)

Parotiditis en Chile: caracterización clínica y molecular de dos casos en una población altamente inmunizada / Parotiditis in Chile: clinical and molecular characterization of two cases in a highly vaccinated population

Resumen El virus de la parotiditis produce una infección benigna caracterizada por un aumento de volumen parotídeo que, antes de la introducción de la vacuna tres vírica, afectaba principalmente a niños y adolescentes. Luego de que esta vacuna se implementara en el Programa Nacional de Inmunizaciones, se produjo una notable disminución en su incidencia. Además, ocasionó un cambio en la edad y presentación clínica, siendo más frecuente en adultos jóvenes con mayor riesgo de complicaciones. Presentamos dos casos clínicos de parotiditis en adultos jóvenes confirmados por serología y en uno de ellos, por biología molecular. Se caracterizó el virus como del genotipo G, como el descrito en los brotes en E.U.A y Europa, diferente al virus contenido en la vacuna. El virus parotídeo sigue circulando en nuestro país y debemos mantenernos alerta ante eventuales brotes. Se hace relevante optimizar el diagnóstico etiológico por serología o técnicas de biología molecular con fines clínicos y epidemiológicos.

Mumps virus usually produces a benign infection characterized by increased parotid volume which, prior to vaccination, mainly affected children and adolescents. After the introduction of measles, mumps and rubella (MMR) vaccine, mumps incidence decreased dramatically. This intervention also produced a change in its clinical presentation, moving to young adult patients, with an increased risk of complications. We report two clinical mumps cases in young adults with different clinical presentations. In both cases, serologic assays were assessed and, in one case, a polymerase chain reaction (PCR) was performed in order to confirm the diagnosis. The isolated virus was characterized and identifed as G genotype, the same genotype observed during outbreaks in United States and Europe, and different to the vaccinal strain. Mumps virus is currently circulating in Chile and it is important to be aware of possible outbreaks. Viral diagnosis can be difficult, particularly in populations with high vaccination coverage. Therefore, the access to etiologic study through PCR and serology becomes more relevant in order to optimize clinical management and secondary prevention measures.

[Case of sialadenitis by iodinated contrast medium in a dialysis patient].

BACKGROUND: Sialadenitis by iodinated contrast medium (i.c.m) oriodine mumps (IM) is a rare and late benign manifestation that occurs independently of intravenous or endoarterial administration modality. If renal function is normal, i.c.m. does not reach salivary glands concentrations able to induce sialadenitis. However, a critical glomerular filtration reduction may lead to salivary ducts edema and glandular swelling after i.c.m. injection. We report a rare case report of IM in a patient on chronic hemodialysis. METHODS: A 72-year-old woman affected by chronic kidney disease on chronic hemodialysis, underwent to endoscopic removal of a rectal cancer. For disease staging, a total body TC with i.c.m. was performed. The following morning, patient showed a soft and aching bilateral paroditidis swelling. Salivary glands ultrasound was diagnostic for sialadenitis. The patient was rapidly treated with betamethasone following by a 240 minutes post-dilution online hemodiafiltration session. RESULTS: Within the next 24h, a complete remission of IM was obtained. CONCLUSION: In our patient, a compensatory hyperactivity of the sodium / iodine symporter (NIS) on salivary gland cells may have played a crucial role in IM induction. An high efficiency hemodialysis session within the few following hours after i.c.m injection is a fundamental tool in patients on renal replacement treatment to prevent IM that is an epiphenomenon of i.c.m. accumulation.

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.

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.

Tuberculous Lymphadenitis and Parotitis.

Tuberculous lymphadenitis is the most common extrapulmonary manifestation of disseminated tuberculosis (TB). It is considered to be the local manifestation of the systemic disease that has disseminated to local lymph nodes, but a high index of suspicion is needed for the diagnosis, because there are several infectious and noninfectious diseases that can mimic the same clinical picture. In recent years, different diagnostic methods have been introduced, including fine-needle aspiration cytology, which has emerged as a simple outpatient diagnostic procedure that replaced the complete excisional node biopsy, and a number of molecular methods which have greatly improved diagnostic accuracy. This chapter covers the most actual knowledge in terms of epidemiology, clinical manifestations, pathogenesis, and treatment and emphasizes current trends in diagnosis of tuberculous lymphadenitis. TB parotid gland involvement is extremely rare, even in countries in which TB is endemic. Because of the clinical similarity, parotid malignancy and other forms of parotid inflammatory disease always take priority over the rarely encountered TB parotitis when it comes to differential diagnosis. As a result, clinicians often fail to make a timely diagnosis of TB parotitis when facing a patient with a slowly growing parotid lump. This chapter highlights the most important features of this uncommon disease.

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.

Parotid swelling and chronic recurrent multifocal osteomyelitis of mandible in children.

OBJECTIVE: Chronic recurrent multifocal osteomyelitis (CRMO) is an aseptic inflammatory disorder of unknown etiology. CRMO involving the mandible is a diagnostic challenge due to a propensity for solitary involvement and lack of definitive radiologic or pathologic diagnosis. The purpose of this study is to report clinical and imaging findings in cohort of children with CRMO of the mandible. MATERIALS AND METHODS: A retrospective search of the Picture Archiving Communication System (PACS) and accompanying reports was performed for studies with suspected osteomyelitis of the mandible. The electronic medical record, histopathology results, and imaging findings were also reviewed. Inclusion criteria included presentation and course as well as surgical biopsy consistent with CRMO. Four children satisfied our inclusion criteria. Mean age was 9.1 years (range 3.5-12). M:F ratio=1:3. RESULTS: Patients presented with mild pain and swelling in the parotid region (n=4), mildly elevated sedimentation rate (n=3), and negative biopsy culture (n=4). A positive response to non-steroidal anti-inflammatory medication was noted in all patients. Imaging demonstrated a predominately sclerotic lesion in the posterior mandible associated with solid periosteal reaction in all patients. Cortical expansion (n=3) was commonly present. The clinical course was prolonged in the patients in whom a diagnosis of CRMO was not initially entertained. CONCLUSIONS: Parotid mass or swelling is a common presentation of CRMO involving the mandible. The possibility of this diagnosis should be raised when typical clinical and imaging features are present. Whole body imaging may be helpful to evaluate for additional lesions. While biopsy is necessary with isolated mandibular involvement, repeated biopsies and prolonged antibiotic therapy should be avoided.

[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.

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.

Recurrent parotitis as a first manifestation in a child with primary sjogren's syndrome / Parotitis recurrente como primera manifestación en un niño con el síndrome de sjögren primario

Recurrent parotitis is an acute, severe inflammation of one or both parotid glands, the major salivary glands in young children. We report the case of a seven-year old boy with Primary Sjogrens syndrome (PSS) who presented with 15 episodes of painful recurrent bilateral swellings of the parotid glands over a four-year period.

La parotitis recurrente es una inflamación aguda, severa y reiterada de una o ambas glándulas parótidas - la mayor de las glándulas salivares - en los niños pequeños. Se reporta el caso de un niño de siete años de edad con el síndrome de Sjögren primario (SSP). El niño presentó 15 episodios de dolorosas inflamaciones bilaterales recurrentes de las glándulas parótidas por un período de cuatro años.