Bilateral parotitis as the initial presentation of childhood sarcoidosis

The differential diagnosis of bilateral parotid gland enlargement in children includes infectious, inflammatory, and neoplastic disorders. We present the case of a 13-year-old male who presented with a 5-week history of bilateral parotid swelling. On exam, both parotid glands were nontender, smooth, and diffusely enlarged. He had slightly elevated inflammatory markers, but other lab results were normal. A neck CT revealed symmetric enlargement of the parotid, submandibular, and sublingual glands. A chest CT revealed scattered peripheral pulmonary nodules and bilateral hilar adenopathy. A parotid gland biopsy showed multiple noncaseating granulomas with multinucleated giant cells surrounded by lymphocytes, consistent with the diagnosis of sarcoidosis. Special stains for acid-fast and fungal organisms were negative. Using this illustrative case, we discuss the differential diagnosis of bilateral salivary gland enlargement in children and review the etiology, diagnosis, clinical manifestations, and treatment of pediatric sarcoidosis.     

Major salivary gland disorders in children and adolescents

The aim of this study was a review of 67 children treated for salivary gland tumors in the ENT Department Medical University of Gdańsk since 1974 to 2008. In that group we observed 52 children with tumor of the parotid gland, 13 with tumor of submandibular gland and 2 with subligual gland tumors. Tumor-like lesions were stated the largest group of tumors and were observed in 33 (49%) of children, mainly of chronic sialadenitis (17 cases). Predominant number of tumors was observed in scholarship children. The most common symptoms were slow enlargement of tumor and swelling of the salivary gland. In the group of benign tumors most common was pleomorphic adenoma (19 cases) and in the group of malignant tumors most common was clear cell carcinoma (2 cases) and rhabdomyosarcoma (2 cases). In this report the authors describe basic epidemiological and clinical data (age, sex, duration of symptoms, histological structure of tumor, diagnostic methods, methods and results of treatment) and observations connected with differences of salivary gland tumors in that group of patients.     

Parotid gland enlargement in HIV infection: clinical/imaging findings

Parotid gland enlargement with or without facial paralysis may be the presenting clinical manifestation of AIDS. Submandibular involvement may occur as well. Failure to recognize this association may lead to inappropriate or unnecessary intervention in the investigation and treatment of the salivary gland enlargement. In this paper, we discuss the role of imaging techniques in the investigation of salivary gland enlargement, and identify the specific radiologic signs found in HIV infection. Demonstration of intraparotid or submandibular gland cysts with focal intraparotid mass lesion(s) on imaging may help to differentiate parotid gland enlargement due to HIV infection from the many other causes of parotid enlargement.     

The benign lymphoepithelial cyst and a classification system for lymphocytic parotid gland enlargement in the pediatric HIV population

OBJECTIVES/HYPOTHESIS: The objectives of this study are to present a series of parotid gland benign lymphoepithelial cysts (BLEC) in HIV-positive children and to propose a three-tiered classification system for HIV-associated lymphocytic parotid gland enlargement. STUDY DESIGN: The authors conducted a retrospective case series and literature review. METHODS: The authors conducted a retrospective chart review of four pediatric patients with HIV-associated parotid gland BLEC who presented to a tertiary care university medical center. RESULTS: Four pediatric HIV-positive patients (four girls; age range, 7-17 years [mean age, 12.8 years]) were diagnosed with parotid gland BLEC. Two patients presented with acute parotitis and the others presented with asymptomatic enlargement of the parotid glands. Three patients had bilateral parotid gland BLEC. The other patient demonstrated persistent generalized lymphadenopathy (PGL) of the intraparotid and cervical lymph nodes and early BLEC limited to the left parotid gland. One patient also displayed parotid gland microcalcifications and cystic changes in the adenoids, neither of which have been described previously in the setting of HIV-associated BLEC. Computed tomography was performed on all patients, and one patient underwent fine needle aspiration to confirm the diagnosis. All patients opted for observation and antiretroviral medication therapy as long-term treatment. Based on these findings and a review of the literature, we propose a three-tiered classification system for lymphocytic parotid gland enlargement in the HIV population: 1) PGL, 2) benign lymphoepithelial lesions (BLEL), and 3) BLEC. CONCLUSIONS: This series equals the largest pediatric series of HIV-associated parotid gland BLEC in the English literature. One patient in our series also demonstrated PGL; there were no cases of BLEL. A classification system based on morphology is proposed to help resolve the confusion in terminology used to describe this entity. Most pediatric HIV-infected patients with parotid gland BLEC can be treated with observation and antiretroviral medication therapy. For others, who are symptomatic or more concerned about their cosmetic appearance, sclerotherapy may offer a reasonable option. Radiation therapy and surgery should be reserved for select cases.     

Sialadenosis of the parotid gland after chronic nicotine use

The effect of chronic nicotine consumption on the morphology of the rat parotid gland was investigated. After nicotine-loading for 90 days with an average serum nicotine concentration of 78 +/- 10 ng/ml a significant increase of acinar cell volume was observed. The acinar cells contained an increased number of enlarged light, immature secretory granules. These findings were confirmed by morphometric analysis. Further an increase of the granular endoplasmatic reticulum, an enlargement of the Golgi complexes and an oedematous swelling of intraglandular autonomous nerves were found. Similar findings have been observed in the parotid gland of animals and also of humans chronically treated with the beta-adrenergic drugs aludrin and isoproterenol, and have been termed "sialadenosis". It seems likely that the observed morphological alterations are caused by a stimulation of glandular beta-adrenoceptors via a nicotine-induced release of catecholamines from the adrenals.     

Recurring bilateral parotid gland swelling: two cases of sialodochitis fibrinosa

Salivary gland swelling is a commonly encountered clinical symptom, but the establishment of a diagnosis is occasionally difficult. Here, we present two sialodochitis fibrinosa patients with recurring bilateral parotid swelling. In both patients, secretion of mucous plugs containing numerous eosinophils was observed from Stensen's ducts. As expected, the level of interleukin-5 in the saliva was much higher than that in the serum. One patient had no medical history of allergic disease; the other had allergic rhinitis which had never been associated with parotid gland swelling. Microbiological examination was unable to isolate significant bacterial specimens from the mucous plugs. Thus, although allergy and/or bacterial infection are reportedly implicated as causes of sialodochitis fibrinosa, there may exist other possibilities for its pathogenesis. Interleukin-5 seems to play a crucial role in the pathogenesis of sialodochitis fibrinosa.     

Immunocytochemical identification of HIV (p24) antigen in parotid lymphoid lesions

Antibodies to specific human immunodeficiency virus (HIV) polypeptides are important laboratory markers of HIV infection. We have used an antibody to the major structural gag protein p24 of HIV-1 virus to immunochemically localize this capsid antigen in lymphoid cells from seven of eight patients at risk for HIV infection and who presented with parotid lymphadenopathy and lymphoepithelial cysts of the parotid gland. A clinicopathological assessment of these two manifestations as they relate to HIV infection is also presented.     

Cystic parotid lesions in patients at risk for the acquired immunodeficiency syndrome

Twenty-three patients at risk for the acquired immunodeficiency syndrome presented with cystic lesions of the parotid gland. Fourteen patients had unilateral parotid cysts and nine had bilateral enlargement. Ten patients were positive for antibodies to the human immunodeficiency virus (HIV), three were negative for antibodies to the human immunodeficiency virus, and ten refused testing. Fine-needle aspiration cytology and computed tomography were helpful in making the diagnosis of benign lymphoepithelial cyst. Superficial parotidectomy confirmed the diagnosis. Concurrent malignancy (Kaposi's sarcoma) was diagnosed in one patient from a parotid specimen. Follow-up has ranged from four months to five years.     

New concepts in the treatment of chronic recurrent parotitis

The release of glandular kallikrein into the interstitium of the parotid gland appears to play an important role in the occurrence of the inflammatory interstitial edema in chronic recurrent parotitis. This provides fresh impetus for the treatment of this parotid disease with a kallikrein inhibitor. In our present study, seven patients with acute exacerbated chronic recurrent parotitis were treated with the kallikrein inhibitor aprotinin (Trasylol, Bayer AG, Leverkusen). With this therapy all patients were free of pain 12 h after the start of the therapy and most salivary gland function had returned to normal by 48 h after beginning treatment. Within this period of time, concomitant swelling of the affected parotid gland disappeared completely in five patients and resolved in the other two patients after 1 week.     

New concepts in the treatment of chronic recurrent parotitis

Summary The release of glandular kallikrein into the interstitium of the parotid gland appears to play an important role in the occurrence of the inflammatory interstitial edema in chronic recurrent parotitis. This provides fresh impetus for the treatment of this parotid disease with a kallikrein inhibitor. In our present study, seven patients with acute exacerbated chronic recurrent parotitis were treated with the kallikrein inhibitor aprotinin (Trasylol, Bayer AG, Leverkusen). With this therapy all patients were free of pain 12 h after the start of the therapy and most salivary gland function had returned to normal by 48 h after beginning treatment. Within this period of time, concomitant swelling of the affected parotid gland disappeared completely in five patients and resolved in the other two patients after 1 week.     

The diagnostic value of nuclear magnetic resonance tomography in Sj?gen's disease]

Sj?gren's syndrome is characterised by xerostomia, xerophthalmia and recurrent parotid swelling. There is a high coincidence with rheumatoid diseases. A variety of clinical and laboratory parameters offers important information for diagnosis, but none of them is specific for this disease. 36 patients with clinical diagnosis of Sj?gren's syndrome were examined in a prospective study to evaluate the diagnostic significance of this technique. 34 patients showed an inhomogeneous, nodular pattern in the parotid gland. The signal intensity was very high, especially in the T2-weighted sequences. There was a significant correlation of the radiologic changes with the course of the disease. Four patients with the clinical diagnosis of sialadenosis demonstrated bilateral enlarged glands with a homogeneous signal pattern. Patients with acute purulent parotitis demonstrated enlargement of the gland without an alteration in the signal pattern. Patients with chronic recurrent parotitis and patients with radiogenic sialadenitis showed little inhomogeneous increase of signal intensity. Our results indicate that MRI is presently the radiological method of choice in the evaluation of Sj?gren's syndrome.     

Doxycycline sclerosis of benign lymphoepithelial cysts in patients infected with HIV

Benign lymphoepithelial cysts (BLCs) are a widely recognized cause of parotid gland swelling in HIV-infected patients. Although they are neither invasive nor associated with malignant degeneration, BLCs can become large and disfiguring. Multiple modalities have been used to control these cysts, but no ideal treatment has been identified. The current study examines the efficacy of doxycycline as a BLC sclerosant in eight patients, and nine BLCs (bilateral BLC in one patient). Follow-up ranged from 12 to 17 months in all cases. Doxycycline sclerosis controlled further cyst growth in 100% of cases with no serious complications. The BLCs became negligible or unnoticeable in two patients, and in six patients (seven BLCs) the cyst became fibrosed and showed no evidence of further growth over the follow-up period. Although further studies are needed to determine the long-term efficacy of this treatment modality, doxycycline sclerosis appears to offer a simple, safe, cost-effective, office-based therapeutic option for the treatment of BLCs in patients infected with HIV.     

The ENT manifestations of HIV infection in children

This study identified the ENT symptoms of 66 HIV infected children over an 8 year period (1986-1993) at Great Ormond Street Hospital for Children. The incidence, nature and age of onset of ENT symptoms were investigated; 91% of the children had ENT symptoms, the most common being cervical lymphadenopathy, oro-oesophageal candidiasis and otitis media. The HIV infected children suffered from the common ENT diseases of childhood. They also presented with specific conditions such as diffuse parotid swelling. Therefore, their clinical features differed from HIV infected adults as well as non-infected children. An increasing incidence of paediatric HIV infection was demonstrated by the study. Most were due to vertical transmission. ENT surgeons are likely to see more HIV infected children in future, either with the usual ENT diseases of childhood (to which they seem more susceptible) or with HIV-specific conditions. Although the diagnosis of HIV may be known, the ENT condition could be the initial presentation suggestive of immunodeficiency.     

Multicentric parotid cysts and cervical adenopathy in AIDS patients. A newly recognized entity: CT and MR manifestations

Patients with AIDS or ARC may present with parotid gland enlargement either initially or during the course of their disease. Of nine such patients, eight had unilateral gland enlargement, and one had bilateral disease. CT and MR scanning revealed that all patients except one had bilateral multiple intraparotid cysts. In addition, four of the nine patients clinically had cervical adenopathy, while all patients had adenopathy on scanning. We believe that the association of parotid cysts and cervical adenopathy is another manifestation of AIDS or ARC and, when found on scanning, should alert the clinician to the probable underlying condition.     

Intralesional bleomycin injections in the treatment of benign lymphoepithelial cysts of the parotid gland in HIV‐positive patients: Case reports

Benign lymphoepithelial cysts (BLCs) of the parotid gland are associated with human immunodeficiency virus. These cysts may grow large and uncomfortable, causing significant cosmetic embarrassment for the patient. Several treatment options have been described with varying successes. We report successful treatment of three adults with bleomycin. All three patients presented with bilateral parotid enlargements. They received a total dose of between 180 U and 270 U of bleomycin over a period of 6 to 10 weeks. They all responded completely without morbidity or recurrence. Bleomycin is effective in the treatment of BLCs of the parotid glands in HIV‐positive patients. Laryngoscope, 2010     

Chronic parotitis: a challenging disease entity

Chronic parotitis is a troubling clinical condition characterized by repeated infection and inflammation of the parotid gland caused by decreased salivary flow or obstruction. Unilateral swelling, pain, and other associated symptoms occur during acute exacerbations of the disease. A variety of laboratory and radiographic tools are available to aid in the diagnosis. Multiple treatment options have been proposed, ranging from conservative medical management to surgical interventions. We present 2 patients with bilateral chronic parotitis who attempted prolonged medical management and ultimately required surgical parotidectomy for control of their disease.     

Salivary gland biometry in female patients with eating disorders

High-resolution greyscale ultrasound is a generally accepted diagnostic tool for salivary gland enlargement, although no standard biometrical data for the sizes of unaffected parotid and submandibular glands exist. A lot of case reports describe non-inflammatory swelling of salivary glands as symptoms of eating disorders like anorexia nervosa or bulimia nervosa. They might be the only visible sign for the disease. With our prospective study we tried to quantify and compare biometrically the salivary gland in eating disordered patients, obese and normal-weight, healthy controls. A total of 70 females between 16 and 40 years of age were included of which 25 were healthy controls. All subjects underwent three-dimensional high resolution B-scan sonography of the parotid and submandibular gland. Gland volumes were calculated and a multiple regression analysis was done to investigate the influence of an eating disorder on salivary gland size. A significant difference in the size of the parotid glands was seen between our three study groups of adipose and eating disordered patients and healthy, normal-weight controls. The parotid gland volumes were seen in the subgroup bulimia nervosa (BN), (parotid volume = 63,708 mm(3)) where the mean value was more than twice as high as in the control group (31,059 mm(3)). By using body mass index and group characteristic the parotid gland volume as the dependent variable could be estimated with an r (2) of 0.327 in multiple regression analysis. Only the parotid glands were significantly enlarged, while the submandibular glands show no signs of enlargement. Eating disorders influence size of parotid glands, still the factors and the pathogenesis leading to this enlargement seem to be different in adiposity on the one and different types of eating disorders on the other hand. Standard gland volumes could be established, comparable to former initial reports. Submandibular gland remains unaffected by these alterations. We could show that high-resolution ultrasound assessment of parotid gland volume adds a complementary quantitative parameter of organ affection in eating disorders (anorexia/bulimia nervosa).     

Cat-scratch disease localisation in the parotid gland

INTRODUCTION: Cat scratch disease is an infection due to Bartonella henselae. It is one of the principal causes of benign chronic adenopathy in children or young adults. CASE REPORT: A 36-year-old woman presented with a left parotid pre-auricular swelling suggesting a pleiomorphic adenoma. The history and complementary tests (ultra sonography, MRI, cytopuncture, B. henselae serology) led to a diagnosis of cat scratch disease. Doxycilline was efficient within 15 days. DISCUSSION: The parotid localization of cat scratch disease is very rare. When a patient presents with parotid swelling, the distinction between a tumor and lymph node is not easy. Performing complementary tests can be in balance with a quicker but sometimes inappropriate surgical indication. The diagnosis is made even more difficult because of delayed specific serologic tests as for B. henselae.     

人类免疫缺陷病毒感染及艾滋病在耳鼻咽喉头颈外科的临床表现

目的 提高对人类免疫缺陷病毒感染者及艾滋病患者在耳鼻咽喉头颈外科临床表现的认识,以便能及时诊治并做好必要的防护.方法 分析55例坦桑尼亚首诊于耳鼻咽喉头颈外科的HIV感染者及艾滋病患者的临床表现及其预后.55例患者中男23例,女32例;年龄3～67岁,中位年龄31岁.结果 55例患者中腮腺肿胀17例次(30.9%);鼻腔、口腔、咽部卡波西肉瘤19例次(34.5%);慢性化脓性中耳炎12例次(21.8%);颈部肿块11例次(20.0%);咽部肿物7例次(12.7%);口腔及咽部黏膜糜烂溃疡6例次(10.9%);口腔白色念珠菌感染2例次(3.6%).HIV感染者或艾滋病患者经治疗后耳鼻咽喉科感染状况可缓解,但易反复发作,且逐渐加重.结论 HIV感染者及艾滋病患者可首诊于耳鼻咽喉头颈外科.对耳鼻咽喉科感染等治疗缓解后反复发作者,应除外是否合并HIV感染或是否为艾滋病患者.

Abstract：

Objective To improve the recognition of clinical manifestation of human immunodeficiency virus (HIV) infection and acquired immunodeficiency syndrome (AIDS) in otorhinolaryngology head and neck surgery, and to emphasize the early diagnosis, correct treatment and effective protection. Methods Fifty-five patients with HIV infection and AIDS encountered in the department of otorhinolaryngology head and neck surgery in Tanzania were included in this study. There were 23 males and 32 femals with age ranged from 3 -67 years, medium 31 year. The clinical manifestation and prognosis of the patients were analyzed and summed up. Results Parotid glands swelling was the most commonly seen clinical manifestation in the 55 cases. Among the cases reported here, 17 (30.9%) showed parotid glands swelling, 19(34. 5% ) had Kaposi's sarcoma in nasal cavity or oral cavity or pharynx,12(21.8% ) were accompanied with chronic suppurative otitis media, 11 (20.0%) had masses in the neck,7 ( 12.7% ) had masses in the pharynx, 6 ( 10.9% ) were seen to have ulcer in oral and pharyngeal mucosa,2(3.6% ) showed infection due to candidaalbicans. The infection in most cases could be controlled and symptoms relieved, but recurrence could occur over time and got worse gradually. Conclusions Many patients with HIV infection and AIDS first see a doctor in otorhinolaryngology head and neck surgery.Patients who had the above clinical manifestations in repeated poor results should be observed carefully,early diagnosis and treatment are important to prevent transmission, prolong life and maximize the quality of life of the patients.     

Treatment of oral ranula in HIV-positive patient

HIV-associated salivary gland disease refers to the pathology in head and neck lesions such as ranula, salivary gland swelling, xerostomia, and benign lymphoepithelial cysts in the parotid gland. Here, we present a unique case of the ranula patient with HIV infection treated with OK-423 sclerotherapy. Case report: The patient was a 42-year-old Japanese male with a few months history of oral floor swelling. Computed tomography (CT) showed a low-density area limited within the right floor of the mouth. Magnetic resonance imaging (MRI) revealed a distinct T2-high intensity area localized on the same location. The puncture fluid was bloody mucus, and the cytology was no malignancy. We diagnosed a simple ranula. He was, however, found to be HIV-antibody positive at the examination before treatment by chance. He was referred to the department of infectious diseases and definitively diagnosed HIV infection by western blot. We chose OK-432 sclerotherapy because of its minimally invasive and the risk of HIV infecting medical staff. Two times OK-432 injection made the lesion disappear. Conclusion: The case indicated that OK-432 sclerotherapy could be effective for ranula related to HIV.