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Cholinergic-drug induced sicca syndrome in Parkinson's disease: a case report and a review of the literature.

A 67-year-old woman developed severe sicca manifestations after initial treatment of Parkinson's disease with an anti-cholinergic drug, which prompted us to look for the presence of Sjögren's syndrome. The results of sialography, labial salivary gland biopsy, Rose-Bengal test as well as the presence of antinuclear antibody were consistent with the diagnosis of Sjögren's syndrome. The sicca symptoms diminished by cessation of the anti-cholinergic drug, and the parkinsonian features were controlled by levodopa. We suggest that Sjögren's syndrome should be considered, if patients with Parkinson's disease complain severe xerostomia.

Aged↗

[Primary Gougerot-Sjögren syndrome in a 13-year-old girl].

UNLABELLED: Sjögren's syndrome is uncommon in children, and occurs most often in association with autoimmune diseases (secondary Sjögren's syndrome). We describe the clinical and biological features of a 13-year-old girl with primary Sjögren's syndrome, revealed by recurrent parotitis. CASE REPORT: This adolescent girl was referred for investigation of multiple episodes of bilateral parotid swelling since age nine, without systemic symptoms. Examination was unremarkable except for enlarged and painless parotid glands. Laboratory investigations, measurement of saliva production, parotid sialography, labial salivary gland biopsy, revealed Sjögren's syndrome without associated disease. Hydroxychloroquine was prescribed with clinical improvement. CONCLUSION: Recurrent parotitis in children is an uncommon condition. The onset of parotid swelling at five years or over deserves screening for dysimmune disorders, sarcoidosis or Sjögren's syndrome. Diagnosis of Sjögren's syndrome is based on laboratory evidence of autoimmune disorders and minor salivary gland biopsy.

Adolescent↗

Primary Sjögren syndrome in childhood: report of a case and review of the literature.

Sjögren syndrome (SS) in childhood is a rare and possibly underdiagnosed condition. The purpose of this study is to report a case of primary SS (PSS) in a 4-year-old Venezuelan girl and to review the pertinent literature. The patient presented with bilateral recurrent parotid enlargement, predominantly on the right side. She did not complain of dry mouth or eyes; however, decreased stimulated salivary flow rate and positive Schirmer and rose bengal tests were obtained. Sialography, sonograms, and a computed tomography scan of the parotid glands revealed pathologic changes consistent with SS. Anti-SS-A and anti-SS-B antibodies were present. Evaluation for antibodies against cytomegalovirus, Epstein-Barr virus, and HIV rendered negative results. Histopathologic examination of incisional biopsies of the right parotid and labial minor salivary glands revealed focal periductal lymphocytic infiltrate and sialoduct ectasia. Taken together, these findings are consistent with the diagnosis of juvenile PSS. The salient features of this rare disease are summarized on the basis of a comprehensive review of the epidemiologic, clinical, and serologic findings of the previously reported cases of PSS in children.

Child, Preschool↗

The management of sialolithiasis in 2 children through use of extracorporeal shock wave lithotripsy.

The management of salivary calculi in children may prove difficult for a number of reasons. Traditional investigations require ionizing radiation and in cases of sialography may be poorly tolerated. Similarly, any surgical treatment is likely to require general anesthesia and, in a number of cases, removal of the affected gland, which is associated with attendant risks. As an alternative, ultrasound and extracorporeal shock wave lithotripsy therapy offers low morbidity and outpatient procedures that are well tolerated, as illustrated by these 2 cases.

Adolescent↗

Parotid gland calculus. Report of a case.

A patient, was admitted with multiple calculi in the left parotid gland. Plain radiography and sialography confirmed the diagnosis. A partial parotidectomy was done and the calculi were removed.

Child↗

The parapharyngeal space.

A series of 73 lesions presenting in the parapharyngeal space is presented. There were 53 (73 per cent) patients with deep lobe parotid benign or malignant tumours, five (7 per cent) patients with tumours of neurogenous origin, seven (9 per cent) patients with abscesses, five (7 per cent) patients with enlarged lymph nodes, two with branchial cysts, and one with a lipoma. The anatomy emphasizes the expansile nature of the space and also the close proximity of vital structures. The importance of the use of combined CAT scan and sialography is discussed in the diagnosis of these patients. The various prognostic factors are mentioned and a description is given of the best way to approach the parapharyngeal space without damaging the facial nerve. With careful planning and preparation of the planned operation, the morbidity of the facial nerve may be considerably reduced.

Abscess↗

An unusual cause of obstructive sialadenitis.

A case of bilateral recurrent submandibular sialadenitis is presented in a patient with decreased salivary flow. The condition was caused by trapping of the submandibular papillae between the lingual flange of the denture and the enlarged genial tubercles. Removal of these causes led to resolution of her symptoms. The value of plain radiography, sialography and scintigraphy is discussed.

Aged↗

A superfemale with primary Sjögren's syndrome which involved systemic organs.

A 52-year-old Japanese woman complicated by a sex chromosomal anomaly as a superfemale, a mosaic of XXXXX/XXXX/XXX/XX/XO, with mild mental retardation, was hospitalized for dry mouth, dry eyes, and proteinuria. The sialography of the right parotid gland showed a globular-type gland enlargement. A definite diagnosis of primary Sjögren's syndrome (SS) was made, and further examinations revealed not only typical sicca syndrome but also systemic extraglandular lymphocytic infiltration; interstitial pneumonitis, glomerular- and interstitial nephritis, superficial gastritis, thyroiditis, and a severe excitation conductive impairment of heart. We report a very rare case of superfemale with primary SS which involved systemic organs.

Bronchi↗

Chronic non-specific parotid sialadenitis.

Chronic non-specific sialadenitis of the parotid gland is an insidious inflammatory disorder which is characterised by intermittent, often painful, swelling of the gland. The disease tends to progress and may lead to the formation of a fibrous mass. The purpose of this paper is to review our experience in the surgical management of patients with chronic non-specific parotid sialadenitis. In a consecutive series of 100 patients treated for benign parotid disease, 19 were found to have chronic non-specific sialadenitis; 10 were male and 9 female. Mean age was 46 years and the mean duration of symptoms was 4.6 years. Sialography was performed in two-thirds of the patients and 17 patients were treated by superficial parotidectomy. Thirteen patients developed temporary facial nerve weakness and three Frey's syndrome. Three patients complained of temporary paraesthesia of the cheek, and two developed painful neuroma of the greater auricular nerve. Although there were two cases of infection of the parotid duct remnant, no recurrence of deep lobe sialadenitis or fistula formation was noted. Histologically, 3 lesions showed mild chronic sialadenitis, the rest had widespread involvement of the gland, and prolonged duration of symptoms was associated with extensive and severe involvement of the gland. Superficial parotidectomy has a very high success rate, with minimal long-term complications, and should be offered early in established cases, to reduce unnecessary morbidity.

Adult↗

[Chronic recurrent parotitis in childhood].

Juvenile recurrent parotitis (j.r.p.) is distinguished from the adult form by the course of the disease, therapeutic considerations and prognosis. Children suffering from j.r.p. are between 2 and 15 years of age; the male: female ratio is 1.5:1. In the present paper the author describes the different pathogenic theories of j.r.p. in accordance with the relevant literature. Besides malformation of the glandular duct like stenosis and ectasis, functional factors like the character of salivary secretion are suspected as being responsible for the disease. Other authors assume that a viral genesis, allergic factors, a physiological immaturity of the immune response, or family history may be the causes. In the evaluation of children with j.r.p. the patients' history with recurrent swelling episodes of the parotid gland(s) followed by quiescent periods is indicative. Further procedures like ultrasonography, sialography and MRI are discussed according to their diagnostic value. Therapeutic possibilities include antibiotics in the acute stage of the disease as well as parotidectomy in severe cases. Radiotherapy cannot be recommended because of its side effects (facial dysplasia, tumour induction). In most cases, the disease ends at puberty.

Child↗

[Hypertrophy of the masseter muscle. Clinical aspects--etiology--therapy].

Masseteric muscle hypertrophy is a problem occurring in daily practice. Main complaints are isolated swelling of the masseteric muscle, myalgia, and functional disturbances of the parotid gland. Diagnosis is effected by inspection, palpation and anterior posterior radiograph. Sialography, CT, EMG are rarely used. Main points in the discussion of the aetiology are congenital genesis, the psychological factor, habits and occlusal disorders. The therapy consists of surgical reduction of the hypertrophied muscle as well as of conservative methods.

Bruxism↗

[Clinical, histopathologic and immunohistochemical studies of chronic sialectatic parotitis in childhood and adolescence].

UNLABELLED: Chronic sialectatic parotitis (CSP) causes problems in differential diagnosis and therapy. CSP shows the typical clinical features of chronic recurrent parotitis and will be investigated histopathologically only after ultimative parotidectomy. The etiology and pathogenesis of these unspecific inflammations is still unknown. Therefore no causal therapy is available and a lot of different trials (sialogoga, gland massage, infrared light, antibiotics, antiphlogistics, Trasylol, duct occlusion, duct ligation, gland denervation, radiotherapy) are not successful in the long run. MATERIAL AND METHOD: The salivary gland registry of the University of Hamburg (1965-1996) contains 22 infants and juvenile patients showing very severe courses of CSP. These cases have been investigated clinical (ultrasound, sialography), histopathological (paraffin embedded sections, histomorphometry of the ectatic duct lumina) and immunohistochemical (CK-MNF, AKTIN, KiM4) in a retrospective study to research the pathogenesis of CSP. RESULTS: Recurrent and always very dolent parotid swelling occurs between the age of 3 and 14 years for the first time. The courses vary from 3 months until 25 years. Local findings as well as ultrasound and sialographic features allow no certain differentiation of chronic recurrent parotitis. Conservative therapy fails in each case and leads to the necessity of surgical treatment. Histopathological three different stages of development can be observed: Initial stages show regular lobular architectonic structure of the parotid gland parenchyme with duct ectasies surrounded by slight inflammation of lymphocytes and plasmacells. Advanced stages are characterized by an increase of periductal inflammation and the appearance of lymphfollicels. Nearly complete lymphatic transformation of the parenchyme with destruction of the lobular formation dominates the terminal "immunologic" stage. Some cases show multiple myoepithelial islands within this lymphatic stroma typically observed in benign lymphoepithelial lesions. Whether bacteria nor primary obstructive changes can be observed. The histomorphometric analyses of the average and maximal luminal duct diameters show marked increase of 39% respectively 46% from and- vanced to terminal stages of CSP. Therefore the pathognomonic duct ectasies seem to depend on the progredient inflammation and are not due to a hereditary malformation of the duct system. Immunohistochemical terminal stages show follicular lymphatic hyperplasia (KiM4) expressing overshooting humoral immune reaction of MALT. CONCLUSION: Concerning the pathogenesis CSP corresponds to a immunopathological disorder of MALT and seems to be a prestage of benign lymphoepithelial lesion. Consequently important changes in the diagnosis and therapy of CSP lead to early histopathological investigation to differentiate the stage of inflammation. In stage III conservative parotidectomy should be carried out because spontaneous healing can not be expected. In contrast initial cases should be treated at first by glucocorticoids and immunosuppressives.

Adolescent↗

[Sonography in salivary calculi--indications and status].

In 23 patients with sialolithiasis the value of real time sonography was investigated. The calculi were correctly diagnosed in 91% of the patients. 3 of 4 calculi could be localized in the main duct of the salivary gland. Because of the high accuracy and the possibility to detect non opaque stones, sonography should be performed, if on plain film the localisation of the calculi is questionable, before sialography is done. In sonolithotrypsy, sonography can be used to document the extraglandular localisation of intraductal calculi.

Humans↗

Pediatric sialolithiasis.

Pediatric sialolithiasis is a rare condition. This article characterizes 15 cases in children between 5 and 14 years of age. The diagnoses of this condition were made with routine radiograph and ultrasound, as well as with sialography whenever possible. Sialoendoscopy was performed as a diagnostic and treatment modality. Thirteen of the 15 affected children were boys, and 12 of 15 cases occurred in the submandibular gland. We were able to diagnose 67% by our imaging methods; the remainder were diagnosed by clinical examination.

Adolescent↗

A case of IgG4-related autoimmune disease with multiple organ involvement.

A 52-year-old male was admitted with autoimmune pancreatitis (AIP), showing mononuclear cell infiltration in both the pancreas and salivary glands with both normal sialography and anti-SS-A/SS-B antibodies. Although the AIP improved with glucocorticoid treatment, subsequent abdominal computed tomography (CT) revealed a nodular shadow in the bilateral kidneys, which was confirmed as interstitial nephritis by renal biopsy. The patient's serum immunoglobulin G4 (IgG4) level was 10 times higher than the upper limit of the normal range. IgG4-positive mononuclear cell infiltration was detected in the salivary gland, pancreas, and kidney. A new entity proposed as 'IgG4-related autoimmune disease' was considered.

Autoimmune Diseases↗

Parotid gland ultrasonography as a diagnostic tool in primary Sjögren's syndrome.

The diagnostic value of parotid gland ultrasonography (Acuson 128, 7 MHz transducer) was studied in 62 patients with primary Sjögren's syndrome (SS) and in 69 controls of similar age and sex distribution. Different degrees (mild, evident or gross) of parenchymal inhomogeneity (PIH) were the most important sonographic changes in SS; they occurred in 83.9% of the patients. The sonographic results (the presence or absence of PIH) were in accordance with the parotid sialographic and scintigraphic findings and the histology of the minor salivary glands in 87.3, 84.7 and 84.3% of the cases, respectively. Of the degrees of PIH, only evident and gross PIH are thought to be of true diagnostic value for SS. On the basis of the good agreement between the sonographic and sialographic results, consideration of the introduction of parotid sonography as an alternative to sialography is suggested in SS if the latter method cannot be performed.

Adult↗

Post-traumatic parotid fistulae and sialoceles. A prospective study of conservative management in 51 cases.

The management of parotid sialoceles and fistulae have been unsatisfactory in the past, and numerous methods of treatment with varying success and morbidity have been described. The present prospective study reports results of conservative therapy in 51 patients over a 3-year period. In 50 patients, the injury healed upon conservative management. During the early phase of the study, a limited conservative regimen through which the patients received nothing orally for 5 days only was used. During the latter part of the study, patients were administered nothing orally until complete healing of the injury. In terms of the time it took for healing of the injury, the differences of the two regimens (24 +/- 4 vs. 9.4 +/- 0.9 days) was highly significant (p less than 0.001). The response to conservative management depended on the severity of injury as demonstrated by sialography. Injury to minor intraparotid ducts (G1) healed in significantly less time compared with that to a major intraparotid duct (G2) or ductal injuries (p less than 0.001). There was no difference between the healing of G2 injury (10.3 +/- 1.8 days) and partial ductal transections (10.5 +/- 2.2 days) (p greater than 0.05). There was a significantly greater delay in healing with complete duct transections (21.5 +/- 3.7 days) compared with partial duct transections and G2 injuries (10.2 +/- 2.1 days) (p less than 0.01). There was no difference in the mean period for healing between salivary fistulae and sialoceles (p greater than 0.05). It is concluded that a new classification of parotid fistulae based on sialographic findings has prognostic and therapeutic value. Furthermore, the excellent results achieved with conservative therapy in this study suggest that it may be the initial treatment of choice for parotid fistulae.

Acute Disease↗

Benign pleomorphic adenomas of the salivary glands: surface coil MR imaging versus CT.

High resolution magnetic resonance (MR) imaging of three salivary gland tumors was performed at 1.5 T and compared with CT. Two of the three tumors were well seen on CT. However, one required CT sialography to separate tumor from the remaining parotid parenchyma, and, in the other, soft tissue invasion could not be excluded on the basis of CT. Magnetic resonance imaging demonstrated sharp tumor margins with no evidence of invasion. All three tumors were pathologically proven benign pleomorphic adenomas confirming the MR findings. On T2 weighted images, the two large adenomas demonstrated inhomogeneity that was not observed on CT.

Adenoma, Pleomorphic↗