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1.
Eur J Pediatr ; 2024 Jun 28.
Article in English | MEDLINE | ID: mdl-38943011

ABSTRACT

Anterior and posterior drooling are prevalent comorbidities in children with neurodevelopmental disabilities. Considering the heterogeneity of the patient population and the multifactorial aetiology of drooling, an interdisciplinary and individualised treatment approach is indispensable. However, no tool for stepwise decision-making in the treatment of paediatric drooling has been developed previously. Within the Radboudumc Amalia Children's Hospital, care for children with anterior and/or posterior drooling secondary to neurodevelopmental disabilities is coordinated by a saliva control team with healthcare professionals from six disciplines. In alignment with international literature, published guidelines, and evidence gained from two decades of experience and research by our team, this paper proposes an algorithm reflecting the assessment and treatment approach applied in our clinic. First, directions are provided to decide on the necessity of saliva control treatment, taking type of drooling, the child's age, and the severity and impact of drooling into account. Second, the algorithm offers guidance on the choice between available treatment options, highlighting the importance of accounting for child characteristics and child and caregiver preferences in clinical (shared) decision-making. CONCLUSIONS: With this algorithm, we aim to emphasise the importance of repeated stepwise decision-making in the assessment and treatment of drooling in children during their childhood, encouraging healthcare professionals to apply a holistic approach. WHAT IS KNOWN: • Children with anterior or posterior drooling secondary to neurodevelopmental disabilities comprise a heterogeneous group, necessitating an individualised treatment approach. • No stepwise decision-making tool is available for the treatment of paediatric drooling. WHAT IS NEW: • Deciding on the necessity of saliva control treatment should be a conscious process, based on type of drooling, age, and drooling severity and impact. • Type of drooling, age, cognition, oral motor skills, self-awareness, posture, diagnosis, and child/caregiver preferences need to be considered to decide on the optimal treatment.

2.
Eur J Paediatr Neurol ; 51: 24-31, 2024 May 16.
Article in English | MEDLINE | ID: mdl-38776696

ABSTRACT

BACKGROUND: Despite the established efficacy of glycopyrronium bromide in reducing drooling among children with neurodevelopmental disabilities, evidence on its impact on the daily lives of children and parents and effectiveness in a real-world setting are scarce, especially among long-term users. This study explored timing and duration of glycopyrronium treatment, effect and impact on daily life, and occurrence of side effects to inform clinical practice. METHODS: This was a retrospective cohort study at a national referral centre for drooling, including 61 children with nonprogressive neurodevelopmental disabilities, treated with glycopyrronium for anterior and/or posterior drooling between 2011 and 2021. Data were obtained from medical records and supplemented by structured telephone interviews with parents. RESULTS: Anterior drooling severity decreased in 82% of the included children. Changes in the impact of drooling on burden of care, social interaction, and self-esteem were reported in 55%, 31%, and 36%, respectively. Side effects were noted for 71% of cases, yet only 36% of parents deemed these as outweighing the positive impact of treatment. A substantial majority (77%) of the included children were long-term users (≥6 months). Among these, 38% of parents reported decreasing effectiveness and 27% noticed more prominent side effects over time. CONCLUSIONS: Glycopyrronium demonstrated potential in mitigating the impact of drooling on daily life, although variations were observed in the specific aspects and extent of improvement. The real-world context of our study provides important insights for refining clinical practices, emphasizing the need for balanced consideration of treatment benefits and potential side effects to facilitate shared decision-making.

3.
Eur J Pediatr ; 183(1): 83-93, 2024 Jan.
Article in English | MEDLINE | ID: mdl-37924348

ABSTRACT

Paediatric anterior drooling has a major impact on the daily lives of children and caregivers. Intraglandular botulinum neurotoxin type-A (BoNT-A) injections are considered an effective treatment to diminish drooling. However, there is no international consensus on which major salivary glands should be injected to obtain optimal treatment effect while minimizing the risk of side effects. This scoping review aimed to explore the evidence for submandibular BoNT-A injections and concurrent submandibular and parotid (i.e. four-gland) injections, respectively, and assess whether outcomes could be compared across studies to improve decision making regarding the optimal initial BoNT-A treatment approach for paediatric anterior drooling. PubMed, Embase, and Web of Science were searched to identify relevant studies (until October 1, 2023) on submandibular or four-gland BoNT-A injections for the treatment of anterior drooling in children with neurodevelopmental disabilities. Similarities and differences in treatment, patient, outcome, and follow-up characteristics were assessed. Twenty-eight papers were identified; 7 reporting on submandibular injections and 21 on four-gland injections. No major differences in treatment procedures or timing of follow-up were found. However, patient characteristics were poorly reported, there was great variety in outcome measurement, and the assessment of side effects was not clearly described.   Conclusion: This review highlights heterogeneity in outcome measures and patient population descriptors among studies on paediatric BoNT-A injections, limiting the ability to compare treatment effectiveness between submandibular and four-gland injections. These findings emphasize the need for more extensive and uniform reporting of patient characteristics and the implementation of a core outcome measurement set to allow for comparison of results between studies and facilitate the optimization of clinical practice guidelines. What is Known: • There is no international consensus on which salivary glands to initially inject with BoNT-A to treat paediatric drooling. What is New: • Concluding on the optimal initial BoNT-A treatment based on literature is currently infeasible. There is considerable heterogeneity in outcome measures used to quantify anterior drooling.and clinical characteristics of children treated with intraglandular BoNT-A are generally insufficiently reported. • Consensus-based sets of outcome measures and patient characteristics should be developed and implemented.


Subject(s)
Botulinum Toxins, Type A , Sialorrhea , Humans , Child , Sialorrhea/drug therapy , Sialorrhea/etiology , Neurotoxins/pharmacology , Neurotoxins/therapeutic use , Submandibular Gland , Botulinum Toxins, Type A/therapeutic use , Botulinum Toxins, Type A/pharmacology , Treatment Outcome
4.
Dev Med Child Neurol ; 66(7): 919-930, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38140924

ABSTRACT

AIM: To develop robust multivariable prediction models for non-response to (1) submandibular botulinum neurotoxin A (BoNT-A) injections and (2) concurrent submandibular and parotid (four-gland) injections, to guide treatment decisions for drooling in children with neurodevelopmental disabilities, including cerebral palsy. METHOD: This was a retrospective cohort study including 262 children (155 males/107 females, median age 7 years 11 months [IQR 5 years 1 month], range 4 years 0 months - 17 years 11 months) receiving submandibular injections and 74 children (52 males/22 females, median age 7 years 7 months [IQR 4 years 3 months], range 4 years 9 months - 18 years 8 months) receiving four-gland injections. Multivariable logistic regression analyses were used to estimate associations between candidate predictors and non-response 8 weeks after injection. RESULTS: Ninety-six children (37%) were non-responders to submandibular injections, for which developmental age was the strongest predictor (adjusted odds ratio [aOR] 2.13; 95% confidence interval [CI] 1.02-4.45 for developmental age <4 years or 4-6 years with IQ <70). Other characteristics that showed a trend towards an increased risk of non-response were diagnosis, sex, and head position. Thirty-four children (46%) were non-responders to four-gland injections, for which tongue protrusion (aOR 3.10; 95% CI 1.14-8.43) seemed most predictive, whereas multiple preceding submandibular injections (aOR 0.34; 95% CI 0.10-1.16) showed a trend towards being protective. Predictors were, however, unstable across different definitions of non-response and both models (i.e. submandibular and four-gland) had insufficient discriminative ability. INTERPRETATION: Potential predictors of non-response to BoNT-A injections were identified. Nevertheless, the developed prediction models seemed inadequate for guidance of treatment decisions. WHAT THIS PAPER ADDS: Developmental age seemed most predictive of non-response to submandibular botulinum neurotoxin A injections. Non-response to concurrent submandibular and parotid injections was best predicted by tongue protrusion and number of previous injections. Multivariable prediction models including these clinical characteristics were unable to discriminate well. Predictors differed when non-response was defined using alternative outcome measures.


Subject(s)
Botulinum Toxins, Type A , Neurodevelopmental Disorders , Sialorrhea , Submandibular Gland , Humans , Sialorrhea/drug therapy , Sialorrhea/etiology , Male , Female , Botulinum Toxins, Type A/administration & dosage , Botulinum Toxins, Type A/pharmacology , Child , Child, Preschool , Adolescent , Retrospective Studies , Submandibular Gland/drug effects , Neuromuscular Agents/administration & dosage , Neuromuscular Agents/pharmacology , Cerebral Palsy/complications , Cerebral Palsy/drug therapy , Parotid Gland
5.
Perspect Med Educ ; 12(1): 418-426, 2023.
Article in English | MEDLINE | ID: mdl-37868074

ABSTRACT

Introduction: Official documentation of specialty training provides comprehensive and elaborate criteria to assess residents. These criteria are commonly described in terms of competency roles and entrustable professional activities (EPA's), but they may also implicitly encompass virtues. Virtues are desirable personal qualities that enable a person, in this case, a medical specialist, to make and act on the right decisions. We articulate these virtues and explore the resulting implied ideal of a medical professional. Method: We applied a two-staged virtue ethical content analysis to analyze documents, specific to the Dutch training program of the Ear, Nose, and Throat (ENT) specialty. First, we identified explicit references to virtues. Next, we articulated implicit virtues through interpretation. The results were categorized into cardinal, intellectual, moral, and professional virtues. Results: Thirty virtues were identified in the ENT- training program. Amongst them, practical wisdom, temperance, and commitment. Furthermore, integrity, curiosity, flexibility, attentiveness, trustworthiness and calmness are often implicitly assumed. Notable findings are the emphasis on efficiency and effectiveness. Together, these virtues depict an ideal of a future medical specialist. Conclusion: Our findings suggest that competency-frameworks and EPA's implicitly appeal to virtues and articulate a specific ideal surgeon. Explicit attention for virtue development and discussion of the role and relevance of implied ideal professionals in terms of virtues could further improve specialty training.


Subject(s)
Medicine , Surgeons , Humans , Virtues , Morals
6.
Cancer Cytopathol ; 131(11): 701-707, 2023 11.
Article in English | MEDLINE | ID: mdl-37519238

ABSTRACT

BACKGROUND: The Milan System for Reporting Salivary Gland Cytopathology (MSRSGC) is developed to aid diagnosis and management of salivary gland tumors. This study evaluates the time-to-treatment initiation (TTI) for parotid gland tumors in the Netherlands and relates these to the MSRSGC classification. Subsequently, the use of the MSRSGC in the Netherlands is evaluated. METHODS: Data regarding fine-needle aspiration cytology (FNAC) and histopathological resections of the parotid were gathered from the Dutch nationwide pathology data bank (PALGA). The TTI was calculated for each MSRSGC category and type of treating center. FNACs performed from 2018 to 2021 were gathered from PALGA to estimate how frequently the MSRSGC classification was applied. RESULTS: Median TTI in days were 86 for nondiagnostic (MSRSGC I), 75 for nonneoplastic (MSRSGC II), 65 for atypia of unknown significance (AUS) (MSRSGC III), 89 for benign (MSRSGC IVa), 52 for salivary gland neoplasm of unknown malignant potential (SUMP) (MSRSGC IVb), 31 for suspected malignant (MSRSGC V), and 30 for malignant (MSRSGC VI) categories. Significant variation in the TTI between the types of treating centers was found for the nondiagnostic, nonneoplastic, AUS, SUMP, and suspected malignant categories. In the first 3 years after the introduction of the MSRSGC, the pathologist stated the MSRSGC classification in 6.4% of all reports. CONCLUSIONS: The median TTI for most categories is long, and there is significant interhospital variation in TTI. Preoperative risk stratification and treatment prioritization in parotid gland surgery in the Netherlands should be improved. The MSRSGC could contribute to this. Until 2021, the MSRSGS classification was implemented on a limited scale in the Netherlands.


Subject(s)
Parotid Gland , Salivary Gland Neoplasms , Humans , Parotid Gland/surgery , Parotid Gland/pathology , Retrospective Studies , Salivary Glands/pathology , Salivary Gland Neoplasms/diagnosis , Cytodiagnosis
7.
J Clin Med ; 12(4)2023 Feb 12.
Article in English | MEDLINE | ID: mdl-36836008

ABSTRACT

The aim of this study was to evaluate our institutions airway management and complications after submandibular duct relocation (SMDR). We analysed a historic cohort of children and adolescents who were examined at the Multidisciplinary Saliva Control Centre between March 2005 and April 2016. Ninety-six patients underwent SMDR for excessive drooling. We studied details of the surgical procedure, postoperative swelling and other complications. Ninety-six patients, 62 males and 34 females, were treated consecutively by SMDR. Mean age at time of surgery was 14 years and 11 months. The ASA physical status was 2 in most patients. The majority of children were diagnosed with cerebral palsy (67.7%). Postoperative swelling of the floor of the mouth or tongue was reported in 31 patients (32.3%). The swelling was mild and transient in 22 patients (22.9%) but profound swelling was seen in nine patients (9.4%). In 4.2% of the patients the airway was compromised. In general, SMDR is a well-tolerated procedure, but we should be aware of swelling of the tongue and floor of the mouth. This may lead to a prolonged period of endotracheal intubation or a need for reintubation which can be challenging. After extensive intra-oral surgery such as SMDR we strongly recommend a extended perioperative intubation and extubation after the airway is checked and secure.

8.
Clin Otolaryngol ; 48(3): 430-435, 2023 05.
Article in English | MEDLINE | ID: mdl-36585381

ABSTRACT

OBJECTIVE: Parotid surgery is historically performed as an inpatient procedure and suctions drains are predominantly used during surgery. Recent literature provides evidence that outpatient parotid surgery is safe and effective. Our study aims to describe the results of drainless outpatient parotidectomy and outpatient parotidectomy with drain placement and compare their outcomes. DESIGN: Retrospective cohort study. SETTING: Bi-institutional retrospective cohort study. PARTICIPANTS: Patients that underwent outpatient drain-less parotidectomy and patients that underwent outpatient parotidectomy with post-operative drain placement. MAIN OUTCOME MEASURES: Complication rates, unplanned post-operative visits, unplanned prolonged stay. RESULTS: Three hundred eighty patients underwent outpatient parotidectomy with drain placement and 31 patients underwent outpatient drainless parotidectomy in two different hospitals. The incidence of haematoma (drain: 3.1% vs. drainless: 0%, p = 1), infection (drain: 14.3% vs. drainless: 13.8%, p = 1) and salivary fistula (drain: 5.6% vs. drainless: 3.4, p = 1) were comparable between both groups. Seroma or sialocele was more frequently seen in the drain-less group (27.6% vs. 6.2%, p < .001), but were all managed conservatively. Within 10 days after surgery, unplanned visits seemed more frequent in the drain group, although the difference was not statistically significant (14.9% vs. 3.4%, p = .16). CONCLUSIONS: Outpatient parotid surgery with or without the use of a post-operative drain is safe, practical and feasible. Same-day discharge with and without drain placement yield comparable outcomes. However, the results need to be interpreted cautiously as this study was limited by a small cohort of parotidectomies without drain placement. Future studies should further compare both approaches.


Subject(s)
Parotid Neoplasms , Salivary Gland Diseases , Humans , Retrospective Studies , Outpatients , Postoperative Complications/epidemiology , Parotid Gland/surgery , Parotid Neoplasms/surgery
9.
Int J Pediatr Otorhinolaryngol ; 164: 111377, 2023 Jan.
Article in English | MEDLINE | ID: mdl-36403383

ABSTRACT

AIM: Submandibular gland excision (SMGE) is suitable for the management of drooling in patients with non-progressive neurodisabilities. We aimed to investigate the long-term effects of SMGE. METHOD: Patients who had SMGE between 2007 and 2018 were included. Main outcomes were a Visual Analogue Scale (VAS), Drooling Severity (DS), and Drooling Frequency (DF) collected at baseline, 8 weeks, 32 weeks and with a median of 313 weeks after SMGE (long-term). Secondary outcomes were satisfaction with the procedure, Drooling Quotient (DQ) and adverse events (AEs). RESULTS: We included thirty-five patients in the long-term analysis with a mean age of 14.5 years. A baseline VAS score of 80.4 was found, which improved on the long-term (mean difference -21.8, t(26) = 4.636, p < 0.0005)). DS and DF decreased significantly at the long-term compared to baseline (Z = -4.361, p < 0.0001 for DS, Z = -3.065, p = 0.002 for DF). Twenty-three out of 35 (66%) patients would recommend the procedure to peers. INTERPRETATION: This study indicates a long-term stable effect on drooling after SMGE in patients with anterior drooling. Recurrence of drooling occurs due to unknown reasons, nevertheless most caregivers and/or patients are still satisfied and would recommend the procedure to others.


Subject(s)
Cerebral Palsy , Neurodevelopmental Disorders , Sialorrhea , Humans , Child , Adolescent , Sialorrhea/surgery , Sialorrhea/complications , Cross-Sectional Studies , Submandibular Gland/surgery , Salivary Glands , Treatment Outcome , Cerebral Palsy/complications
10.
Cancer Cytopathol ; 130(3): 189-194, 2022 Mar.
Article in English | MEDLINE | ID: mdl-34847276

ABSTRACT

BACKGROUND: The Milan System for Salivary Gland Cytopathology (MSRSGC) is a categorical system for salivary gland fine-needle aspiration cytopathology (FNAC) developed to aid clinicians in the management of salivary gland lesions. This classification is widely studied and validated, especially in cohorts that consist of mostly parotid gland lesions. However, only sparse literature describes the use of this classification for submandibular gland lesions in particular. METHODS: All patients in the Netherlands that underwent a submandibular gland resection between January 1, 2006, and January 1, 2017, with a FNAC before resection were identified with the use of the Dutch Pathology Registry database (PALGA). All FNAC results were retrospectively classified according to the MSRSGC. The risk of malignancy was calculated for all the MSRSGC categories. The sensitivity and specificity of the MSRSGC classification were calculated for submandibular gland FNAC. RESULTS: A total of 837 patients who underwent 975 FNAC aspirates from the submandibular glands were included in the analysis. Risks of malignancy for each of the MSRSGC categories were 14.4% in nondiagnostic, 4.4% in nonneoplastic, 37.0% in atypia of unknown significance, 3.9% in benign neoplasms, 40.7% in salivary gland neoplasms of unknown malignant potential, 76.2% in suspected malignant, and 91.3% in malignant cytology results. The sensitivity for diagnosing malignant submandibular gland tumors was 71.6% and specificity was 98.4%. CONCLUSIONS: The results of the present study validate the use of this classification for submandibular gland lesions. Risks of malignancy vary according to the anatomical subsites of the salivary gland lesions. LAY SUMMARY: The risks of malignancy of the various Milan System for Salivary Gland Cytopathology (MSRSGC) categories vary according to the anatomical subsite of the salivary gland lesion. The proposed management techniques of the MSRSGC are valid for use with submandibular gland lesions.


Subject(s)
Salivary Gland Neoplasms , Submandibular Gland , Biopsy, Fine-Needle/methods , Humans , Retrospective Studies , Salivary Gland Neoplasms/diagnosis , Salivary Gland Neoplasms/pathology , Salivary Gland Neoplasms/surgery , Salivary Glands/pathology , Submandibular Gland/pathology
11.
J Voice ; 36(1): 128-133, 2022 Jan.
Article in English | MEDLINE | ID: mdl-32434679

ABSTRACT

OBJECTIVES: To assess the influence that several factors, such as the amount of obtained biopsies, difficult procedures, biopsy site and the experience of the attending physician, have on accuracy of flexible endoscopic biopsy (FEB). MATERIALS AND METHODS: 203 FEB procedures for benign or malignant laryngopharyngeal lesions were prospectively included. During the procedure, three representative biopsies (macroscopically containing vital tumor tissue and not only necrosis or healthy tissue) were obtained. The accuracy of each biopsy was separately analyzed. Difficulties during the procedures leading to failure of acquiring three representative biopsies were recorded and classified into tumor, patient and procedural factors. Histological results of FEB were defined correct when consistent with clinical context, additional biopsies or Positron emission tomography-computed tomography (PET-CT) revealed equivalent pathology, or the lesion was stable or resolved in >6 months follow-up. RESULTS: The first representative biopsy yielded a correct diagnosis in 65% of the cases. After the second representative biopsy, 78% was correctly diagnosed. The contribution of the third and fourth representative biopsies to accuracy was 3%. The overall accuracy of FEB was 85%. Difficult procedures were more likely to result in misdiagnosis, whereas biopsy site or experience of the attending physician did not influence results. CONCLUSIONS: FEB was accurate in diagnosing laryngopharyngeal lesions when at least two representative biopsies were obtained. Accuracy of FEB could be further improved by limiting possible constraints during the procedures, for example by selecting, informing, and anesthetizing patients carefully.


Subject(s)
Hypopharynx , Positron Emission Tomography Computed Tomography , Biopsy , Humans
12.
Int J Pediatr Otorhinolaryngol ; 150: 110894, 2021 Nov.
Article in English | MEDLINE | ID: mdl-34474251

ABSTRACT

OBJECTIVES: This study aims to evaluate the medium to long-term (1-12 years) effectiveness of two-duct ligation (2-DL) in patients with nonprogressive neurodisabilities. METHODS: Main outcomes included a Visual Analogue Scale (VAS), Drooling Severity (DS) and Drooling Frequency (DF), collected at baseline, 32 weeks postintervention and 1-12 years postintervention. Secondary outcomes were adverse events (AEs), and satisfaction. RESULTS: Forty-two patients were analyzed (mean age 17 years, mean baseline VAS 82). VAS decreased significantly from baseline to long-term (n = 30. Mean difference -36.5, CI -47.0 to -26.0, p ≤ 0.001; -26.1, CI -36.2 to -15.9, p ≤ 0.001). However, long-term VAS significantly increased compared to VAS at 32 weeks (+10.4, CI 1.0-19.8, p = 0.031). Out of 42 patients, 64% would recommend 2-DL to peers. CONCLUSION: There is a significant subjective 2-DL effect on drooling severity in the medium to long-term as reported by patients and caregivers, but there is also a certain degree of recurrence in this time span, and 33% of the patients required subsequent treatment. However, the majority of patients and/or caregivers would recommend 2-DL to peers.


Subject(s)
Cerebral Palsy , Sialorrhea , Adolescent , Cross-Sectional Studies , Humans , Neoplasm Recurrence, Local , Sialorrhea/etiology , Sialorrhea/surgery , Submandibular Gland , Treatment Outcome
13.
Dev Med Child Neurol ; 63(11): 1351-1359, 2021 11.
Article in English | MEDLINE | ID: mdl-33997959

ABSTRACT

AIM: To compare the effect of bilateral submandibular duct ligation and botulinum neurotoxin A (BoNT-A) on drooling severity and its impact on daily life and care in children and adolescents with moderate-to-severe drooling. METHOD: This was a randomized, interventional, controlled trial in which 53 children and adolescents (31 males, 22 females, mean age 11y, range 8-22y, SD 2y 10mo) with cerebral palsy (58.5%) or other non-progressive developmental disorders (41.5%) were randomized to BoNT-A (n=26) or bilateral submandibular duct ligation (n=27). A parent questionnaire on the severity of drooling in specific positions and daily activities and the impact of drooling on daily life and care was filled out at baseline and 8 and 32 weeks posttreatment. RESULTS: Both BoNT-A and bilateral submandibular duct ligation had a positive effect on daily care, damage to electronic equipment and/or furniture, social interactions, and self-esteem. However, bilateral submandibular duct ligation had a significant greater and longer-lasting short- (8wks) and medium-term (32wks) effect on daily care, reducing damage to electronic devices, and improving social interactions and satisfaction with life in general. INTERPRETATION: This randomized controlled trial confirms reduced drooling by both BoNT-A and bilateral submandibular duct ligation, but provides new evidence on improved well-being through a reduction in drooling. Even though there is a greater risk of complications and morbidity after bilateral submandibular duct ligation, compared to BoNT-A there was a significantly greater and longer-lasting positive effect on most outcomes. What this paper adds Bilateral botulinum neurotoxin A (BoNT-A) and submandibular duct ligation had a positive effect on the well-being of individuals with moderate-to-severe drooling. Bilateral submandibular duct ligation had a greater effect on the impact of drooling during daily care than BoNT-A. Bilateral submandibular duct ligation reduced damage to electronic devices and improved social interactions and satisfaction with life.


Subject(s)
Botulinum Toxins, Type A/therapeutic use , Developmental Disabilities/therapy , Quality of Life , Salivary Ducts/surgery , Sialorrhea/therapy , Adolescent , Child , Developmental Disabilities/drug therapy , Developmental Disabilities/surgery , Female , Humans , Male , Sialorrhea/drug therapy , Sialorrhea/surgery , Treatment Outcome , Young Adult
14.
Cancer Cytopathol ; 129(9): 719-728, 2021 09.
Article in English | MEDLINE | ID: mdl-33908189

ABSTRACT

BACKGROUND: Differentiating between malignant and benign salivary gland tumors with fine-needle aspiration cytology (FNAC) can be challenging. This study was aimed at testing the validity of the Milan System for Reporting Salivary Gland Cytopathology (MSRSGC) and at assessing possible differences in the sensitivity and specificity of parotid gland FNAC between dedicated head and neck (H&N) centers, subdivided into head and neck oncology centers (HNOCs) and head and neck oncology affiliated centers (HNOACs), and general hospitals (GHs). METHODS: The Dutch Pathology Registry (PALGA) database was searched for patients who had undergone a salivary gland resection between January 1, 2006, and January 1, 2017, and had a preoperative FNAC result. The FNAC reports were retrospectively assigned to MSRSGC categories. The risk of malignancy (ROM) was calculated for each category. The sensitivity and specificity for diagnosing malignancy were calculated and compared among HNOCs, HNOACs, and GHs. RESULTS: In all, 12,898 FNAC aspirates were evaluated. The ROMs for each category were as follows: 12.5% in MSRSGC I, 10.3% in MSRSGC II, 29% in MSRSGC III, 2.3% in MSRSGC IVa, 28.6% in MSRSGC IVb, 83% in MSRSGC V, and 99.3% in MSRSGC VI. The sensitivity of FNAC was highest in HNOCs (88.1%), HNOACs scored lower (79.7%), and GHs had a sensitivity of 75.0%. CONCLUSIONS: The MSRSGC is a valid tool for reporting parotid gland FNAC; therefore, these results strongly advocate its use. On the basis of the higher sensitivity of FNAC in dedicated H&N centers, the authors recommend that GHs use the presented management strategies to help to minimize the chances of a preoperative misdiagnosis.


Subject(s)
Parotid Gland , Salivary Gland Neoplasms , Biopsy, Fine-Needle , Humans , Parotid Gland/pathology , Pathology/methods , Reproducibility of Results , Retrospective Studies , Salivary Gland Neoplasms/pathology , Salivary Glands/pathology
15.
Neuromuscul Disord ; 30(11): 925-929, 2020 11.
Article in English | MEDLINE | ID: mdl-33077317

ABSTRACT

We report a case of a patient with concurrent myotonic dystrophy and recurrent pleomorphic adenoma and hypothesize the association between both diseases. A 58-year-old man with classic myotonic dystrophy type 1 was diagnosed with pleomorphic adenoma. Appropriate treatment was commenced. Massive recurrences occurred within 15, 28 and 22 months respectively, after repeated surgical removal. Three case reports on similar occurrences of synchronous myotonic dystrophy and pleomorphic adenoma are discussed and an association between both disease entities is hypothesized. A conceivable association between myotonic dystrophy and pleomorphic adenoma is hypothesized by upregulation of the Wnt/Beta-catenin signaling pathway, initiated by a decreased expression of microRNA, pleomorphic adenoma gene 1 induced Beta-catenin accumulations and alterations in tumor suppressor genes and oncogenes due to RNA processing defects induced by the expanded repeat in the DMPK gene.


Subject(s)
Adenoma, Pleomorphic/complications , Myotonic Dystrophy/complications , Salivary Gland Neoplasms/complications , Adenoma, Pleomorphic/diagnosis , Humans , Magnetic Resonance Imaging , Male , MicroRNAs , Middle Aged , Myotonin-Protein Kinase , Parotid Gland/pathology , Salivary Gland Neoplasms/diagnosis , Up-Regulation , beta Catenin
16.
Cancer ; 126(17): 3982-3990, 2020 09 01.
Article in English | MEDLINE | ID: mdl-32634271

ABSTRACT

BACKGROUND: Timely and efficient diagnostic workup of patients with head and neck cancer (HNC) is challenging. This observational study describes the implementation of an optimized multidisciplinary oncological diagnostic workup for patients with HNC and its impact on diagnostic and treatment intervals, survival, costs, and patient satisfaction. METHODS: All patients with newly diagnosed HNC who underwent staging and treatment at the Radboud University Medical Center were included. Conventional workup (CW) in 2009 was compared with the fast-track, multidisciplinary, integrated care program, that is, optimized workup (OW), as implemented in 2014. RESULTS: The study included 486 patients with HNC (218 with CW and 268 with OW). The time-to-treatment interval was significantly lower in the OW cohort than the CW cohort (21 vs 34 days; P < .0001). The 3-year overall survival rate was 12% higher after OW (72% in the CW cohort vs 84% in the OW cohort; P = .002). After correction for confounders, the 3-year risk of death remained significantly lower in the OW cohort (hazard ratio, 1.73; 95% confidence interval, 1.14-2.63; P = .010). Total diagnostic costs were comparable in the 2 cohorts. The general satisfaction score, as measured with the Consumer Quality Index for Oncological Care, was significantly better in a matched OW group than the CW group (9.1 vs 8.5; P = .007). CONCLUSIONS: After the implementation of a fast-track, multidisciplinary, integrated care program, the time-to-treatment interval was significantly reduced. Overall survival and patient satisfaction increased significantly, whereas costs did not change. This demonstrates the impact and improved quality of care achieved by efficiently organizing the diagnostic phase of HNC management.


Subject(s)
Chemotherapy, Adjuvant , Head and Neck Neoplasms/diagnosis , Head and Neck Neoplasms/therapy , Time-to-Treatment , Cohort Studies , Female , Head and Neck Neoplasms/epidemiology , Head and Neck Neoplasms/pathology , Humans , Male , Middle Aged , Patient Satisfaction , Progression-Free Survival , Proportional Hazards Models , Survival Rate
17.
Dev Med Child Neurol ; 62(7): 861-867, 2020 07.
Article in English | MEDLINE | ID: mdl-32149393

ABSTRACT

AIM: To assess: (1) the effect on drooling of bilateral submandibular duct ligation as surgical therapy after the administration of submandibular botulinum neurotoxin A (BoNT-A) for excessive drooling and (2) the predictive value of treatment success with BoNT-A on treatment success after bilateral submandibular duct ligation. METHOD: This was a within-participant retrospective observational study in which 29 children with severe drooling (15 males, 14 females) received BoNT-A treatment at a mean age of 9 years 6 months (SD 2y 5mo), followed by bilateral submandibular duct ligation at a mean age of 10 years 11 months (SD 2y 4mo). Fifteen children were diagnosed with cerebral palsy (CP), with 12 children classified in Gross Motor Function Classification System levels IV and V. The 14 children without CP had non-progressive developmental disorders. The primary drooling severity outcomes were the Visual Analogue Scale (VAS; subjective assessment) and drooling quotient (objective assessment). Measurements were taken before each intervention and again at 8 and 32 weeks. RESULTS: The VAS was significantly lower after bilateral submandibular duct ligation at follow-up compared to BoNT-A treatment (mean difference -33, p≤0.001; 95% confidence interval [CI]=-43.3 to -22.9). The mean drooling quotient did not significantly differ between BoNT-A treatment and bilateral submandibular duct ligation at follow-up (3.3, p=0.457; 95% CI=-4.35 to 9.62) or between 8 and 32 weeks (4.7, p=0.188; 95% CI=-2.31 to 11.65). INTERPRETATION: BoNT-A treatment and bilateral submandibular duct ligation are both effective treatment modalities for drooling. At 32-week follow-up, subjective drooling severity after bilateral submandibular duct ligation was significantly lower compared to previous BoNT-A injections in participants. However, treatment success with BoNT-A is no precursor to achieving success with bilateral submandibular duct ligation. WHAT THIS PAPER ADDS: Bilateral submandibular duct ligation is an effective therapy for drooling after treatment with botulinum neurotoxin A (BoNT-A). Treatment success with BoNT-A is not a predictor of successful therapy with bilateral submandibular duct ligation.


Subject(s)
Botulinum Toxins, Type A/pharmacology , Cerebral Palsy , Neuromuscular Agents/pharmacology , Outcome Assessment, Health Care , Salivary Ducts/surgery , Sialorrhea , Submandibular Gland/surgery , Botulinum Toxins, Type A/administration & dosage , Cerebral Palsy/complications , Cerebral Palsy/drug therapy , Cerebral Palsy/surgery , Child , Female , Humans , Male , Neuromuscular Agents/administration & dosage , Retrospective Studies , Sialorrhea/drug therapy , Sialorrhea/etiology , Sialorrhea/surgery
18.
Otolaryngol Head Neck Surg ; 162(4): 446-457, 2020 Apr.
Article in English | MEDLINE | ID: mdl-32093572

ABSTRACT

OBJECTIVE: An increased interval between symptomatic disease and treatment may negatively influence oncologic and/or functional outcomes in head and neck cancer (HNC). This systematic review aims to provide insight into the effects of time to treatment intervals on oncologic and functional outcomes in oral cavity, pharyngeal, and laryngeal cancer. DATA SOURCES: PubMed, EMBASE, and Cochrane library were searched. REVIEW METHODS: All studies on delay or time to diagnosis or treatment in oral, pharyngeal, and laryngeal cancer were included. Quality assessment was performed with an adjusted version of the Newcastle-Ottawa scale. Outcomes of interest were tumor volume, stage, recurrence, survival, patient-reported outcome measures (PROMs), toxicity, and functionality after treatment. RESULTS: A total of 51 studies were included. Current literature on the influence of delay in HNC is inconsistent but indicates higher stage and worse survival with longer delay. The effects on PROMs, toxicity, and functional outcome after treatment have not been investigated. The inconsistencies in outcomes were most likely caused by factors such as heterogeneity in study design, differences in the definitions of delay, bias of results, and incomplete adjustment for confounding factors in the included studies. CONCLUSION: Irrespective of the level of evidence, the unfavorable effects of delay on oncologic, functional, and psychosocial outcomes are undisputed. Timely treatment while maintaining high-quality diagnostic procedures and decision making reflects good clinical practice in our opinion. This review will pose practical and logistic challenges that will have to be overcome.


Subject(s)
Laryngeal Neoplasms/diagnosis , Laryngeal Neoplasms/therapy , Mouth Neoplasms/diagnosis , Mouth Neoplasms/therapy , Pharyngeal Neoplasms/diagnosis , Pharyngeal Neoplasms/therapy , Time-to-Treatment , Humans , Treatment Outcome
19.
J Neurol ; 267(5): 1508-1515, 2020 May.
Article in English | MEDLINE | ID: mdl-32025794

ABSTRACT

BACKGROUND: Drooling is dependent on various clinical variables. However, while drooling proves refractory to two-duct ligation in 40% of patients, predictors for treatment success are sparse and to date there is little evidence why some respond well while others are non-responders. We aim to find predictors for treatment success and study the effectiveness of two-duct ligation for drooling in neurodisabilities. METHODS: Fifty-four patients with moderate to severe drooling who had undergone two-duct ligation were screened for inclusion. Four patients were excluded due to missing or unreliable primary outcomes. The average age at the time of surgery was 12 years. Predictors were evaluated for treatment success which was defined as ≥ 50% visual analog scale for severity of drooling and/or drooling quotient reduction from baseline. Treatment effect was measured after 8 and 32 weeks compared to baseline. RESULTS: Age (more mature), adequate posture (no anteflexion), and normal speech are predictors for treatment success. Compared to baseline, drooling quotient was significantly lower at 8 (difference 18.6%, 95% confidence interval 12.3-24.9%) and 32 weeks (difference 10.1%, 95% confidence interval 3.9-16.4%). Compared to baseline, visual analog scale was significantly lower at 8 (difference 45.0, 95% confidence interval 37.0-52.9) and 32 weeks (difference 32.9, 95% confidence interval 25.0-40.7). CONCLUSIONS: Age, adequate posture, and a normal speech are predictors for treatment success, are easily determined pre-operatively, and help the clinician providing patient-specific probability of treatment success. There is a significant subjective and objective decrease of drooling after two-duct ligation.


Subject(s)
Cerebral Palsy/complications , Neurodevelopmental Disorders/complications , Oral Surgical Procedures , Outcome Assessment, Health Care , Sialorrhea/etiology , Sialorrhea/surgery , Submandibular Gland/surgery , Adolescent , Child , Clinical Decision-Making , Female , Follow-Up Studies , Humans , Ligation , Male , Oral Surgical Procedures/adverse effects , Oral Surgical Procedures/methods , Prognosis , Retrospective Studies , Severity of Illness Index , Sialorrhea/physiopathology
20.
Laryngoscope ; 130(6): 1503-1507, 2020 06.
Article in English | MEDLINE | ID: mdl-31498454

ABSTRACT

OBJECTIVE: Patients with laryngeal pathology are often treated with CO2 laser surgery, usually in the operating room under general anesthesia. Although office-based laser surgery using several other laser types has been investigated, prospective studies on office-based CO2 laser surgery are scarce. Our goal was to investigate the feasibility of office-based CO2 laser surgery for benign and premalignant laryngeal pathology by analyzing completion rate, safety, effect on voice quality, and success rate (i.e., no residual or recurrent disease). METHODS: A prospective cohort study was performed of 30 consecutive procedures. Inclusion started in June 2016 and was completed in August 2018. Adult patients with clinically benign or premalignant laryngeal lesions who could not undergo transoral laser microsurgery in the operating room under general anesthesia were included. Reasons were either contraindications for general anesthesia, previously failed therapeutic laryngoscopy under general anesthesia, and preference of a procedure under topical anesthesia by the patient. The mean follow-up was 9 months. RESULTS: Thirty procedures were performed in 27 patients (24 males) with an average age of 62 years. Twenty-nine (97%) procedures were fully completed without complications. The mean preoperative Voice Handicap Index (VHI) score (VHI 44) significantly decreased 2 months (VHI 28, P = 0.032) and 6 months (VHI 14, P < 0.001) after the procedure. Almost two-thirds of patients showed no residual or recurrent disease at their follow-up visits. CONCLUSION: Office-based CO2 laser surgery is a feasible and safe procedure that results in significant voice-quality improvement. Almost two-thirds of patients did not require further treatment. LEVEL OF EVIDENCE: 2 Laryngoscope, 130:1503-1507, 2020.


Subject(s)
Ambulatory Surgical Procedures , Laryngeal Diseases/surgery , Lasers, Gas/therapeutic use , Precancerous Conditions/surgery , Feasibility Studies , Female , Humans , Male , Middle Aged , Prospective Studies
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