Scurvy masquerading as IgA vasculitis.

BACKGROUND: Vitamin C deficiency, or scurvy, is rare but poses risks for children with poor diets, limited resources, or malabsorption issues. It may also be common in children with restrictive or selective dietary habits in children with global developmental delay, autism spectrum disorder, and physical disabilities. Symptoms include fatigue, irritability, joint and muscle pain, joint swellings, edema, swollen gums, easy bruising, and delayed wound healing. Early recognition and prompt intervention are essential to prevent the progression of symptomatic vitamin C deficiency in children. CASE PRESENTATION: We present a case of a 13-year-old boy with developmental delay secondary to Lennox Gastaut syndrome referred for suspected recurrent, severe, and atypical IgA vasculitis. He presented with irritability, loss of appetite, petechial and ecchymotic lower limb lesions, unilateral gum swelling, severe arthritis, peripheral oedema, severe weight loss, anaemia, and raised inflammatory markers. Multiple investigations were performed before the diagnosis of scurvy was made. A surgical finding of friable gingival tissue with multiple loose teeth, a skin biopsy with follicular hyperkeratosis and extravasated perifollicular red blood cells, and a typical X-ray finding led to the diagnosis of scurvy. CONCLUSION: Scurvy should be given careful consideration as a differential diagnosis in patients presenting with musculoskeletal issues, mucocutaneous complaints, and constitutional symptoms such as malaise, asthenia, irritability, and loss of appetite. A focused and detailed dietary history looking for a lack of good sources of vitamin C can be an easy indicator of this differential. Imaging studies revealing the typical features can also help make the diagnosis. Pathology of the skin revealing pathognomonic features can add to the certainty of the diagnosis. In the absence of all else, the rapid response to treatment with an appropriate dose of vitamin C has a diagnostic and therapeutic role.

Effects of maxillomandibular advancement on respiratory function and facial aesthetics in obstructive sleep apnoea patients with versus without maxillomandibular deficiency.

The aim of this study was to compare the effects of maxillomandibular advancement (MMA) on respiratory function between obstructive sleep apnoea (OSA) patients with and without maxillomandibular deficiency, and to compare the changes in facial aesthetics after MMA between the two groups. MMA-treated patients who had both baseline and follow-up polysomnography (PSG) data and lateral cephalograms were enrolled in this retrospective study. In addition to PSG and cephalometric data, patient satisfaction with postoperative breathing and facial aesthetics, and overall satisfaction with the treatment were assessed. Twenty-one patients were classified as not having maxillomandibular deficiency (without-deficiency group) and 40 patients as having maxillomandibular deficiency (with-deficiency group). The improvements in respiratory parameters (e.g., apnoea-hypopnoea index) and patient satisfaction with postoperative breathing were comparable in the two groups (P = 0.094-0.713). The changes in facial profile measurements (e.g., nasal prominence, nasolabial angel, and lip positions relative to the true vertical line) and patient satisfaction with postoperative facial aesthetics were also comparable in the two groups (P = 0.148-0.983). In conclusion, no significant difference in the effects of MMA on respiratory function and facial aesthetics between OSA patients with and without maxillomandibular deficiency was observed.

[Patient-specific joint prothesis for ankylotic temporomandibular joints]. / Individueel vervaardigde prothesen voor ankylotische kaakgewrichten.

A 49-year-old woman who suffered from severe obstructive sleep apnea (OSA) was referred to the department of Oral-, Maxillofacial Surgery department due to progressive limitation of the mouth opening and chronic pain in both temporomandibular joints. Based on clinical and radiological examinations, the patient was diagnosed with recurrent ankylosis of the temporomandibular joints. The patient was treated with 2 patient-specific implants of the temporomandibular joint combined with a Le Fort I osteotomy, and a genioplasty including a genioglossus advancement. This treatment may have advantages for the patient such as a lower recurrence rate of ankylosis, improved maximal mouth opening, pain reduction and improved aesthetic results.

Prevalence of post-traumatic neuropathic pain after digital nerve repair and finger amputation.

INTRODUCTION: Post-traumatic neuropathic pain is a major factor affecting the quality of life after finger trauma and is reported with considerable variance in the literature. This can partially be attributed to the different methods of determining neuropathic pain. The Douleur Neuropathique 4 (DN4) has been validated to be a reliable and non-invasive tool to assess the presence of neuropathic pain. This study investigated the prevalence of neuropathic pain after finger amputation or digital nerve repair using the DN4 questionnaire. METHODS: Patients with finger amputation or digital nerve repair were identified between 2011 and 2018 at our institution. After a minimal follow-up of 12 months, the short form DN4 (S-DN4) was used to assess neuropathic pain. RESULTS: A total of 120 patients were included: 50 patients with 91 digital amputations and 70 patients with 87 fingers with digital nerve repair. In the amputation group, 32% of the patients had pain, and 18% had neuropathic pain. In the digital nerve repair group, 38% of the patients had pain, and 14% had neuropathic pain. Secondly, of patient-, trauma-, and treatment-specific factors, only the time between trauma and surgery had a significant negative influence on the prevalence of neuropathic pain in patients with digital nerve repair. CONCLUSION: This study shows that persistent pain and neuropathic pain are common after finger trauma with nerve damage. One of the significant prognostic factors in developing neuropathic pain is treatment delay between trauma and time of digital nerve repair, which is of major clinical relevance for surgical planning of these injuries.

Prevention is better than cure: Surgical methods for neuropathic pain prevention following amputation - A systematic review.

BACKGROUND: Pain after amputation can be known as residual limb pain (RLP) or phantom limb pain (PLP); however, both can be disabling in daily life with reported incidences of 8% for finger amputations and up to 85% for major limb amputations. The current treatment is focused on reducing the pain after neuropathic pain occurs. However, surgical techniques to prevent neuropathic pain after amputation are available and effective, but they are underutilized. The purpose of the review is to investigate the effects of techniques during amputation to prevent neuropathic pain. METHODS: A systematic review was performed in multiple databases (Embase, Medline, Web of Science, Scopus, Cochrane, and Google Scholar) and following the PRISMA guidelines. Studies that reported surgical techniques to prevent neuropathic pain during limb amputation were included. RESULTS: Of the 6188 selected studies, 13 eligible articles were selected. Five articles reported techniques for finger amputation: neurovascular island flap, centro-central union (CCU), and epineural ligatures, and flaps. For finger amputations, the use of prevention techniques resulted in a decrease of incidences from 8% to 0-3% with CCU being the most beneficial. For major limb amputations, the incidences for RLP were decreased to 0 to 55% with TMR and RPNI and compared to 64-91% for the control group. Eight articles reported techniques for amputations on major limbs: targeted muscle reinnervation (TMR), targeted nerve implantation, concomitant nerve coaptation, and regenerative peripheral nerve interface (RPNI). CONCLUSIONS: Based on the current literature, we state that during finger and major limb amputation, the techniques to prevent neuropathic pain and PLP should be performed.

Impact of orthognathic surgery on quality of life in patients with different dentofacial deformities: longitudinal study of the Oral Health Impact Profile (OHIP-14) with at least 1 year of follow-up.

OBJECTIVE: The objective of this study was to assess the impact of orthognathic surgery for dental facial deformities on oral health-related quality of life (OHRQoL) in the immediate postoperative period up to at least 1 year after surgery. STUDY DESIGN: This prospective study evaluated data from 85 patients. OHRQoL was assessed using the Dutch version of the Oral Health Impact Profile questionnaire (OHIP-14NL) preoperatively (T0), each day for 7 days postoperatively (T1-T7) and 4 weeks (T8), 6 months (T9), and at least 1 year (T10) after surgery. The total OHIP score was calculated for each patient, with higher OHIP scores indicating a worse impact on oral health. Patients also completed an extra questionnaire about self-care, discomfort, and experienced pain (rated on a 10-point scale) in the postoperative period (T1-T10). RESULTS: The mean OHIP score increased sharply at T1 compared to T0 but decreased significantly in the first postoperative week. The mean OHIP score at T8 was still higher than before surgery. However, at T9 and T10, the mean OHIP score was significantly lower than at T0 (P < .05). No significant difference in OHIP score was found between gender, age, type of surgery, and indication for surgery. Pain significantly decreased from T6 to T0. The OHIP and pain scores significantly positively correlated at every time point except T9. CONCLUSION: The findings indicate that OHRQoL is reduced from baseline in the immediate postoperative period but improves over time. By 1 year, OHRQoL improves significantly after orthognathic surgery in patients with dentofacial deformities.

[Intra-oral Perineurioma, a case report]. / Perineurioma: een zeldzame intraorale wekedelentumor.

A 48 year old woman was referred by her general practitioner to an oral and maxillofacial surgeon because of an asymptomatic, slow growing intra-oral tumor since three years. There were no sensory and motor symptoms. A well-defined tumor of 5 cm in diameter was located in the right cheek between the zygomaticus arch and the labial commissure. The skin and the intra-oral mucosa were intact without any change in colour or texture. The MRI showed a solitary mass with benign characteristics. A transoral radical excisional biopsy was performed under general anesthesia. Histopathological examination revealed a rare soft tissue perineurioma tumor.

Stability of fixation methods in large mandibular advancements after sagittal split ramus osteotomy: an in vitro biomechanical study.

Sagittal split ramus osteotomy (SSRO) with large mandibular advancements is a common surgical procedure and could be indicated for patients with sleep apnoea. As a large variety of fixation methods is used for the stabilisation of SSRO, a biomechanical test model was used to analyse which fixation technique was most stable. For this in vitro study, 80 polyurethane hemimandibles with a prefabricated SSRO were used as substrates. Loads in Newtons were recorded at displacements of the mandibular incisive edge at 1mm, 3mm and 5mm. The samples were divided into two groups: mandibular advancements of 10mm and 15mm. In both groups, four fixation techniques were used: (A) one four-hole miniplate; (B) two four-hole miniplates; (C) one four-hole miniplate plus one bicortical screw; and (D) three bicortical screws in an inverted-L arrangement. In group 1, three bicortical screws resulted in the best stability, and in group 2, two miniplates resulted in the best stability. The use of two miniplates did not show significant differences between both groups. Other fixation methods showed more stability with 10mm advancements. This study therefore suggests that in SSRO with advancements exceeding 10mm, the use of two miniplates is the optimal means of providing rigid fixation.

Elevated Coronary Artery Calcium scores are associated with tooth loss.

AIM: This study explores the association between Coronary Artery Calcium (CAC) scores and dental pathology such as missing teeth, the (peri-apical) health status and restoration grade of the teeth, and the grade of alveolar bone loss seen on a dental panoramic radiograph (Orthopantomograph-OPG). MATERIALS AND METHODS: In this retrospective cross-sectional study, data was collected from three hospitals spread in the Netherlands. Patients were included when a CAC score and an OPG were available, both recorded within a maximum period of 365 days from 2009-2017. The CAC score was measured on a CT scan, using the Agatston method. To assess dental pathology, the number of missing teeth, the number of dental implants, alveolar bone loss, caries, endodontic treatments, peri-apical radiolucencies, bone loss at implants, impacted teeth and dental cysts, were determined on the OPG. All observers were calibrated. The electronic health records provided information about: gender, age, smoking, Diabetes Mellitus, hypercholesterolemia, hypertension and Body Mass Index (BMI). RESULTS: 212 patients were included. We found a statistically significant association between the number of missing teeth and the CAC score. When modeling age, sex, and other well-known risk factors for cardiovascular disease, the significant correlation was no longer present after multivariate correction. Furthermore, the results showed a trend for more teeth with peri-apical lesions and a higher percentage of mean alveolar bone loss in the group with the highest CAC scores. CONCLUSION: This study showed that being edentulous or missing teeth is correlated to higher CAC scores however failed to be an independent predictor of atherosclerotic cardiovascular diseases. The number of (missing) teeth is an easily accessible marker and could be used as a marker for atherosclerotic cardiovascular disease (ACVD) risk by almost any healthcare worker. The current study needs to be considered as an explorative pilot study and could contribute to the design of further (prospective) studies on the relationship between dental pathology and coronary artery calcification by adding clinical information and extra cardiovascular biomarkers.

Cervicofacial non-tuberculous mycobacterial lymphadenitis: clinical determinants of incomplete surgical removal.

In patients with non-tuberculous mycobacterial cervicofacial lymphadenitis, incomplete surgical removal of infected lymph nodes leads to delayed healing and a higher recurrence rate, with eventual spontaneous drainage through the skin. However, complete surgical removal is not always achievable due to the extent of the infected tissue and proximity to vulnerable structures, such as the facial or accessory nerve. The aim of this study was to identify the clinical determinants of the (in)ability to perform complete surgical removal. The electronic health records of patients aged 0-15 years with bacteriologically proven non-tuberculous mycobacterial cervicofacial lymphadenitis, who underwent surgical treatment and preoperative sonographic imaging, were analysed. This was a case-control study. A total of 103 patients met the inclusion criteria. Most of the infections were unilateral, submandibular, and caused by Mycobacterium avium. Multiple logistic regression analysis revealed that higher age (odds ratio 1.24, 95% confidence interval 1.04-1.47) and fistulization (odds ratio 3.15, 95% confidence interval 1.13-8.75) were significantly associated with a limited ability to surgically remove all infected tissue. However, a larger sonographic lymph node size was not significantly associated. These findings could aid clinicians when informing the parent(s)/guardian(s) of the patient preoperatively and in properly estimating the intraoperative and postoperative course.

Outcome of a rabbit model for late irradiation effects in mandibular oral mucosa and bone: A pilot study.

BACKGROUND/AIM/OBJECTIVE: Late side effects of radiotherapy (RT) in the treatment for head and neck (HN) malignancies involve an inadequate healing response of the distressed tissue due to RT-induced hypovascularity. The aim of this study was to develop a pilot model in which vascular alterations associated with the onset of late irradiation (IR) injury could be measured in rabbit oral mucosa and mandibular bone. MATERIALS AND METHODS: Eight male New Zealand white rabbits were divided over four treatment groups. Group I-III received four fractions of RT (5.6 Gy, 6.5 Gy, and 8 Gy, respectively) and Group IV received 1 fraction of 30 Gy. Oral microcirculatory measurements were performed at baseline (before RT) and once a week during 11 consecutive weeks after RT assessing perfusion parameters, that is, total vessel density (TVD), perfused vessel density (PVD), proportion of perfused vessels (PPV), and microvascular flow index (MFI). Post-mortem histopathology specimens were analyzed. RESULTS: Five weeks after RT, TVD, and PVD in all groups showed a decrease of >10% compared to baseline, a significant difference was observed for Groups I, II, and IV (P<0.05). At T11, no lasting effect of decreased vessel density was observed. PPV and MFI remained unaltered at all-time points. Group IV showed a marked difference in scattered telangiectasia such as microangiopathies, histological necrosis, and loss of vasculature. CONCLUSION: No significant lasting effect in mucosal microcirculation density due to IR damage was detected. Observed changes in microcirculation vasculature and histology may align preliminary tissue transition towards clinical pathology in a very early state associated with late IR injury in the oral compartment. RELEVANCE FOR PATIENTS: Enhancing knowledge on the onset of late vascular IR injury in the HN region could help the development, monitoring, and timing of therapies that act on prevention, discontinuation, or repair of radiation pathology.

Splintless orthognathic surgery in edentulous patients-a pilot study.

The aim of this pilot study was to evaluate the accuracy and predictability of a splintless treatment protocol for edentulous patients undergoing orthognathic surgery in four consecutive cases. All operations were virtually planned, followed by computer-aided design of individual osteotomy guides and patient-specific fixation implants, which were three-dimensionally printed in titanium. In order to evaluate the discrepancy between the planned and the achieved postoperative result, the postoperative outcome was compared to the virtual treatment plan. Rotational and translational movement and discrepancies with the planned movements were quantified for the maxilla; the advancement was quantified for the mandible. For the maxilla, there was a mean translation discrepancy of 0.6mm. With regard to rotation, there was a mean discrepancy of 1.9°, 0.1°, and 0.4° for pitch, yaw, and roll, respectively. The mean discrepancy in translation of the mandible was 0.4mm. The results of this pilot study indicate that the splintless treatment protocol for orthognathic surgery in edentulous patients presented here is accurate and predictable.

Fixation methods in sagittal split ramus osteotomy: a systematic review on in vitro biomechanical assessments.

The aim of this systematic review was to assess the stability of rigid internal fixation (RIF) techniques in sagittal split ramus osteotomy (SSRO) based on in vitro biomechanical assessments, with particular interest in large mandibular advancements. In general, RIF methods can be divided into three groups: bicortical screws, miniplates, and a combination of the two. An electronic search of the PubMed, CINAHL, and Embase databases was performed, and studies published between January 2003 and March 2018 were screened for inclusion. Comparative studies with an in vitro experimental design, using biomechanical assessments to measure the stability of RIF methods in SSRO, were included. Of 104 unique studies identified in the initial search, 24 were included. Twenty-two of these 24 studies analyzed an advancement of the mandible of 7mm or less. The use of a single four-hole or six-hole miniplate was less stable than the use of bicortical screws, hybrid techniques, double miniplates, or grid plates. Two studies analyzed advancements of 10mm, for which two miniplates placed in parallel and a grid plate showed most stability. Although there was agreement between studies with regard to results, more biomechanical studies are required to quantify the stability of fixation methods in larger mandibular advancements.

[Maxillary osteonecrosis after denosumab treatment]. / Verlies van de bovenkaak door denosumab.

A 58-year-old patient with a history of intravenous use of denosumab was referred to the department of oral and maxillofacial surgery with inadequate recovery after a series of extractions. During physical and radiological examination, several sites of exposed necrotic bone with purulent discharge were seen, and the maxilla appeared fractured at the level of Le Fort 1. Treatment consisted of intravenous administration of antibiotics with consecutive sequestrectomy of the maxilla under general anaesthesia. While Medication Related Osteonecrosis of the Jaw (MRONJ) is most commonly seen in patients treated with bisphosphonates, in the past decade, it has become apparent that other types of medication, most notably denosumab, can cause comparable disorders. Treatment of MRONJ strongly depends on the stage the disease is in. In patients with a history of denosumab or bisphosphonate use (administered either intravenously or orally in combination with corticosteroids), abnormal radiological findings, or developing osteonecrosis, referral to a department of oral and maxillofacial surgery is recommended.

[An (un)conventional autogenous tooth transplantation]. / Een (on)conventionele autotransplantatie.

A 15-year-old boy was referred to an Oral and Maxillofacial Surgery department because of a persistent tooth 85 and a malposition of tooth 45. Following additional examination, it was concluded that the root formation of tooth 45 was already completed. Furthermore, it was horizontally impacted against the inferior border of the mandible. The decision was made to extract tooth 85 and to autotransplant tooth 45. Post-operatively, tooth 45 was fixed to the orthodontic equipment already in place and later treated endodontically. The success rate of this kind of autotransplantation of teeth with already closed apices (94%) is comparable in the literature to the success rate of autotransplantation of teeth with open apices (98%). Autotransplantation should be considered, especially in patients who are still growing and for whom dental implants do not (yet) offer a solution.

Open treatment of condylar fractures via extraoral approaches: A review of complications.

BACKGROUND: There is considerable controversy about whether condylar fractures should be treated open or closed. Even when there is a clear indication for open treatment, the appropriate approach opens another debate. PURPOSE: To provide a clear overview on the complications of extraoral approaches to condylar fractures. METHODS: Systematic literature search of all indexed years on PubMed, Medline, and Embase. For detailed analysis, we selected 70 studies. RESULTS: Of the 2783 patients who were studied with respect to facial nerve function, 338 (12%) experienced some form of weakness. Most (95%) achieved full recovery. In 17 of the 338 patients (5%), these paralyses were permanent. A hematoma occurred in 1.7%, an infection of the wound in ≥2.9%, Frey syndrome in 1.1%, a sialocele in 2.6%, a salivary fistula in ≥4.8%, sensory disturbance of the great auricular nerve in 7.9%, and an unsatisfactory scar in ≥1.6% of the patients. CONCLUSION: Due to the great diversity in fractures, approaches, and surgical techniques, it is difficult to objectively compare surgical techniques for condylar fractures and their complications. Based on the literature studied in this review, we propose a treatment protocol with respect to open treatment approaches.

Clinical outcomes in the treatment of unilateral condylar fractures: a cross-sectional study.

The treatment of mandibular condyle fractures has been the subject of considerable discussion, especially whether open or closed treatment should be used. There is a need for practical,evidence-based guidelines, and both objective and subjective parameters should be measured. To date, fewstudies have considered clinically relevant subjective parameters. This study was performed to evaluate the outcomes of the treatment of condylar fractures using the Mandibular Function Impairment Questionnaire (MFIQ) and the Diagnostic Criteria for Temporomandibular Disorders (DC/TMD) and to compare the outcomes of open and the closed treatment. Patients with condylar fractures and at least 1year of follow-up were examined. These patients completed the MFIQ and other questionnaires, and were examined according to the DC/TMD. Seventy-four of 171 eligible patients participated in this study. The mean MFIQ score was 10.70 (standard error 2.9) in the open group and 4.96 (standard error 1.3) in the closed group (P=0.023), an outcome in favour of the closed treatment group. Examination according to the DC/TMD did not reveal a significant prevalence of TMD complaints. Closed treatment appears to be a safe and appropriate modality for most unilateral condylar fractures. Although the open group in general showed similar outcomes, this treatment should be reserved for limited indications.

Fulminant presentation of oral mucosal leishmaniasis as severe stomatitis and periodontitis.

This case report shows an atypical presentation of mucosal leishmaniasis infantum in the oral cavity resulting in severe stomatitis and periodontitis. The patient was immunocompromised because of rheumatoid arthritis for which he used prednisone and methotrexate. He was treated with intravenous liposomal amphotericin B and recovered within four weeks.

Open treatment of unilateral mandibular condyle fractures in adults: a systematic review.

Since the introduction of rigid internal fixation devices, more and more surgeons favour an open approach to treating condylar fractures of the mandible in adult patients. Different indications for open treatment have been published. Open treatment is associated with surgical complications because of the technique employed. The aim of this systematic review was to provide an overview of the studies published exclusively on open treatment, and to summarize the existing open treatment modalities and their clinical outcomes. A total of seventy studies were selected for detailed analysis. Most studies reported good results with regard to the outcome measures of open treatment. Surgical complications including hematoma, wound infection, weakness of the facial nerve, sialocele, salivary fistula, sensory disturbance of the great auricular nerve, unsatisfactory scarring, and fixation failure were reported in the studies. This review suggests that because of the high level of methodological variance in the relevant studies published to date, among other factors, there are currently no evidence-based conclusions or guidelines that can be formulated with regard to the most appropriate open treatment. Establishment of such standards could potentially improve treatment outcomes.