Orocervical Actinomycosis in a Patient With Neck Swelling and Occipital Headache: A Case Report

Transsigmoid and extreme lateral approaches for removal of jugular foramen tumors

Skull Base Surgery with Minimal Resources.

Lyme arthritis first was described in the United States in 1977 after a report of a cluster of children with arthritis living in Old Lyme, Connecticut.1 In the subsequent 45 years, Lyme disease has come to be recognized as an infectious disease that if left untreated may involve multiple organ systems, including the skin, joints, heart, and nervous system, and is referred to as Lyme borreliosis. The Infectious Diseases Society of America issued guidelines for the management of Lyme disease based on a systematic review of the scientific literature in 2000 and 2006.2,3 The American Academy of Neurology issued guidelines in 2007 for the treatment of nervous system Lyme disease.4 Practice guidelines were affirmed in 2010.5 The current guidelines were published in 2020 by a panel representing the Infectious Diseases Society of America, American Academy of Neurology, and American College of Rheumatology, using the Grading of Recommendations, Assessment, Development and Evaluations approach to determine both certainty of evidence and strength of the recommendations.6 More than 33 individuals representing 9 societies, as well as patient representatives and a health care consumer representative, participated in the process.This report extracts recommendations from the 2020 guidelines for the management of pediatric Lyme disease that differ from previous guidelines and reviews treatment recommendations that remain unchanged. Changes in the guidelines include the use of doxycycline in children <8 years of age for postexposure prophylaxis after a nymphal Ixodes scapularis tick bite and for treatment of neuroborreliosis. In addition, we review treatment recommendations for erythema migrans, arthritis, and cardiac disease that remain unchanged. Finally, we note the current status of second-generation vaccine development for the prevention of Lyme disease.In the United States, Lyme borreliosis is caused by the spirochete Borrelia burgdorferi sensu stricto (B burgdorferi in the strict sense), except for a small number of cases caused by Borrelia mayonii in the upper Midwest. Transmission occurs during a bite by an infected tick of the species I scapularis in eastern and upper midwestern states or Ixodes pacificus in the west coast states. Person-to-person transmission of B burgdorferi sensu stricto, including vertical transmission resulting in congenital disease or via transfusion of blood products, has not been documented.Approximately 35 000 cases of Lyme borreliosis are reported annually to the Centers for Disease Control and Prevention Lyme disease surveillance program, although because of likely underreporting, the true number is estimated to be at least three- to 12-fold higher (>400 000 cases annually).7 Lyme borreliosis presents a significant disease burden for those living in the endemic regions of the United States, including the Northeast, Mid-Atlantic, and upper Midwest (Fig 1). Minimal, if any, disease occurs in other areas of the United States except in the west coast states, where a low incidence is recognized.Worldwide, at least 20 different genospecies of Borrelia are recognized and collectively, are referred to as B burgdorferi sensu lato (B burgdorferi in the general sense), although 3 genospecies account for most cases of Lyme borreliosis in humans: B burgdorferi sensu stricto, Borrelia afzelii, and Borrelia garinii. The latter 2 species are found in Europe and Asia and cause certain clinical syndromes such as acrodermatitis chronica atrophicans and borrelial lymphocytoma, which are not seen with B burgdorferi sensu stricto infection in North America. On the other hand, Lyme arthritis is much more common in North America than in Europe.Postexposure prophylaxis is recommended for adults largely on the basis of results from a chemoprophylaxis trial published in 2001.8 Trial results demonstrated that if specific factors are satisfied (the tick is identified as Ixodes species, the bite occurs in a highly endemic area, and the tick was attached for at least 36 hours), the benefit of prophylaxis outweighs the risk of a complication from doxycycline. Doxycycline traditionally has not been used in children <8 years of age because of concern about staining of permanent teeth. However, available data indicate this complication has not been associated with doxycycline, in contrast to older tetracyclines.6 Thus, current guidelines for postexposure prophylaxis (or treatment) of Lyme borreliosis include the use of doxycycline regardless of age.9 Doxycycline is administered as a single dose at 4.4 mg/kg for children up to a maximum of the adult dose of 200 mg (≥45 kg).Single-dose postexposure prophylaxis with amoxicillin is not recommended because of its short half-life (∼60 minutes) relative to that of doxycycline (16–22 hours). Prophylactic antibiotic therapy is not recommended for a tick bite that is equivocal or considered low risk, but the person should be followed for development of erythema migrans or other manifestations of infection. Other tick-borne infections, such as Babesia microti, likely are transmitted in <24 hours, emphasizing the need for prompt tick removal. Whenever possible, avoiding exposure to or prompt removal of ticks is preferred to postexposure prophylaxis.A person with a potential tick exposure in an endemic area who develops erythema migrans should be treated on the basis of the clinical findings. Laboratory testing is not recommended because most people will be seronegative during the early stage of Lyme borreliosis.Selection of an antimicrobial agent for the treatment of erythema migrans (amoxicillin, doxycycline, cefuroxime axetil, or azithromycin) should be based on the following considerations: the presence of extracutaneous manifestations of infection, particularly neurologic involvement (for which doxycycline is the drug of choice); drug allergy history; drug adverse effects; ability to minimize sun exposure (photosensitivity may be associated with doxycycline use); frequency of administration (doxycycline and cefuroxime are administered twice a day, amoxicillin is administered 3 times a day); and the likelihood of coinfection with Anaplasma phagocytophilum or an Ehrlichia muris–like agent (which are sensitive to doxycycline but not to β-lactam antibiotics (Table 1). In most circumstances, a child <8 years of age is treated with oral amoxicillin, and a patient >8 years of age is treated with oral doxycycline.Treatment of erythema migrans results in resolution of the rash within several days of antibiotic initiation and almost always prevents the development of later stages of disease. Among untreated adult patients who had erythema migrans before the cause of the disease was known, ∼5% developed Lyme carditis (mainly atrioventricular conduction abnormalities), 15% develop early neuroborreliosis within weeks, and ∼60% develop monoarticular or pauciarticular arthritis (mostly large joints, particularly the knees) within months.10Patients with possible Lyme arthritis should undergo serum antibody testing rather than polymerase chain reaction or culture of blood or synovial tissue. For patients with Lyme arthritis, oral antibiotic therapy for 28 days is recommended. As with therapy for erythema migrans, children <8 years of age are usually treated with oral amoxicillin and those >8 years of age are treated with oral doxycycline. For patients with Lyme arthritis who have minimal or no response to the initial course of oral antibiotic therapy, a 2- to 4-week course of intravenous (IV) ceftriaxone is recommended over a second course of oral antibiotic. Although rare, especially in prepubertal children, a postinfectious proliferative synovitis may develop after oral and IV therapy. Referral to a rheumatologist is recommended for consideration of treatment with disease-modifying antirheumatic drugs.An electrocardiogram is recommended for patients with signs or symptoms consistent with Lyme carditis, including dyspnea, edema, palpitations, lightheadedness, chest pain, or syncope. Patients with PR interval prolongation >300 milliseconds or other arrhythmias or signs of myopericarditis should be hospitalized for continuous electrocardiographic monitoring. Hospitalized patients with Lyme carditis should be treated with IV ceftriaxone, followed by an oral antibiotic once evidence of clinical improvement is present. An outpatient with Lyme carditis may receive an oral antibiotic instead of an IV antibiotic.Treatment of neuroborreliosis often is the same whether meningitis is present, so the decision to perform a lumbar puncture should be individualized. Performance of a cerebrospinal fluid analysis in patients with suspected neurologic disease is indicated for excluding bacterial, viral, or other etiologies of inflammation. In addition, children with neuroborreliosis may experience a pseudotumor-like syndrome, which can be diagnosed on the basis of the finding of increased intracranial pressure. Children who present with developmental, behavioral, or psychiatric disorders are not recommended to undergo routine testing for Lyme borreliosis, as B burgdorferi infection is unlikely to cause such disorders. For patients with Lyme borreliosis–associated meningitis or cranial neuropathy, oral antibiotic therapy with doxycycline generally is recommended over IV treatment because of ease of administration, a lower likelihood of adverse effects, and evidence of equivalent outcome in European studies.11 For patients with the very rare condition of Lyme disease encephalomyelitis (parenchymal involvement of the brain or spinal cord), IV ceftriaxone is recommended over oral antibiotic therapy. Routine use of corticosteroid therapy for children <16 years of age with cranial neuropathy is not recommended on the basis of lack of evidence of benefit.12Once diagnosed, Lyme borreliosis is a treatable disease, but if misdiagnosed or untreated, the infection can result in complications, excessive health care usage, inappropriate long-term antibiotic administration, and overuse of diagnostic testing. Thus, the burden of disease in endemic areas demonstrates the need for effective prevention strategies. Two recombinant vaccines based on the outer surface protein A (OspA) of the spirochete were developed in the 1990s, and one was licensed for prevention of Lyme borreliosis in 1998 for people 15 to 70 years of age living in areas with high rates of disease.13,14 The licensed vaccine (LYMErix; GlaxoSmithKline) was evaluated in a 3-dose series in a randomized placebo-controlled trial wherein 2 doses were administered in the first year with a booster dose 1 year later. Vaccine efficacy was >75% in the second season of the trial, confirming that serum concentration of antibody against OspA correlated with protection against B burgdorferi infection. However, unsubstantiated concerns about adverse reactions contributed to poor sales of the vaccine, and the vaccine was voluntarily withdrawn by the manufacturer in 2002.15In early-phase trials for preexposure disease prevention, researchers currently are evaluating both active immunization with a modified recombinant OspA vaccine or passive immunization with a long-half-life monoclonal antibody. An investigational, multivalent, subunit, adjuvanted Lyme vaccine (VLA15; produced in a collaboration between Valneva and Pfizer) has received fast-track designation by the US Food and Drug Administration and is being evaluated in an observer-blind, placebo-controlled phase 2 trial (ClinicalTrials.gov identifier: NCT04801420) of seropositive and seronegative participants ≥5 years of age treated with a 2- and 3-dose regimen.16As an alternative to active immunization, seasonal passive administration of a protective antibody at the start of tick season could be an effective strategy for prophylaxis in endemic areas.17 A monoclonal antibody that binds to a protective epitope on OspA may provide protection similar to that of a vaccine. A single dose of a long-half-life antibody at the beginning of the season theoretically would offer immediate protection that persists for the duration of seasonal exposure risk, but the injection would need to be repeated in each subsequent year.The authors thank Paul M. Lantos, MD, for helpful comments regarding this manuscript.

Introduction and aim. Pulmonary actinomycosis is a rare chronic infection caused by Actinomyces species, often mimicking tuberculosis, malignancy, or other chronic lung diseases both clinically and radiologically. These similarities frequently lead to diagnostic delays. We present a rare case of pulmonary actinomycosis in a patient with long-standing bronchiectasis who developed acute hemoptysis during anticoagulant therapy. Description of the case. A 59-year-old woman with a ten-year history of bronchiectasis and recent mechanical aortic valve replacement presented with persistent hemoptysis while on warfarin. Thoracic computed tomography revealed bronchiectatic changes and opacities in the right middle lobe. Bronchoscopy showed bloody, encrusted material, but no definitive diagnosis was made. Due to ongoing symptoms and radiological suspicion of malignancy, an urgent right middle lobectomy was performed. Histopathological analysis confirmed pulmonary actinomycosis, revealing filamentous organisms via PAS, Gram, and Grocott’s staining. Conclusion. This case highlights the importance of considering pulmonary actinomycosis in patients with bronchiectasis and unexplained hemoptysis, particularly when standard treatment fails. Surgical resection may be essential for both diagnosis and treatment. The case underscores how structural lung disease and anticoagulant use can reveal rare infections otherwise masked by chronic symptoms.

N OTHERWISE HEALTHY 24-year-old man presented with progressive left neck swelling of 2‐3 weeks duration. Physical examination was remarkable for a diffusely enlarged and tender left thyroid gland with a 3 � 3 � 2 cm sized mass. The patient’s work-up revealed normal leukocyte count of 4960 cells per microliter with normal differentiation with hemoglobin of 14.2 g/dL, hematocrit of 42.8%, and platelet count of 267,000 cells per microliter. Tests of thyroid function revealed thyroxine of 4.4 � g/dL (normal, 4.5‐12 � g/dL), triiodothyronine of 0.67 ng/mL (normal, 0.6‐1.7 ng/mL), and thyrotropin (TSH) of 1.1508 � U/mL (normal, 0.4‐5 � U/mL) with antithyroglobulin antibody of below 20 (normal, below 40 IU/mL), negative antithyroid microsome antibody and TSH receptor antibody of 9.20 U/L. Thyroid scan with 99m Tc pertechnetate (Fig. 1) showed no enlarged thyroid glands with inhomogeneous decreased thyroid uptake in left lobe of thyroid gland. Thyroid ultrasonography (Fig. 2) showed an ill-defined area of heterogenous hypoechogenicity (arrows) in the left lobe of thyroid gland and left lateral neck obliterating the margin of left lobe. Precontrast-enhanced computed tomography scan of the neck (Fig. 3A) demonstrated an ill-defined low-density lesion (arrows) in the left thyroid lobe and adjacent soft tissue, lateral to left thyroid cartilage and medial to left sternocleidomastoid muscle. On contrast-enhanced scan (Fig. 3B), the lesion (arrows) was heterogeneousely enhanced with obliteration of the fat plane between the left thyroid gland. Rim-like enhancement of multiple enlarged lymph nodes along the bilateral internal jugular chains was noted. Findings are suggestive of inflammatory condition involving the left thyroid lobe and adjacent soft tissue. After the ultrasound-guided aspiration, left lobectomy of thyroid gland was performed. Histopathology (Fig. 4) demonstrated gram-positive, branching, thin, filamentous-like organisms suggestive of actinomyces, and the characteristic sulfur granules (arrows), consistent with an actinomycosis abscess of the left thyroid lobe and left lateral neck. Actinomycosis is an uncommon disease caused by Actinomyces species, gram-positive anaerobic bacteria and normal inhabitants in the oral cavity and respiratory and digestive tracts. Clinical manifestation occurs in one of three forms: cervicofacial, abdominal‐pelvic, or pulmonary actinomycosis. Cervicofacial form accounts for approximately 50% of the cases and is an exceedingly uncommon entity. Also, thyroid involvement is even more unusual. Imaging findings include an enhancing soft-tissue mass with a low-attenuating area associated with inflammation in the adjacent soft tissue including the muscles. If imaging is performed in the acute phase, actinomycosis may be seen as a localized mass with moderate homogeneous contrast enhancement (1‐3). We report a rare case of acute thyroiditis of actinomycosis with imaging findings of computed tomography (CT) and ultrasonogram.

Imaging interpretation of skull base lesions can be challenging because of their infrequent occurrence, the complex nature of the skull base and the ability of normal anatomical variations to mimic pathology. Since the majority of skull base lesions are accidentally detected and inaccessible for biopsy, the clinicians heavily rely upon the accurate image interpretation by the radiologist. The main goals of the image analysis are to distinguish normal anatomical variations form true pathology, recognize medically treatable conditions and to differentiate benign “do-not-touch” lesions from malignant processes that might require aggressive treatment. The imaging features and preferential location of the different malignant skull base lesions and their benign counterparts will be discussed in this chapter. After the diagnosis of a malignancy is established, the radiologist’s main role is the delineation of the extent of the suspected malignant skull base lesion for treatment planning.

Tick-borne encephalopathies constitute a broad range of infectious diseases affecting the brain and other parts of the CNS. The causative agents are both viral and bacterial. This review focuses on the current most important tick-borne human diseases: tick-borne encephalitis (TBE; including Powassan encephalitis) and Lyme borreliosis. Rocky Mountain spotted fever (RMSF) and Colorado tick fever (CTF), less common tick-borne diseases associated with encephalopathy, are also discussed. TBE is the most important flaviviral infection of the CNS in Europe and Russia, with 10 000-12 000 people diagnosed annually. The lethality of TBE in Europe is 0.5% and a post-encephalitic syndrome is seen in over 40% of affected patients, often producing a pronounced impairment in quality of life. There is no specific treatment for TBE. Two vaccines are available to prevent infection. Although these have a good protection rate and good efficacy, there are few data on long-term immunity. Lyme borreliosis is the most prevalent tick-borne disease in Europe and North America, with >50 000 cases annually. Localised early disease can be treated with oral phenoxymethylpenicillin (penicillin V), doxycycline or amoxicillin. The later manifestations of meningitis, arthritis or acrodermatitis can be treated with oral doxycycline, oral amoxicillin or intravenous ceftriaxone; intravenous benzylpenicillin (penicillin G) or cefotaxime can be used as alternatives. The current use of vaccines against Lyme borreliosis in North America is under discussion, as the LYMErix vaccine has been withdrawn from the market because of possible adverse effects, for example, arthritis. RMSF and CTF appear only in North America. RMSF is an important rickettsial disease and is effectively treated with doxycycline. There is no treatment or preventative measure available for CTF.

<p>Prikazan je slujaj poznice stare 52 godine sa abdominalnom aktinomykozom, kod koje su se symptoms zapatili dva mecea pre hospitalizationizacije. Abdominal actinomycosis is rare, chronic, granulomatous infection, koju karakterime pojaja ''sumpornih granula''. It usually occurs in patients with surgical interventions or invasive procedures, manifesting tumors in the abdomen. The diagnosis is often difficult to establish. U manje od 10% is placed preoperatively. Definitive diagnosis is based on makroskopskom and histološkom pregledu sampler zavrentih tsikava. Lečenje is carried out intravenskom primenom coché doza penicillin u durati od 2-6 nedelja, a potom peroralna antibiotska terapija traje još 6-12 meseci.</p>

A CASE OF PRIMARY PULMONARY BOTRYOMYCOSIS IN A PATIENT WITHOUT ANY UNDERLYING ILLNESS

Evaluation of skull base lesions is challenging. On the one hand, the skull base is not directly accessible for clinical evaluation, and an underlying lesion is suspected or can be only roughly outlined based on neurological deficits. On the other, cross-sectional radiological studies are excellent in demonstrating a skull base lesion and its extent, but their evaluation is intimidating to the majority of the radiologists. There are three main reasons for the intimidation of the radiologists: The anatomical complexity of the skull base, the ability of normal anatomical structures to mimic pathology and the rarity of skull base lesions preventing dedicated training throughout residency and even during fellowship. In addition, inappropriate choice of an imaging study, imaging parameters and or sequences may amplify the insecurity of the radiologist.KeywordsInternal Carotid ArterySkull BaseCavernous SinusSphenoid SinusJugular ForamenThese keywords were added by machine and not by the authors. This process is experimental and the keywords may be updated as the learning algorithm improves.

We study the occurrence and management of the trigeminocardiac reflex (TCR) during neurosurgical procedures for lesions of the skull base. Two hundred patients underwent neurosurgical procedures for various skull base lesions and were evaluated retrospectively for the occurrence of the TCR during surgery. This phenomenon was defined as the onset of bradycardia lower than 60 beats/minute and hypotension with a drop in mean arterial blood pressure of 20% or more due to intra-operative manipulation or traction on the trigeminal nerve. Sixteen patients (8%) had a TCR intra-operatively (7 vestibular schwannomas, 5 sphenoid wing meningiomas, 3 petroclival meningiomas, 1 intracavernous epidermoid cyst). In all 16 patients with a TCR the postoperative courses presented no complications that could be directly related to this intra-operative phenomenon. Due to the intracranial course of the trigeminal nerve several surgical procedures at the anterior, middle and posterior skull base may elicit the trigeminocardiac reflex. Continuous monitoring of hemodynamic parameters allows the surgeon to interrupt surgical manoeuvres immediately upon the occurrence of the TCR. This technique is sufficient for the heart rate and the arterial blood pressure to return to normal levels without the necessity of additional anticholinergic medication.

The skull base is one of the most complex anatomical regions and forms the floor of the cranial cavity. Skull base surgery involves open, microscopic, and endoscopic approaches to the anterior, middle, or posterior cranial fossa. A multispecialty team approach is essential in treating patients with skull base lesions. Traditionally, rhinologists are involved in providing access to anterior skull base lesions while otologists are involved in the treatment of lesions of the posterior skull base. This is the case in most skull base centers today. In this article, we share a new perspective of an integrated skull base unit where a team of otolaryngologists and neurosurgeons treat anterior, middle, and posterior skull base pathologies. The rationale for this approach is that most technical skills required in skull base surgery are interchangeable and apply whether an endoscopic or microscopic approach is used. We show how the different skills apply to the different approaches and share our experience with an integrated skull base unit.

The authors report about a 3-years experience with helical CT and 3-D surface reconstruction applied in neurosurgical patients. All examinations were performed in addition to preexisting diagnostic CT, MRI, or angiography. The aim of this study was to assess the clinical value of this method with regard to planning of the surgical approach to anterior, middle, and posterior skull base and spinal lesions. 75 examinations of 55 patients were analysed and ranked as follows: A = examination with significant additional information for neurosurgical planning of skull base or spinal procedures or for postoperative evaluation of the neurosurgical approach, B = examination with some useful information for the neurosurgical planning or postoperative control, however, without significant advantage as compared to established diagnostic methods, C = examination without significant additional information. Classification was performed independently by two experienced surgeons. Examinations of anterior, middle, and posterior skull base lesions including cerebral aneurysms were in the majority rated as helpful and significantly informative, (A = 21, B = 24, C = 9, n = 54). Three-dimensional imaging of the spine was of clinical value only in specific cases (A = 6, B = 6, C = 9, n = 21). The authors conclude that three-dimensional imaging is a valuable diagnostic tool for pre- and postoperative imaging of tumorous and vascular lesions adjacent to the skull base, allowing for optimal surgical approaches with minimal invasiveness.

Actinomycosis is a rare chronic infectious disease caused by Actinomyces species. Orocervicofacial actinomycosis is the most common presentation (50%), followed by thoracic disease (15%–20%) and abdominopelvic disease (20%). It poses great diagnostic dilemma due to its variable presentations. We present a case of a 45-year-old female who presented with complaints of abdominal pain, abdominal fullness, constipation, weight loss and loss of appetite for the past 1 year. On examination, the patient was found to have palpable mass in lower abdominal region measuring about 10 cm × 12 cm, nontender, immobile mass. Patient was seen initially at another hospital, where ovarian malignancy was suspected in view of large size and radiological findings. Laparotomy was done but in view of adhesions and vascularity, multiple biopsies were taken and abdomen was closed. The patient was diagnosed to have actinomycosis based on histopathology and was treated accordingly. This case stresses on the diagnostic importance of clinical suspicion of actinomycosis in patients presenting with long-standing abdominal complaints, especially with history of previous surgeries.

Background:Skull base lesions are not uncommon, but their management has been challenging for surgeons. There is large no of bony tumors at the skull base which has not been studied in detail as a group. These tumors are difficult not only because of their location but also due to their variability in the involvement of important local structure. Through this retrospective analysis from a Tertiary Care Centre, we are summarizing the details of skull base bony lesions and its management nuances.Materials and Methods:The histopathologically, radiologically, and surgically proven cases of skull base bony tumors or lesions involving bone were analyzed from the neurosurgery, neuropathology record of our Tertiary Care Institute from January 2009 to January 2014. All available preoperative and postoperative details were noted from their case files. The extent of excision was ascertained from operation records and postoperative magnetic resonance imaging if available.Results:We have surgically managed 41 cases of skull base bony tumors. It includes 11 patients of anterior skull base, 13 middle skull base, and 17 posterior skull base bony tumors. The most common bony tumor was chordoma 15 (36.6%), followed by fibrous dysplasia 5 (12.2%), chondrosarcoma (12.2%), and ewings sarcoma-peripheral primitive neuroectodermal tumor (EWS-pPNET) five cases (12.2%) each. There were more malignant lesions (n = 29, 70.7%) at skull base than benign (n = 12, 29.3%) lesions. The surgical approach employed depended on location of tumor and pathology. Total mortality was 8 (20%) of whom 5 patients were of histological proven EWS-pPNET.Conclusions:Bony skull base lesion consists of wide variety of lesions, and requires multispecialty management. The complex lesions required tailored approaches surgery of these lesions. With the advent of microsurgical and endoscopic techniques, and use of navigation better outcomes are being seen, but these lesions require further study for development of proper management plan.