IgG4-Related Disease With Tuberculosis: A Case Report and Retrospective Review of Patients in a Single Center.

Background: IgG4-related disease (IgG4-RD) is a recently recognized systemic fibro-inflammatory disease of unknown cause involving many organs including pancreas, salivary glands, and lymph nodes. Chronic tuberculosis (TB) infection has been reported in IgG4-RD, but the prevalence of TB infection has not been evaluated in IgG4-RD. Methods: Characterization of a patient with IgG4-RD by physical examination, laboratory tests, magnetic resonance imaging (MRI) and histological examination. TB infection was evaluated by medical history, radiological examinations, sputum examinations, tubercular skin test (TST) and interferon gamma (IFN-γ) release assay test (IGRA). Medical records of IgG4-RD patients were reviewed in our institute from February 2015 to September 2020 to explore the prevalence of TB infection in IgG4-RD. Results: We described a 40-year-old Chinese man presented with headache and diplopia. Physical examination revealed bitemporal hemianopsia and limited abduction of both eyes. MRI revealed uniformly enhancing mass overlying clivus with dural tail sign. Laboratory data revealed elevation of IgG4 (1.9g/L), and TB-IGRA demonstrated significantly elevated IFN-γ (414.21 pg/ml). The clivus lesion was subtotally removed and IgG4 was strongly positive on immunohistochemical staining. The diagnosis of IgG4-RD was established, and the patient received treatment of corticosteroids, methotrexate, and cyclophosphamide with isoniazid prophylaxis. Consequently, the mass shrank remarkably within 3 months. A similar concurrence of TB disease or latent TB infection (LTBI) and IgG4-RD was present in 17/47 (36.2%) patients in our institute. Conclusion: High frequency of TB/LTBI presented in patients with IgG4-RD. Patients with IgG4-RD and LTBI should be closely monitored for resurgence of TB. Whether TB represents a risk for IgG4-RD should be further investigated in prospective cohort.

Delayed implant infection with Cutibacterium acnes (Propionibacterium acnes) 30 years after silicone sheet orbital floor implant.

Purpose: To report a case of delayed implant infection with Cutibacterium acnes (C. acnes, previously known as Propionibacterium acnes) 30 years after silicone sheet orbital floor implant.Methods: Case report with orbital imaging.Results: A 61-year-old male with a history of traumatic orbital floor fracture right eye (OD) repaired using a silicone sheet orbital floor implant 30 years prior, presented with 6 months of painless blepharoptosis and diplopia OD. On examination, there was 3 mm right upper eyelid blepharoptosis and hyperglobus. There was no globe proptosis, dysmotility, or compression and no cutaneous erythema, hyperthermia, discharge, or tenderness to palpation. Orbital magnetic resonance imaging (MRI) revealed a cystic mass in the inferior orbit in the region of the floor implant, measuring 25 mm in diameter and 10 mm in thickness. By MRI, T1-weighted images revealed a hypointense signal within the mass and T2-weighted images showed hyperintense signal with a flat hypointensity centrally representing the floor implant. Microbiologic cultures grew C. acnes.Conclusions: C. acnes can manifest several decades after placement of an orbital prosthetic implant, leading to delayed infection.

Intracranial actinomycosis of odontogenic origin masquerading as auto-immune orbital myositis: a fatal case and review of the literature.

BACKGROUND: Actinomycetes can rarely cause intracranial infection and may cause a variety of complications. We describe a fatal case of intracranial and intra-orbital actinomycosis of odontogenic origin with a unique presentation and route of dissemination. Also, we provide a review of the current literature. CASE PRESENTATION: A 58-year-old man presented with diplopia and progressive pain behind his left eye. Six weeks earlier he had undergone a dental extraction, followed by clindamycin treatment for a presumed maxillary infection. The diplopia responded to steroids but recurred after cessation. The diplopia was thought to result from myositis of the left medial rectus muscle, possibly related to a defect in the lamina papyracea. During exploration there was no abnormal tissue for biopsy. The medial wall was reconstructed and the myositis responded again to steroids. Within weeks a myositis on the right side occurred, with CT evidence of muscle swelling. Several months later he presented with right hemiparesis and dysarthria. Despite treatment the patient deteriorated, developed extensive intracranial hemorrhage, and died. Autopsy showed bacterial aggregates suggestive of actinomycotic meningoencephalitis with septic thromboembolism. Retrospectively, imaging studies showed abnormalities in the left infratemporal fossa and skull base and bilateral cavernous sinus. CONCLUSIONS: In conclusion, intracranial actinomycosis is difficult to diagnose, with potentially fatal outcome. An accurate diagnosis can often only be established by means of histology and biopsy should be performed whenever feasible. This is the first report of actinomycotic orbital involvement of odontogenic origin, presenting initially as bilateral orbital myositis rather than as orbital abscess. Infection from the upper left jaw extended to the left infratemporal fossa, skull base and meninges and subsequently to the cavernous sinus and the orbits.

Rare presentation of cryptococcal meningitis in an immunocompetent patient.

Cryptococcal meningitis is an opportunistic infection predominantly affecting immunocompromised patients but rarely can affect the immunocompetent. We describe a 53-year-old Caucasian man who presented complaining of a 2-week history of severe bilateral eye pain and diplopia. His only known risk factor was that he lived in a horse farm and recently shot bats and pigeons in his barn. He visited an outside hospital during this time without a diagnosis established. After further deliberation, we obtained a lumbar puncture (LP) which revealed an opening pressure (OP) of 27 cm H2O. Cerebrospinal fluid (CSF) and fungal cultures confirmed the presence of Cryptococcus neoformans The patient was diagnosed with C. neoformans-mediated meningoencephalitis and was initiated on the appropriate induction anti-fungal therapy. This case emphasises the need for clinicians to remain vigilant and consider cryptococcal meningitis in immunocompetent individuals even when classic symptoms of meningitis are absent.

Typhoid fever with isolated left lateral rectus palsy.

A 17-year-old young woman presented to Patan Hospital, Kathmandu, Nepal, with high-grade fever and headache for 4 days and non-projectile vomiting for 1 day. She also had blurred vision with dizziness on and off. There was no abnormal physical finding. Enteric fever was suspected, and she was empirically started on azithromycin (20 mg/kg) for 7 days. She became afebrile after 2 days and was followed up in 7 days with diplopia since 5 days. At this time, the blood culture was positive for Salmonella serovar typhi. On examination, there was isolated left lateral rectus palsy which accounted for her diplopia. Methylprednisolone (1 mg/kg) was prescribed which was tapered over 1 month and gradually her diplopia subsided. We hypothesise that vasculitic change in the blood vessel supplying the left abducens nerve could be causing the diplopia.

Tuberculosis versus pyogenic meningitis in a Pakistani population.

BACKGROUND: Research has been going on to formulate diagnostic criteria for TBM. Two criteria that have been studied and validated in high TB prevalence areas are the Youssef criteria (Rule 1) and Thwaites criteria (Rule 2). In our study we aimed to compare the different features of TBM and acute bacterial meningitis. METHODS: This retrospective study was done at Northwest General Hospital & Research Centre (NWGH&RC), Peshawar, Pakistan. Patients who were clinically diagnosed with TB meningitis or bacterial meningitis at the time of presentation were included in the study. RESULTS: Lab parameters for both groups were compared using independent sample T tests. We plotted ROC curves for Rule 1 and Rule 2. For Rule 1, at cut off value 2 it has a sensitivity of 97.5% and a specificity of 47.2%. For Rule 2, area at cut off value 3.5, sensitivity was 95% and specificity was 23.5%. We also plotted CSF protein to glucose ratio of our sample on an ROC curve and looked for measures of sensitivity and specificity. At cut off point 2 the sensitivity was 93% and specificity was 66.66%. CONCLUSION: It should be noted that although sensitivity for all three indices were high, specificity of all three tests was not very encouraging. We would like to emphasize that these indices can be useful in screening for patients with suspected TBM but they do not have the specificity to act as the sole test for initiation and continuance of therapy.

Severe invasive listeriosis--case report.

Listeriosis is a rare food borne infection which, in the invasive form, presents as bloodstream infection, central nervous system infection, materno-fetal infection, or focal infection. Certain immunosuppressive conditions have been identified as risk factors for severe invasive disease. The invasive forms of listeriosis are associated with a high case fatality rate. We present the case of a 62-year-old male with an unremarkable medical history admitted to the Iasi Infectious Diseases Hospital for fever. headache, ataxia, and diplopia. Physical examination revealed high temperature, confusion, relative bradycardia, and signs of meningeal irritation. Laboratory test showed leukocyt osis with neutrophilia. pathological CSF findings (high WBC count with predominance of neutrophils, low glucose and high protein levels), increased liver enzymes (ALAT, ASAT, AP, gammaGT), and important renal impairment (normal levels at presentation). No abnormalities at chest x-ray, cranial CT and abdominal ultrasound. CSF and blood cultures were positive for Listeria monocytogenes. Under antibiotics (ampicillin and ciprofloxacin), the course was marked by respiratory failure requiring mechanical ventilation, coma, hypotension, tachycardia. and death 12 days after admission. The particularity of this case consists in the association of the two classical forms of invasive listeriosis, meningitis and bacteriemia, with a focal infection. acute hepatitis, and a course marked by multiple organ dysfunction syndromes and exitus in a previously apparently healthy individual.

Isolated abducens nerve palsy in a patient with scrub typhus.

Abducens nerve palsies associated with infectious diseases are infrequent. Scrub typhus is an acute, febrile, infectious illness caused by Orientia tsutsugamushi (formerly Rickettsia tsutsugamushi), an obligate-intracellular gram-negative bacterium. The organism has been reported to be capable of entering the nervous system, causing meningitis and focal neurologic abnormalities. We report a case of isolated abducens nerve palsy associated with a scrub typhus infection.

Multiple cranial nerve involvement caused by Brucella melitensis.

A case of neurobrucellosis complicated by optic, abducens and vestibulocochlear nerve palsies is reported. Brucella melitensis was isolated in the cerebrospinal fluid, and the patient was diagnosed with retrobulbar neuritis. Despite medical treatment, the patient developed optic atrophy. Multiple cranial nerve involvement should be kept in mind in patients presenting with blurred vision and double vision, especially in regions where brucellosis is endemic.

Orbital abscess from an odontogenic infection.

An orbital abscess is a rare but serious complication of an odontogenic infection, which can lead to loss of vision or worse. This paper presents a case of orbital abscess secondary to an infection from the upper molar teeth, which extended to the retobulbar and posterosuperior region of the orbit, close to the superior orbital fissure. The infection spreaded to the pterygopalatine and infratemporal fossa and then to the orbit via the inferior orbital fissure. This paper reviews the clinical presentation, differential diagnosis, route of spread, value of serial CT scanning, treatment and possible complications.

Nasopharyngeal tuberculosis: a case report presenting with diplopia.

Tuberculosis (TB) is a common infectious disease worldwide. It can affect many organs, the most common is lungs infection. Upper respiratory tract involvement is uncommon and the least common regional involvement is nasopharyngeal region. The authors report a case of nasopharyngeal tuberculosis who came with diplopia which is a very rare presentation. Nasopharyngeal tuberculosis is a rare condition that is found in only reported cases in the literature. In Thailand, the present case is the second case of this condition with diplopia. Nasopharyngeal examination reveal mass or ulceration. The diagnostic tool is lesional biopsy. Histology is necessary to diagnose tuberculosis and to exclude other conditions especially nasopharyngeal cancer Nasopharyngeal tuberculosis has a good prognosis after treatment.

[Botulism in Casablanca. (11 cases)]. / Le botulisme à Casablanca. (A propos de 11 cas).

Botulism is a rare but severe disease. Whereas until 1980, only one case of botulism had been reported in our department, in 1999, a real botulism epidemic took place in Morocco. To our knowledge, it's the first outbreak of that kind in Morocco. We report here an epidemiologic and descriptive study of 11 patients suffering from botulism, admitted at the Infectious Diseases department and in the Medical Intensive Care Unit of Ibn Rochd University Hospital, from August, the 10th to October, the 1st, 1999. Clinical diagnosis of botulism was made, at the admission, on ocular signs (diplopia, ptosis), swallowing troubles and/or muscle weakness. There was no fever, no trouble of conscience and normal reflexes, at the early stage of the disease. The average age of patients was of 23.9 years +/- 12.07. Three patients were first admitted in the Medical Intensive Care Unit. The period before symptom appearance varied between 7 and 96 hours. Dysphagia sore throat, dry mouth and dysphonia were always found in all patients, with normal conscience. The fever was noted in 3 cases, polypnea in 3 cases leading to respiratory assistance in 2 cases. Neurologic findings were dominated by ptosis and hypotonia. The search of botulism toxin B in blood was positive in 6 cases. The electromyography showed clear signs of botulism. The evolution was favourable in 10 cases. Respiratory complications were found in 2 cases and infectious complications in 4 cases. One patient died. The period of hospitalization varied between 10 to 24 days with an average stay of 15.8 days. Eating "mortadella" has been noticed in 7 patients) and investigations permitted to identify the factory of "mortadella" as well as the toxin's type B responsible for these poisoning. It appears clearly that it is important to reinforce hygiene controls. Physicians and specialists in public health must be aware of the severity of this illness, knowing that the recovery is shortened when the treatment is administered on an early stage of the disease.

Bilateral abducens nerve paresis associated with anti-GQ1b IgG antibody.

PURPOSE: To describe two cases with bilateral abducens nerve paresis associated with serum anti-GQ1b IgG antibody. METHODS: Case reports. RESULTS: Two boys, aged 12 and 10 years, experienced acute onset of diplopia after preceding symptoms and signs of infection. In both boys, examination showed only bilateral abducens nerve paresis. Although routine laboratory data and magnetic resonance imaging demonstrated no pathologic findings, titer of anti-GQ1b IgG antibody in the sera of both patients was increased. Diplopia and signs of bilateral abducens nerve paresis disappeared in 6 weeks without any specific treatment. CONCLUSION: The anti-GQ1b IgG antibody in the sera of both patients probably contributed to the bilateral abducens nerve paresis.

Clinical evidence of extraocular muscle fiber-type specificity of botulinum toxin.

OBJECTIVE: To compare the effects of botulinum toxin on static and dynamic aspects of eye movements, and thereby elucidate the mechanisms of its action on eye muscles. BACKGROUND: Laboratory evidence indicates that static alignment and saccades are subserved by different extraocular muscle fiber types, and botulinum toxin may cause specific dysfunction of the fibers controlling static alignment. Diplopia is a well-known side effect of periorbital botulinum toxin injections in humans, and may be a clinical correlate of the laboratory findings. METHODS: Search coil recording of eye movements was performed in one patient with systemic botulism, and in three patients with diplopia following periorbital injection of botulinum toxin A. RESULTS: In the patient with acute botulism, eye movement alignment, range, and saccadic velocity profiles were abnormal. In three patients with iatrogenic diplopia, static alignment was abnormal but movement range and saccadic velocities were within normal limits. Edrophonium improved the range of movements and saccadic velocities in the patient with systemic botulism but was ineffective in reversing ocular misalignment in the one iatrogenic patient to whom it was administered. CONCLUSIONS: Precise alignment is subserved by orbital singly innervated muscle fibers, and the effects of botulinum toxin are greatest on these fibers. This predilection is apparent when the toxin dose is very small, as must have been the case in our patients with iatrogenic diplopia. The lack of a response to edrophonium probably reflects structural damage to muscle fibers. In contrast, larger doses of toxin produce an acute dysfunction of all extraocular muscle fiber types, which is responsive to edrophonium and consequently reflects partial blockade at the neuromuscular junction.

Skull base osteitis following fungal sinusitis.

Aspergillus sp. sinusitis is not uncommon in immunocompromised patients but is unusual in patients who are not immunocompromised. The disease may occur as a saprophytic condition, as an allergic sinusitis or as a potentially lethal invasive disease. The differentiation between non-invasive and invasive Aspergillus sp. sinusitis is crucial and this distinction is fully discussed. The treatment options are also considered. Invasive disease requires aggressive treatment with long-term antifungal agents in sufficient doses combined with wide surgical excision. We present a patient who presented with invasive Aspergillus fumigatus sinusitis and subsequently developed cranial neuropathies and skull base osteitis. She was initially treated with oral itraconazole (400 mg daily) for 18 months but due to lack of response this was changed to a new experimental oral azole (voriconazole) which was continued for a further 14 months. She has since remained well for the last five years.