Recurrent Respiratory Infections Research Articles

A rare pediatric neurometabolic disorder: Leigh syndrome

Subacute necrotizing encephalomyelopathy called Leigh Disease is a rare inherited pediatric neurological disorder generally involving infants. The condition primarily involves the central nervous system and the most involved structures are basal ganglia, thalamus, and brainstem. In this disorder, there is a progressive loss of mental and movement abilities associated with abnormal muscle tones, weakness, visual loss, respiratory failure, and strikingly, we got raised lactate levels in the blood, cerebral spinal fluid, and other body fluids. The prognosis of this condition is bad as there is no effective treatment and finally, the patient succumbs to death mostly due to respiratory failure. Here, we present a case of Leigh disease in an 8-month-old boy having chief complaints of delayed motor (weakness), and cognitive development with a previous history of recurrent respiratory infection. Upon recent radiological imaging, gross neurological abnormalities were seen which suggested the possibility of Leigh disease which was further confirmed by raised body fluid lactate levels.

Equine common variable immunodeficiency: Lessons from 100 clinical cases

SummaryThe clinical manifestation of recurrent fevers and infections alerts the clinician to the possibility of an underlying immunodeficiency. Common variable immunodeficiency (CVID) in the horse is a rare late‐onset, nonfamilial immunological disorder of B cell depletion and/or dysfunction with resultant inadequate antibody production. The most common clinical presentations in horses with CVID are recurrent upper and/or lower respiratory infections, meningitis and/or ataxia, cholangiohepatitis, infectious colitis, infectious dermatitis and severe gastrointestinal parasitism. Immune‐mediated and lymphoproliferative conditions are additional clinical features. The diagnosis of CVID in horses is based on a persistent hypogammaglobulinaemia primarily caused by a serum IgG concentration below 10.00 g/L in at least two different measurements, often accompanied by a serum IgM concentration below 0.50 g/L. Most horses with CVID show a persistent peripheral blood B cell distribution below 6% of total circulating lymphocytes, indicating severe B cell paucity or depletion, but the B cell distribution can be within the normal reference interval. Post‐mortem findings add diagnostic information about the distribution of B and T cells in lymphoid tissues. Clinical management of horses with immunodeficiency is intense and expensive, and these factors weigh on the difficult decision of elective euthanasia. To date, no genetic mutation has been identified in horse patients with CVID, and the large number of breeds of single‐affected individuals in a same herd or immediate lineage from various parts of the United States and the world do not point at an obvious inheritable mechanism of disease or environmental risk factors. This article describes the clinical and immunological findings in 100 equine cases, and comparisons with the disease in human patients.

Activated phosphoinositide 3-kinase δ syndrome caused by PIK3CD mutations: expanding the phenotype

BackgroundGermline heterozygous gain-of-function (GOF) mutations in the PIK3CD gene lead to a rare primary immunodeficiency disease known as activated phosphoinositide 3-kinase (PI3K) δ syndrome type 1(APDS1). Affected patients present a spectrum of clinical manifestations, particularly recurrent respiratory infections and lymphoproliferation, increased levels of serum immunoglobulin (Ig) M, Epstein-Barr virus (EBV) and cytomegalovirus (CMV) viremia. Due to highly heterogeneous phenotypes of APDS1, it is very likely that suspected cases may be misdiagnosed.MethodsHerein we reported three patients with different clinical presentations but harboring pathogenic variants in PIK3CD gene detected by trio whole-exome sequencing (trio-WES) and confirmed by subsequent Sanger sequencing.ResultsTwo heterozygous mutations (c.3061G > A, p.E1021K and c.1574 A > G, p.E525G) in PIK3CD (NM_005026.3) were identified by whole exome sequencing (WES) in the three patients. One of two patients with the mutation (c.3061G > A) presented with abdominal pain and diarrhea as the first symptoms, which was due to intussusception caused by multiple polyps of colon. The patient with mutation (c.1574 A > G) had an anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV)-like clinical manifestations, including multisystemic inflammation, acute nephritic syndrome, and positive perinuclear ANCA (p-ANCA), thus the diagnosis of ANCA-AAV was considered.ConclusionsOur study expands the spectrums of clinical phenotype and genotype of APDS, and demonstrates that WES has a high molecular diagnostic yield for patients with immunodeficiency related symptoms, such as respiratory infections, multiple ecchymosis, ANCA-associated vasculitis, multiple ileocecal polyps, hepatosplenomegaly, and lymphoid hyperplasia.Trial registrationRetrospectively registered.

'Don't let it hold you back' - The experience of transition to adulthood in young people with primary ciliary dyskinesia: An interpretative phenomenological analysis.

Primary ciliary dyskinesia (PCD) is a rare, chronic genetic condition with variable features arising from motile cilia dysfunction, including recurrent respiratory infections, sinonasal disease, reduced hearing, infertility and situs inversus. The aim of the study was to understand the experiences of young people with PCD as they transition into adulthood and adult healthcare services. An interpretative phenomenological analytical method was applied. Semi-structured interviews were conducted with three participants aged 18-24 years. Four interconnected group experiential themes were identified: (1) reconceptualising a stigmatised identity, (2) sharing the journey to independence, (3) entering adulthood with newfound autonomy, (4) anticipating an uncertain future. Overall, we found that transition for young people with PCD presents as a complex period marked by identity-formation, creating systems of support and becoming an autonomous adult. Facilitation of personalised and integrated approaches to care should be prioritised. Our findings are important to help health professionals provide appropriate, anticipatory support.

Identification of a novel RPGR mutation associated with retinitis pigmentosa and primary ciliary dyskinesia in a Slovak family: a case report

BackgroundThe mutations in the RPGR (retinitis pigmentosa GTPase regulator) gene are the most common cause of X-linked retinitis pigmentosa (XLRP), a rare genetic disorder affecting the photoreceptor cells in the retina. Several reported cases identified this gene as a genetic link between retinitis pigmentosa (RP) and primary ciliary dyskinesia (PCD), characterised by impaired ciliary function predominantly in the respiratory tract. Since different mutations in the same gene can result in various clinical manifestations, it is important to describe a correlation between the gene variant and the observed phenotype.MethodsTwo young brothers from a non-consanguineous Slovak family with diagnosed retinal dystrophy and recurrent respiratory infections were examined. Suspected PCD was diagnosed based on a PICADAR questionnaire, nasal nitric oxide analysis, transmission electron microscopy, high-speed video microscopy analysis, and genetic testing.ResultsWe identified a novel frameshift RPGR mutation NM_001034853: c.309_310insA, p.Glu104Argfs*12, resulting in a complex X-linked phenotype combining PCD and RP. In our patients, this mutation was associated with normal ultrastructure of respiratory cilia, reduced ciliary epithelium, more aciliary respiratory epithelium, shorter cilia, and uncoordinated beating with a frequency at a lower limit of normal beating, explaining the clinical manifestation of PCD in our patients.ConclusionThe identified novel pathogenic mutation in the RPGR gene expands the spectrum of genetic variants associated with the X-linked PCD phenotype overlapping with RP, highlighting the diversity of mutations contributing to the disorder. The described genotype–phenotype correlation can be useful in clinical practice to recognise a broader spectrum of PCD phenotypes as well as for future research focused on the genetic basis of PCD, gene interactions, the pathways implicated in PCD pathogenesis, and the role of RPGR protein for the proper functioning of cilia in various tissues throughout the body.

P895 Analysis of the efficacy of intravenous ustekinumab maintenance treatment in patients with refractory Inflammatory Bowel Disease

Abstract Background Ustekinumab (UST) is a monoclonal antibody that inhibits the p40 subunit of IL-12 and IL-23, approved for the treatment of Crohn's Disease (CD) and Ulcerative Colitis (UC). Its administration is intravenous during induction and subcutaneous during maintenance. Several studies have assessed the efficacy of reinduction and intensification of this treatment in patients with loss of response. However, there are few studies evaluating the effectiveness of intravenous UST for maintenance. The aim of the study is to evaluate the efficacy and safety of maintenance with intravenous UST in patients with Inflammatory Bowel Disease (IBD). Methods Observational, retrospective, and single-center study. All patients receiving maintenance with intravenous UST until June 2023 were reviewed. Clinical response was evaluated with Harvey-Bradshow Index for CD and partial Mayo for UC. Biochemical response was evaluated with calprotectin and C-reactive protein. Results A total of 29 patients with IBD (24 CD and 5 UC) were included. The mean follow-up of patients was 449 days (74-948). The cohort's characteristics are summarized in Table 1. In all cases, intravenous maintenance was performed with UST 130mg/4 weeks. In 4 cases (13.8%), maintenance was initiated directly with intravenous UST. In 9 (31%) patients due to primary failure to UST and in 16 (55.2%) patients due to secondary failure. The clinical response to intravenous UST maintenance in patients with CD at week 8 and 16 and at one year of follow-up was 87.5% (n=21/24), 83.3% (n=20/24), 73.3% (n=11/15) respectively. For UC, the response was 60% (n=3/5) at week 8 and 16 and at one year. The drug survival was 723 days (± 74) (Figure 1). No predictive variable for treatment response was identified. Two patients (both with CD) experienced recurrent respiratory infections as an adverse effect, without the need to discontinue UST. Conclusion Maintenance treatment with intravenous UST can be useful and safe in patients with severe IBD and/or failure of subcutaneous UST maintenance.

Surgical treatment for severe pediatric tracheobronchomalacia: the 20-year experience of a single center

ObjectiveIn children with tracheobronchomalacia, surgical management should be reserved for the most severe cases and be specific to the type and location of tracheobronchomalacia. The goal of this study is to describe the presentation and outcomes of children with severe tracheobronchomalacia undergoing surgery. MethodsRetrospective case series of 20 children operated for severe tracheobronchomalacia at a tertiary hospital from 2003 to 2023. Data were collected on symptoms age at diagnosis, associated comorbidities, previous surgery, age at surgery, operative approach, time of follow-up, and outcome. Surgical success was defined as symptom improvement. ResultsThe most frequent symptoms of severe tracheobronchomalacia were stridor (50 %), cyanosis (50 %), and recurrent respiratory infections (45 %). All patients had one or more underlying conditions, most commonly esophageal atresia (40 %) and prematurity (35 %). Bronchoscopy were performed in all patients. Based on etiology, patients underwent the following procedures: anterior aortopexy (n = 15/75 %), posterior tracheopexy (n = 4/20 %), and/or posterior descending aortopexy (n = 4/20 %). Three patients underwent anterior aortopexy and posterior tracheopexy procedures. After a median follow-up of 12 months, 16 patients (80 %) had improvement in respiratory symptoms. Decannulation was achieved in three (37.5 %) out of eight patients with previous tracheotomy. The presence of dying spells at diagnosis was associated with surgical failure. ConclusionsIsolated or combined surgical procedures improved respiratory symptoms in 80 % of children with severe tracheobronchomalacia. The choice of procedure should be individualized and guided by etiology: anterior aortopexy for anterior compression, posterior tracheopexy for membranous intrusion, and posterior descending aortopexy for left bronchus obstruction.

Effectiveness of inosine pranobex in the treatment and prevention of infectious diseases (system review)

The purpose of the article is a systematic review of studies conducted in recent decades in children and adults, with an emphasis on domestic works, on the use of inosine pranobex preparations, in the treatment of infectious diseases. The clinical and immunological effectiveness of inosine pranobex in the treatment and prevention of relapses in patients with recurrent respiratory viral infections against the background of persistent herpesviruses types 4-6 has been demonstrated. Using the example of multicenter comparative as well as placebo-controlled studies, not only the effectiveness, but also the safety of long-term courses of therapy is shown. A promising direction is the use of inosine pranobex in the treatment of COVID-19 and post-Covid conditions, taking into account its immunomodulatory effect without the risk of increasing the “cytokine storm”, as well as studies of the direct antiviral effect on the SARS-CoV-2 virus in vitro.

Dietary habits, physical activity, and self-reported rhinosinusitis in children and adolescents.

Pediatric paranasal rhinosinusitis is one of the more common pediatric diseases of the upper respiratory tract and it entails significant morbidity. Most commonly, it is caused by a viral infection of the nasal mucosa, which spreads through the natural passages within the cavities of the paranasal sinuses, leading to inflammation of the mucosa that lines the nasal cavity and paranasal sinuses. The objective of this cohort study was to assess whether there is a correlation between pediatric rhinosinusitis, physical activity, and selected dietary habits among pupils aged 6 to 16 years from elementary schools in Wrocław, Poland. This study - as part of the pro-health program "Let us Get the Kids Moving" - is also aimed at establishing factors that potentially predispose children to developing RS. The survey study was conducted on a group of 2,458 children and adolescents from elementary schools in Wrocław. The age of the examined children ranged from 6 to 17 years (mean = 10.8 years; standard deviation = 2.7). Rhinosinusitis was more common in the children aged 13-17 years than in those aged 6-9 years (6.4% vs. 1.5%; p < 0.001) or 10-12 years (6.4 vs. 2.6%; p < 0.001). The study revealed a significant positive correlation between rhinosinusitis development and several variables: age > 11 years, attending swimming classes fewer than 1-2 times a week, using a computer, consuming milk, salty snacks, and carbonated sweet drinks, consuming fruit fewer than 1-2 times a week, not attending physical education classes, eating fewer than 4 meals, and not eating breakfast at home (p < 0.05). It is of great importance to establish preventive measures against recurrent upper respiratory tract infections that may predispose children to rhinosinusitis. Introducing healthier, traditional dietary habits and regular physical activity in children and adolescents may result in normal and adequate immune response and proper functioning of the inflammatory control system.

Recurrent respiratory tract infections in children might be associated with vitamin A status: a case-control study.

Recurrent respiratory tract infections (RRTIs) are common in children and its development might be associated with vitamin A deficiency according to recent research. The aim of this study was to understand the relation between vitamin A status and RRTIs in children, and the relation between dietary intake of vitamin A and RRTIs. 2,592 children aged 0.5-14 years from Heilongjiang province of China participated in the survey. The RRTI group consisted of 1,039 children with RRTIs, while 1,553 healthy children were included in the control group. The levels of serum vitamin A were determined by high performance liquid chromatography (HPLC); dietary information was collected with the Food Frequency Questionnaire (FFQ). Serum vitamin A concentration in the RRTI group was significantly lower than that in the control group (0.27 ± 0.09 mg/L vs. 0.29 ± 0.09 mg/L) (P < 0.01). The levels of vitamin A was obviously associated with the occurrence of RRTIs. The odds ratios (ORs) for vitamin A insufficiency and deficiency were 1.32 (95% CI: 1.09-1.60) and 1.95 (95% CI: 1.50-2.55) respectively; whereas 1.48 (95% CI: 1.13-1.94) and 6.51 (95% CI: 4.18-10.14) respectively, in children with current respiratory tract infection (RTI) symptoms. Even an insufficient intake of animal liver was associated with lower RRTIs [OR: 0.45 (95% CI: 0.38-0.53)], while only an excessive intake of meat had the same effect [OR: 0.85 (95% CI: 0.68-1.06)]. Low serum vitamin A concentration was associated with high incidence of RRTIs in children in northeast China; low serum vitamin A concentrations and the current RTI symptoms were associated with the development of RRTIs; and low intakes of vitamin A-rich foods were also associated with the development of RRTIs.

P27 Successful treatment of severe eczema and plane warts with Dupilumab in a child with Dedicator of Cytokinesis 8 (DOCK8) deficiency (Autosomal recessive form of hyper-IgE syndrome)

Abstract A six-year-old girl first presented with severe eczema at the age of 6 months, which was difficult to control with topical treatments. She was found to have severely elevated total IgE and multiple food allergies. Due to recurrent ear and chest infections, and persistent varicella zoster, she was screened for a primary immunodeficiency and found to have a homozygous exon 29-46 deletion in the DOCK8 gene. Her eczema remained challenging, with coalescing warts on her limbs. She started Methotrexate with an initial good response; however, the frequency of infections and hospitalizations increased. After the diagnosis of DOCK8 deficiency was confirmed, she started Dupilumab, resulting in the complete resolution of her eczema and warts. She is awaiting haematopoietic stem cell transplant (HSCT) . DOCK8 deficiency is an autosomal recessive hyper-IgE syndrome that presents early in life with severe eczema, skin abscesses, multiple food allergies, recurrent respiratory tract infections, and severe viral (including skin) infections. Older patients invariably develop bronchiectasis and are at high risk of malignancy. Patients have a combined immunodeficiency, with T-cell lymphopenia, reduced thymic output and T-cell skewing to a Th2 response. Children with this condition often present to dermatologists because of severe eczema, which is usually difficult to treat with standard topical treatments. The decision to treat with systemic immunosuppression is often challenging due to recurrent infections. Although HSCT is the curative treatment for DOCK8 deficiency, Dupilumab can be used to reduce inflammation and infections, and improve quality of life in patients awaiting HSCT.

“A Complex Conundrum”- Fanconi-Bickel Syndrome

Fanconi-Bickel Syndrome (FBS) is a rare form of Glycogen Storage Disease (GSD) type 11 characterised by massive hepatomegaly due to the buildup of glycogen and severe hypophosphatemic rickets due to a proximal renal tubular dysfunction. Since 1940, it has been initially known as hepatorenal glycogenesis with proximal renal tubular dysfunction. It is due to a pathogenic mutation of the GLUT-2 (glucose transporter) gene. Herein, present case report a young toddler who is the firstborn of 3rd-degree consanguinity, presented with rickets, recurrent respiratory tract infections, and hepatomegaly, and was subsequently diagnosed with FBS with the help of genetic studies, showing a mutation in the GLUT-2 gene. With less than 200 cases reported so far, this child represents a unique case with recurrent respiratory infections and developmental delay as presentations along with rachitic features.

A comprehensive analysis of anthropometric indicators in preschool children suffering from recurrent respiratory infections.

Aim: To determine the state of relationship between anthropometric indicators and susceptibility to recurrent respiratory infections in preschool children. Materials and Methods: A total of 143 children (73 boys and 70 girls) aged 12-59 months, undergoing inpatient treatment on acute respiratory infection, were involved in the clinical study. The number of acute respiratory infection episodes during a previous year of their lives was taken into account. Besides, the basic indicators of physical development were assessed in the children, including: 1) body weight; 2) body length; 3) chest circumference; 4) body mass index; 5) body surface area; 6) Vervek's index. Results: Cross-tabulation and rank correlation analysis did not demonstrate any interdependence between the susceptibility of the children examined to recurrent respiratory infections and their anthropometric indicators. Simultaneously, linear regression analysis showed that in the children aged 12-23 months, resistance index depended on their age and body length. The relative importance of the combined effect of the two above-mentioned indicators among all other potential risk factors for recurrent respiratory infections was 32.2%. Conclusions: The detailed analysis of the findings outlined the methodological basis for further studies of the association between the incidence of acute respiratory infections in preschool children and their physical development. Multivariate statistical calculations of various risk factors for recurrent respiratory infections, including abnormal anthropometric indicators, are likely to increase the informational value of subsequent examinations.

Serum Levels of Selected Cytokines and Chemokines and IgG4 in Children With Recurrent Respiratory Tract Infections.

Background: Respiratory tract infections are a common health problem. Cytokines/chemokines play a critical role in the regulation of the immune system. Their defective production may predispose to recurrent respiratory tract infections (RRIs), and an excessive immune response may lead to chronic inflammation and cause damage to the respiratory tract. Another biomarker of respiratory infections may be immunoglobulin-IgG4. Its meaning has still been little explored. We wanted to assess the suitability of the levels of biomarkers tested: interleukin (IL)-17A, IL-18, IL-23, normal T cells expressed and secreted (RANTES), and induced protein (IP)-10, as well as immunoglobilun G4 (IgG4) to predict recurrent infections. Methods: The study group (SG) included a total of 130 children (68 girls, 62 boys) between 3 and 17 years of age with RRI. The control group (CG) included 86 healthy children with no symptoms of inflammatory or allergic diseases (44 girls and 42 boys) of the same age. Blood samples were collected in fasting state and then serum samples were frozen and stored until biomarker assay. Results: Serum RANTES, IL-18, IL-23, and IgG4 concentration were higher in all children with recurrent infections vs. those in the CG (p < 0001). Serum levels of IL-17A and IP-10 were also significantly higher in the SG than in the CG, but only in the youngest children. Among the six serum markers, RANTES demonstrated the highest area under the receiver operating characteristic curve (area under curve) value (0.998, 95% confidence interval [CI]: 0.98-1.0, p < 0.001) for the diagnosis of RRIs, followed by IL-23 (0.99, 95% CI 0.966-0,999, p < 0.001) and IL-18 (0.957, 95% CI 0.921-0.980, p < 0.001). Conclusions: RANTES, IL-23, and IL-18 could be strong predictors of respiratory infections recurrence in children.

Pathogenic TNFRSF13B Variant in an Adult Japanese Patient with Common Variable Immunodeficiency.

Common variable immunodeficiency (CVID) is a primary B cell immunodeficiency disorder. Symptoms do not develop immediately after birth, and patients are often diagnosed in childhood and adulthood. These patients often develop autoimmune diseases and malignant tumors. We herein report a 50-year-old woman with severe hypogammaglobulinemia and recurrent respiratory tract infections who was diagnosed with CVID. Target sequencing showed a TNFRSF13B heterozygous frameshift variant. The patient had many comorbidities, probably caused by a CVID-induced immune imbalance. Physicians who treat adult patients are often unaware of CVID. CVID should be recognized as a differential diagnosis in hypogammaglobulinemia and recurrent infections.

Prevalence of Congenital Heart Disease in Children with Recurrent Respiratory Tract Infection

Prevalence of Congenital Heart Disease in Children with Recurrent Respiratory Tract Infection

Cystic Fibrosis – A Case Report Illustrating the Importance of Access to Cystic Fibrosis Care and Therapy

Abstract A 9-year-old Hispanic female, initially diagnosed with asthma, presented with recurrent respiratory infections, poor growth, a persistent cough, and chest congestion. Targeted gene sequencing testing confirmed cystic fibrosis (CF) with two F-508del mutations. Following referral to a specialized CF center, the patient received comprehensive clinical care, including patient and parent education, nutritional support, CF-specific therapies, and concurrent management of a pulmonary exacerbation. The introduction of CF modulator therapy resulted in substantial improvements in body mass index and forced expiratory volume in 1 s. This case highlights the importance of early diagnosis and access to CF care in improving clinical outcomes, especially in underserved populations.

Risk Factors for Lower Respiratory Tract Infection and Recurrent Lower Respiratory Tract Infection in Children

Risk Factors for Lower Respiratory Tract Infection and Recurrent Lower Respiratory Tract Infection in Children

Complications of untreated hypogammaglobulinemia in a patient with common variable immunodeficiency – A case report

Common variable immunodeficiency (CVID) is a primary immunodeficiency disorder characterized by decreased or undetectable serum levels of immunoglobulin G (IgG), decreased levels of IgA isotypes, and poor specific antibody production in response to vaccines and pathogens. Owing to hypogammaglobulinemia, patients have increased susceptibility to infections and a higher risk of malignancies. Here we present a case study of patient with a diagnosis of CVID, who refused immunoglobulin replacement therapy despite plenty of complications. Patient suffered from recurrent respiratory and digestive tract infections, diarrhea, malnutrition, infection of the central nervous system, and autoimmune complications. The complications of untreated hypogammaglobulinemia eventually led to the death of the patient at the age of 63 years. Patients with CVID have an increased susceptibility to chronic infections and subsequent complications; hence, proper treatment with immunoglobulin replacement therapy should be administered. Owing to the increased risk of autoimmunity and malignancy, regular surveillance should be conducted in patients with CVID. Understanding the natural history of untreated hypogammaglobulinemia emphasizes the need for early diagnosis, appropriate treatment, and vigilant follow-up in patients with CVID.

Intermittent esotropia and ataxia in a child with late diagnosis of empyema in the sphenoid sinus - a case report

Background: Acute bacterial rhinosinusitis is a common condition often affecting children with inflammation of the mucous membrane in one or more nasal sinuses. Isolated sphenoid sinus infections can exhibit atypical or no symptoms before potentially severe suppurative complications develop. Awareness of early signs of complications is crucial to avoid delays in referral, diagnosis, and treatment. We present the first detailed report of a child with sphenoid sinus empyema with the symptoms intermittent unilateral esotropia and limb and gait ataxia. This case emphasizes the need to recognise atypical presentations, aiding early diagnosis and treatment to prevent severe, possibly life-threatening complications. Case presentation: A 7-year-old girl, previously healthy, presented with a months-long decline in health marked by general malaise, significant weight loss, and recurrent upper respiratory infections. Despite multiple primary care visits no antibiotics were prescribed because infection parameters were found to be normal. Subsequently, the emergence of intermittent double vision prompted referral to the pediatric emergency department. Examination revealed a wide-based gait, ataxic finger-nose-finger and knee-heel tests, and intermittent esotropia of the left eye. Neurological examination was otherwise unremarkable. Imaging of the cerebrum was performed on the suspicion of intracranial pathology and found isolated empyema within the left sphenoid sinus. Endoscopic sinus surgery and antibiotic treatment led to a full recovery within a month. The accommodative esotropia, likely triggered by the infection, was identified, and successfully treated with spectacles. Conclusion: This case recognises isolated empyema in the sphenoid sinus as a potential cause of intermittent esotropia and limb and gait ataxia. It serves as a reminder for clinicians to be vigilant for early signs of suppurative complications to commence prompt diagnosis and treatment to prevent delayed recognition and severe complications. It advocates the inclusion of images of the sphenoid sinus to accompany the conventional scans of the cerebrum, as patients risk referral on the suspicion of intracranial pathology on the basis of neurological symptoms.