BRIEF OBSERVATION
Exercise-induced Chest Pain: An Atypical Manifestation of Eosinophilic Esophagitis Jasmin Kahn,a Christian Bussmann, MD,b Christoph Beglinger, MD,a Alex Straumann, MD,a Petr Hruz, MD, PhDa a Department of Gastroenterology and Hepatology, University Hospital Basel, Basel, Switzerland; bInstitute Pathology Viollier, Basel, Switzerland.
ABSTRACT BACKGROUND: Eosinophilic esophagitis is a chronic-inflammatory immune-mediated disease of the esophagus. The most common symptoms of eosinophilic esophagitis are dysphagia for solids and bolus obstruction. Approximately half of the patients complain about spontaneously occurring chest pain. However, some patients with eosinophilic esophagitis suffer from, in addition to the described symptoms, exercise-induced chest pain. METHODS AND RESULTS: Here we report on several patients presenting with exercise-induced chest pain, with 2 patients presenting exclusively only with this symptom. After a comprehensive evaluation including exclusion of gastroesophageal reflux disease and heart disease, a diagnosis of eosinophilic esophagitis based on endoscopical and histological criteria was unequivocally established. The condition improved rapidly after initiation of a treatment with topical corticosteroids. CONCLUSION: Eosinophilic esophagitis should be considered in the differential diagnosis in patients presenting with exercise-induced chest pain, particularly in younger male individuals. Ó 2015 Elsevier Inc. All rights reserved. The American Journal of Medicine (2015) 128, 196-199 KEYWORDS: Eosinophilic esophagitis; Exercise-induced chest pain; Symptoms SEE RELATED EDITORIAL p. 103
Eosinophilic esophagitis is a chronic immune-mediated inflammatory disorder of the esophagus.1 The pathophysiological mechanisms are not understood completely, although it seems that the eosinophilic inflammation in patients with eosinophilic esophagitis is almost always triggered by dietary proteins and less likely by aeroallergens.2,3 Clinicopathologically, eosinophilic esophagitis is defined by esophagus-related symptoms, in combination with a dense esophageal eosinophilia, which are unresponsive to acid
Funding: None. Conflicts of Interest: None. Authorship: PH and AS conceptualized and designed the clinical question, drafted the initial manuscript, and approved the final manuscript as submitted. JK evaluated the patient data, drafted the initial manuscript, revised the manuscript, and approved the final manuscript as submitted. CB provided the histological evaluation, critically reviewed, revised the manuscript and approved the final manuscript as submitted. Requests for reprints should be addressed to Petr Hruz, MD, PhD, Department of Gastroenterology and Hepatology, University Hospital Basel, Petersgraben 4, Basel 4031, Switzerland. E-mail address: [email protected] 0002-9343/$ -see front matter Ó 2015 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.amjmed.2014.08.007
suppression with proton pump inhibitors (PPI).1,4 The most prominent symptoms in adolescents, and adults are dysphagia for solids, bolus impaction, and spontaneously occurring chest pain resembling reflux disease.1 In our Swiss eosinophilic esophagitis cohort, 825 (M ¼ 631, F ¼ 194) adolescent and adult patients are currently enrolled. A systematic analysis of symptoms in these patients revealed that dysphagia, long-lasting food impaction, and spontaneously occurring chest pain was present in 94.8% (n ¼ 782), 34.2% (n ¼ 282), and 38.4% (n ¼ 317) of patients. Interestingly, we found a subgroup of patients (n ¼ 10; 1.21%) having exercise-induced chest pain in absence of any signs of cardiovascular or pulmonary disease. We report here on a series of patients having a diagnosis of eosinophilic esophagitis according to the established criteria complaining about exercise-induced chest pain. The demographic parameters and clinical findings of all patients are summarized in detail in the Table. Two of these patients complained exclusively of exercise-induced chest pain and are presented as a case report in the following section.
Kahn et al
Exercise-induced Chest Pain and Eosinophilic Esophagitis
CASE REPORT
197
Case 2
A 16-year-old boy presented to his family physician with exercise-induced sensation of constriction in the throat and A 22-year-old professional hockey player was admitted to a hyper salivation, starting some weeks before his visit. The hospital with chest pain after a strong strike/hit on the breastpatient was evaluated by an ENT specialist and diagnosed bone during an ice hockey game. Clinical examination and with pharyngitis and laryngitis, which was treated with a thoracic computed tomography scan PPI (esomeprazole 40 mg once a were unremarkable, and the patient day) and a single dose of an antiCLINICAL SIGNIFICANCE was discharged with suspicion of a mycotic (diflucan 400 mg). thoracic concussion. Almost 1 week Eosinophilic esophagitis is a chronic inSubsequently, the patient was later his physical performance was referred to our institution, where flammatory immune-mediated disease of still substantially reduced due to an he described exclusively exercisethe esophagus. unpleasant feeling of choking, induced throat tightness and Eosinophilic esophagitis presents with reflux-like symptoms, and dyspnea increased salivation, but no occurring regularly, a few minutes growing incidence and prevalence in dysphagia. His personal history after beginning any physical effort. industrialized countries. revealed an allergic asthma, a polA series of comprehensive cardiolen allergy, and an oral allergy Affects predominantly middle-aged men, logical, pneumological, and ear, syndrome. Laboratory testing with a male-to-female ratio of 3:1. nose, and throat (ENT) examinarevealed an elevated serum immutions did not reveal any relevant Presents with dysphagia and bolus noglobulin E level (330 U/mL; pathological findings. Treatment norm 60 eosinophils/highand thoracic oppression, limiting his physical performance power field and focally eosinophilic microabscesses. Sympdrastically. The patient was referred for evaluation to our toms resolved within a few days after initiation of oral topical institution, where he presented with exclusively exercisecorticosteroids (Axotide 250 Diskus). Fluticasone 1000 mg induced, oppression-like chest sensations, but no dysphagia. bid was given for 10 days followed by a maintenance treatPersonal history revealed an allergic asthma, urticaria, and an ment with fluticasone 250 mg bid. Symptoms reappeared oral allergy syndrome on consumption of chicken, meat, or 2 weeks after the patient stopped the maintenance treatment. bananas. The family history, physical examination, and laboAnother endoscopic evaluation demonstrated an active ratory analyses were unremarkable except for a slightly eosinophilic esophagitis with edema, longitudinal furrows, increased total immunoglobulin E value (255 U/mL; norm: and moderate white exudates in the distal esophagus, with an 150 eosinophils/high-power esophagitis with longitudinal furrows in the proximal part of the field. No signs of a gastroesophageal reflux disease were esophagus and with histological esophageal eosinophilia. observed. The treatment with oral topical corticosteroids Functional esophageal examinations, including high-resolution (Axotide 250 Diskus) was reinitiated with complete resolumanometry and a 24-hour impedance-pH monitoring, were tion of symptoms. unremarkable. A treatment with swallowed topical corticosteroids (fluticasone 1000 mg bid; Axotide 250 Diskus; GlaxoSmithKDISCUSSION line, Brentford, Middlesex, UK) was initiated. All symptoms Eosinophilic esophagitis was first reported not more than 20 resolved within a few days and under a maintenance treatment years ago; to this day, not all clinical facets of this chronicwith topical corticosteroids (Axotide 250 Diskus, fluticasone inflammatory disease are elucidated. Meanwhile, it has 250 mg bid), the patient was again able to play ice hockey. After become clear that dysphagia for solids, food impaction, and discontinuation, symptoms reappeared within a few weeks. spontaneously occurring chest pain are the most prominent Another upper endoscopy demonstrated endoscopical signs of symptoms in adolescent and adult eosinophilic esophagitis eosinophilic esophagitis with furrowing, white exudates, and patients.1 edema (Figure A). Histology confirmed a severe eosinophilic In our cohort we identified 10 patients with confirmed infiltration in the proximal and distal esophagus, with >100 diagnosis of eosinophilic esophagitis complaining about eosinophils/high-power field (Figure B). Treatment was exercise-induced chest pain. The differential diagnosis for reinitiated and symptoms disappeared rapidly. this symptom includes musculoskeletal, gastrointestinal,
Case 1
198
The American Journal of Medicine, Vol 128, No 2, February 2015
Table
Demographics and Clinical Findings of Patients with Exercise-induced Chest Pain (n ¼ 10)
Demographics Age in years at diagnosis; median (range) Duration of symptoms (months); median (range) Affected relatives Sex; male:female BMI kg/m2, median (range) History of allergies; AA ¼ allergic asthma, ARC ¼ allergic rhinoconjunctivitis, OAS ¼ oral allergy syndrome Laboratory findings Peripheral blood eosinophils eos /mm3; norm:
Exercise-induced Chest Pain: An Atypical Manifestation of Eosinophilic Esophagitis Jasmin Kahn,a Christian Bussmann, MD,b Christoph Beglinger, MD,a Alex Straumann, MD,a Petr Hruz, MD, PhDa a Department of Gastroenterology and Hepatology, University Hospital Basel, Basel, Switzerland; bInstitute Pathology Viollier, Basel, Switzerland.
ABSTRACT BACKGROUND: Eosinophilic esophagitis is a chronic-inflammatory immune-mediated disease of the esophagus. The most common symptoms of eosinophilic esophagitis are dysphagia for solids and bolus obstruction. Approximately half of the patients complain about spontaneously occurring chest pain. However, some patients with eosinophilic esophagitis suffer from, in addition to the described symptoms, exercise-induced chest pain. METHODS AND RESULTS: Here we report on several patients presenting with exercise-induced chest pain, with 2 patients presenting exclusively only with this symptom. After a comprehensive evaluation including exclusion of gastroesophageal reflux disease and heart disease, a diagnosis of eosinophilic esophagitis based on endoscopical and histological criteria was unequivocally established. The condition improved rapidly after initiation of a treatment with topical corticosteroids. CONCLUSION: Eosinophilic esophagitis should be considered in the differential diagnosis in patients presenting with exercise-induced chest pain, particularly in younger male individuals. Ó 2015 Elsevier Inc. All rights reserved. The American Journal of Medicine (2015) 128, 196-199 KEYWORDS: Eosinophilic esophagitis; Exercise-induced chest pain; Symptoms SEE RELATED EDITORIAL p. 103
Eosinophilic esophagitis is a chronic immune-mediated inflammatory disorder of the esophagus.1 The pathophysiological mechanisms are not understood completely, although it seems that the eosinophilic inflammation in patients with eosinophilic esophagitis is almost always triggered by dietary proteins and less likely by aeroallergens.2,3 Clinicopathologically, eosinophilic esophagitis is defined by esophagus-related symptoms, in combination with a dense esophageal eosinophilia, which are unresponsive to acid
Funding: None. Conflicts of Interest: None. Authorship: PH and AS conceptualized and designed the clinical question, drafted the initial manuscript, and approved the final manuscript as submitted. JK evaluated the patient data, drafted the initial manuscript, revised the manuscript, and approved the final manuscript as submitted. CB provided the histological evaluation, critically reviewed, revised the manuscript and approved the final manuscript as submitted. Requests for reprints should be addressed to Petr Hruz, MD, PhD, Department of Gastroenterology and Hepatology, University Hospital Basel, Petersgraben 4, Basel 4031, Switzerland. E-mail address: [email protected] 0002-9343/$ -see front matter Ó 2015 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.amjmed.2014.08.007
suppression with proton pump inhibitors (PPI).1,4 The most prominent symptoms in adolescents, and adults are dysphagia for solids, bolus impaction, and spontaneously occurring chest pain resembling reflux disease.1 In our Swiss eosinophilic esophagitis cohort, 825 (M ¼ 631, F ¼ 194) adolescent and adult patients are currently enrolled. A systematic analysis of symptoms in these patients revealed that dysphagia, long-lasting food impaction, and spontaneously occurring chest pain was present in 94.8% (n ¼ 782), 34.2% (n ¼ 282), and 38.4% (n ¼ 317) of patients. Interestingly, we found a subgroup of patients (n ¼ 10; 1.21%) having exercise-induced chest pain in absence of any signs of cardiovascular or pulmonary disease. We report here on a series of patients having a diagnosis of eosinophilic esophagitis according to the established criteria complaining about exercise-induced chest pain. The demographic parameters and clinical findings of all patients are summarized in detail in the Table. Two of these patients complained exclusively of exercise-induced chest pain and are presented as a case report in the following section.
Kahn et al
Exercise-induced Chest Pain and Eosinophilic Esophagitis
CASE REPORT
197
Case 2
A 16-year-old boy presented to his family physician with exercise-induced sensation of constriction in the throat and A 22-year-old professional hockey player was admitted to a hyper salivation, starting some weeks before his visit. The hospital with chest pain after a strong strike/hit on the breastpatient was evaluated by an ENT specialist and diagnosed bone during an ice hockey game. Clinical examination and with pharyngitis and laryngitis, which was treated with a thoracic computed tomography scan PPI (esomeprazole 40 mg once a were unremarkable, and the patient day) and a single dose of an antiCLINICAL SIGNIFICANCE was discharged with suspicion of a mycotic (diflucan 400 mg). thoracic concussion. Almost 1 week Eosinophilic esophagitis is a chronic inSubsequently, the patient was later his physical performance was referred to our institution, where flammatory immune-mediated disease of still substantially reduced due to an he described exclusively exercisethe esophagus. unpleasant feeling of choking, induced throat tightness and Eosinophilic esophagitis presents with reflux-like symptoms, and dyspnea increased salivation, but no occurring regularly, a few minutes growing incidence and prevalence in dysphagia. His personal history after beginning any physical effort. industrialized countries. revealed an allergic asthma, a polA series of comprehensive cardiolen allergy, and an oral allergy Affects predominantly middle-aged men, logical, pneumological, and ear, syndrome. Laboratory testing with a male-to-female ratio of 3:1. nose, and throat (ENT) examinarevealed an elevated serum immutions did not reveal any relevant Presents with dysphagia and bolus noglobulin E level (330 U/mL; pathological findings. Treatment norm 60 eosinophils/highand thoracic oppression, limiting his physical performance power field and focally eosinophilic microabscesses. Sympdrastically. The patient was referred for evaluation to our toms resolved within a few days after initiation of oral topical institution, where he presented with exclusively exercisecorticosteroids (Axotide 250 Diskus). Fluticasone 1000 mg induced, oppression-like chest sensations, but no dysphagia. bid was given for 10 days followed by a maintenance treatPersonal history revealed an allergic asthma, urticaria, and an ment with fluticasone 250 mg bid. Symptoms reappeared oral allergy syndrome on consumption of chicken, meat, or 2 weeks after the patient stopped the maintenance treatment. bananas. The family history, physical examination, and laboAnother endoscopic evaluation demonstrated an active ratory analyses were unremarkable except for a slightly eosinophilic esophagitis with edema, longitudinal furrows, increased total immunoglobulin E value (255 U/mL; norm: and moderate white exudates in the distal esophagus, with an 150 eosinophils/high-power esophagitis with longitudinal furrows in the proximal part of the field. No signs of a gastroesophageal reflux disease were esophagus and with histological esophageal eosinophilia. observed. The treatment with oral topical corticosteroids Functional esophageal examinations, including high-resolution (Axotide 250 Diskus) was reinitiated with complete resolumanometry and a 24-hour impedance-pH monitoring, were tion of symptoms. unremarkable. A treatment with swallowed topical corticosteroids (fluticasone 1000 mg bid; Axotide 250 Diskus; GlaxoSmithKDISCUSSION line, Brentford, Middlesex, UK) was initiated. All symptoms Eosinophilic esophagitis was first reported not more than 20 resolved within a few days and under a maintenance treatment years ago; to this day, not all clinical facets of this chronicwith topical corticosteroids (Axotide 250 Diskus, fluticasone inflammatory disease are elucidated. Meanwhile, it has 250 mg bid), the patient was again able to play ice hockey. After become clear that dysphagia for solids, food impaction, and discontinuation, symptoms reappeared within a few weeks. spontaneously occurring chest pain are the most prominent Another upper endoscopy demonstrated endoscopical signs of symptoms in adolescent and adult eosinophilic esophagitis eosinophilic esophagitis with furrowing, white exudates, and patients.1 edema (Figure A). Histology confirmed a severe eosinophilic In our cohort we identified 10 patients with confirmed infiltration in the proximal and distal esophagus, with >100 diagnosis of eosinophilic esophagitis complaining about eosinophils/high-power field (Figure B). Treatment was exercise-induced chest pain. The differential diagnosis for reinitiated and symptoms disappeared rapidly. this symptom includes musculoskeletal, gastrointestinal,
Case 1
198
The American Journal of Medicine, Vol 128, No 2, February 2015
Table
Demographics and Clinical Findings of Patients with Exercise-induced Chest Pain (n ¼ 10)
Demographics Age in years at diagnosis; median (range) Duration of symptoms (months); median (range) Affected relatives Sex; male:female BMI kg/m2, median (range) History of allergies; AA ¼ allergic asthma, ARC ¼ allergic rhinoconjunctivitis, OAS ¼ oral allergy syndrome Laboratory findings Peripheral blood eosinophils eos /mm3; norm:
