Central Annals of Sports Medicine and Research

Cite this article: Ashkenazi E, Yacobovich J, Eliakim A, Nemet D (2020) Prolonged Progressive Exercise Intolerance in a Healthy Young Basketball Player - An Unusual Case. Ann Sports Med Res 7(5): 1163.

*Corresponding authorDan Nemet, Department of Pediatrics, Meir Medical Center, 59 Tchernichovski St. Kfar-Saba, 44281, Israel, Email: dnemet@gmail.com

Submitted: 06 Setpember 2020

Accepted: 20 September 2020

Published: 23 September 2020

ISSN: 2379-0571

Copyright© 2020 Ashkenazi E, et al.

OPEN ACCESS

Keywords•Effort thrombosis; Pulmonary emboli; Exercise

intolerance

Abstract

Self-reported exercise intolerance is a common complaint in youth. The most common cause of exercise intolerance, apart from poor physical fitness, is exercise-induced bronchoconstriction (EIB), which may occur in children with asthma and even in some healthy children. We present a case of exercise intolerance in a healthy 16-year old male, professional youth league basketball player, admitted to our pediatric department with complaints of prolnged (6 months) progressive exercise intolerance for the past 6 months. A comprehansive ambulatory workup was performed, yet no plain X-ray was done. The patient was found to have effort thrombosis (Paget-Schroetter syndrome) with multiple bilateral pulmonary embolisms. High index of suspicion in young athletes with similar complaints should raise the possible diagnosis of effort thrombosis and secondary PE. Meticulous history taking and a plain chest X-ray may have revealed the diagnosis earlier.

Case Report

Prolonged Progressive Exercise Intolerance in a Healthy Young Basketball Player - An Unusual CaseElya Ashkenazi1,3, Joanne Yacobovich1-3, Alon Eliakim1,3, and Dan Nemet1,3*1Department of Pediatrics, Meir Medical Center, Israel2Pediatric Hematology/Oncology Center, Schneider Children’s Medical Center of Israel, Israel3Sackler School of Medicine, Tel Aviv University, Israel

INTRODUCTION Self-reported exercise intolerance is a common complaint in

youth. The most common cause of exercise intolerance, apart from poor physical fitness, is exercise-induced bronchoconstriction (EIB), which may occur in children with asthma and even in some healthy children [1]. Other disorders, such as chronic cardiac or lung disease, congenital airway malformations, exercise induced hyperventilation or exercise induced laryngeal obstruction, may also result in respiratory difficulties during exercise in non-athletes [1]. In athletes, without known asthma, the prevalence of self-reported exercise-induced respiratory events is also high and may reach up to 30 percent [2].

We present a case of exercise intolerance in a healthy 16-year old male, professional youth league basketball player, admitted to our pediatric department with complaints of prolonged, progressive exercise intolerance for the past 6 months.

CASE PRESENTATIONA previously healthy 16-year-old boy, professional youth

league basketball player was admitted to our pediatric department complaining on progressive effort induced dyspnea during 6 months prior to his hospitalization, without chest pain or palpitations, muscular pain or weakness. Initially, his exercise intolerance started 20 minutes after strenuous aerobic exercise, however during the last two months prior to hospitalization; he was unable to perform more than 5 minutes of intense aerobic exercise. In addition, he complained of painless swelling of his right arm. He described a sense of pressure in his arm which was, at times, associated with discoloration without a history of recent trauma.

Upon admission, his parents mentioned that they observed a difference in arm size with right arm hypertrophy. Two years prior to his hospitalization, the patient noticed swelling in his right axillary area and a small lump was palpated. An ultrasound (US) was performed which demonstrated an enlarged superficial vein with no other pathological findings.

Prior to his hospitalization, due to the exceptional and worsening dyspnea in a young healthy athlete, he underwent several ambulatory medical exams that included: ECG at rest, cardiac ergometry, echocardiogram, and pulmonary function tests both at rest and with exercise, all interpreted to be normal.

On physical examination upon admission, his heart rate was 54 beats per minute, blood pressure 136/78 mmHg, and oxygen saturation was 96%. A pigeon chest appearance was noted and internally rotated shoulders were observed, lung and heart auscultation were normal as well as abdominal exam. A 4 cm difference in the biceps circumference between two arms was noted. Firm swelling of the right arm from the axillary region to below the elbow was obvious, with a bluish discoloration and colder temperature. Marked superficial veins in the shoulder and upper right chest area were noted.

INVESTIGATIONSThe following medical evaluations were peformed:

Lung X-ray demonstrated a left costo-phrenic angle blunting, and a normal cardiac silhouette and otherwise clear lung fields (Figure 1).

Duplex ultrasonography of the right arm demonstrated deep

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vein thrombosis (DVT) of the axillary, brachial and subclavian veins.

Due to the history of dyspnea the suspicion of pulmonary embolism (PE) arised and a CT angiography demonstrated multiple bilateral pulmonary embolisms with signs of ischemia in the lingula.

CT venography was performed for suspected thoracic outlet syndrome (TOS) and subsequent Paget Schroetter Syndrome (PSS), no anatomical abnormality was demonstrated in the thoracic outlet of the right shoulder. Additionally, a bone directed X-ray test ruled out presence of a cervical rib.

Pulmonary function examinations were normal. A cardiologic evaluation showed sinus bradycardia on ECG, with a normal echocardiogram. Neither tests showed signs of increased pulmonary vascular resistance.

Hypercoagulability testing: Increased D-Dimer up to 2348 ng/mL (normal range: 400 ng/ml) was found. Coagulation function tests were normal. Anti-cardiolipin IgG and IgM, beta-2 glycoprotein IgG and IgM, Lupus anticoagulant RVVT+KCT, antithrombin, protein S, protein C, activated protein C, lipoprotein a, homocysteine and prothrombin 20210 mutation – were all in the normal range.

TREATMENT As the reported symptomatology of our patient was

prolonged, and the dyspnea started six months prior to admission, we decided to treat the patient conservatively, initially with enoxaparin-sodium followed by bridging to warfarin. Due to his age direct oral anti-coagulants were not initiated.

FOLLOW UP AT 6 MONTHSA follow up CT angiography demonstrated mild improvement

in the pulmonary embolisms and the lingular infarct. Venous

pathologies corresponding to Paget-schroetter syndrome were reported.

A dynamic MRA demonstrated no anatomical abnormalities corresponding to thoracic outlet syndrome, a 3 cm irregularity in the proximal part of the right subclavian vein was found, this irregularity was unaffected by the position of the arm.

DISCUSSIONWe present a case of a healthy, 16-year-old basketball player

who presented to our department with history of prolonged, progressive exercise dyspnea and swelling of the right arm. Examinations revealed an effort thrombosis (Paget-Schroetter syndrome) with multiple bilateral pulmonary embolisms.

Although we are not the first to describe this condition in young athletes, there are certain exceptional aspects of this case leading to a delay in diagnosis and treatment and should be discussed.

From the beginning of his exercise intolerance complaints our patient went through a rather inclusive ambulatory workup for almost 6 months under the working diagnosis of exercise induced asthma/bronchoconstriction (EIA/EIB).

EIB is indeed the leading cause of exercise intolerance in both athletes and non-athletes. A joint task force of the European Respiratory Society (ERS) and European Academy of Allergy and Clinical Immunology (EAACI) defined exercise induced asthma (EIA) as symptoms and signs of asthma occurring in an asthmatic patient after exercise, whereas EIB was defined as a reduction in FEV1 of at least 10 percent after a standardized exercise test [2]. Our patient didn’t meet either of these definitions. Moreover, it is uncommon for a young athlete with no history of asthma to develop asthma at the age of 16. His reported exercise intolerance pattern was not typical for EIB, and no physical findings suggesting asthma were reported (e.g. wheezing or cough).

There are several cardio-respiratory causes that may lead to exercise intolerance, including disruption of oxygen transport (due to heart failure, cardiac arrhythmia, pulmonary hypertension or severe anemia), disorders of gas exchange in the lungs (such as interstitial lung disease or pulmonary edema) or disorders of ventilation [1].

Our patient was seen by a pediatric cardiologist, pediatric pulmonologist and a sports medicine specialist that performed a cardiac stress-test; exercise challenge test for asthma as well as echocardiogram, yet no chest X-ray was performed.

Upper extremity deep venous thrombosis (UEDVT) is an uncommon vascular problem, occurring primarily in young, healthy, active people. Although the history and symptoms are often unremarkable, the condition can lead to acute and long-term complications if not correctly recognized and appropriately treated [3]. From an epidemiological perspective, although it is regarded as the most common vascular condition among athletes, the general incidence of UEDVT is low (approximately 2\100,000 persons per year) [4]. The mechanism of injury is widely thought to be associated with repetitive strenuous upper extremity activities, hence the name “effort thrombosis” also known as Paget-Schrotter syndrome (PSS) [3,4]. A recent study identified

Figure 1 Chest X-ray, left costophrenic blunting is noted.

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32 adult high level athletes with effort thrombosis over a 10-year period, 14 of which were baseball players [5].

Although effort thrombosis is rare, immediate diagnosis can improve outcome and be lifesaving. Sportive activities can be initiated when treatment is started very early [6]. Hull and Harris postulated that elite athletes are predisposed to develop Virchow’s classic triad of hypercoagulability, vascular fluid stasis, and vascular damage with consequent venous thrombosis [7]. Furthermore, in a research of elite throwing athletes, the site of the thrombosis was the thoracic outlet, with compression of the vein at the level of the first rib [8]. The average age of professional players with upper extremity DVT is significantly lower (26 years) than players with other VTEs [9]. Professional athletes are predisposed to several acquired conditions that increase the risk of thrombosis; tissue trauma from contact during play, hemo-concentration from dehydration, circulatory stasis secondary to bradycardia, and long period of immobilization from either travel or injury [7].

Several studies have explored the relationship between exercise and the blood coagulation cascade, finding increases in pro-thrombotic markers and factor VIII as well as simultaneous activation of the fibrinolytic system and increased platelet aggregation [10].

There are no evidence-based guidelines when to allow return to regular and/or competitive sportive activities with or without anticoagulation [9].

CONCLUSIONSThis report illustrates the case of a young basketball teenager

with prolonged, progressive complaints of exercise intolerance who was eventually found to have a primary UEDVT and secondary PE. Meticulous history taking and a plain chest X-ray may have revealed the diagnosis earlier. A high index of suspicion in young athletes with similar complaints (in particularly in sports involving intensive use of the upper arms such as basketball, volleyball, tennis and baseball pitching) should raise the possible diagnosis of effort thrombosis and secondary PE.

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2. KH Carlsen, SD Anderson, L Bjermer, S Bonini, V Brusasco, W Canonica, et al. Treatment of exercise-induced asthma, respiratory and allergic disorders in sports and the relationship to doping. Allergy. 2008; 63: 492-505.

3. Wendy L Hurley, Sonya A Comins, Richard M Green, John Canizzaro. Atraumatic subclavian vein thrombosis in a collegiate baseball player: a case report. J Athl Train. 2006; 41: 198-200.

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6. Nathan A Mall, Geoffrey S Van Thiel, Wendell M Heard, George A Paletta, Charles Bush-Joseph, Bernard R Bach Jr. Paget-schrotter syndrome: A review of effort thrombosis of the upper extremity from a sport medicine perspective. Sports Health. 2013; 5: 353-6.

7. Hull CM, Harris JA. Venous thromboembolism in physically active people: consideration for risk assessment, mainstream awareness and future research. Sport Med. 2015; 45: 1365-1372.

8. Gregory S DiFelice, George A Paletta Jr, Barry B Phillips, Rick W Wright. Effort Trhombosis in the elite throwing athlete. Am J Sports Med. 2002; 30: 708-12.

9. Meghan Bishop, Matthew Astolfi, Eric Padegimas, Peter DeLuca, Sommer Hammoud. Venous thromboembolism within professional American sport leagues. Orthop J Sports Med. 2017; 5: 2325967117745530.

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Ashkenazi E, Yacobovich J, Eliakim A, Nemet D (2020) Prolonged Progressive Exercise Intolerance in a Healthy Young Basketball Player - An Unusual Case. Ann Sports Med Res 7(5): 1163.

Cite this article

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Prolonged Progressive Exercise Intolerance in a Healthy Young Basketball Player - An Unusual CaseAbstractIntroduction Case PresentationInvestigationsFigure 1TreatmentFollow up at 6 months ConclusionsReferences