Takotsubo syndrome triggered by emotional and

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W. Elikowski et al.

OPISY PRZYPADKÓW / CASE REPORTS

Takotsubo syndrome triggered by emotional and physical stress due to coincidental immobilization resembling the experimental model of the disease – a case report WALDEMAR ELIKOWSKI1, MAŁGORZATA MAŁEK-ELIKOWSKA2, NATALIA FERTAŁA1, MAGDALENA ZAWODNA1, ALINA BASZKO3, ZOFIA TRYPUĆ1 3

Department of Internal Medicine, Józef Struś Hospital, Poznań, Poland; 22nd Department of Cardiology, Medical University of Poznań, Poland; Radiology Unit, Józef Struś Hospital, Poznań, Poland

Takotsubo syndrome triggered by emotional and physical stress due to coincidental immobilization resembling the experimental model of the disease – a case report

Zespół takotsubo sprowokowany przez stres emocjonalny i fizyczny związany z przypadkowym unieruchomieniem przypominającym eksperymentalny model choroby – opis przypadku

Elikowski W1, Małek-Elikowska M2, Fertała N1, Zawodna M1, Baszko A3, Trypuć Z1.

Elikowski W1, Małek-Elikowska M2, Fertała N1, Zawodna M1, Baszko A3, Trypuć Z1.

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Department of Internal Medicine, Józef Struś Hospital, 22nd Department of Cardiology, Medical University of Poznań, Poland; 3Radiology Unit, Józef Struś Hospital, Poznań, Poland

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The term takotsubo syndrome (TTS) is derived from a comparison of left ventricular (LV) appearance in its apical form to the shape of the pot which is a trap used for octopus catching. In the classical animal model on rats, its occurrence can be triggered by experimental immobilization (IMO). Former observations of capture myopathy, seen in animals after extreme exertion, showed coexistence of rhabdomyolysis related to skeletal muscle injury and myocardial damage. The authors describe a case of a 66-year-old obese female in whom TTS was triggered by an uncommonly stressful event when she had been trapped in the bathtub for several hours. The woman lives alone and has impaired functionality due to degenerative disease of the spine and hips. On this unlucky day, she decided to have a bath after which she was unable to get out of the bathtub despite repeated efforts. She was very frustrated and helpless having to spend all the evening and night in that cul-de-sac-like situation. She was rescued in the morning by neighbors and firemen who transferred her to hospital. On admission she was exhausted and suffering from dyspnea, she negated any chest pain; there were bloody abrasions on her elbows. Due to negative T waves in ECG and elevated troponin I, echocardiography was performed which revealed LV apical ballooning. The high level of creatine phosphokinase (CPK) was suggestive of rhabdomyolysis. Following heart failure therapy, the , patient s clinical state, as well as echocardiographic parameters, improved gradually. Normalization of LV function and CPK values was observed after 11 days.

Nazwa zespołu takotsubo (TTS) nawiązuje do porównania prezentacji lewej komory (LV) w jego koniuszkowej postaci do kształtu dzbana służącego jako pułapka do połowu ośmiornic. W klasycznym modelu zwierzęcym TTS u szczurów, jego wystąpienie można spowodować przez eksperymentalne unieruchomienie (IMO). Wcześniejsze obserwacje miopatii powysiłkowej, spotykanej u zwierząt po ekstremalnym wysiłku, dowodzą współistnienia zależnej od uszkodzenia mięśni szkieletowych rabdomiolizy oraz uszkodzenia mięśnia sercowego. Autorzy opisali przypadek 66-letniej otyłej kobiety, u której TTS został sprowokowany przez niecodzienną sytuację stresową, związaną z jej uwięzieniem przez wiele godzin w wannie. Kobieta mieszka sama i ma ograniczoną sprawność wskutek zmian zwyrodnieniowych kręgosłupa i stawów biodrowych. Feralnego dnia postanowiła wykąpać się, jednak nie była w stanie wyjść z wanny pomimo wielokrotnie podejmowanych prób. Była bardzo zła i bezsilna spędzając w tych przymusowych warunkach cały wieczór i noc. Rano została uratowana przez sąsiadów i strażaków, którzy zawieźli ją do szpitala. W chwili przyjęcia kobieta była wyczerpana, skarżyła się na duszności, bólów zamostkowych nie zgłaszała; stwierdzano krwiste otarcia łokci. Ze względu na ujemne załamki T w EKG i podwyższone stężenie troponiny I wykonano badanie echokardiograficzne, w którym wykazano balonowanie koniuszka LV. Wysokie stężenie kinazy fosfokreatynowej (CPK) sugerowało obecność rabdomiolizy. Po zastosowaniu leczenia niewydolności serca obserwowano stopniową poprawę stanu klinicznego chorej oraz obrazu echokardiograficznego. Normalizację funkcji LV oraz stężenia CPK stwierdzono po 11 dniach.

Key words: takotsubo syndrome/cardiomyopathy, experimental model, immobilization, capture myopathy, rhabdomyolysis

Słowa kluczowe: zespół/kardiomiopatia takotsubo, model eksperymentalny, unieruchomienie, miopatia powysiłkowa, rabdomioliza

Pol Med J, 2018, 2018; XLV (268); 154–157

Pol Merkur Lekarski; XLV (268); 154–157

The term takotsubo syndrome (TTS), previously named takotsubo cardiomyopathy, is derived from a comparison of left ventricular (LV) echocardiographic or ventriculographic appearance in its apical form to the shape of the pot which is a trap used for octopus catching [5]. Numerous physical and emotional factors that can trigger TTS development have been described [5,21,22]. Broken heart syndrome, a TTS synonym, underlies the importance of a strong negative emotion in the pathogenesis of the disease; however, TTS as a happy heart syndrome after excessive positive emotion is also possible [5].

The first descriptions of the disease are believed to come from the early 1990s from Japan [4,18]; however, a decade before, a pathological study on stress cardiomyopathy in victims of homicidal assaults without internal injuries was published [3]. Still earlier, in the 1970s, pathological myocardial changes suggestive of stress-induced cardiomyopathy were described in experiments on rabbits crowded in a cage [31] and on restrained pigs [9,10]. In the classical animal TTS model on rats, its occurrence can be observed after experimental immobilization (IMO), which reproduces emotional (and physi-

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Oddział Chorób Wewnętrznych, Szpital im. Józefa Strusia w Poznaniu; 2II Klinika Kardiologii, Uniwersytet Medyczny w Poznaniu; 3Zakład Radiologii, Szpital im. Józefa Strusia w Poznaniu

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Takotsubo syndrome triggered by emotional and physical stress due to coincidental immobilization ...

cal) stress [25-29]. Rats are restrained by securing them on their backs to a board using adhesive tape. Sometimes, immobilization or restraint constitute the conditions preceding TTS occurrence in humans [17,20]. On the other hand, in capture myopathy seen in animals after extreme exertion, coexistence of rhabdomyolysis due to skeletal muscle injury and stressrelated myocardial damage can be observed [1,2].

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heart failure symptoms. Normalization of ECG was delayed; after 2 months shallow inverted T waves in precordial leads were still found (fig.2C). After that period, during a control appointment, her cardiac as well as general state remained stable; only the scars after elbow injuries were still present (fig.1C). Completing her history, the patient confessed that she had been

Figure 1. Patient’s trap – her bathtub (A), bloody abrasions on elbows on admission (B), scars on elbows after 2 months (C) Rycina 1. Pułapka chorej – jej wanna (A), krwiste otarcia na łokciach przy przyjęciu (B), blizny na łokciach po 2 miesiącach (C)

The purpose of this paper is to present a particular case of TTS, whose development resembled unexpectedly the experimental model of the disease.

in chronic stress due to her husband’s cancer disease resulting in death a year before; also, ten years earlier she had lost her son.


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Figure 2. ECG on admission (A), before discharge (B), after 2 months (C) Rycina 2. EKG przy przyjęciu (A), przed wypisem (B), po 2 miesiącach (C)

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A 66-year-old obese female (BMI – body mass index – 34) with a history of hypertension was admitted to hospital due to exhaustion and severe dyspnea, after an uncommonly stressful event when she had been trapped in the bathtub for several hours. The woman lives alone and has impaired functionality due to degenerative disease of the spine and hips. A day before admission she decided to have a bath after which she was unable to get out of the bathtub despite repeated efforts. She was very frustrated and helpless having to spend all the evening and night in that cul-de-sac-like situation. She was rescued in the morning by neighbors and firemen who transferred her to hospital. On admission, she negated any chest pain; apart from pulmonary congestion and gallop heart rhythm, there were bloody abrasions on her elbows that she sustained after many failed attempts to get out of the bathtub (fig.1B). Due to negative T waves in ECG (fig.2A) and elevated troponin I (2831 ng/l, normal value £56 ng/l) and brain natriuretic peptide – BNP (397 pg/ml, normal value £100 pg/ml) echocardiography was performed which revealed LV apical ballooning with a decreased ejection fraction (EF 30%) and global longitudinal strain (GLS 10%) (fig.3A). The high level of creatine phosphokinase – CPK (31959 IU/l, normal value 26-192 IU/L) was suggestive of rhabdomyolysis as a result of the patient’s exertion and skeletal muscle injury. Creatinine concentration as well as glomerular filtration rate (GFR) were within normal ranges on admission and during hospitalization. Following heart failure therapy, the woman’s clinical state, as well as echocardiographic parameters, improved gradually. Return of troponin I and BNP to normal values was seen after 7 and 10 days, respectively. Normaliza-tion of LV function (EF 58%, GLS -19%; fig.3B) and CPK activity (148 IU/l) was observed after 11 days. 128-row computed tomography of coronary arteries revealed mild coronary artery disease (CAD) with non-obstructive changes (fig.4). She was discharged without

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Figure 3. Echocardiography: LV apical ballooning in 4-chamber view and decreased global and regional (in apical segments) longitudinal strain in bull’s eye presentation (A), LV function normalization after 11 days (B) Rycina 3. Echokardiografia: obraz balonowania koniuszka LV w projekcji 4-jamowej oraz obniżone całkowite i regionalne (w segmentach koniuszkowych) odkształcanie podłużne w prezentacji „bull’s eye” (A), normalizacja funkcji LV po 11 dniach (B)


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Figure 4. 128-row computed tomography of coronary arteries in 3-dimensional and multiplanar reformatted images – mild (non-obstructive) coronary artery stenosis in LAD, CX and RCA. LAD – left anterior descending artery, CX – circumflex artery, RCA – right coronary artery Rycina 4. 128-rzędowa tomografia komputerowa tętnic wieńcowych w rekonstrukcji trójwymiarowej i wielopłaszczyznowej – łagodne (nieistotne) zwężenia LAD, CX i RCA. LAD – tętnica zstępująca przednia, CX – tętnica okalająca, RCA – prawa tętnica wieńcowa

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Before Sato’s [18] and Dote’s [4] publications, which resulted in TTS definition, important studies on sympathetic stimulation and brain-heart relations were described in an IMO animal model [12]. Further experimental observations by Ueyama et al. on rats showed IMO stress-induced immediate-early gene expression via activation of adrenoreceptors [25,26], elevation of ST segments in ECG [27] and reversed LV contractile disturbances [28,29]. Gene expression was rapidly upregulated in the endothelial, myocardial and smooth muscle cells of the coronary vessels. Experimentally provoked LV dysfunction appeared as apical ballooning or global hypokinesis; however, not all animals were susceptible to developing TTS. Earlier treatment with beta-blockers prevented LV dysfunction. Observations on wild and domestic animals showed that extreme exertion leading to skeletal muscle injury (capture myopathy) and resulting in rhabdomyolysis coexists sometimes with severe heart failure [1]. Based on patients’ descriptions from the literature, some authors have suggested that rhabdomyolysis may favor TTS development [8,11,24], but it seems rather that both diagnoses have resulted from the same cause, e.g. excessive exertion, fall [13], traffic injury [20] or malnutrition [23]. IMO/restraint and rhabdomyolysis coexist usually with adrenergic excitation. Both may occur simultaneously in the animal world [7], and in humans, like in the case presented, when the woman trapped in her bath tried desperately to get out of the trap. It is quite probable that immobilization or restraint do not rarely constitute the conditions preceding TTS occurrence in humans. The conditions, similar to the IMO model, can be observed, e.g. in a traffic (car, train, ship) accident [17], in those buried under earth or snow, in restrained aggressive or delirious patients [15] or crime victims (restraint and confinement) [19], following a home fall in aged, especially solitary living persons [13,21]. Recently, these authors have observed TTS in a woman who held on to an overhead leaking drainpipe in her house during a downpour and was forced not to move for an hour. Other coexisting TTS triggering factors

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DISCUSSION




Takotsubo syndrome triggered by emotional and physical stress due to coincidental immobilization ...

important in such patients are trauma, fear or pain. LV dysfunction develops usually immediately after the provocative factors have appeared, but a delayed reaction is also possible. There are no data on the prevalence of death or acute heart failure as a TTS manifestation following physical restraint in hospitalized patients [14]. Most cases of rhabdomyolysis are seen in elderly after a fall, in some of them with immobilization [32]; such patients are probably at risk of TTS. Mental and physical stress can overlap; to differentiate which one is more essential can be impossible. Additionally, predisposition and chronic stress should also be taken into consideration; longterm stressful circumstances can lead to vulnerability to acute new mental or physical stressors and, subsequently, to an onset of TTS [30]. Extreme exertion interchangeably with resignation may result in alternating sympathetic and parasympathetic stimulation, which especially predisposes to TTS occurrence. When it comes to non-obstructive CAD found in the patient presented, it should be noted that CAD prevalence, assessed by coronary angiography, in TTS patients has been reported to be within the range of 10-29% [5,16,22]. Intravascular ultrasound use can reveal mild atherosclerotic lesions in most TTS patients [6]. Furthermore, significant coronary artery stenosis does not exclude TTS diagnosis, which is in contradiction to the classical Mayo Clinic criteria but with accordance to the current International Takotsubo Diagnostic Criteria [5].

CONCLUSIONS Immobilization or restraint stress may result in intensive sympathetic or alternating sympathetic/parasympathetic stimulation leading to TTS development as well as in the experimental model, wild and domestic animal world, and in humans. Capture myopathy with rhabdomyolysis may coexist with immobilization/restraint stress. Chronic stress may result in a decreased threshold to new stress stimuli and can allow augmented response to emotional or physical triggering factors.

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