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Pseudopheochromocytoma induced by anxiolytic withdrawal.

Páll A, Becs G, Erdei A, Sira L, Czifra A, Barna S, Kovács P, Páll D, Pfliegler G, Paragh G, Szabó Z - Eur. J. Med. Res. (2014)

Bottom Line: Setting of the diagnosis of pseudopheochromocytoma treatment was initiated with alpha- and beta-blockers resulting in reduced frequency of symptoms.The patient's clinical condition improved rapidly and the dosage of alpha- and beta-blockers could be decreased.We conclude that the withdrawal of an anxiolytic therapeutic regimen may generate sympathetic overdrive resulting in life-threatening paroxysmal malignant hypertension and secondary encephalopathy.

View Article: PubMed Central - PubMed

Affiliation: Division of Emergency Medicine, Institute of Internal Medicine, University of Debrecen Medical Center, Nagyerdei krt, 98, Debrecen, 4032, Hungary. szaboz.med@gmail.com.

ABSTRACT

Background: Symptomatic paroxysmal hypertension without significantly elevated catecholamine concentrations and with no evidence of an underlying adrenal tumor is known as pseudopheochromocytoma.

Methods: We describe the case of a female patient with paroxysmal hypertensive crises accompanied by headache, vertigo, tachycardia, nausea and altered mental status. Previously, she was treated for a longer period with alprazolam due to panic disorder. Causes of secondary hypertension were excluded. Neurological triggers (intracranial tumor, cerebral vascular lesions, hemorrhage, and epilepsy) could not be detected.

Results: Setting of the diagnosis of pseudopheochromocytoma treatment was initiated with alpha- and beta-blockers resulting in reduced frequency of symptoms. Alprazolam was restarted at a daily dose of 1 mg. The patient's clinical condition improved rapidly and the dosage of alpha- and beta-blockers could be decreased.

Conclusions: We conclude that the withdrawal of an anxiolytic therapeutic regimen may generate sympathetic overdrive resulting in life-threatening paroxysmal malignant hypertension and secondary encephalopathy. We emphasize that pseudopheochromocytoma can be diagnosed only after exclusion of the secondary causes of hypertension. We highlight the importance of a psychopharmacological approach to this clinical entity.

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Related in: MedlinePlus

During the first clinical evaluation of the patient a very severe attack could be observed. The patient became unconscious, her systolic blood pressure rapidly rose above 230 mmHg. Similar trends were observed during the repeated paroxysms. BP: blood pressure.
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Fig1: During the first clinical evaluation of the patient a very severe attack could be observed. The patient became unconscious, her systolic blood pressure rapidly rose above 230 mmHg. Similar trends were observed during the repeated paroxysms. BP: blood pressure.

Mentions: The patient presented herself at the Division of Rare Diseases for further etiologic examinations. During the first clinical evaluation a very severe attack could be observed. The patient became unconscious and her blood pressure rapidly rose to 230/100 mmHg, with a regular heart rate of 160 to 180/minute (Figure 1). Moreover, focal muscle twitching appeared on her left face, and excessive lacrimation and flushing could also be observed. Because of her unstable clinical condition she was immediately admitted to our ICU. Before the administration of additional medications, the patient’s severe clinical condition improved significantly without assistance. By the end of the paroxysm the frequency of sinus rhythm decreased to 90/minute and her blood pressure was also normalized. After the crisis, no signs of arrhythmia or long-standing neurological defects could be observed. No other significant clinical abnormalities could be found during further clinical examinations. Afterwards, during the first week of hospitalization she had attacks two to four times a day. These paroxysmal hypertensive crises lasted for three to five minutes and then disappeared spontaneously without any medical interventions. Between the paroxysms the patient was asymptomatic. Because of the repeated attacks, a combination of alpha- and beta-adrenoceptor blockers was given, which was able to lower her blood pressure and heart rate during the paroxysms, but not the frequency of the attacks. Holter electrocardiography recordings and twelve-lead surface electrocardiograms revealed sudden onset of episodic sinus tachycardia without any signs of further atrial or ventricular arrhythmias (Figure 2). Renal Doppler ultrasound examination was performed to exclude renovascular disease. It revealed physiological blood flow in both renal arteries, with no significant difference regarding resistive indices (0.67 versus 0.7, respectively). Although previous examinations were not able to prove any endocrinological background of the paroxysms, a repeated laboratory testing of pheochromocytoma and carcinoid was performed. Laboratory results of our patient are shown in Table 2. An elevated serum chromogranin A level appeared, but it proved to be a false positive result due to concomitant proton-pump inhibitor (PPI) therapy (after cessation of the PPI, the chromogranin A level was in the normal range). Surprisingly, an adenoma could be detected in the left adrenal gland during computed tomography. Due to the repeated severe clinical symptoms, we were obliged to start treatment of the pheochromocytoma; thus, a cardio-selective beta-blocker (bisoprolol 5 mg twice daily) in combination with an alpha-adrenoceptor-blocker (doxazosine 4 mg once daily) was initiated. During this time, urine concentrations of 5-hydroxyindoleacetic acid (5-HIAA), metanephrine, normetanephrine and dopamine were found to be normal. Although, we measured slightly elevated serum concentrations of noradrenalin and dopamine during an attack, the levels did not fulfill the criteria for pheochromocytoma (Table 3). To ensure the safe exclusion of pheochromocytoma, an iodine-131-metaiodobenzylguanidine (131I-MIBG) scan was also performed, which did not reveal any abnormalities relating to adrenal gland dysfunction (Figure 3). Furthermore, hyperaldosteronism as a very rare cause of paroxysmal hypertension could also be excluded. Further laboratory tests helped to exclude any hormonal abnormalities; thus the aforementioned adrenal gland adenoma was regarded as an ‘incidentaloma’. Thyroid laboratory tests showed the effective hormonal substitution of hypothyroidism secondary to the previous thyroidectomy. After excluding the possibility of endocrine disorders we focused on anxiolytic medication. For this reason psychiatric examination was performed and alprazolam was re-administered in a daily dose of 1 mg (0.5 mg twice daily). We could demonstrate an immediate clinical improvement; furthermore, the daily dose of alpha- and beta-blockers could also be decreased. During the administration of alprazolam at a daily dose of 1 mg, sleepiness and fatigue occurred, therefore we decreased the daily dose to 0.5 mg. Consequently, the paroxysmal increase in blood pressure reappeared so further therapy of 1 mg was necessary for maintenance. After a four-week period on the ICU, the patient was discharged though still with mild symptoms but with an improved quality of life. The systolic and diastolic blood pressures and heart rate collected after the discharge of our patient were inserted into the manuscript (Table 4).Figure 1


Pseudopheochromocytoma induced by anxiolytic withdrawal.

Páll A, Becs G, Erdei A, Sira L, Czifra A, Barna S, Kovács P, Páll D, Pfliegler G, Paragh G, Szabó Z - Eur. J. Med. Res. (2014)

During the first clinical evaluation of the patient a very severe attack could be observed. The patient became unconscious, her systolic blood pressure rapidly rose above 230 mmHg. Similar trends were observed during the repeated paroxysms. BP: blood pressure.
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4196012&req=5

Fig1: During the first clinical evaluation of the patient a very severe attack could be observed. The patient became unconscious, her systolic blood pressure rapidly rose above 230 mmHg. Similar trends were observed during the repeated paroxysms. BP: blood pressure.
Mentions: The patient presented herself at the Division of Rare Diseases for further etiologic examinations. During the first clinical evaluation a very severe attack could be observed. The patient became unconscious and her blood pressure rapidly rose to 230/100 mmHg, with a regular heart rate of 160 to 180/minute (Figure 1). Moreover, focal muscle twitching appeared on her left face, and excessive lacrimation and flushing could also be observed. Because of her unstable clinical condition she was immediately admitted to our ICU. Before the administration of additional medications, the patient’s severe clinical condition improved significantly without assistance. By the end of the paroxysm the frequency of sinus rhythm decreased to 90/minute and her blood pressure was also normalized. After the crisis, no signs of arrhythmia or long-standing neurological defects could be observed. No other significant clinical abnormalities could be found during further clinical examinations. Afterwards, during the first week of hospitalization she had attacks two to four times a day. These paroxysmal hypertensive crises lasted for three to five minutes and then disappeared spontaneously without any medical interventions. Between the paroxysms the patient was asymptomatic. Because of the repeated attacks, a combination of alpha- and beta-adrenoceptor blockers was given, which was able to lower her blood pressure and heart rate during the paroxysms, but not the frequency of the attacks. Holter electrocardiography recordings and twelve-lead surface electrocardiograms revealed sudden onset of episodic sinus tachycardia without any signs of further atrial or ventricular arrhythmias (Figure 2). Renal Doppler ultrasound examination was performed to exclude renovascular disease. It revealed physiological blood flow in both renal arteries, with no significant difference regarding resistive indices (0.67 versus 0.7, respectively). Although previous examinations were not able to prove any endocrinological background of the paroxysms, a repeated laboratory testing of pheochromocytoma and carcinoid was performed. Laboratory results of our patient are shown in Table 2. An elevated serum chromogranin A level appeared, but it proved to be a false positive result due to concomitant proton-pump inhibitor (PPI) therapy (after cessation of the PPI, the chromogranin A level was in the normal range). Surprisingly, an adenoma could be detected in the left adrenal gland during computed tomography. Due to the repeated severe clinical symptoms, we were obliged to start treatment of the pheochromocytoma; thus, a cardio-selective beta-blocker (bisoprolol 5 mg twice daily) in combination with an alpha-adrenoceptor-blocker (doxazosine 4 mg once daily) was initiated. During this time, urine concentrations of 5-hydroxyindoleacetic acid (5-HIAA), metanephrine, normetanephrine and dopamine were found to be normal. Although, we measured slightly elevated serum concentrations of noradrenalin and dopamine during an attack, the levels did not fulfill the criteria for pheochromocytoma (Table 3). To ensure the safe exclusion of pheochromocytoma, an iodine-131-metaiodobenzylguanidine (131I-MIBG) scan was also performed, which did not reveal any abnormalities relating to adrenal gland dysfunction (Figure 3). Furthermore, hyperaldosteronism as a very rare cause of paroxysmal hypertension could also be excluded. Further laboratory tests helped to exclude any hormonal abnormalities; thus the aforementioned adrenal gland adenoma was regarded as an ‘incidentaloma’. Thyroid laboratory tests showed the effective hormonal substitution of hypothyroidism secondary to the previous thyroidectomy. After excluding the possibility of endocrine disorders we focused on anxiolytic medication. For this reason psychiatric examination was performed and alprazolam was re-administered in a daily dose of 1 mg (0.5 mg twice daily). We could demonstrate an immediate clinical improvement; furthermore, the daily dose of alpha- and beta-blockers could also be decreased. During the administration of alprazolam at a daily dose of 1 mg, sleepiness and fatigue occurred, therefore we decreased the daily dose to 0.5 mg. Consequently, the paroxysmal increase in blood pressure reappeared so further therapy of 1 mg was necessary for maintenance. After a four-week period on the ICU, the patient was discharged though still with mild symptoms but with an improved quality of life. The systolic and diastolic blood pressures and heart rate collected after the discharge of our patient were inserted into the manuscript (Table 4).Figure 1

Bottom Line: Setting of the diagnosis of pseudopheochromocytoma treatment was initiated with alpha- and beta-blockers resulting in reduced frequency of symptoms.The patient's clinical condition improved rapidly and the dosage of alpha- and beta-blockers could be decreased.We conclude that the withdrawal of an anxiolytic therapeutic regimen may generate sympathetic overdrive resulting in life-threatening paroxysmal malignant hypertension and secondary encephalopathy.

View Article: PubMed Central - PubMed

Affiliation: Division of Emergency Medicine, Institute of Internal Medicine, University of Debrecen Medical Center, Nagyerdei krt, 98, Debrecen, 4032, Hungary. szaboz.med@gmail.com.

ABSTRACT

Background: Symptomatic paroxysmal hypertension without significantly elevated catecholamine concentrations and with no evidence of an underlying adrenal tumor is known as pseudopheochromocytoma.

Methods: We describe the case of a female patient with paroxysmal hypertensive crises accompanied by headache, vertigo, tachycardia, nausea and altered mental status. Previously, she was treated for a longer period with alprazolam due to panic disorder. Causes of secondary hypertension were excluded. Neurological triggers (intracranial tumor, cerebral vascular lesions, hemorrhage, and epilepsy) could not be detected.

Results: Setting of the diagnosis of pseudopheochromocytoma treatment was initiated with alpha- and beta-blockers resulting in reduced frequency of symptoms. Alprazolam was restarted at a daily dose of 1 mg. The patient's clinical condition improved rapidly and the dosage of alpha- and beta-blockers could be decreased.

Conclusions: We conclude that the withdrawal of an anxiolytic therapeutic regimen may generate sympathetic overdrive resulting in life-threatening paroxysmal malignant hypertension and secondary encephalopathy. We emphasize that pseudopheochromocytoma can be diagnosed only after exclusion of the secondary causes of hypertension. We highlight the importance of a psychopharmacological approach to this clinical entity.

Show MeSH
Related in: MedlinePlus