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23 January 2024: Articles  USA

Cystocerebral Syndrome in a Patient with Altered Mental Status

Unusual clinical course, Challenging differential diagnosis, Unusual or unexpected effect of treatment

Badar Sabeen12ABCDEF, Joseph Allen White ORCID logo34BCDE*, Sandy D. Espinosa Hernandez1ACDF, Abdulhusein Kapasi35BCDF, Temilola Majekodunmi ORCID logo3EF, Sri Harsha Boppana6CDF, Isaac Trelles2CDF, Marco Ruiz2CDF, Odalys Frontela1CDF

DOI: 10.12659/AJCR.942264

Am J Case Rep 2024; 25:e942264

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Abstract

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BACKGROUND: Cystocerebral syndrome is delirium occurring in the elderly that results from urinary retention and acute bladder distension. Urinary retention can cause cerebral issues, such as altered mental status, without having an infection present. The pathophysiology is possibly due to increased catecholamine secretion while attempting to micturate. Due to its presenting symptoms, most physicians begin cerebrovascular workup, performing unnecessary and, often, invasive imaging studies. Although easily overlooked as a differential diagnosis, cystocerebral syndrome is an easily treatable cause of delirium and should be considered during treatment of elderly patients with delirium.

CASE REPORT: The patient was an 89-year-old man with a medical history of chronic obstructive airway disease, dementia, hypertensive disorder, and gastroesophageal reflux disease who presented with altered mental status secondary to urinary incontinence. The computed tomography scan without contrast showed a large volume of impacted stool in the cecum, with a distended urinary bladder. This case report describes his presentation, medical treatment, and outcome and discusses areas of gap improvement.

CONCLUSIONS: To date, there are only a handful of published articles on cystocerebral syndrome. This case report aims to add the awareness of bladder distention as an etiology of cystocerebral syndrome to the body of knowledge in the scientific community in the hope that patients will be identified and treated earlier, more safely, and at a reduced cost. Cystocerebral syndrome needs to be extensively addressed in research, and physicians should consider it one of the important differential diagnoses of delirium among elderly men.

Keywords: Delirium, Urinary Retention, Neurocognitive Disorders

Background

Cystocerebral syndrome is a term used to describe delirium in elderly patients that is caused by acute urinary retention [1]. This syndrome can include symptoms of agitation, confusion, paranoia, decreased responsiveness, and increased difficulty in redirecting the patient [2]. In 1990, Blackburn and Dunn were the first to describe acute urinary retention presenting as delirium in elderly men [3]. In 1991, Lim and Carter described the pathophysiology of cystocerebral syndrome as possibly an increase in catecholamine hormone from the bladder while attempting to micturate, which leads to delirium [4]. Other factors can lead to delirium, especially in the elderly, such as infection, metabolic disturbance, medications, intoxication, vascular causes, and the presence of dementia [5], and literature shows that geriatricians are more aware of cystocerebral syndrome than are their physician counterparts [6]. This case report aims to bridge the knowledge gap by bringing awareness to internists, primary care physicians, neurologists, and physicians as a whole on this easily diagnosed and treatable pathology known as cystocerebral syndrome. However, it is not sufficient to treat the symptom, one must exercise due diligence in identifying and addressing the cause of the urinary retention itself.

Case Report

An 89-year-old man with a medical history of advanced chronic obstructive airway disease on home oxygen, dementia, hyper-tensive disorder, gastroesophageal reflux disease, chronic constipation, and benign prostatic hyperplasia, which was treated with a transurethral procedure several years ago, presented to the Emergency Department with altered mental status and urinary incontinence. The dementia was diagnosed after a left hip replacement 3 years prior and had slowly worsened since then. The daughter reported that her father was doing fine prior to his symptom onset. He communicated with her and could use the phone to call her when she was at work. He could walk with a walker at home, feed himself, and control his urine and stool.

According to the patient’s daughter (who was the main historian and the patient’s proxy), the patient became agitated 2 days prior to presentation and had “not been his usual self”. His agitation was associated with confusion, visual hallucinations, and urinary incontinence.

The patient had no recent illness (including COVID-19), sick contacts, or changes in routine or environment and did not ingest anything unusual. There was no history of fever, cough, nausea, vomiting, abdominal pain, diarrhea, or weakness. However, the patient was short of breath, which was attributed to his chronic obstructive airway disease-emphysema. The patient had been an active smoker from his teenage years and smoked 1 to 1.5 packs a day.

Vital signs were as follows on admission: body temperature, 36.6°C; heart rate, 78 beats per min; respiratory rate, 20 breaths per min; and O2 saturation, 99% on room air or supplemental O2. During the hospital stay the patient’s respiratory rate ranged from 17 to 20 breaths per min, and the O2 saturation ranged from 97% to 99%. On evaluation, the patient was awake, confused, agitated, and disorientated to time, place, and person. Physical examination and laboratory findings were unremarkable.

Imaging studies with electrocardiogram revealed a sinus rhythm with right bundle branch block. Chest X-ray showed stable chronic lung changes, with no evidence of acute cardiopulmonary disease. A non-contrast computer tomography (CT) scan of the brain revealed chronic encephalomalacia in the right frontal and left frontal lobes, which was unchanged from a prior examination (Figure 1). A CT scan of the abdomen showed no acute changes from a prior evaluation 2 years prior, apart from moderate to large volume stool burden in the cecum (Figure 2).

The urinary bladder was decompressed after the CT scan, and about 1000 mL of clear urine was removed, without hematuria. An admitting diagnosis of vascular dementia was made, and the patient was admitted for further workup and symptomatic treatment.

On hospital day 2, there was no neurological improvement. The patient was placed on valproic acid 125 mg oral twice daily and olanzapine 2.5 mg intramuscular injection every 8 h as needed for agitation. The psychiatry team was consulted because of the patient’s agitation and psychotic features, such as visual hallucinations. The nurse reported anuria in the patient, and a Foley catheter was re-inserted, with 1100 mL of urine output. The patient showed significant improvement in mental status and decreased agitation following the procedure. He was able to recognize present family members and hold conversations. The Urology Service was consulted, and on hospital day 3, an ultrasound of the kidneys showed a large bilateral simple cyst, a bilateral small non-obstructing nephrolithiasis, and Foley catheter in place within a mostly decompressed bladder. Mirtazapine 2.5 mg oral at bedtime was started. The prostrate-specific antigen level was 0.28, and urine output was adequate (Figure 3).

By day 4, the patient was oriented to person, disoriented to time and place, but calm, cooperative, and able to obey commands. The psychiatry team started the patient on quetiapine 25 mg oral 3 times daily. The geriatric team was consulted and reported acute altered mental status (delirium) secondary to urinary retention with overflow incontinence, most likely due to fecal impaction. A repeat non-contrast CT scan of the brain showed stable chronic encephalomalacia in the right frontal and left frontal lobes, with no acute changes. No further diagnostic procedures, magnetic resonance imaging, or other imaging of the brain were performed. A repeat abdominal X-ray showed a heavy stool burden with a non-obstructive bowel gas pattern (Figure 4). The patient was continued on laxatives, and an enema was prescribed.

From hospital days 5 to 8, the patient continued to improve cognitively and remained unagitated. The antipsychotic medications were adjusted accordingly. He remained oriented to person, place and time, and could hold conversations steadily.

A discharge plan to hospice care was underway, when on day 9, the patient became agitated, confused, and disoriented to person, place, and time. At this time, the patient had not had a bowel movement since admission and a repeat enema proved futile. A sigmoidoscopy and cystoscopy were not attempted, as the patient was termed high-risk and could not obtain cardiology clearance. Following a discussion with the proxy, the patient was eventually transferred to hospice care.

On day 10, the patient remained confused, agitated, and dis-oriented to person, time, and place. The patient had been constipated for more than 10 days and did not respond to the enema and increased laxative dose. An X-ray of the abdomen prior to discharge on day 10 showed prominent loops of the large bowel with stool burden. The patient’s colonos-copy screening was held due to a high risk for cardiovascular complications due to severe aortic stenosis, per the cardiology team. Due to his advanced dementia and other comorbidities, noninvasive stool de-impaction was recommended, and hence, no further invasive measures were pursued. The hospice service discussed transfer of the patient to hospice care, the patient’s proxy agreed, and the primary team arranged and transferred the patient to hospice. On follow-up, the patient’s proxy stated that he had a bowel movement and had urinated since discharge and was back to his original baseline status. The patient could now feed himself and walk with a walker at home. At the time of this report, he was urinating every day and having bowel movements every 2 to 4 days.

Discussion

The purpose of this case report is to bring awareness to the diagnosis of cystocerebral syndrome, as it can present as something else. This diagnosis is more likely when a patient comes in with urinary retention that is not due to a urinary tract infection. Urinary retention can cause cerebral issues, such as altered mental status, without having an infection present. The other reason can be that, based on the knowledge of how patients present, physicians may insist or assume that it is something else and not think about cystocerebral syndrome as a differential diagnosis. Our patient did not have any weakness, the reflexes were fine, and there were no central nervous system issues, such as motor tone. Altered mental status is a unique symptom of this diagnosis, and the CT scan should show something if it was due to some sort of dementia; however, our patient’s CT scan was normal when compared with a previous one, and was unchanged. Awareness is key and should be part of the differential diagnosis for elderly patients presenting similarly to acute urinary retention, most commonly due to a urinary tract infection, without signs and symptoms of infection.

Acute urine retention can cause delirium in elderly men. Rapid recognition of this phenomenon can lead to a significant improvement in the patient’s mental status. In this case report, the patient showed significant improvement, where the patient was at 95% of his mental status baseline, once his urinary retention was resolved. This was resolved by the insertion of a urinary catheter. The underlying cause of urinary retention was known by the physicians, which was the patient’s benign prostatic hyperplasia and/or chronic constipation. The patient was cleared as a high risk for colonoscopy and cystoscopy interventions. The physicians decided to try to relieve the patient’s constipation through comfort measures with certain medications and by using a fleet enema. However, the patient relapsed again with delirium symptoms as his constipation persisted, causing acute urinary retention again. The relapse of these symptoms seen once his constipation continued shows evidence that acute urinary retention can cause acute altered mental status in the elderly man.

Moreover, giving patients antipsychotic medication, such as valproic acid, which has an anticholinergic effect, to control the patient’s agitation was adding another risk factor for urinary retention. The patient continued to have confusion and agitation as the urinary retention existed. However, since the prominent symptom was altered mental status in this patient’s scenario, invasive imaging studies, such as a CT scan of the brain, were ordered by physicians twice to exclude acute vascular issues. Those imaging results could not explain the cause of delirium (Figures 1, 2, and 4). Cystocerebral syndrome not only creates confusion for the patient and family members, but also creates confusion for physicians if it is not diagnosed early. Physicians need to put acute urinary retention on the top of the list of differential diagnoses of delirium in an elderly male patient.

Conclusions

Cystocerebral syndrome is one of the differential diagnoses of delirium among elderly men and it can be diagnosed and treated. This case report shows with clear evidence that, once all the other diagnoses have been ruled out, acute urinary retention can lead to altered mental status in an elderly male patient. The symptoms can be treated symptomatically if the differential diagnosis of cystocerebral syndrome is considered as well. However, because it is not common for physicians to put cystocerebral syndrome at the top of the deferential diagnosis, this can lead to running invasive medical tests, although the history and physical examination itself can yield the urological cause. Moreover, few studies have addressed this phenomenon, and it is uncommonly reported in the international literature. Cystocerebral syndrome needs to be extensively addressed in research, and physicians should consider it one of the important differential diagnoses of delirium among elderly men.

References:

1.. Liem PH, Carter WJ, Cystocerebral syndrome: A possible explanation: Arch Intern Med, 1991; 151(9); 1884-86

2.. Thelmo FL, Tzarnas S, Rosal NR, Cystocerebral syndrome: An updated review and a new proposed mechanism for an often forgotten cause of delirium: Cureus, 2020; 12(10); e11034

3.. Blackburn T, Dunn M, Cystocerebral syndrome. Acute urinary retention presenting as confusion in elderly patients: Arch Intern Med, 1990; 150(12); 2577-78

4.. Blè A, Zuliani G, Quarenghi C, Gallerani M, Fellin R, Cystocerebral syndrome: A case report and literature review: Aging (Milano), 2001; 13(4); 339-42

5.. Francis J, Delirium and acute confusional states: prevention, treatment, and prognosis: UpToDate, 2023, Waltham, MA, UpToDate

6.. Waardenburg IE, Delirium caused by urinary retention in elderly people: A case report and literature review on the “cystocerebral syndrome”: J Am Geriatr Soc, 2008; 56(12); 2371-72

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923