Introduction
Acute colonic pseudo-obstruction (Ogilvie's syndrome, OS) is a disease characterized by acute enlargement of the colon in the absence of a mechanical cause preventing the flow of small or large intestinal contents (Vanek and Al-Salti, Reference Vanek and Al-Salti1986). The development of acute colonic pseudo-obstruction has no definite causes; however, many clinical conditions which are common in older adults (Conner et al., Reference Conner, Nassereddin and Mitchell2020) and at increased risk for it, such as advanced age, comorbidities, electrolyte imbalance, polypharmacy, and immobility, have been identified (Ates Bulut et al., Reference Ates Bulut, Soysal and Isik2018). The main clinical feature is abdominal distension which is reported in 80% of the patients. Additionally, nausea-vomiting and constipation are also reported in 60% and 50% of the patients, respectively (Vanek and Al-Salti, Reference Vanek and Al-Salti1986; Jetmore et al., Reference Jetmore, Timmcke and Gathright1992; Ates Bulut et al., Reference Ates Bulut, Soysal and Isik2018; Conner et al., Reference Conner, Nassereddin and Mitchell2020).
On the other hand, corticobasal syndrome (CBS) is a chronic progressive neurodegenerative disorder characterized by the involvement of various combinations of basal ganglia dysfunction signs such as akinesia, asymmetric rigidity, dystonia, and as well as cortical ones such as apraxia, myoclonus, alien-limb phenomena, and cognitive impairment. Constipation, one of the signs of autonomic failure, may also be observed in patients with atypical parkinsonian syndromes (Deutschländer et al., Reference Deutschländer, Ross and Dickson2018), and constipation causing fecal impaction and accompanying a variety of conditions may cause delirium in older adults with chronic progressive neurological diseases. These diseases like CBS are one of the most common predisposing conditions for delirium. Besides, now, CBS and other atypical parkinsonism syndromes have been reported not only to carry a heavy symptom burden equivalent to that of patients with advanced cancer but also have a higher burden of multimorbidity and risk of becoming wheelchair-dependent or bedridden. Additionally patients with CBS need palliative or hospice care, eventually (Bükki et al., Reference Bükki, Nübling and Lorenzl2016; Wiblin et al., Reference Wiblin, Lee and Burn2017) and intestinal motility problems, such as constipation, colonic pseudo-obstruction, as well as delirum, may be common in these patients receiving palliative care and can cause extreme suffering and discomfort.
Therefore, identifying and managing the possible underlying cause is crucial for patient outcomes, particularly, in those who potentially need palliative care or already have benefited from a palliative care service. To the best of our knowledge, clinical manifestation of delirium presented as OS in a patient with CBS has not been previously reported.
Case
An 80-year-old female patient was admitted to the emergency room with constipation for the last five days and change in consciousness for the last two days. She has been treated with levodopa-benserazide (625 mg/day), amantadine (150 mg/day), and levothyroxine (50 μg/day) due to CBS and hypothyroidism for 3 years in our unit. There was no history of new surgery or medication.
In the physical examination performed in the emergency department, her blood pressure was 110/70 mmHg, pulse rate was 112 per minute; and altered mental status, disorientation, fluctuations in consciousness, distraction, asymmetric limb, and axial rigidity, abdominal distention were determined and there were no bowel sounds. The blood test results performed at the hospital admission were as follows: white blood cell count 7000/L, hemoglobin 12.1 g/dL, glucose 116 mg/dL, urea 24 mg/dL, creatinine 0.64 mg/dL, sodium 142 mM/L, potassium 3.8 mM/L, total calcium 8.9 mg/dL, magnesium 0.87 mg/dL, serum TSH 1.39 m(IU)/L, free T4 1.19 ng/dL, all of them were within normal limits. Plain abdominal radiographs showed enlargement of the colon loops and air-fluid levels (Figure 1). After the mechanical cause of obstruction was ruled out, she was transferred to our geriatric clinic because of delirium and OS superimposed to CBD.
Fig. 1. Plain abdominal radiographs showed an enlargement of the colon loops and air-fluid levels (before treatment).
In our clinic, conservative therapy was initiated with fluids, lactulose, and enema. In the clinical observation of the case, despite the conservative and supportive treatment, colonic discharge was not provided and delirium continued. However, she has required no additional treatment due to delirium. During the follow-up, she was not able to defecate despite repeated enema application. Therefore, 48 h after the hospitalization, neostigmine (1 mg/30 min) infusion was started and repeated two times. After the second dose of neostigmine administration, we observed an increase in intestinal movement and then stool discharge was started. After stool discharge, an improvement was observed in the patient's delirium clinic. Abdominal distension completely regressed on the 5th day of hospitalization (Figure 2). The patient was discharged with fiber and lactulose treatment.
Fig. 2. Decrease in the width of the colon loops and loss of air-fluid levels on abdominal radiography (after treatment).
Discussion
Herein, we report a case of OS presented with delirium in an older lady with CBD. In this case, we consider that CBD is a predisposing factor in delirium development, and OS is a precipitating factor. The treatment of the etiological causes of delirium is very important because it allows the rapid treatment of the cases by preventing the use of potentially harmful drugs or inappropriate interventions.
OS is common in patients over the age of 60 years (Vanek and Al-Salti, Reference Vanek and Al-Salti1986), and the pathophysiology is still unknown. It may be caused by an increased sympathetic intestinal innervation or a decreased parasympathetic tone, or both, which results in functional obstruction of the colon (Jain and Vargas, Reference Jain and Vargas2012). On the other hand, CBD is a rare neurodegenerative disease, in which the signs of autonomic failure can be seen like other parkinsonian disorders. For this reason, in patients with CBD, it should be kept in mind that accompanying comorbid conditions, related drugs, and age-related changes may lead to the development of OS as well as autonomic dysfunction such as deterioration in bowel and bladder control (Isik et al., Reference Isik, Kolukisa and Ergun2013). Although OS was firstly described in 1948 (Ogilvie, Reference Ogilvie1948), this is the first CBD case that presented with OS, as far as we are concerned. Pharmacological treatment of OS aims to counteract intestinal sympathetic–parasympathetic dysregulation. Neostigmine therapy turned out to be effective for acute colonic pseudo-obstruction that does not respond to conservative treatment (Turégano-Fuentes et al., Reference Turégano-Fuentes, Muñoz-Jiménez and Del Valle-Hernández1997; Ponec et al., Reference Ponec, Saunders and Kimmey1999). Therefore, in patients with OS who do not benefit from the supportive and symptomatic treatment despite potential side effects of neostigmine, it is very important to apply it by taking the necessary precautions for possible complications of the drug. We suggest an initial conservative treatment and drug therapy approach after excluding the causes of mechanical obstruction in patients with a diagnosis of CBD.
Health-care professionals including palliative care physicians should be skilled in a holistic approach addressing many distinctive challenges of life-limiting chronic neurological illnesses in these frail older adults, such that OS may cause acute colonic pseudo-obstruction and should be reviewed from this point of view before the surgical procedure.
Finally, OS may be superimposed to CBD in older patients, and OS in such patients may play a role as a precipitating factor for the development of delirium. Before surgical management, supportive care is crucial for the management of OS in CBD patients and neostigmine treatment should not be delayed in patients who do not benefit from supportive treatment. In addition, the elimination of delirium with the removal of colonic pseudo-obstruction in our case is an indicator of the importance of the etiological approach in delirium treatment.
Funding
This research received no specific grant from any funding agency, commercial or not-for-profit sectors.
Conflicts of interest
The authors of this manuscript declare that there is no conflict of interest.
