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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 2  |  Page : 91-94

Intermittent sympathetic stimulation: A clinical clue of Intussusception with altered mental status


1 Department of Pediatrics, Bharati Vidyapeeth Medical College, Pune, Maharashtra, India
2 Department of Pediatrics, Rangadore Hospital, Bengaluru, Karnataka, India

Date of Submission21-Sep-2020
Date of Decision10-Nov-2020
Date of Acceptance21-Nov-2020
Date of Web Publication10-Mar-2021

Correspondence Address:
Dr. Arun Thomas
Department of Pediatrics, Rangadore Hospital, Bengaluru, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpcc.jpcc_150_20

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  Abstract 


Neurological symptoms are documented in intestinal pathologies; however pure neurological presentation in abdominal conditions are rare and presenting with intermittent autonomic disturbances are extremely rare. Here we describe two cases with intestinal obstruction and intussusception who had intermittent hypertension and tachycardia. Diagnosis in both the children were initially challenging, especially so in the first case as the child presented with only neurological symptoms. The exact pathogenesis of this sympathetic overdrive is not known and requires more studies to evaluate the cause. In both the kids, once the primary pathology was treated, the children became neurologically better and has been fine on follow up.

Keywords: Hypertension, intussusception, sympathetic stimulation


How to cite this article:
Shankar GH, Thomas A, Santhikrishna V, Venkatesha GA. Intermittent sympathetic stimulation: A clinical clue of Intussusception with altered mental status. J Pediatr Crit Care 2021;8:91-4

How to cite this URL:
Shankar GH, Thomas A, Santhikrishna V, Venkatesha GA. Intermittent sympathetic stimulation: A clinical clue of Intussusception with altered mental status. J Pediatr Crit Care [serial online] 2021 [cited 2021 Apr 20];8:91-4. Available from: http://www.jpcc.org.in/text.asp?2021/8/2/91/311051




  Introduction Top


Medicine is a science of uncertainty and an art of probability (Sir W. Osler). As the field of medicine grows in experience over time, we find that even relatively common illnesses may surprise us in the way they present and progress.

As early as 1947, a case series of 95 children with Intussusception described “listlessness” as a symptom often unrecognized and receiving only scant mention in literature.[1] Since then, neurologic symptoms have often been appreciated and reported either in conjunction with gastrointestinal symptoms, or being the dominant feature of the presentation or very rarely, being the sole manifestation of Intussusception. We, hereby report a clinical sign that has not been previously described in literature that clinicians need to be aware of, whilst managing children with intussusception and other intestinal pathology.


  Case Reports Top


Case 1

A 6-month-old previously well female child and developmentally normal presented with sudden onset of lethargy and increased sleepiness lasting for 2 h. At the beginning of the illness, the child had excessive crying for a few minutes. There was no history of fever/vomiting/any other symptoms apart from lethargy. She had passed stools the same morning which were normal. No history of trauma or accidental toxic ingestion or clinical seizure.

On arrival to the emergency department, the baby was drowsy with a Glasgow Coma Scale (GCS) of 10/15– opening eyes to painful stimulus, crying with pain, and localizing pain. No abnormal findings were present on examination of cardiovascular, respiratory systems, and per abdominal examination. Central nervous system (CNS) examination was not revealing as well except for extensor plantar response bilaterally (which can be normal for the age). Random Blood sugar at arrival was 119 mg/dl. The child was shifted to pediatric intensive care unit (PICU) and put on continuous cardiorespiratory monitoring, with noninvasive blood pressure (NIBP) recorded every 5 min.

The preliminary diagnostic considerations included CNS disease – postictal state, nonconvulsive status epilepticus, infection, and accidental/nonaccidental traumatic brain injury. Metabolic diseases, sepsis were the other possibilities. Venous blood gases with lactate were normal. An initial set of investigations was sent to look for a cause – complete blood counts, C-reactive protein (CRP), electrolytes, liver function test, and renal functions – which were all normal.

The baby was given a dose of empirical antibiotics for presumed sepsis/meningitis. Considering the possibility of nonconvulsive status epilepticus, she was loaded with levetiracetam. Over the next couple of hours, infant was noted to have NIBP recordings well above 99th centile up to 120/70 mmHg, with heart rate (HR) 130/min. Four limb blood pressure revealed similar recordings in all the limbs. The baby was very drowsy at the time. No clinical suggestion of pain was present. These high readings persisted for 15 min and then resolved back to normal values in the range of 90-95/55-60 mmHg with HR around 100/min. No clinical suggestion of pain/bradycardia/abnormal movements of limbs or eyes was observed during this period, nor any change in the breathing pattern or pupil reaction.

Plan was made to do an emergency head CT with contrast– which was normal. After about an hour, the child continued to have low GCS and transient hypertensive readings were noted again (without the child being irritable). At this point, owing to unexplained altered mental status in an infant with intermittent hypertension (presumably correlating with periodic abdominal pain), urgent USG abdomen was requested which revealed ileocolic intussusception. Several such episodes of transient sympathetic hyperactivity were observed by the time patient underwent pneumatic reduction. Sensorium normalized following the reduction. Oral feeds were initiated without any new concerns. A repeat ultrasound was normal, and child was discharged on D2 of admission. Currently doing well on follow-up at 6 and 12 months.

Case 2

A 1 year 6 months old, previously well male child with no significant past history came with complaints of nonbilious, nonprojectile vomiting of 2 days' duration and 1 day of diarrhea associated with poor feeding and decreased urine output for preceding 8 h. At the time of presentation to the ER, the child was drowsy with a GCS of 12/15. The child was irritable on abdominal palpation and there was mild abdominal distension, otherwise systemic examination was unremarkable. However, over next 2 h, sensorium worsened and he remained persistently drowsy with GCS of 10/15. Worryingly, there was sinus tachycardia (up to 220 beats/min) with intermittent hypertension (blood pressure rising up to 150/96 mm Hg) with BP crossing the 99th centile for age, sex, and height. Arterial access was established for invasive blood pressure monitoring. 12-lead ECG confirmed sinus tachycardia which was not responsive to fluid challenge. Tachycardia was persistent with HR staying above 180 beats/min. There were multiple episodes of hypertension associated with worsening tachycardia lasting up to ½ h without any manifestation of pain with periods of normal blood pressures lasting for 2–3 h. The initial differential diagnosis considered were sepsis with meningitis, compromised bowel loops or urosepsis. Although intestinal obstruction was suspected, the child had been passing stools and the vomiting was non bilious and bowel sounds were present albeit sluggish. Investigations revealed neutrophilic leukocytosis with CRP of 150 mg/dl and hence he was empirically started on IV Ceftriaxone, amikacin, and metronidazole after sending blood and urine cultures. X-ray erect abdomen showed hugely dilated bowel loops suggestive of possible intestinal obstruction. Ultrasound abdomen confirmed ileoileal intussusception with dilated bowel loops. Although it was ileoileal intussusception, since child continued to be clinically symptomatic, he was taken for pneumatic reduction under fluoroscopic guidance. There was no evidence of intussusception during fluoroscopy suggestive of spontaneous reduction and hence child was shifted back to PICU. However, as child continued to be drowsy with intermittent hypertension, computed tomography (CT) brain, and lumbar puncture was done which was normal. Repeat USG abdomen done the following morning showed recurrence of intussusception and contrast-enhanced CT abdomen confirmed this above finding with associated mesenteric lymphadenopathy. The child was taken for emergency exploratory laparotomy and child was found to have an ileal band acting as lead point for intussusception causing intestinal obstruction. Band resection and lymph node resection was done. Following surgery, the child was extubated in the theatre and child was shifted back to PICU with normal sensorium. Child improved hemodynamically and there were no more episodes of hypertension. Cultures were sterile and antibiotics were stopped after total 5 days' duration and child was discharged on postoperative day 3.


  Discussion Top


Neurologic symptoms are now commonly known to occur in Intussusception, more so in infants.[2] The cause of neurologic manifestations in Intussusception is not definitely known. Several mechanisms have been proposed, including endogenous opioids,[3] transmural release of toxic metabolic products from an ischemic bowel,[4] fluid and electrolyte abnormalities due to GI symptoms and behavioral responses of infants to severe abdominal pain.[5] It is of interest to note that Pumberger et al.[4] noted impaired mental state in conditions other than Intussusception (volvulus, incarcerated inguinal hernia, and vitelline bands), causing traction and strangulation of mesentery and bowel.

Since a neurologic presentation causes more alarm in caregivers, it tends to be associated with a shorter duration of presentation to medical care. However, it is recognized that duration of symptoms has no correlates with the likelihood of neurologic involvement.[2] Lethargy is the most commonly reported symptom and its incidence ranges from 16% to 38% in different case series.[2],[5],[6],[7],[8] Various descriptions may be found in literature, including acute changes in levels of consciousness, profound apathy, listlessness, restlessness, miosis, generalized hypotonia, moderately impaired reaction to painful stimuli, seizures.[3],[4],[9] One of the authors has previously published a case report highlighting the neurologic presentation of intussusception as the sole finding.[10] Isolated neurologic symptomatology, as in our first case, is extremely rare (<3% in a large case series[9]) and presents a significant diagnostic challenge to the treating physician. This has led to recommendations of including USG abdomen and stool occult blood in any child with an unexplained altered mental status.[11]

Pain is associated with increase in sympathetic tone leading to hypertension with tachycardia.[12] Though nonspecific, it may be useful in recognizing inadequately treated pain in critically ill ventilated children on sedatives and muscle relaxants. However, its value in nonintubated children with neurologic disease and altered mental status is difficult to assess since it may represent multiple pathologic processes including seizure, raised ICP along with nociception.

Pain assessment in infants is not straightforward. The pain scales are based on behavioral changes and physiologic variables.[13] In our case series, behavioral changes were looked for in both the children during episodes of intermittent sympathetic hyperactivity, but were absent. Since the classic description in intussusception is intermittent pain as part of the triad including vomiting and red currant jelly stools, these brief episodes of tachycardia and hypertension were interpreted as physiologic responses to pain. In these children, as we were in a dilemma about the diagnosis and working up toward a neurologic cause, the recurrent episodes of sympathetic overactivity even when there were no other signs possibly indicating pain (behavioral and clinical), alerted us to consider this as a sign of pain and alerted us to workup for intussusception. We think that sympathetic overactivity is probably unique in its periodicity in this particular context and is, to the best of our knowledge not described in literature, probably due to rarity of exclusive neurologic presentation of Intussusception. Clinicians may utilize this clinical sign as a clue when faced with similar diagnostic conundrum.

The sign of intermittent sympathetic stimulation in the form of tachycardia and/or hypertension can be a clinical clue for a neurological presentation of intussusception.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Obberhelman HA, Condon JB. Acute intussusception in infants and children. Surg Clin North Am 1947;27:3-22.  Back to cited text no. 1
    
2.
Kleizen KJ, Hunck A, Wijnen MH, Th Draaisma JM. Neurological symptoms in children with intussusception. Acta Paediatr 2009;98:1822-4.  Back to cited text no. 2
    
3.
Tenenbein M, Wiseman NE. Early coma in intussusception: Endogenous opioid induced? Pediatr Emerg Care 1987;3:22-3.  Back to cited text no. 3
    
4.
Pumberger W, Dinhobl I, Dremsek P. Altered consciousness and lethargy from compromised intestinal blood flow in children. Am J Emerg Med 2004;22:307-9.  Back to cited text no. 4
    
5.
Reijnen JA, Festen C, Joosten HJ, Wieringen PM. Atypical characteristics of a group of children with intussusception. Acta Paediatr 1990;79:675-9.  Back to cited text no. 5
    
6.
Luks FI, Yazbeck S, Perreault G, Desjardins JG. Changes in the presentation of intussusception. Am J Emerg Med 1992;10:574-6.  Back to cited text no. 6
    
7.
Kaiser AD, Applegate KE, Ladd AP. Current success in the treatment of intussusception in children. Surgery 2007;142:469-75.  Back to cited text no. 7
    
8.
Buettcher M, Baer G, Bonhoeffer J, Schaad UB, Heininger U. Three-year surveillance of intussusception in children in Switzerland. Pediatrics 2007;120:473-80.  Back to cited text no. 8
    
9.
Domínguez-Carral J, Puertas-Martín V, Carreras-Sáez I, Maraña-Pérez AI, Escobar-Delgado T, García-Peñas JJ. Neurological symptoms in children with intussusception. An Pediatr (Barc) 2014;80:293-8.  Back to cited text no. 9
    
10.
Venkatesha GA, Natarajan A. Neurological presentation of intussusception: Case discussion and literature review. Case Reports 2009;2009:bcr0420091730. http://dx.doi.org/10.1136/bcr.04.2009.1730.  Back to cited text no. 10
    
11.
Maldonado L, Takagishi J. Four-month-old infant with intussusception presenting as altered mental status. SAGE Open Med Case Rep 2014;2:2050313X14558903.  Back to cited text no. 11
    
12.
Piriyapatsom A, Bittner EA, Hines J, Schmidt UH. Sedation and paralysis. Respir Care 2013;58:1024-37.  Back to cited text no. 12
    
13.
Franck LS, Greenberg CS, Stevens B. Pain assessment in infants and children. Pediatr Clin North Am 2000;47:487-512.  Back to cited text no. 13
    




 

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