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Table of Contents
CASE REPORT
Year : 2019  |  Volume : 10  |  Issue : 1  |  Page : 42-44

Gastric ascariasis mimicking rapunzel syndrome in an epileptic patient: Learning points galore


1 Department of Medicine, Sanjay Gandhi Memorial Hospital, Government of NCT Delhi, New Delhi, India
2 Department of Obstetrics and Gynaecology, Sanjay Gandhi Memorial Hospital, Government of NCT Delhi, New Delhi, India

Date of Submission27-Jun-2018
Date of Decision26-Jul-2018
Date of Acceptance25-Aug-2018
Date of Web Publication17-Oct-2018

Correspondence Address:
Dr. Shalini Singh
BW- 70- C, Shalimar Bagh, Delhi - 110 088
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/INJMS.INJMS_5_18

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  Abstract 


Rapunzel syndrome refers to a very rare condition in which swallowed hair forms a gastric trichobezoar that has a long tail extending into the small bowel. It presents with early satiety, malnutrition, obstructive symptoms, and sometimes, gastric-outlet obstruction. Despite the varied manifestations of Ascaris infestation in man, gastric ascariasis is rare which is demonstrated in the stomach by radiology and by endoscopic examination. We present a rare case of trichobezoar in a 14 years epileptic female with recurrent seizure episodes and no prior history of psychiatric illness or worm infestation where the final diagnosis of gastric ascariasis was made on upper gastrointestinal-endoscopy revealing the presence of worms at the level of the fundus and body of the stomach. Endoscopically some worms were removed, and the rest passed down dead in stools after antihelminthic treatment. Recovery was uneventful and free of complications. Frequent seizures could also be attributed to malabsorption of anti-epileptic drugs because of gastric ascariasis. A prompt diagnosis (tissue diagnosis and direct visualization) always scores above characteristic radiologic findings and appropriate therapy can reduce comorbidities.

Keywords: Gastric ascariasis, gastric outlet obstruction, Rapunzel syndrome, trichobezoar


How to cite this article:
Passi P, Kanwar S, Chaurasiya S, Vats S, Kumar R, Singh S. Gastric ascariasis mimicking rapunzel syndrome in an epileptic patient: Learning points galore. Indian J Med Spec 2019;10:42-4

How to cite this URL:
Passi P, Kanwar S, Chaurasiya S, Vats S, Kumar R, Singh S. Gastric ascariasis mimicking rapunzel syndrome in an epileptic patient: Learning points galore. Indian J Med Spec [serial online] 2019 [cited 2019 Mar 20];10:42-4. Available from: http://www.ijms.in/text.asp?2019/10/1/42/243626


  Introduction Top


Bezoars are concretions of indigestible material found in the gastrointestinal (GI) tract and are generally classified according to their composition into trichobezoars (hair), phytobezoars (plant material), and pharmacobezoars (pharmaceutical products).[1] Many bezoars are asymptomatic, so the number of reported cases underestimates their prevalence in the general population. GI infestation with Ascaris lumbricoides is common in the tropical countries, particularly in children. A wide range of clinical presentations are reported for GI ascariasis in adults and children. Up to 25% of the world's population, mostly in the third world countries, is infested with the worm.[2]

In the present case, the rarity of mimicking a trichobezoar, finally diagnosed on upper GI endoscopy to be gastric ascariasis, is not well mentioned in the literature. Hence, the case is being presented because of its rarity.


  Case Report Top


A 14-year-old female brought to the emergency department in active generalized tonic–clonic seizures, was stabilized with intravenous sedatives and antiepileptic therapy. Her parents gave a history of recurrent episodes of seizures for the last 5 years, despite being compliant and on two different anti-epileptic drugs in full adult dose from a super-specialty hospital. There was no prior history of any psychiatric illness, worm infestation, any chronic drug intake (except anti-epileptic drugs), or any other chronic illness. The patient also complained of decreased appetite with abdominal pain and fullness after meals associated with frequent vomitings for the last 2 months. Provisional diagnosis was made of seizure disorder with severe acute gastritis. The patient underwent general and systemic examinations which were unremarkable. Electroencephalography showed generalized epileptic form sharp waves. Magnetic resonance imaging of the brain showed no abnormality. Serum valproate levels (34.8) and serum eptoin levels (2.7) were found to be low and Serum amylase level was raised (234 mg/dl) whereas all other biochemical and hematological reports were within normal range. In view of recurrent vomitings and pain in the abdomen and low blood levels of antiepileptics, a contrast-enhanced computed tomography (CT) of the whole abdomen was done which showed a large heterogeneous intraluminal density with mottled gas pattern in stomach suggesting trichobezoar [Figure 1] with mild prominence of small bowel loops. Upper GI-endoscopy was done to confirm trichobezoar, which showed gastric erosions with duodenal worms [Figure 2], and worms were removed. The patient was started on antihelminthic drugs, and there was a history of passage of dead worms in stool within 3 days. Patient's repeat contrast enhanced computed tomography (CT) whole abdomen was done which showed no evidence of any intra-luminal density or trichobezoar. The patient got relieved of chronic pain abdomen and disturbed episodes of vomitings post-anti-helminthic treatment and was continued on anti-epileptic treatment and discharged. On follow-up, the patient is seizure free with adequate blood levels of anti-epileptic drugs.
Figure 1: Contrast enhanced computed tomography abdomen suggesting trichobezoar

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Figure 2: Upper gastrointestinal-endoscopy showing removal of worms with gastric erosions

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  Discussion Top


Rapunzel syndrome refers to a very rare condition mostly occurring in emotionally disturbed, depressed, or mentally retarded in which swallowed hair forms a gastric trichobezoar that has a long tail extending into the small bowel. The clinical presentation of bezoars is related to their location and usually includes nausea, vomiting, loss of appetite, early satiety, abdominal discomfort or pain, and weight loss, malnutrition, obstructive symptoms, and sometimes gastric outlet obstruction. Abdominal CT imaging, while being diagnostic when an intraluminal mass with a mottled gas pattern is seen, could offer useful information to exclude a neoplastic cause of rapid weight loss associated with decreased appetite, such as pancreatic or gastric cancer. Endoscopy, however, provides the most accurate tool for the identification and classification of bezoars.[3]

Gastric ascariasis is a rarity in view of continuous gastric peristaltic activity and the acid milieu of the stomach. The intestine has an immense capacity for dilatation and can possibly accommodate >5000 worms without any symptoms. There are varied manifestations of Ascaris infestation in man, likely gastric outlet obstruction or perforation, vomiting, loss of appetite, early satiety, abdominal discomfort, or pain.[4],[5],[6] Gastric ascariasis can be demonstrated in the stomach by radiology and by endoscopic examination.

The present case was being evaluated for intra-abdominal neoplasm or tuberculosis [in view of recurrent vomitings, low blood levels of antiepileptics, abdominal fullness and discomfort after meals associated with weight loss and decreased appetite] when diagnosis of trichobezoar was made. On further evaluation, by upper GI endoscopy, the final diagnosis of gastric ascariasis was revealed. The patient was started on anti-helminthic drugs and repeat contrast enhanced CT whole abdomen showed no evidence of trichobezoar. A single dose is usually curative in 85% of the patients but taking into account the worm count, efficacy, and adverse effects of the drugs multiple doses can be administered.[7] The patient got relieved of chronic pain abdomen and disturbed episodes of vomitings.

The rarity of mimicking a trichobezoar, finally diagnosed on the upper GI endoscopy to be gastric ascariasis, is not well mentioned in literature. Hence, the case is being presented because of its rarity.


  Conclusion Top


Rapunzel syndrome is a rare entity that may be complicated by life-threatening events. Gastric ascariasis, a rarity in view of continuous peristaltic activity and the acid milieu of the stomach, can cause gastric outlet obstruction or perforation. Frequent seizures and decreased blood levels of anti-epileptics despite full dose and patient compliance could also be attributed to malabsorption because of gastric ascariasis. In present case, the rarity of mimicking of a trichobezoar, finally diagnosed by upper GI endoscopy, we strongly recommend endoscopy to be routinely done in each case for the identification and classification of bezoars, as a prompt diagnosis (tissue diagnosis and direct visualization) always scores above characteristic radiologic findings and an appropriate therapy can reduce comorbidities.

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

None.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Williams RS. The fascinating history of bezoars. Med J Aust 1986;145:613-4.  Back to cited text no. 1
    
2.
Refeidi A. Live Ascaris lumbricoide s in the peritoneal cavity. Ann Saudi Med 2007;27:118-21.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Andrus CH, Ponsky JL. Bezoars: Classification, pathophysiology, and treatment. Am J Gastroenterol 1988;83:476-8.  Back to cited text no. 3
    
4.
Louw JH. Abdominal complications of Ascaris lumbricoides infestation in children. Br J Surg 1966;53:510-21.  Back to cited text no. 4
    
5.
Basavaraju SV, Hote PJ. Acute GI and surgical complications of Ascaris lumbricoide s infection. Infect Med 2003;20:154-9.  Back to cited text no. 5
    
6.
Peker K, Kılıç K. Endoscopic diagnosis in Ascaris lumbricoides case with pyloric obstruction. Turkiye Parazitol Derg 2011; 35:210-3.  Back to cited text no. 6
    
7.
Adegnika AA, Zinsou JF, Issifou S, Ateba-Ngoa U, Kassa RF, Feugap EN, et al. Randomized, controlled, assessor-blind clinical trial to assess the efficacy of single- versus repeated-dose albendazole to treat Ascaris lumbricoides, Trichuris trichiura, and hookworm infection. Antimicrob Agents Chemother 2014; 58:2535-40.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2]



 

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