Tropical Doctor

HSV-1 oesophagitis presenting with persistent hiccoughs in

an immunocompetent host: A case report

Journal: Tropical Doctor

Manuscript ID TD-21-0103

Manuscript Type: Case Report

Date Submitted by the

10-Feb-2021
Author:
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Complete List of Authors: Paul, Aritra; Nilratan Sircar Medical College and Hospital,
Ghose, Aruni; Medway NHS Foundation Trust

Keyword: Asia < Location, Viral infection < Disease

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3 MANUSCRIPT TITLE
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6 HSV-1 oesophagitis presenting with persistent hiccoughs in an immunocompetent host: A
7 case report
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10 MANUSCRIPT INFORMATION
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Article Type : Case Report
14 Total word count (manuscript) : 494
15 Total word count (title) : 13
16 Number of references : 4
17 Number of figures : 1 (2 illustrations)
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23 AUTHORS:
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25 Aritra Paul 1, Aruni Ghose 2
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Affiliations:
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1 Nil Ratan Sircar Medical College and Hospital, Kolkata, India.
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32 2 Medway NHS Foundation Trust, Gillingham, Kent, UK.
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37 CORRESPONDING AUTHOR:
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39 Dr. Aruni Ghose
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41 Telephone number: +44 7570839307
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44 Email id: aruni.ghose@nhs.net
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46 Address: Medway NHS Foundation Trust, Windmill Road, Gillingham, Kent, ME7 5NY.
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3 Abstract
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8 HSV-1 oesophagitis as an aetiology of persistent hiccoughs is a rarity in immunocompetent
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10 hosts and entails an exhaustive diagnostic workup, since it does not present with any of the
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typical oesophagitis symptoms. Our patient presented with persistent hiccoughs that were
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15 resistant to treatment with baclofen. Oesophagogastroduodenoscopy with biopsy confirmed
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17 the diagnosis of HSV-1 oesophagitis. The hiccoughs subsided within 48 hours of acyclovir
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therapy.
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24 Case Report
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29 A 54-year-old man presented to casualty with cough, persistent hiccoughs and intermittent
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31 vomiting for one week. The hiccoughs persisted during his sleep. There was no history of
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33 fever, breathlessness, dysphagia or heartburn. There were no other systemic complaints. He
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was a non-smoker, non-alcoholic and was on daily thyroxine for hypothyroidism.
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40 Systemic examination was unremarkable apart from regular hiccoughs 4-6 times every
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minute.
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47 Blood tests including complete blood count, renal profile, and liver function tests were within
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49 normal limits. ECG and Chest X-ray were normal. Abdominal ultrasonography showed grade
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52 I fatty liver and multiple stones in the gall bladder with no evidence of cholecystitis or
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54 pericholecystic inflammation. CECT (Contrast Enhanced Computed Tomography) chest and
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56 abdomen revealed gallstone disease, right sided inguinal hernia and renal calculus (4.5mm).
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Sputum gram stain and culture showed no growth.
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6 The patient was started on conservative treatment with intravenous rabeprazole and sucralfate
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8 syrup. Intravenous ondansetron and metoclopramide did not give any relief to his continuous
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10 hiccoughs. Subsequently oral baclofen 10 mg and domperidone 10mg thrice daily along with
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clonazepam 0.25mg was started which also did not improve his symptoms.
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17 He underwent an upper GI endoscopy which showed a small hiatus hernia with severe
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oesophagitis complicated by deep linear ulcers in the distal oesophagus [Figure 1a].
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24 The oesophageal biopsy sample revealed mucosal ulceration with acute inflammatory cell
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26 exudate admixed with few giant epithelial cells, suggestive of herpes simplex virus (HSV)
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29 infection [Figure 1b]. Viral serology for HSV-1 was positive, while that for CMV and HIV
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31 were negative.
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He was commenced on acyclovir 800 mg three times daily.
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40 Within 48 hours, his hiccoughs subsided and he was discharged. He was advised to continue
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acyclovir for 7 days. At one month he was asymptomatic when reviewed in the clinic. Follow
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45 up endoscopy after 6 months showed complete resolution of oesophagitis, gastritis and
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47 duodenitis.
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52 Discussion
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56 Hiccough or Singultus is an involuntary, sudden contraction of the diaphragm and external
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intercostal muscles, producing a spasmodic inspiratory effort that is ceased abruptly by
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3 glottic closure, generating the distinctive ‘hic’ sound. Persistent hiccoughs last for more than
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6 48 hours and are generally not self-limiting.
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10 Multiple pharmacological measures failed to terminate the hiccoughs in our patient. Gastro-
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esophageal reflux disease has a strong association with hiccoughs [1-2], but our patient did
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15 not respond to intravenous rabeprazole and oral sucralfate. Upper gastro-intestinal endoscopy
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17 and subsequent biopsy confirmed HSV-1 oesophagitis. HSV-1, a common cause of
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esophagitis in immunocompromised individuals has also been reported in immunocompetent
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22 patients as ours [3].The hiccoughs subsided within 48 hours of initiation of anti-viral therapy
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24 with acyclovir.
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29 Mulhall et al, [4] have described a similar case in an immunocompetent individual presenting
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31 with persistent hiccoughs following coronary bypass surgery which resolved after treatment
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33 with acyclovir. The absence of classical symptoms of chest pain, odynophagia, dysphagia and
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fever was also unusual.
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40 Our case highlights the rare association of HSV oesophagitis in an immunocompetent patient
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causing persistent hiccoughs. To the best of our knowledge only one such case report exists
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45 in literature prior to ours.
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3 AUTHORS’ CONTRIBUTIONS
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8 A.P., and A.G. conceived and designed the project. A.P., and A.G. collected the data. A.P.,
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10 and A.G.analyzed and interpreted the data. A.P. drafted the manuscript. All authors read and
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approved the final manuscript.
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17 ETHICS AND PATIENT CONSENT
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22 The authors confirm that the ethics of this work have been reviewed by the ethics committee
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24 and an informed consent has been obtained from the patient.

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28 FUNDING
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The authors confirm that no source of funding was involved in this work.
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35 ACKNOWLEDGEMENT
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The authors want to acknowledge the kind peer review and guidance of Dr. Suvadip
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40 Chatterjee, MD, MRCP, FRCP, CCT- Gastroenterology (UK), Senior Consultant
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42 Gastroenterologist affiliated to Peerless Hospital, Kolkata, West Bengal, India.
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45 CONFLICT OF INTEREST
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49 None Declared.
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52 REFERENCES
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56 1.Koçkar C, İşler M, Cüre E, Şenol A, Baştürk A. Two Cases of Hiccups due to
57 Gastroesophageal Reflux Disease. Eur J Gen Med 2009;6:262-4.
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3 2.Rey E, Moreno C, Rodriguez-Artalejo F et al. Association between typical and atypical
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symptoms of gastroesophageal reflux: A population-based study in Spain. Gastroenterology
6 2003;124:A166
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8 3.Ramanathan J, Rammouni M, Baran J, Khatib R. Herpes simplex virus esophagitis in the
9 immunocompetent host: an overview. Am J Gastroenterol 2000;95:2171-6.
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4.Mulhall B, Nelson B, Rogers L, Wong R. Herpetic esophagitis and intractable hiccups
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(singultus) in an immunocompetent patient. Gastrointest Endosc 2003;57:796-7.
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17 FIGURE LEGENDS
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21 Figure 1a- Oesophagogastroduodenoscopy showing punched out ulcers in distal oesophagus
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24 inflammation.
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26 Figure 1b- Oesophageal biopsy showing inflammatory cells and epithelial giant cells typical
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of HSV-1 oesophagitis.
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Figure 1a- Oesophagogastroduodenoscopy showing punched out ulcers in distal oesophagus extending up to
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gastroesophageal junction, with fibrinous exudates and surrounding inflammation.

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Figure 1b- Oesophageal biopsy showing inflammatory cells and epithelial giant cells typical of HSV-1
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