Dear Editor, Herpes zoster (HZ), commonly known as shingles, is an infection caused by the reactivation of varicella-zoster virus that remains dormant in the sensory ganglion following the primary infection of varicella (chicken pox). The typical clinical presentation of herpes zoster includes a painful vesicular rash along the sensory nerve distribution. However, there have been rare cases where atypical manifestations occur, posing a diagnostic challenge for clinicians. Our patient was a 44-year-old male who developed persistent hiccups as the primary symptom of herpes zoster. The patient initially complained of sudden onset of intractable hiccups of five days duration. He also reported mild right-sided non-radiating chest pain and heartburn simultaneously that were partially relieved by antacids. There was no other significant history. His symptoms prompted him to visit a hospital where he was evaluated. All the baseline investigations including electrocardiogram (ECG) were within normal limits. He was diagnosed as a case of intractable hiccups. However, five days after the onset of hiccups, he developed a characteristic painful vesicular rash on the right side of the chest and back in a band-like distribution. On examination, he was afebrile, and his other vital signs were within normal limits. Frequent hiccups were noticed throughout the examination. Numerous vesiculobullous lesions on an erythematous base were seen with a distribution that precisely followed the right thoracic-5 and 6 dermatomes [Figure 1]. There was no lymphadenopathy. The remainder of the physical examination, including that of the abdomen, was unremarkable. A Tzanck smear cytology from a vesicle showed multinucleated giant cells. On the basis of clinical findings, he was diagnosed as a case of HZ. He was prescribed tablet valacyclovir 1 gm thrice daily and tablet baclofen 10 mg thrice daily. There was significant improvement in the lesions and his hiccups resolved within the next two days.Figure 1: (a and b) Numerous vesiculobullous lesions on an erythematous base were seen with a distribution that precisely followed the right thoracic-5 and 6 dermatomesHiccups typically considered benign and a self-limiting condition are rarely associated with herpes zoster infection.[1] Herpes is caused by the reactivation of the latent varicella-zoster virus and commonly presents as a painful dermatomal rash.[1,2] The prodromal symptoms include pain, burning, and tingling, which is followed by the eruption of vesicles. If left untreated it can lead to complications such as post-herpetic neuralgia, paralysis, and encephalitis.[2] The sudden closure of the glottis results in involuntary spasmodic contraction of the diaphragm. The reflex pathways then mediate hiccups. The afferent limbs of the reflex mediating hiccups consist of the vagus and phrenic nerves, the pharyngeal plexus (C2–C4), and the sympathetic chain (T6–T12), while the efferent pathway consists of the phrenic motor nerve (C3–C5). The exact role of the central nervous system is still not fully understood. Hiccups can result from irritation at any point along this reflex route.[1] Because T5–T6 is the dermatome affected by HZ in this case, it is likely that the afferent limb was affected by the virus with subsequent nerve irritation resulting in stimulation of the diaphragm. It is postulated that the phrenic nerve may become inflamed and irritated as a result of the viral infection causing persistent hiccups.[3] An elevated hemi-diaphragm suggestive of diaphragmatic paralysis can be seen in chest radiography while a paradoxical movement of the diaphragm during inspiration in fluoroscopy suggests a phrenic nerve irritation and damage.[4] Our case did not have such findings in the chest x-ray. Diagnosis is clinical by assessing the presence of the identifiable rash or any indications of a past occurrence of rash (i.e. hyperpigmented scarring).[5] Disseminated zoster with visceral organ involvement and zoster sine herpete, two rare uncommon forms of HZ are more frequently observed in immunocompromised patients, and exemplify extreme manifestation of the virus.[3] Zoster sine herpete presents with severe dermatomal pain without the typical cutaneous eruption and sporadic microbiologic zoster findings. About 10% of immunocompromised people have visceral involvement from the disseminated form of HZ compared with 2% of patients who are immunocompetent. Meningoencephalitis, pneumonitis, hepatitis, and pericarditis have all been recorded as complications of HZ.[4] In conclusion, this case demonstrates a rare occurrence of hiccups as the primary manifestation of herpes zoster and highlights the importance of considering this diagnosis, even in the absence of typical clinical presentation to help prevent delays in diagnosis and potential complications associated with herpes zoster. Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given consent for images and other clinical information to be reported in the journal. The patient understands that names and initials will not be published and due efforts will be made to conceal patient identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.
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Das et al. (2024) studied this question.
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