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BACKGROUND: Hiccups (medically termed, "singultus"), when intractable, can cause significant medical consequences such as aspiration, malnutrition, and depression, leading to poor quality of life. Several case reports have shown that vagus nerve stimulator (VNS) implantation can help treat central idiopathic intractable hiccups. However, we present a contrary case of a patient who developed intractable singultus following VNS placement for medically refractory epilepsy. CASE PRESENTATION: We report a 71-year-old male patient with drug-resistant epilepsy who underwent VNS implantation and developed intractable hiccups shortly thereafter. The hiccups were severe and persistent, such that the patient developed a Mallory-Weiss tear, which required intensive care, invasive intubation and mechanical ventilation, and a prolonged rehabilitation course. Despite multiple therapies including phrenic nerve block and Nissen fundoplication, the patient's hiccups persisted and only stopped once the VNS was permanently deactivated. CONCLUSIONS: Little is known about the incidence of hiccups after VNS implantation. We present one case of hiccups as a direct consequence of VNS implantation. The clinical impact of this report is significant given the relative unfamiliarity of hiccups as an adverse effect of VNS implantation. Neurologists and epileptologists, who present VNS implantation as a surgical option for seizure control to their patients, should be aware of the possibility of singultus development and its significant physical and emotional ramifications.
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Epilepsia Resistente a Medicamentos , Soluço , Estimulação do Nervo Vago , Masculino , Humanos , Idoso , Soluço/etiologia , Soluço/terapia , Qualidade de Vida , Estimulação do Nervo Vago/efeitos adversos , Convulsões/complicações , Resultado do TratamentoRESUMO
How to cite this article: Hirolli D, Panda R, Baidya DK. Bygone Ether: Theriac to Obstinate Hiccups-Food for Thought! Indian J Crit Care Med 2022;26(7):884.
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In this topical review, we discuss the history of the area postrema syndrome, with special attention given to early studies aimed at identifying the area postrema and its function, possible early cases of the syndrome and its current relevance in neuroimmunology and demyelinating diseases. In 1896, Retzius named a structure in the posterior medulla oblongata as the area postrema. The work of Borison in the middle of the 20th century led to the elucidation of its function as a "vomiting center." The historical medical literature is filled with excellent examples that could be described as "area postrema syndrome." While severe and bilateral optic neuritis and transverse myelitis still constitute the classic components of neuromyelitis optica spectrum disorder (NMOSD), intractable vomiting and hiccups due to area postrema involvement is now recognized as essentially pathognomonic, indeed a shiny pearl in neuroimmunology and demyelinating diseases.
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Área Postrema/fisiopatologia , Soluço/fisiopatologia , Náusea/fisiopatologia , Neuromielite Óptica/fisiopatologia , Vômito/fisiopatologia , Soluço/etiologia , Soluço/história , História do Século XIX , História do Século XX , História do Século XXI , Humanos , Náusea/etiologia , Náusea/história , Neuromielite Óptica/complicações , Neuromielite Óptica/história , Síndrome , Vômito/etiologia , Vômito/históriaRESUMO
Gangrenous cholecystitis (GC) is a serious complication of acute cholecystitis that has been associated with increased morbidity. Patient with GC can present with a wide variety of non-specific clinical, laboratory, and imaging characteristics, making the diagnosis challenging. This disease requires emergent treatment, which is why a quick and reliable diagnosis is essential for the wellbeing of the patient. The authors herein present a case of GC in a patient whose initial complaint was intractable hiccups, and provide a thorough review of the literature of cases of GC with atypical presentations.
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Colecistite Aguda/diagnóstico , Colecistite Aguda/cirurgia , Vesícula Biliar/patologia , Vesícula Biliar/cirurgia , Idoso , Colecistectomia Laparoscópica/efeitos adversos , Colecistite Aguda/etiologia , Gangrena , Humanos , Masculino , Tomografia Computadorizada por Raios X , UltrassonografiaAssuntos
Soluço/etiologia , Neuromielite Óptica/complicações , Feminino , Humanos , Pessoa de Meia-IdadeRESUMO
Hiccups, a common and usually self-limiting condition, are caused by involuntary, spasmodic contractions of the diaphragm and intercostal muscles, followed by the sudden closure of the glottis. While most cases resolve spontaneously, persistent hiccups (lasting 48 hours to one month) and intractable hiccups (lasting more than one month) require medical attention. Intractable hiccups, although rare, can significantly impair a patient's quality of life. The etiology of intractable hiccups is diverse, but they are often associated with serious underlying medical conditions, such as severe renal dysfunction and uremia. We present the case of a 72-year-old male patient with stage IV chronic kidney disease (CKD) who developed intractable, violent hiccups following a mild COVID-19 infection. Despite treatment attempts with chlorpromazine and baclofen, the hiccups persisted for five months and only resolved after the initiation of hemodialysis. Interestingly, the patient's renal function deteriorated significantly during the period of hiccup persistence, suggesting a possible link between the hiccups and the progression of CKD, likely exacerbated by COVID-19. This case highlights the challenges of managing intractable hiccups in patients with advanced CKD and emphasizes the importance of addressing underlying metabolic derangements in such complex clinical scenarios. Moreover, it contributes to the growing evidence supporting the role of dialysis in resolving intractable hiccups associated with severe renal dysfunction.
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A 63-year-old man who presented to the hospital with altered mental status and decreased responsiveness was found to have severe symptomatic hyponatremia with a sodium level of 96 mmol/L and pneumonia. The patient was admitted to the medical intensive care unit for septic shock and acute severe hyponatremia. He was intubated for airway protection, and treated with 3% hypertonic saline bolus and antibiotics. After four days, sodium levels were corrected to 128 mmol/L, and the patient was extubated and downgraded to the medical floor. This case demonstrates one of the lowest recorded sodium lab values ever and the patient was successfully treated and discharged home with appropriate outpatient appointments.
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Hiccups, also known as singultus, are involuntary spasms of the diaphragm muscle followed by laryngeal closure involving a reflex arc. It is a relatively common phenomenon, usually transient and self-limiting. However, in medical settings, it could be much more serious and is often a sign of underlying pathology. When hiccups last for over 48 hours, they are referred to as persistent hiccups, and if they persist for more than a month, they are known as intractable hiccups. Current pharmacologic treatment of persistent or intractable hiccups mainly includes antidopaminergic drugs, which specifically antagonize the dopamine D2 receptor. Here, we present the case of a 54-year-old gentleman who was admitted under our care with a posterior circulation stroke specifically affecting the medulla. He was symptomatic with severe, persistent hiccups interfering with sleep and oral intake and unresponsive to all standard medications. After nearly two weeks, a trial of hydrocortisone was given, to which he responded dramatically. To the best of our knowledge, this is the only case of hiccups that has been successfully treated with hydrocortisone. The remarkable improvement seen in our patient when treated with hydrocortisone suggests hydrocortisone could be a useful agent in post-stroke hiccups that are unresponsive to traditional treatment for hiccups.
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Background Hiccups are a common physiologic reflex resulting from intermittent and involuntary spasmodic contraction of the diaphragm and intercostal muscles. While most cases are self-limited, lasting less than 48 hours, rare pathologies may result in prolonged symptoms. Hiccups can be disruptive and uncomfortable, leading many to seek management strategies using common home remedies. Few methods for terminating hiccups have been published in the scientific literature. We report the efficacy of the Hiccup relief using Active Prolonged Inspiration (HAPI) technique, which combines phrenic and vagal nerve stimulation with transient hypercapnia for hiccup relief. Methods Twenty patients with self-limited hiccups and one patient with prolonged hiccups were successful in eliminating hiccups using HAPI. In this method, patients are instructed to inspire maximally. Once at the peak of inspiration, they continue to attempt to inspire with an open glottis for a total of 30 seconds. This is followed by a slow expiration and resumption of normal respiration. Results In all cases, patients reported immediate hiccup relief. Conclusion These findings suggest the HAPI technique is a simple and viable method for hiccup relief. Further studies are needed to validate effectiveness.
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Hiccups are a minor nuisance for most people and usually resolve within minutes. However, for some, they can persist for years and result in severe symptoms and even death. This case report describes a patient found to have a mediastinal lipoma resulting in intractable hiccups. The discussion highlights the pathophysiology, etiologies, and treatments of hiccups.
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Isolated area postrema syndrome (APS) is a rare neurological presentation of, neuromyelitis optica spectrums disorder (NMOSD), recognizable by uncontrollable hiccups, nausea, or vomiting. When it occurs as the first presentation of NMOSD, it may present as a diagnostic challenge as the condition may be frequently attributed to gastrointestinal pathology, and the subsequent diagnostic delay may result in debilitating neurological sequelae such as optic neuritis or myelitis. We report such a case of isolated APS in a young woman who presented with a clinical picture of bouts of vomiting and intractable hiccups causing considerable distress and was finally diagnosed to be a case of seronegative NMOSD.
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Background: Intractable hiccups (IH) due to syringomyelia or syringomyelia/syringobulbia associated with Chiari type I malformations (CMI) are extremely rare. Here, we present two patients who presented with IH; one had a CMI with syringomyelia/syringobulbia, and the other, with CMI and syringomyelia. Case Description: The first patient was an 18-year-old female who presented with IH attributed to a holocord syrinx and syringobulbia involving the right dorsolateral medulla. The second patient was a 22-year-old female with a C3-5 syringomyelia. Both patients successfully underwent foramen magnum decompressions that improved their symptoms, while subsequent magnetic resonance studies confirmed shrinkage of their syringobulbia/syringomyelia cavities. Conclusion: IH was due to cervical syringomyelia/syringobulbia in one patient and cervical syringomyelia in the other; both were successfully managed with foramen magnum decompressions.
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Postoperative intractable hiccups slow patient recovery and generate multiple adverse effects, highlighting the importance of investigating the pathogenesis and terminating the hiccups in a timely manner. At present, medical and physical therapies account for the main treatments. We encountered a case in which postoperative intractable hiccups after biliary T-tube drainage removal ceased with the application of an ultrasound-guided block of the unilateral phrenic nerve and stellate ganglion. No complications developed, and the therapeutic effect was remarkable. To our knowledge, this approach has not been reported to date. Simultaneously blocking the phrenic nerve and stellate ganglion may be a treatment option for intractable hiccups.
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Efeitos Colaterais e Reações Adversas Relacionados a Medicamentos , Soluço , Humanos , Nervo Frênico/cirurgia , Soluço/etiologia , Soluço/terapia , Gânglio Estrelado/cirurgia , DrenagemRESUMO
Singultus is the sudden onset of erratic diaphragmatic and intercostal muscle contraction immediately followed by laryngeal closure. Pathophysiology involves afferent, central and efferent components. Bilateral phrenic nerve block was performed to a 46-year-old woman with a brain tumor with persistent hiccups, with initially positive response but later symptom recurrence. Bilateral pulsed radiofrequency of the phrenic nerve was performed guided by ultrasonography (US). In the follow-up, absence of hiccups was confirmed. The patient was discharged 24 h later. Persistent or untreatable singultus is an infrequent condition that should not be dismissed. This approach is a safe, accurate and effective therapeutic approach for patient's refractory to conservative treatment. Further studies are needed to establish safety and effectiveness of the treatment.
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Soluço , Tratamento por Radiofrequência Pulsada , Feminino , Humanos , Pessoa de Meia-Idade , Nervo Frênico/diagnóstico por imagem , Soluço/terapia , Soluço/tratamento farmacológico , Tratamento por Radiofrequência Pulsada/efeitos adversos , Ultrassonografia/efeitos adversos , Ultrassonografia de IntervençãoRESUMO
Intractable hiccups are a rare yet debilitating pathology with a broad differential and often indicate a more serious underlying pathology, which can range from neoplasms to structural abnormalities. In this case report, we present a 64-year-old male with seven months of intractable hiccups determined to be caused by eventration of the right hemidiaphragm. The patient was treated with baclofen to treat the hiccups pharmacologically. He was also prescribed voice therapy to establish rescue breathing techniques and reduce laryngospasm. Finally, he was referred to thoracic surgery for further evaluation and potential surgical intervention should his diaphragmatic eventration worsen or cause hypoxemia. To our knowledge, this is the first reported case of an association between diaphragmatic eventration and intractable hiccups. It is important to highlight this addition to the broad differential of intractable hiccups and to emphasize an interdisciplinary approach to workup and treatment of intractable hiccups.
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BACKGROUND: Intractable hiccups can last for more than 1-2 months and can occur, as shown in this case study, due to cervical nerve root compression. CASE DESCRIPTION: A 76-year-old male presented with intractable hiccups and neck pain of 7 months' duration. The patient underwent magnetic resonance imaging studies of the entire neuraxis. The only abnormality found was on the cervical magnetic resonance images that demonstrated left C4 nerve root compression due to the C3- C4 lateral/foraminal osteophyte. Following a left-sided C3-C4 laminoforaminotomy, the hiccups and the neck pain improved. CONCLUSION: A 76-year-old male presented with intractable hiccups and neck pain attributed to a left C3/C4 lateral/foraminal spondylotic ridge. Following a left C3-C4 laminoforaminotomy, the frequency of hiccup attacks remained the same, but their duration was markedly shortened to 30 s, while the neck pain improved.
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Acute optic neuritis (ON) is caused by variety of complex disorders that can be differentiated with the help of history, radiology, and serology. Identification of nonneurological symptoms that occur before the demyelinating event aids in timely diagnosis and prevention of further neurological attacks. We describe a case of unilateral ON with a history of intractable hiccups, nausea, and vomiting, wherein the possibility of area postrema syndrome (APS) was overlooked until the development of visual symptoms. APS recently identified as a hallmark of neuromyelitis optica spectrum disorder is a rare neurologic cause of gastrointestinal symptoms. This atypical presentation of APS results from autoantibodies directed against the aquaporin-4 rich sites, such as area postrema. This case brings to light the importance of eliciting history of intractable hiccups, nausea, and vomiting in a patient with ON. Despite being a commonly encountered symptom, it may rarely raise a suspicion for neuromyelitis optica.
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Hiccup is a common phenomenon experienced by almost everyone in life. Although the exact physiology of this phenomenon remains unknown, it is associated with multiple central and peripheral etiologic causes. Vocal fold granulomas are benign laryngeal lesions typically caused by iatrogenic trauma, voice misuse, or chronic irritation. We present, for the first time, an association between intractable hiccups and vocal fold granulomas with good response to acupuncture and voice therapy in a 62-year-old male patient. This is an important contribution to the literature as the first report describing the co-occurrence of these pathologies in the context of a patient with several treatment failures, including vagal nerve stimulator.
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INTRODUCTION: Intractable hiccups frequently result from an underlying pathology and can cause considerable illness in the patients. Initial remedies such as drinking cold water, induction of emesis, carotid sinus massage or Valsalva manoeuvre all seem to work by over stimulating the Vagus nerve. Pharmacotherapy with baclofen, gabapentin and other centrally and peripherally acting agents such as chlorpromazine and metoclopramide are reserved as second line treatment. Medical refractory cases even indulge in unconventional therapies such as hypnosis, massages and acupuncture. Surgical intervention, although undertaken very rarely, predominantly revolves around phrenic nerve crushing, blockade or pacing. A novel surgical strategy is emerging in the form of Vagus nerve stimulator (VNS) placement with three cases cited in literature to date with varying degrees of success. Here the authors report a case of VNS placement for intractable hiccups with partial success, in accordance with SCARE-2018 guidelines. PRESENTATION OF THE CASE: An 85-year-old gentleman with a 9-year history of intractable hiccups secondary to pneumonia came to our hospital. The hiccups were symptomatic causing anorexia, insomnia, irritability, depression, exhaustion, muscle wasting and weight loss. The patient underwent countless medical evaluations. All examinations and investigations yielded normal results. The patient underwent aggressive pharmacotherapy, home remedies and unconventional therapies for intractable hiccups but to no avail. He also underwent left phrenic nerve blocking and resection without therapeutic success. The patient presented to our hospital and decision for VNS insertion was taken for compassionate reasons considering patient morbidity. The patient demonstrated significant improvement in his symptoms following VNS insertion. DISCUSSION: A temporary hiccup is an occasional happening experienced by everyone. However, intractable hiccups are associated with significant morbidity and often mortality. Several medical, pharmacological, surgical and novel treatment options are available for intractable hiccups. CONCLUSION: VNS insertion is a novel surgical option for the treatment of intractable hiccups.
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The possibilities of coronavirus disease 2019 (COVID-19) to present with atypical manifestations have reported. Information of COVID-19 atypical signs and symptoms is still emerging globally. One of these presentations is persistent hiccups. One of the hypotheses is that COVID-19 has been linked to several neurological manifestations and effects. Some observations noticed phrenic nerve paralysis after COVID-19 infection leading to pulmonary failure. We report one case of COVID-19-positive patient where he presented with persistent hiccups. Many predisposing factors might lead to the development of hiccups in COVID-19 infection such as a history of smoking, phrenic and vagus nerve damage or irritation, high inflammatory markers, lower lobe pneumonia, ground-glass-like appearance on x-rays. We hypothesize that hiccups are the first sign of serious deterioration of patients with COVID-19 and such patients are at high risk of developing kidney injury and intubation.