Central neurogenic hyperventilation in a patient with post-vaccination acute disseminated encephalomyelitis

Monterrubio-Villar, Jesús; Mora-Encinas, José Pedro; Jiménez-Delgado, Juan Diego; Almaraz-Velarde, Rocío

doi:10.1016/j.medcle.2016.03.021

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Magnetic resonance imaging a month later (D–F): extensive infiltration and oedema of the white matter in the cerebral hemispheres and cerebellum (D, E), and non-communicating obstructive hydrocephalus (F)." ] ] ] "textoCompleto" => "Central neurogenic hyperventilation (CNH) is a rare disorder, described in patients in a coma or without a deteriorated level of consciousness caused by the progressive tumoral infiltration of the brain stem, mainly secondary to lymphoma or glioma. After conducting a literature search in PubMed without time restrictions, with the MeSH terms “hyperventilation”, “encephalomyelitis, acute disseminated” and “central nervous system diseases”, we describe the first case of CNH secondary to acute disseminated encephalomyelitis after trivalent flu vaccination.A male, 63 years old with no personal medical history who was admitted to our hospital with dysarthria, dysphagia, peripheral facial weakness and gait disturbance without fever, headache or prodromal signs of infection. The symptoms had gradually appeared and had started approximately two weeks after being vaccinated with a trivalent adjuvant flu vaccine a month before. On admission, the examination showed neither impairment of consciousness nor visual field or ocular motility disorder. The patient had severe dysarthria with palatal and tongue movement restrictions, intention tremor, abnormal heel–knee test and proximal weakness of the left arm. The tendon reflexes were hyperactive, without clonus, while both proprioception and vibratory sensation were preserved.Lab tests, chest X-ray, ECG and Chest-Abdomen CT scan were normal. CSF analysis showed: 11cells/mm3, 28red blood cells/mm3, proteins 61mg/dl and glucose 69mg/dl. There were no neoplastic cells or oligoclonal bands or intrathecal IgG synthesis. The PCR in CSF for herpes simplex, varicella zoster, cytomegalovirus, Epstein–Barr virus, JC and tuberculosis were negative as well as serology for measles, parvovirus B19, Brucella, Toxoplasma, Borrelia, Rickettsia and VDRL. Levels of angiotensin converting enzyme in serum and CSF were normal, and tumour markers and antinuclear antibodies were negative. Cranial MRI showed bilateral focal lesions in the white matter which were hyperintense on T2, with minimal enhancement after contrast (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A–C). During his hospital stay he developed constipation and urinary retention. Taking into account his medical history, lab tests and MRI results, he was diagnosed with post-vaccination acute disseminated encephalomyelitis and treatment was started with a high dose methylprednisolone.<elsevierMultimedia ident="fig0005"></elsevierMultimedia>Due to the emergence of a significant tachypnea he was admitted to intensive care. The arterial blood gas analysis showed the following results: 7.486 pH, PaO2 126mm Hg, PaCO2 12mm Hg, HCO3 14.6mmol/l. The blood lactate was 4.7mmol/l (VN<2mmol/l). Since the patient had hyperpnoea and difficulty to protect the airway due to involvement of the lower cranial nerves, he was intubated and sedated with remifentanil and midazolam, which decreased the respiratory alkalosis. Repeated attempts to disconnect the respirator failed due to severe hyperventilation, ruling out concomitant infection, lung disease or heart disease, therefore intravenous treatment was started with immunoglobulins for 5 days and 5 plasmapheresis sessions were performed. Despite treatment the patient progressively deteriorated and went into a coma. In a second MRI, performed one month after the first, involvement of deep grey substance was observed with extensive oedema of the white matter of both cerebral hemispheres and cerebellum, with brainstem compression and secondary triventricular hydrocephalus (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D–F). With his family's consent, it was decided to limit the life-sustaining treatments and he died shortly after in our unit.The CNH was first described in 1959 by Plum and Swanson.<a class="elsevierStyleCrossRef" href="#bib0055">1</a> It is characterized by a sustained hyperventilation that persists during sleep, a marked decrease in PaCO2, an elevated PaO2 and a high blood pH, together with the absence of cardiac or respiratory diseases or metabolic-drug causes. Neoplasms, mainly the primary CNS lymphoma, are the most common cause of CNH, particularly in tumours infiltrating the protuberance.<a class="elsevierStyleCrossRef" href="#bib0060">2</a> It has also been described in association with other locations, such as the high cervical cord or the bilateral medial thalamus,<a class="elsevierStyleCrossRefs" href="#bib0065">3,4</a> and with non-neoplastic diseases. The underlying pathophysiological mechanism of CNH is not well known, both biochemical and structural factors have been proposed for its genesis.<a class="elsevierStyleCrossRefs" href="#bib0075">5,6</a> There is no effective therapy for this entity, but the intensive treatment of the primary tumour or disease can induce a CNH remission.<a class="elsevierStyleCrossRefs" href="#bib0085">7,8</a> In a patient with multiple sclerosis CNH was successfully controlled by plasmapheresis.<a class="elsevierStyleCrossRef" href="#bib0080">6</a> Symptomatic treatment involves the repeated or continuous intravenous administration of midazolam, propofol and opioids such as morphine or fentanyl.<a class="elsevierStyleCrossRefs" href="#bib0075">5,8</a> Also, the oral administration of opioids may be useful.<a class="elsevierStyleCrossRef" href="#bib0095">9</a> Some patients with severe CNH may require mechanical ventilation to prevent respiratory distress, and even the use of neuromuscular blocking agents.The CNH forecast is ominous, with a survival of only a few months in most of the reported cases. In our patient, the CNH was refractory to treatment of the primary disease, consistent with increased severity and extent of brain and brain stem lesions. Although this adverse reaction to the flu vaccine was not communicated to the pharmacovigilance service, its causation was considered at least possible using the Karch and Lasagna algorithm.<a class="elsevierStyleCrossRef" href="#bib0100">10</a>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "Please cite this article as: Monterrubio-Villar J, Mora-Encinas JP, Jiménez-Delgado JD, Almaraz-Velarde R. Hiperventilación central neurogénica en un paciente con encefalomielitis aguda diseminada posvacunal. Med Clin (Barc). 2015;145:e23–e24." ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1343 "Ancho" => 1798 "Tamanyo" => 317382 ] ] "descripcion" => array:1 [ "en" => "Initial magnetic resonance imaging (A–C): bilateral focal lesions in the white matter (A, B) without hydrocephalus (C). Magnetic resonance imaging a month later (D–F): extensive infiltration and oedema of the white matter in the cerebral hemispheres and cerebellum (D, E), and non-communicating obstructive hydrocephalus (F)." ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Central neurogenic hyperventilation in man" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "F. Plum" 1 => "A.G. 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ISSN: 2387-0206

Launched in 1943, Medicina Clínica is a fortnightly journal aimed at the promotion of clinical research and practice among internal medicine and other specialists. The key characteristics of Medicina Clínica are the scientific and methodological rigor of its manuscripts, the topicality of its contents, and, especially, its practical focus with highly useful information for clinical practice. Medicina Clínica is predominantly interested in publishing original research manuscripts, which are rigorously selected according to their quality, originality, and interest, and also in continued medical education-oriented manuscripts, which are commissioned by the journal to relevant authors (Editorials, Reviews, and Diagnosis and Treatment). These manuscripts contain updated topics with a major clinical or conceptual relevance in modern medicine. The journal adheres to the standards of academic research publications in all aspects including peer-review and ethical principles. Medicina Clínica is included in the General and Internal Medicine category of Thomson Reuters.

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Impact factor

The Impact Factor measures the average number of citations received in a particular year by papers published in the journal during the two preceding years.

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Impact factor 2023

2.6

Citescore

CiteScore measures average citations received per document published.

Citescore 2023

3.1

SJR

SRJ is a prestige metric based on the idea that not all citations are the same. SJR uses a similar algorithm as the Google page rank; it provides a quantitative and qualitative measure of the journal's impact.

SJR 2023

0.371

SNIP

SNIP measures contextual citation impact by wighting citations based on the total number of citations in a subject field.

SNIP 2023

0.511

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Vol. 145. Issue 10.

Pages e23-e24 (November 2015)

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