Letter to the Editor
Diaphragmatic flutter: A case report and literature review
Aleteo diafragmático. Descripción de caso y revisión de la literatura
a Servicio de Neurología, Clínica Comfamiliar Risaralda, Pereira, Colombia
b Servicio de Cirugía, Clínica Comfamiliar Risaralda, Pereira, Colombia
c Servicio de Medicina Interna, Universidad Tecnológica de Pereira, Pereira, Colombia
d Servicio de Radiología, Universidad Tecnológica de Pereira, Pereira, Colombia
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(A) Haematoxylin-eosin stain. Eyelid infiltration of lymphoid tumour showing nodular diffuse growth, made up of small monomorphic cells with no mitotic figures. (B-D) Immunohistochemical profile: (B) CD20+, (C) IgM+, (D) kappa+ chains.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "R. Cuenca Hernández, J.A. Guzman de Villoria Lebiedziejewski, E. Roa Martínez, J. Menarguez Diaz" "autores" => array:4 [ 0 => array:2 [ "nombre" => "R." "apellidos" => "Cuenca Hernández" ] 1 => array:2 [ "nombre" => "J.A." "apellidos" => "Guzman de Villoria Lebiedziejewski" ] 2 => array:2 [ "nombre" => "E." "apellidos" => "Roa Martínez" ] 3 => array:2 [ "nombre" => "J." 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Ramírez, M. Gonzales, J.A. Hoyos, L. Grisales" "autores" => array:4 [ 0 => array:3 [ "nombre" => "J.D." "apellidos" => "Ramírez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "M." "apellidos" => "Gonzales" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:4 [ "nombre" => "J.A." "apellidos" => "Hoyos" "email" => array:1 [ 0 => "juanhope9@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 3 => array:3 [ "nombre" => "L." "apellidos" => "Grisales" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Servicio de Neurología, Clínica Comfamiliar Risaralda, Pereira, Colombia" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Cirugía, Clínica Comfamiliar Risaralda, Pereira, Colombia" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Medicina Interna, Universidad Tecnológica de Pereira, Pereira, Colombia" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Servicio de Radiología, Universidad Tecnológica de Pereira, Pereira, Colombia" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Aleteo diafragmático. Descripción de caso y revisión de la literatura" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 672 "Ancho" => 1400 "Tamanyo" => 112468 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Gadolinium-enhanced brain MRI sequences yielding normal results: (A) axial T2-weighted sequence, (B) sagittal T2-weighted FLAIR sequence.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Diaphragmatic flutter is a rare condition characterised by rhythmic involuntary contractions of the diaphragm and other respiratory muscles innervated by cervical nerve roots. It has been described in people of all ages and both sexes. Its clinical presentation varies greatly and clinical symptoms are quite unusual, which may result in late diagnoses and ineffective treatments. Symptoms include thoracic or abdominal pain and dyskinetic movements in the thorax and abdominal wall. Idiopathic diaphragmatic flutter is the most common presentation, although it has also been described in association with other clinical and surgical entities. Diagnosis is based on strong clinical suspicion in addition to fluoroscopy or electrophysiological studies showing diaphragm movement. No clinical studies have addressed treatment options for this disorder. Current treatment relies on expert opinion and case reports and may be pharmacological or non-pharmacological.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a 17-year-old adolescent who experienced sudden onset of pain in the right lumbar region and right iliac fossa which radiated to the back and lasted 45 days. Pain was associated with involuntary movements of the trunk. She was referred to the emergency department by the neurology department due to exacerbation of pain, presence of predominantly right-sided rhythmic involuntary movements of the trunk, nausea, vomiting, and a burning feeling in the right facial and brachial regions and right hemithorax. Our patient had previously been evaluated by several departments at an outpatient clinic, including the neurology, internal medicine, psychiatry, psychology, and rehabilitation and physical medicine departments. She had also undergone neural therapy and physiotherapy, but symptoms did not improve. Findings from contrast and non-contrast MRI studies of the cervical and thoracic spine were normal (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). She was treated with fluoxetine 20<span class="elsevierStyleHsp" style=""></span>mg/day and with clonazepam drops; the latter improved her symptoms slightly. Our patient had a history of recurrent urinary infections and mixed anxiety-depressive disorder. She was admitted to the emergency department. Mucosa was hydrated and pink, and vital signs were as follows: blood pressure, 110/60<span class="elsevierStyleHsp" style=""></span>mmHg; heart rate, 78<span class="elsevierStyleHsp" style=""></span>bpm; respiratory rate, 17<span class="elsevierStyleHsp" style=""></span>breaths/min; oxygen saturation, 96%; and no fever. Neurological examination showed that the patient was alert and her higher mental functions were intact. Cranial nerves II to XII were unaffected. She presented deep tendon reflexes graded 2+/4+, preserved muscle strength, no Babinski sign, and a burning feeling on the right facial and brachial regions and right hemithorax. Involuntary rhythmic movements of the abdominal muscles were not exacerbated by touch. All other results from the physical examination were normal. According to the mental examination, the patient showed anxiety; modulated, coherent, and resonant emotions; and logical thoughts, including concerns about her state of health, with no other relevant findings. She was admitted to hospital for additional tests including electromyography and nerve conduction studies of abdominal, paraspinal, thoracic, and cervical muscles. Results showed a myoclonic pattern of involuntary contractions in thoracic dermatomes (T4 to T12). The patient was sedated before undergoing a gadolinium contrast MRI scan of the cervical and thoracic spine, which yielded normal results (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). We also performed a complete blood count, HIV and syphilis serology (VDRL), a partial urine test, a lumbar puncture, and tests for creatinine, vitamin B<span class="elsevierStyleInf">12</span> and folic acid, erythrocyte sedimentation rate, C-reactive protein, serum electrolytes (potassium, sodium, magnesium, chloride), thyroid stimulating hormone, free T4, antinuclear antibodies, anti-dsDNA antibodies, extractable nuclear antigens, C3 and C4 complements, blood levels of lead and arsenic, and arterial gas. All tests yielded normal results. Since we suspected diaphragmatic flutter, we evaluated diaphragm movement with fluoroscopy. The study showed repetitive movements of the diaphragm (120<span class="elsevierStyleHsp" style=""></span>movements per minute) and normal diaphragm mobility with inspiration and expiration; these findings are consistent with diaphragmatic flutter. The patient was treated with phenytoin and gabapentin, which provided partial pain relief but failed to improve dyskinetic movements. This slight drug-related improvement led us to perform an ultrasound- and fluoroscopy-guided right phrenic nerve block with bupivacaine. During the procedure, diaphragmatic flutter decreased until disappearing on the right side. Although symptoms improved significantly, they reappeared about 6<span class="elsevierStyleHsp" style=""></span>hours later. Since clinical symptoms persisted, we decided to treat the patient by crushing and clipping the right phrenic nerve using video-assisted thoracoscopic surgery. This procedure achieved substantial and lasting improvement of symptoms, resulting in the patient being discharged.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Diaphragmatic flutter is an infrequent disorder characterised by high-frequency rhythmic involuntary contractions of the diaphragm and other respiratory muscles innervated by cervical nerve roots.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> Antonie van Leeuwenhoek is thought to have provided the first description in 1723 after experiencing the disorder himself. In one of his studies, <span class="elsevierStyleItalic">De structura diaphragmatis: epistola domini Antonii van Leeuwenhoek, R. S. S. ad Societatem Regiam</span>, the author explains that he had palpitations in the thorax. Although his doctor suggested that they were of cardiac origin, van Leeuwenhoek realised that his heart rate did not change when symptoms appeared, and thus concluded that it was the diaphragm and not the heart that caused the palpitations.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The descriptions in the literature are based on case reports, and the largest series are those published by Rigatto and DeMedeiros, and Graber and Sinclair-Smith.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Diaphragmatic flutter has also been called Leeuwenhoek disease, diaphragmatic myoclonus, respiratory myoclonus, belly dancer's syndrome, and belly dancer's dyskinesia.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">1,4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">It has been described in both children and adults.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a> Clinical symptoms are highly variable and the syndrome is very rare, which results in late diagnosis. According to the literature, it can take up to 18 years to diagnose this disorder. It may present with pain, but pain cannot be used to locate damage since it is perceived in the thorax, epigastrium, and lumbar region. In fact, this entity may even be confused with ischaemic heart disease due to the pain location, especially when pain affects the left hemidiaphragm: in these cases, pain radiates to the left arm and patients experience dyspnoea. Abdominal movements are also frequent, which has given rise to the term belly dancer's syndrome. These movements may appear at any location, especially in the upper quadrants, and they fluctuate throughout the day. No precipitating factors are known and contractions may persist during sleep according to some studies. Other reported symptoms are inspiratory stridor, epigastric pulsations, palpitations, dyspnoea, nausea, and vomiting. Pain has sometimes led patients to undergo surgery (appendectomy, cholecystectomy, among others) since they attributed the pain to surgical conditions.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">1–4,6</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">A number of causes may explain onset of diaphragmatic flutter. It has been described in conjunction with central and peripheral nervous system disorders, such as encephalitis and phrenic nerve irritation, pleural disorders (pleurisy), mediastinal disorders (adenopathies), intra-abdominal disorders (peritonitis), heart diseases (rheumatic fever), cardiac and thoracic surgeries (myocardial revascularisation), idiopathic cervical spine trauma, and lung diseases (including the case of a paediatric patient presenting diaphragmatic flutter after an upper respiratory tract infection). Other studies have described cases of diaphragmatic flutter following osmotic demyelination syndrome and secondary to galantamine and clebopride use.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">6–8</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Diagnosis can be based on fluoroscopy since this technique shows real-time diaphragm movement and allows doctors to evaluate its amplitude and rate.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> Electrophysiological studies can also be used for this purpose. Needle electromyography is particularly useful: needle electrodes should be inserted into the diaphragm. Surface electrophysiological studies are less accurate since thoracic wall movements may interfere. Electrophysiological studies have shown that diaphragmatic flutter can maintain adequate ventilation despite suppressing normal breathing.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Patients are usually diagnosed with some type of psychiatric disorder before a correct diagnosis is made and will therefore have been treated with multiple drugs, such as valproic acid, haloperidol, pimozide, and clonidine.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a> Our patient had been treated with clonazepam and fluoxetine. Treatment is based on the descriptions given in different case series. Some articles recommend pharmacological treatment with phenytoin<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> and carbamazepine,<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> while others support such invasive procedures as phrenic nerve block at the C4 level with bupivacaine or methylprednisolone infiltrations. The latter procedure is performed when the disorder is believed to have an inflammatory origin, and it will temporarily resolve diaphragmatic contractions.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a> In our patient, symptoms improved for 6<span class="elsevierStyleHsp" style=""></span>hours after phrenic nerve infiltration with bupivacaine. Some authors suggest a surgical procedure to crush the phrenic nerve, after which symptom improvement has been reported as lasting up to 6 months, or the time it takes the nerve to regenerate. Several studies report positive outcomes of phrenic nerve crush at the C4 level.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">3,6</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Ramírez JD, Gonzales M, Hoyos JA, Grisales L. Aleteo diafragmático. Descripción de caso y revisión de la literatura. Neurología. 2015;30:249–251.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 932 "Ancho" => 1400 "Tamanyo" => 112494 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Gadolinium-enhanced sagittal T2-weighted MRI sequences of the cervical (A) and thoracic (B) spine, with normal results.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 672 "Ancho" => 1400 "Tamanyo" => 112468 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Gadolinium-enhanced brain MRI sequences yielding normal results: (A) axial T2-weighted sequence, (B) sagittal T2-weighted FLAIR sequence.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Diaphragmatic flutter after spinal cord trauma in a child" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "J. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 613 | 25 | 638 |
| 2024 September | 1238 | 40 | 1278 |
| 2024 August | 1123 | 41 | 1164 |
| 2024 July | 1120 | 37 | 1157 |
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| 2023 December | 1278 | 17 | 1295 |
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| 2023 September | 1253 | 42 | 1295 |
| 2023 August | 1222 | 60 | 1282 |
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| 2023 April | 1043 | 39 | 1082 |
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| 2017 January | 127 | 26 | 153 |
| 2016 December | 103 | 30 | 133 |
| 2016 November | 144 | 40 | 184 |
| 2016 October | 128 | 53 | 181 |
| 2016 September | 184 | 61 | 245 |
| 2016 August | 140 | 48 | 188 |
| 2016 July | 69 | 14 | 83 |
| 2016 June | 118 | 33 | 151 |
| 2016 May | 84 | 32 | 116 |
| 2016 April | 81 | 45 | 126 |
| 2016 March | 109 | 43 | 152 |
| 2016 February | 104 | 33 | 137 |
| 2016 January | 86 | 30 | 116 |
| 2015 December | 85 | 29 | 114 |
| 2015 November | 101 | 31 | 132 |
| 2015 October | 101 | 28 | 129 |
| 2015 September | 97 | 20 | 117 |
| 2015 August | 91 | 26 | 117 |
| 2015 July | 62 | 9 | 71 |
| 2015 June | 56 | 15 | 71 |
| 2015 May | 56 | 34 | 90 |
| 2015 April | 11 | 5 | 16 |
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