A case of atopic cough with aphonia showed a prominent response to a histamine H1 receptor antagonist.

A 33-year-old woman admitted to our hospital for further examination of severe non-productive cough lasting for about two months. Her symptom did not ameliorate by treatments including long acting ß2 agonists. She had a medical history of drug allergy to non-steroidal anti-inflammatory drugs. At the initial visit, she could not speak at all and communicated with us in writing. Chest auscultation revealed no wheezes, rhonchi and other crackles. Laboratory findings showed a mild eosinophilia with normal total and specific serum immunoglobulin E. The results of an electrocardiogram, a chest X-ray and a chest CT were unremarkable. A fractional exhaled nitric oxide value was within normal limit. Based on these observations, a diagnosis of atopic cough (AC) was suspected, and we started treatment with a histamine H1 receptor antagonist (H1-RA). She had become able to speak again in association with complete disappearance of cough by eight-weeks after treatment initiation, and her symptoms did not recur even after cessation of treatment. By the confirmation of remarkable clinical improvement in response to a H1-RA, a diagnosis of AC was made. To the best of our knowledge, this is the first report of an AC patient who presented severe cough with aphonia. J. Med. Invest. 70 : 281-284, February, 2023.

CoblationTM-assisted excision of suprastomal granulation tissue.

BACKGROUND: Suprastomal granulation tissue is a common complication of long-term tracheostomy. It may be associated with bleeding, aphonia, airway obstruction and delayed decannulation. METHODS: This study describes the experience of a tertiary paediatric medical centre with CoblationTM-assisted suprastomal granulation tissue excision. RESULTS: Thirteen children (mean age, 5.7 years) who underwent the procedure from 2013 to 2019 because of delayed decannulation or aphonia were included. Lumen obstruction ranged from 50 to 90 per cent, with a mean of 68.8 per cent. After the procedure, decannulation was successfully performed in 7 patients, and voice quality improved in 10 patients. There were no peri- or post-operative complications. CONCLUSION: This is the largest series to date that describes Coblation used for the treatment of suprastomal granuloma. Coblation has advantages of high precision, relatively low temperature (thereby avoiding thermal injury to adjacent tissue), haemostatic resection and feasibility for use for even large granulomas. The promising results should prompt further studies in larger samples.

The clinical syndrome of dystonia with anarthria/aphonia.

OBJECTIVES: In dystonia the formulation of a clinical syndrome is paramount to refine the list of etiologies. We here describe the rare association of dystonia with anarthria/aphonia, by examining a large cohort of patients, to provide a narrow field of underlying conditions and a practical algorithmic approach to reach diagnosis. METHODS: We retrospectively reviewed cases, which were evaluated between 2005 and 2014, to identify those with dystonia combined with marked anarthria and/or aphonia. We reviewed demographic information, clinical characteristics, as well as clinico-genetic investigations. We evaluated video material where available. RESULTS: From 860 cases with dystonia as the predominant motor feature, we identified 32 cases (3.7%) with anarthria/aphonia. Age at neurological symptom onset was variable, but the majority of cases (n = 20) developed symptoms within their first eight years of life. A conclusive diagnosis was reached in 27 cases. Monoamine neurotransmitter disorders, neurodegeneration with brain iron accumulation syndromes, hypomyelination with atrophy of the basal ganglia and cerebellum, and syndromes with inborn errors of metabolism were the most common diagnoses. Brain MRI was crucial for reaching a diagnosis by examining the structural integrity of the basal ganglia, the cerebral cortex, brain myelination and whether there was abnormal metal deposition. Pathophysiological mechanisms underlying anarthria/aphonia included dystonia, corticobulbar involvement, apraxia and abnormalities of brain development. CONCLUSIONS: The spectrum of conditions that may present with the syndrome of dystonia with anarthria/aphonia is broad. Various causes may account for the profound speech disturbance. A practical brain MRI-based algorithm is provided to aid the diagnostic procedure.

Dystonia with aphonia, slow horizontal saccades, epilepsy and photic myoclonus: a novel syndrome?

BACKGROUND: Dystonia with anarthria and/or aphonia is a rare syndromic association. Here we present two cases with slowly progressive, severe generalized dystonia and aphonia, slow horizontal saccades, epilepsy and photic myoclonus. METHODS: Detailed clinical data were collected over two decades in the female (index) patient and for nine years in her similarly affected son. Sanger sequencing followed by exome sequencing was performed. RESULTS: Both patients had leg onset generalized dystonia with gradual rostral spread including prominent facial and oro-mandibular involvement. The index patient was anarthric, her son aphonic. Both had saccadic slowing, more marked for the horizontal plane, and subclinical epileptic activity. The index patient also had photic myoclonus and a combined axonal and demyelinating neuropathy. Known genetic causes of similar syndromes were not identified. CONCLUSION: These cases with caudo-rostrally spreading generalized dystonia with prominent facial and oro-mandibular involvement, severe speech impairment, marked slowing of horizontal saccades, and photic myoclonus likely represent a novel entity.

Membrana laríngea congénita / Congenital laryngeal membrane

La membrana laríngea congénita es una malformación infrecuente de las vías aéreas. La clínica que produce depende del grado de obstrucción que produzca en dichas vías. Debe tenerse en consideración al realizar el diagnóstico diferencial de la dificultad respiratoria del recién nacido. El tratamiento dependerá asimismo del grado de obstrucción de la vía respiratoria. Se describe el caso de una lactante de dos semanas de vida que presenta afonía desde el nacimiento y dificultad respiratoria en el curso de infecciones de las vías aéreas superiores. Mediante laringoscopia directa, se realiza el diagnóstico de membrana laríngea congénita. El tratamiento realizado consiste en varias intervenciones de vaporización con láser de dióxido de carbono (AU)

Congenital laryngeal membrane is a rare malformation of the airways. The symptoms depend on the degree of obstruction that triggers in these pathways. It must be taken into consideration in the differential diagnosis of the newborn respiratory distress. The treatment depends on the degree of airway obstruction. We report the case of an infant 2 weeks old with aphonia from birth and respiratory distress with upper airways infections. She is diagnosed of congenital laryngeal membrane by direct laryngoscopy. The treatment is performed with lasser (AU)

Effect of pregnancy on the speaking voice.

The study aims to investigate the vocal symptoms and acoustic changes in pregnant women pre- and postpartum in comparison to the controls. A total of 25 pregnant women who presented for delivery were enrolled in this study. Twenty-one nonpregnant women were matched as controls. Vocal symptoms such as hoarseness, vocal fatigue, and aphonia were assessed. Acoustic analysis included fundamental frequency (F(0)), habitual pitch, relative average perturbation (RAP), shimmer, noise-to-harmony ratio (NHR), and maximum phonation time (MPT). There were no significant differences in the incidence of vocal symptoms in pregnant women versus controls. However, vocal fatigue was more prevalent in the pregnant group. With respect to the acoustic parameters, there was a significant decrease in the MPT at term. The rest of the variables were comparable. Postpartum, the MPT significantly increased and there was an increase in F(0) and a significant decrease in the voice turbulence index (VTI). Pregnant women have more vocal fatigue and a reduction in MPT compared to the controls. Immediately after delivery, there is a significant increase in MPT.

Changes in speech and language development of a young child after decannulation.

UNLABELLED: This report reviews the speech and language development of a child who, as result of complete subglottic stenosis, was aphonic from birth until 2 years and 11 months of age at which time laryngotracheal reconstruction provided normal respiration. The boy had congenital subglottic stenosis requiring neonatal tracheostomy. The congenital subglottic stenosis progressed to complete subglottic stenosis during the neonatal period. The child's speech and language development was monitored for a 24-week period following airway reconstruction. LEARNING OUTCOMES: The reader will learn about and be able to describe: (1) the speech and language development of a child who was aphonic from birth until 35 months of age, (2) the effects of surgical repair of a tracheotomy on the child's speech and language development, (3) the likely importance of babbling in speech and language development.

Hysterical aphonia--an analysis of 25 cases.

Hysteria is a common neurotic disorder in psychiatric practice. Many of its conversion symptoms have not been studied in detail. In the present prospective study in a tertiary care teaching hospital, 25 cases of hysterical aphonia were analysed. There were 17 females and 8 males. Mean age of presentation was 18.4 years in females and 21.2 years in males. Majority of patients were literate upto primary class, belonging to joint family and had urban background. Duration of symptoms was within 2 weeks. Most common precipitating factor was stress of examination or failure followed by quarrels with peers or spouse. In 20% cases, cause was not known. Comorbid psychiatric disorders were found in 80% cases, the most common being mixed anxiety and depressive disorder (36%) followed by generalized anxiety disorder (20%).

[Voice pathology in patients with allergic rhinitis]. / Zaburzenia glosu u chorych z alergicznym niezytem nosa.

The aim of work was evaluation of voice pathology in patients with allergic rhinitis. Larynx organic pathology were found in 75% patients with coexisting allergic rhinitis in the form of Reinke's oedema, chronic hypertrophic laryngitis, larynx polyp and vocal nodules. It caused serious voice pathology (dysphonia) which was confirmed by an objective spectrographic method. Larynx organic pathology was not in 15% patients. In these cases rhinophonia was found in consequence of resonance nasal defect.

[The treatment of psychogenic aphonia in two adolescents (author's transl)]. / Zur Therapie der psychogenen Aphonie am Beispiel von zwei seltenen Fällen bei Jugendlichen.

Psychogenic aphonia is rare in childhood and adolescence and its management is therefore problematic. Two examples, one of a hypofunctional aphonia in a 14 year old with a stutter and another of psychogenic aphonia confirm the necessity after psychiatric assessment and treatment of speech therapy in order to restore the voice to normal. The place of the laryngologist and speech therapist is discussed.