Revista Portuguesa de Pneumología ISSN: 0873-2159 [email protected] Sociedade Portuguesa de Pneumologia Portugal

Dias, C.; Sousa, L.; Batata, L.; Teixeira, F.; Moita, J.; Moutinho dos Santos, J. CPAP treatment for catathrenia Revista Portuguesa de Pneumología, vol. 23, núm. 2, marzo-abril, 2017, pp. 101-104 Sociedade Portuguesa de Pneumologia Lisboa, Portugal

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Conflicts of interest randomised controlled trial of efficacy, feasibility and costs. Respir Med. 2014;108:1387---95.

The authors have no conflicts of interest to declare. J.C. Winck a,∗, J. Chaves Caminha b

a References Faculdade de Medicina da Universidade do Porto, Portugal b Instituto de Ciências Biomédicas Abel Salazar, 1. Nava S. Behind a mask: tricks, pitfalls and preju- Universidade do Porto, Portugal dices for non invasive ventilation. Respir Care. 2013;58: 1367---76. ∗ Corresponding author. 2. Köhnlein T, Windisch W, Köhler D, Drabik A, Geiseler J, Hartl S, E-mail address: [email protected] (J.C. Winck). et al. Non-invasive positive pressure ventilation for the treat- ment of severe stable chronic obstructive pulmonary disease: a http://dx.doi.org/10.1016/j.rppnen.2015.12.008 prospective, multicentre, randomised, controlled clinical trial. 2173-5115/ Lancet Respir Med. 2014;2:698---705. © 2016 Sociedade Portuguesa de Pneumologia. Published by Elsevier 3. Hazenberg A, Kerstjens HA, Prins SC, Vermeulen KM, Wijk- Espa na,˜ S.L.U. This is an open access article under the CC BY-NC-ND stra PJ. Initiation of home mechanical ventilation at home: a license (http://creativecommons.org/licenses/by-nc-nd/4.0/ ).

CPAP treatment for catathrenia home with the established pressure. The patients were eval- uated at 1 and 3 months of CPAP therapy, in the presence of Catathrenia is a rare, idiopathic disorder classified as their partner. During the evaluation, patients were ques- an isolated symptom of sleep-disordered breathing (SDB). 1 tioned how they would classify their daytime complaints Its prevalence is unknown, 2 and its onset is usually in improvement (scale 0---100). After the first 3 months of adolescence. 3 Affected individuals are frequently unaware CPAP therapy, they were contacted monthly by telephone of their problem, and family members or bed partners to evaluate improvements over the remaining 3 months of commonly report strange sounds while breathing during therapy. sleep. 1,3 Eight patients were included. Five patients had abnor- The hallmark of this disorder is a deep inspiration mal sleepiness (ESS > 10). Six patients had symptoms of followed by prolonged expiration and a monotonous vocal- anxiety disorder and 7 were medicated with psycopharms ization resembling groaning, usually during rapid eye (Table 1). movement sleep. 1 Catathrenia events during the diagnostic and CPAP titra- No pharmacological treatments are available, 4 but some tion polysomnograms are displayed on Table 2. Diagnostic studies have shown partial or complete resolution of events PSG showed a mean respiratory disturbance index (RDI) with continuous positive airway pressure (CPAP) therapy, of 2.8 ± 3.29 events/h, a mean apnea---hypopnea index of especially in patients with a SDB associated. 2,4,5 1.2 ± 1.5 events/h. The mean number of catathrenia events The purpose of this study was to assess the effectiveness was 39.3 ± 26.6. Patient #6 experienced a partial resolution of CPAP treatment and a 6-month CPAP therapy in patients of events, despite an increase in pressure up to 12 cmH 2O. with catathrenia without SDB events associated. This patient did not tolerate a higher pressure. We performed a prospective study of patients with After 1 month of therapy, seven patients had signifi- catathrenia, diagnosed between 2008 and 2014, who under- cantly fewer moaning/groaning episodes and patient #6 went a CPAP titration PSG and subsequently initiated home reported a moderate reduction. After 3 months, seven CPAP therapy for 6 months. There were no exclusion patients reported complete resolution of nocturnal episodes criteria. and patient #6 reported maintenance of the moderate During anamnesis, the evaluated symptoms were: groan- reduction achieved with the first month of therapy. An ing, , choking, apnea, daytime sleepiness (Epworth improvement in daytime complaints was reported by all Sleepiness Scale [ESS]), headache, fatigue, and anxi- patients (mean subjective improvement of 80/100). One ety/depression. The diagnosis was based on an overnight patient maintained an abnormal sleepiness (ESS > 10). Com- polysomnogram (PSG). A catathrenia event was defined as plete resolution of events was maintained during the 6 a deep inhalation followed by prolonged exhalation, and months of follow-up, except for patient #6 who continued to a monotonous vocalization resembling groaning. 1 After the exhibit partial improvement. This patient was subsequently diagnostic study, patients underwent a CPAP titration PSG treated with clonazepam for 6 months, but showed no addi-

to correct catathrenia events. CPAP began at 4 cmH 2O and tional improvements. was progressively increased according to the type of respira- Although CPAP titration has been used in previous studies tory events observed. In the presence of obstructive events, of catathrenia, its goal was to correct apnea, hypopnea, and such as obstructive apnea or hypopnea, the pressure was flow limitation. 2,5 Other studies have reported an improve-

increased by 2 cmH 2O every 15 min, and in the presence of ment in moaning/groaning with CPAP, but the majority of snoring, respiratory effort-related arousals, and/or groan- patients had associated SDB. 2,4 6 ing episodes, it was increased by 1 cmH 2O every 15 min, Iriarte et al. have argued that the pathological mech- until the events reduced in number or disappeared. Once anism underlying catathrenia is mainly obstructive. Our the optimal pressure was achieved, CPAP was initiated at results support this theory, as our patients responded to 102 RESEARCH LETTERS score 4 3 4 ESS 13 20 11 14 14 Cloxazolam --- Paroxetine, Lorazepam, Trazodone Fluoxetine, alprazolam Sertraline, alprazolan Paroxetine Ethyl loflazepate, trazodone Alprazolam Psycopharms II, I, I I, I, I, II, II, nasal uvula palate palate, palate narrow uvula uvula narrow narrow tonsillectomy long High high high deviated long Mallampati short hard hard hard Mallampati septum Mallampati Mallampati Mallampati Mallampati Mallampati Mallampati Craniofacial examination and attack, attack Choking, morning headache, panic Morning headache, panic Tension headache fatigue anxiety Depression Morning headache Apnea, depression Headache, fatigue, depression Choking, morning fatigue, Other symptoms of of of of of of years years years years years age Childhood 25 age age 26 age 32 age groaning 28 Onset Childhood Childhood 15 and Scale. headache Chief complaint Groaning, excessive daytime sleepiness Groaning, fatigue Groaning, snoring, choking Groaning Groaning Groaning, morning fatigue Groaning Groaning Sleepiness patients. ) 2 of Epworth --- (kg/m ESS 45.8 18.2 27.7 39.5 22 26 27.2 BMI 21.4 index; characteristics mass (years) clinical body --- 36 34 25 37 30 31 32 Age 40 and BMI male; F F F F F M M Sex F ---

M Demographic

1 female; --- 8 6 4 5 2 7 3 F 1 Table Patient Documento descargado de http://www.elsevier.pt el 28/03/2017. Copia para uso personal, se prohíbe la transmisión de este documento por cualquier medio o formato. Documento descargado de http://www.elsevier.pt el 28/03/2017. Copia para uso personal, se prohíbe la transmisión de este documento por cualquier medio o formato.

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positive airway pressure. Vetrugno et al. 3 described a series of 10 patients with catathrenia and a normal RDI (as CPAP our patients) who had a post-inspiratory positive rise in 7 7 5 4 5 2 ESS After 12 10 endoesophageal pressure during events, higher than that

movement; observed in expiration in eupnoic breathing, suggesting

eye an expiratory upper airway obstruction. One possible CPAP mechanism that might explain the response observed in our 3 rapid patients is subtotal closure of the glottis during expiration. 0 0 0 0 0 0 0 (s) Maximum duration under 14 --- Our patients presented some differences in relation to

REM previous published series: there was a preponderance of CPAP women; OSA was absent and the majority of patients were

time; sleepy. It is possible that patients with more severe SDB, 0 0 0 0 0 8 0 0 (s) Minimum duration under

PSG catathrenia events were unnoticed or obscured by other

sleep respiratory sounds and therefore not detected. This may

CPAP explain the lower prevalence in men and patients with Titration total OSA in our series. The higher frequency of young women --- 0 0 0 0 0 9 0 0 Catathrenia events under (total) is also probably related to the less severe SDB. As for

TST sleepiness, it may be argued that it may be related to the

O) concomitant psychiatric disorders and medications. Other CPAP 2 during

index; sleepiness causes, recognized to contribute to sleep depri- used Peak 7 6 6 8 9 8 7 titration (cmH sound. 12 vation, were not analyzed in the present study. However, its improvement following efficacious catathrenia treat- 0.2 0.4 6.1 3.1 7 0.3 5 4.8 RDI ment suggest an association between both complaints and groaning

s) warrants further research. While no clear association has a been demonstrated between catathrenia and psychiatric

apnea/hypopnea 1 0 disorders, they marked a bold presence in our study (7/8 --- Clusters duration (mean, 59 23 15 76 56 80 67 patients). Examination by a psychiatric specialist with val- AHI a idated questionnaires would have provided a clearer idea containing of the presence of psychiatric disease in our patients. index; (total) 7 0 5 1 8 3 Clusters We believe CPAP treatment in patients with catathre- 12 18 nia without SDB events can improve nocturnal groaning

expirations episodes and daytime complaints. In a future research, laryngoscopy during sleep may help to elucidate catathrenia Maximum Duration (s) 40 15 23 13 28 34 20 21

disturbance 7

PSG. pathophysiology. prolonged by 6 0.8 7 5 6 9 2 Minimum Duration (s) 15 Conflicts of interest respiratory diagnostic --- The authors have no conflicts of interest to declare. the REM RDI followed PSG NREM. 8 0 4 sleep Catathrenia events during 21 12 25 10 44 during of

pressure; References Diagnostic inspirations stages 1. Sateia M. International classification of sleep disorders. In: airway 1 7 2 0 deep Catathrenia events during non-REM sleep 29 53 48 50 Catathrenia. 3rd ed. Darien, IL: American Academy of Sleep sleep 2

--- Medicine; 2014. p. 141. 2. Abbasi AA, Morgenthaler TI, Slocumb NL, Tippmann-Peikert characterization N3 least positive M, Olson EJ, Ramar K. Nocturnal moaning and groaning- at N2,

1 6 catathrenia or nocturnal vocalizations. Sleep Breath. 2012;16: of sleep Catathrenia events (total) 37 28 65 73 60 44 367---73. N1,

and 3. Vetrugno R, Lugaresi E, Plazzi G, Provini F, D’Angelo R, Mon-tagna continuous series

2 P. Catathrenia (nocturnal groaning): an abnormal respiratory pat- --- a 96 98 95 99 94 98 93 99 Mean O Sat tern during sleep. Eur J Neurol. 2007;14:1236---43.

events 4. Songu M, Yilmaz H, Yuceturk AV, Gunhan K, Ince A, Baytu- CPAP movement; 2 ran O. Effect of CPAP therapy on catathrenia and OSA: a case Min O Sat 89 95 91 91 88 91 88 97

eye report and review of the literature. Sleep Breath. 2008;12: considered 401---5. AHI 3.9 0.5 3.1 0.9 1.2 0 0 0 was

rapid 5. Guilleminault C, Hagen CC, Khaja AM. Catathrenia: parasom- Catathrenia nia or uncommon feature of sleep disordered breathing? Sleep. RDI 7 0.8 8.3 1.3 4.6 0 0.9 0.1 non 2008;31:132---9. 2 polysomnogram; --- cluster

--- 6. Iriarte J, Campo A, Alegre M, Fernández S, Urrestarazu E. A

a Catathrenia: respiratory disorder or ? Sleep Med. 2 4 5 6 7 8 3 NREM Table Patient 1 PSG 2015;16:827---30. Documento descargado de http://www.elsevier.pt el 28/03/2017. Copia para uso personal, se prohíbe la transmisión de este documento por cualquier medio o formato.

104 RESEARCH LETTERS

7. Ott SR, Hamacher J, Seifert E. Bringing light to the sirens of ∗ Corresponding author. night: laryngoscopy in catathrenia during sleep. Eur Resp J. E-mail address: [email protected] (C. Dias). 2011;37:1288---9. http://dx.doi.org/10.1016/j.rppnen.2016.12.008 a,b, b b b b C. Dias ∗, L. Sousa , L. Batata , F. Teixeira , J. Moita , 2173-5115/ b J. Moutinho dos Santos © 2017 Sociedade Portuguesa de Pneumologia. Published by Elsevier a Pulmonology Department, Centro Hospitalar de S. João, Espa na,˜ S.L.U. This is an open access article under the CC BY-NC-ND Porto, Portugal license (http://creativecommons.org/licenses/by-nc-nd/4.0/ ). b Center, Centro Hospitalar e Universitário de Coimbra, Portugal

Concomitant lung cancer and In our sample the majority of patients had a smoking his- interstitial lung disease: A tory. Cigarette smoking is a recognised risk factor for the development of ILD 1 but the pathogenesis of LC in patients challenge in clinical practice with ILD is still unclear. IPF has been considered a neo- proliferative lung disorder since both IPF and cancer share Dear Editor, similar pathogenic hallmarks such as genetic alterations, uncontrolled mesenchymal cell proliferation and tissue inva- Lung cancer (LC) risk is increased in patients with interstitial sion behaviour, and dysregulated intracellular signalling 1 lung disease (ILD) and sometimes both occur concomitantly. pathways. 3 LC incidence is increased 4.96-fold in patients with idio- The treatment choice for ILD patients presenting LC pathic pulmonary fibrosis (IPF) compared with the general is a challenge to the physicians. In our sample some population even after adjusting for age, gender and smoking patients benefited from LC treatment but the pre-existence 2 habit. Idiopathic interstitial pneumonias are also associated of ILD also influenced negatively the prognosis. Voltolini with increased LC risk and connective tissue disease- et al. showed that major lung resection in patients with associated ILD (CT-ILD) may be a predisposing factor for early stage non-small cell LC and ILD was associated with 2 pulmonary malignancy. increased postoperative morbidity and mortality, mainly in A retrospective analysis of ILD patients diagnosed with patients presenting lower preoperative FVC% and DLCO%. LC at our centre in the past 5 years was performed. Charac- There was a higher postoperative mortality for pneumonec- teristics of this cohort were described and outcomes were tomy and lobectomy. No patients died after sublobar resec- also reported. tion. Thus, anatomic surgical resections can be an option Eleven patients were included [median age 68 (range 36 in appropriately selected LC-ILD patients. 4 When planning to 78) years; mostly men (n = 9; 81.8%)]. Almost all patients radiotherapy, it is important to determinate the radiation had smoking history (81.8%; ex-smokers n = 7; active smok- pneumonitis risk. A recent study showed that fatal radiation ers n = 2). The ILDs identified were CT-ILD (n = 5; 45.5%), pneumonitis tended to be more common in patients with combined pulmonary fibrosis and emphysema (CPFE) (n = 2; subclinical ILD and that the presence of extensive fibrosis 18.1%), IPF (n = 1; 9.1%), sarcoidosis (n = 1; 9.1%), cryp- on CT may be a contraindication for thoracic radiotherapy. 5 togenic organising pneumonia (n = 1; 9.1%) and silicosis Stereotactic body radiation therapy (SBRT) could also be an (n = 1; 9.1%). The most prevalent LC histological type was option in LC-ILD patients because of its less invasive nature, adenocarcinoma (n = 5; 45.5%), followed by squamous cell nevertheless there is an increasing body of evidence sug- carcinoma (n = 2; 18.1%) and small cell carcinoma (n = 2; gesting that even SBRT can induce acute exacerbation of 18.1%). Most patients were diagnosed at advanced stages ILD. None of our patients were submitted to SBRT. (IIIB and IV) (n = 7; 63.6%), mainly during clinical and Chemotherapy plays an irreplaceable role against LC, radiological follow-up for the ILD. The tumours were pre- but LC-ILD patients receiving chemotherapy may face risks dominantly in the peripheral lung fields, in relation to of chemotherapy-related acute exacerbation of ILD. The fibrotic areas. Median time from the onset of ILD to the question arises as to whether chemotherapy regimens are onset of LC was 4 (range 0.3 to 249.7) months. Surgical efficacious and safe for the co-morbidity population. So far, resection was performed in 3 patients (27.3%) with stage no consensus has been reached nor has enough evidence IIA and IIIA LC; chemotherapy and/or radiotherapy were been presented to support an optimal treatment strategy given to 6 patients (54.5%) with advanced disease (stage for LC---ILD patients --- these patients are usually excluded III and IV). One patient was refused for radiotherapy due by most clinical trials and the relevant studies are largely to concern about the adverse effects and prognosis. Three single-armed. A previous meta-analysis performed to eval- patients (27.3%) had acute exacerbations of the ILD after uate the safety and efficacy of chemotherapy in non-small LC treatment: 1 patient with CPFE and another with sar- cell LC---ILD patients suggested that chemotherapy might be coidosis presented acute exacerbation after radiotherapy an effective therapy for these patients, but it also might and 1 patient with IPF presented acute exacerbation after be associated with higher incidence of acute exacerbations chemotherapy with pemetrexed. Two of these patients died of ILD. 6 Recently, the role of anti-fibrotic drugs in LC treat- due to respiratory failure. Median survival time from the ment was studied and the results were promising, opening diagnosis of LC was 6.6 months (range 1.2 to 55.6). Three new perspectives on therapeutic options for these complex patients died due to progression of LC. patients. 7