Clinical suspicion is central to diagnosing hypoventilation syndromes. Many of these disorders commonly present with, or develop, alveolar hypoventilation. It is with this knowledge that clinicians should first question and then confirm whether the patient's signs and symptoms can be explained by the diagnosis of alveolar hypoventilation.
Disease states that have been associated with the hypoventilation syndromes include:[1]American Academy of Sleep Medicine. International classification of sleep disorders. 3rd ed, text revision. Darien, IL: American Academy of Sleep Medicine; 2023.
Obesity hypoventilation syndrome (OHS)
Restrictive thoracic disorders, such as patients with chest wall deformities (e.g., kyphoscoliosis, fibrothorax, or thoracoplasty)
Neuromuscular disorders, particularly Duchenne muscular dystrophy and other types of muscular dystrophies and spinal muscular atrophies
Central sleep apnea syndromes, such as idiopathic central sleep apnea, and Cheyne-Stokes respiration (CSR)
Congenital central alveolar hypoventilation, which is a rare disorder
Obstructive airway disease (in particular, COPD).
This topic focuses on OHS, restrictive thoracic disorders, Cheyne-Stokes respiration, and COPD.
See Central sleep apnea, Obstructive sleep apnea, and Muscular dystrophies.
History
Many of the symptoms secondary to the disorder causing hypoventilation are nonspecific and of limited value. In the early stages of the disorder, the patient may be totally asymptomatic. However, as the syndrome progresses, dyspnea on exertion followed by dyspnea at rest is the most common symptom encountered by patients with hypoventilation. Disturbed sleep and daytime hypersomnolence resulting from nocturnal hypoventilation may progress and be associated with symptoms of morning headaches and fatigue. If a disorder causes respiratory muscle weakness, impaired cough and repeated lower respiratory tract infections may also complicate the patient's course. In addition, careful history-taking can allow one to determine the rate of progression of the underlying disorder, in order to initiate appropriate therapeutic interventions.
Congenital central alveolar hypoventilation typically presents in newborns, with symptomatic and asymptomatic children surviving to adulthood.[25]Berry-Kravis EM, Zhou L, Rand CM, et al. Congenital central hypoventilation syndrome: PHOX2B mutations and phenotype. Am J Respir Crit Care Med. 2006;174:1139-1144.
http://www.atsjournals.org/doi/full/10.1164/rccm.200602-305OC#.U1ZtQvldUww
http://www.ncbi.nlm.nih.gov/pubmed/16888290?tool=bestpractice.com
[26]Doherty LS, Kiely JL, Deegan PC, et al. Late-onset central hypoventilation syndrome: a family genetic study. Eur Respir J. 2007;29:312-316.
http://erj.ersjournals.com/content/29/2/312.full
http://www.ncbi.nlm.nih.gov/pubmed/17264323?tool=bestpractice.com
[27]Weese-Mayer DE, Berry-Kravis EM, Ceccherini I, et al. An official ATS clinical policy statement: congenital central hypoventilation syndrome: genetic basis, diagnosis and management. Am J Respir Crit Care Med. 2010;181:626-644.
http://www.atsjournals.org/doi/full/10.1164/rccm.200807-1069ST#.U1Ztf_ldUww
http://www.ncbi.nlm.nih.gov/pubmed/20208042?tool=bestpractice.com
In patients with neuromuscular disease, sleep-disordered breathing, including nocturnal alveolar hypoventilation, presents in childhood.[16]Ragette R, Mellies U, Schwake C, et al. Patterns and predictors of sleep disordered breathing in primary myopathies. Thorax. 2002;57:724-728.
http://www.ncbi.nlm.nih.gov/pubmed/12149535?tool=bestpractice.com
[28]Culebras A. Sleep disorders and neuromuscular disease. Semin Neurol. 2005;25:33-38.
http://www.ncbi.nlm.nih.gov/pubmed/15798935?tool=bestpractice.com
Patients with obesity hypoventilation are usually middle-aged.[23]Mokhlesi B, Tulaimat A. Recent advances in obesity hypoventilation syndrome. Chest. 2007;132:1322-1336.
http://journal.publications.chestnet.org/article.aspx?articleid=1085427
http://www.ncbi.nlm.nih.gov/pubmed/17934118?tool=bestpractice.com
Patients with COPD and those with CSR are usually in the age range of 40 to 60 years, but this can be variable. In patients with OHS, there is a 2:1 male-to-female ratio.[23]Mokhlesi B, Tulaimat A. Recent advances in obesity hypoventilation syndrome. Chest. 2007;132:1322-1336.
http://journal.publications.chestnet.org/article.aspx?articleid=1085427
http://www.ncbi.nlm.nih.gov/pubmed/17934118?tool=bestpractice.com
Physical examination
The value of the physical exam is not only in characterizing the cause of hypoventilation (e.g., chest wall deformity, obesity [BMI ≥30 kg/m²], severe COPD) but also in detailing the severity of the complications that result from it (e.g., the presence of cor pulmonale). While a precipitating event such as a respiratory tract infection can trigger acute respiratory failure at any time, most patients' physical exam reflects the more usual, gradual progressive development of alveolar hypoventilation over months or years. As a result of diurnal CO₂ retention and associated hypoxemia, patients may demonstrate signs of cor pulmonale, including an increased pulmonic component of the second heart sound (P2), and lower-extremity edema. An increased P2 can be seen with most causes of hypoventilation syndrome.[16]Ragette R, Mellies U, Schwake C, et al. Patterns and predictors of sleep disordered breathing in primary myopathies. Thorax. 2002;57:724-728.
http://www.ncbi.nlm.nih.gov/pubmed/12149535?tool=bestpractice.com
[23]Mokhlesi B, Tulaimat A. Recent advances in obesity hypoventilation syndrome. Chest. 2007;132:1322-1336.
http://journal.publications.chestnet.org/article.aspx?articleid=1085427
http://www.ncbi.nlm.nih.gov/pubmed/17934118?tool=bestpractice.com
[28]Culebras A. Sleep disorders and neuromuscular disease. Semin Neurol. 2005;25:33-38.
http://www.ncbi.nlm.nih.gov/pubmed/15798935?tool=bestpractice.com
Patients with OHS have a BMI ≥30 kg/m², with an increasing prevalence with increasing BMI.[23]Mokhlesi B, Tulaimat A. Recent advances in obesity hypoventilation syndrome. Chest. 2007;132:1322-1336.
http://journal.publications.chestnet.org/article.aspx?articleid=1085427
http://www.ncbi.nlm.nih.gov/pubmed/17934118?tool=bestpractice.com
Lower-extremity edema and right-sided third heart sound (S3 gallop) are evident with most causes of hypoventilation syndrome.[16]Ragette R, Mellies U, Schwake C, et al. Patterns and predictors of sleep disordered breathing in primary myopathies. Thorax. 2002;57:724-728.
http://www.ncbi.nlm.nih.gov/pubmed/12149535?tool=bestpractice.com
[23]Mokhlesi B, Tulaimat A. Recent advances in obesity hypoventilation syndrome. Chest. 2007;132:1322-1336.
http://journal.publications.chestnet.org/article.aspx?articleid=1085427
http://www.ncbi.nlm.nih.gov/pubmed/17934118?tool=bestpractice.com
[28]Culebras A. Sleep disorders and neuromuscular disease. Semin Neurol. 2005;25:33-38.
http://www.ncbi.nlm.nih.gov/pubmed/15798935?tool=bestpractice.com
Other conditions, such as CSR, may present with physical findings suggestive of left-sided congestive heart failure (CHF), such as an S3 gallop, and inspiratory crackles on examination of the lungs. A left-sided fourth heart sound (S4 gallop) can be seen in patients with CSR due to CHF.
Confirmatory investigations
An arterial blood gas analysis is required in all patients to document the presence of an elevated CO₂ and confirm the diagnosis. It is the definitive test used to confirm the diagnosis of alveolar hypoventilation and to document the extent of associated hypoxemia.[29]Ozsancak A, D'Ambrosio C, Hill NS. Nocturnal noninvasive ventilation. Chest. 2008;133:1275-1286.
http://journal.publications.chestnet.org/article.aspx?articleid=1085836
http://www.ncbi.nlm.nih.gov/pubmed/18460530?tool=bestpractice.com
If hypoventilation becomes more severe, hypercapnia or hypoxemia becomes more evident, and respiratory failure may ensue, requiring ventilatory support.
Pulmonary function tests, including spirometry, measurement of lung volumes, and measurements of respiratory muscle strength, give important clues as to the cause and severity of the disease underlying the hypoventilation. In patients with neuromuscular disease, sleep-disordered breathing is evident when the FVC declines to <65% of predicted.[30]Alves RS, Resende MB, Skomro RP, et al. Sleep and neuromuscular disorders in children. Sleep Med Rev. 2009;13:133-148.
http://www.ncbi.nlm.nih.gov/pubmed/18534877?tool=bestpractice.com
In patients with OHS, the restrictive pattern is accompanied by a decrease in the expiratory reserve volume.[31]Mokhlesi B, Kryger MH, Grunstein RR. Assessment and management of patients with obesity hypoventilation syndrome. Proc Am Thorac Soc. 2008;5:218-225.
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2645254
http://www.ncbi.nlm.nih.gov/pubmed/18250215?tool=bestpractice.com
Respiratory muscle strength is known to be decreased in patients with restrictive thoracic disorders, which correlates with the development of sleep-disordered breathing.[16]Ragette R, Mellies U, Schwake C, et al. Patterns and predictors of sleep disordered breathing in primary myopathies. Thorax. 2002;57:724-728.
http://www.ncbi.nlm.nih.gov/pubmed/12149535?tool=bestpractice.com
It is also decreased in patients with OHS due to a combination of abnormal respiratory mechanics and weak respiratory muscles.[32]Koenig SM. Pulmonary complications of obesity. Am J Med Sci. 2001;321:249-279.
http://www.ncbi.nlm.nih.gov/pubmed/11307867?tool=bestpractice.com
Because many of the disorders associated with the hypoventilation syndrome initially demonstrate more significant hypoxemia during sleep (particularly REM sleep) an overnight polysomnogram is often indicated. In addition, many disorders have associated obstructive and central sleep-disordered breathing events that would require appropriate treatment if recognized. A polysomnogram is indicated in patients with chest wall abnormalities and neuromuscular disease to identify patients who would benefit from nocturnal ventilation.[33]Sawicka EH, Branthwaite MA. Respiration during sleep in kyphoscoliosis. Thorax. 1987;42:801-808.
http://www.ncbi.nlm.nih.gov/pubmed/3424256?tool=bestpractice.com
[34]Barthlen GM. Nocturnal respiratory failure as an indication of noninvasive ventilation in the patient with neuromuscular disease. Respiration. 1997;64(suppl 1):S35-S38.
http://www.ncbi.nlm.nih.gov/pubmed/9380959?tool=bestpractice.com
[35]Pradella M. Sleep polygraphic parameters in neuromuscular diseases. Arq Neuropsiquiatr. 1994;52:476-483.
http://www.ncbi.nlm.nih.gov/pubmed/7611939?tool=bestpractice.com
[36]Bourke SC, Gibson GJ. Sleep and breathing in neuromuscular disease. Eur Respir J. 2002;19:1194-1201.
http://erj.ersjournals.com/content/19/6/1194.full
http://www.ncbi.nlm.nih.gov/pubmed/12108875?tool=bestpractice.com
[37]Lofaso F, Quera-Salva MA. Polysomnography for the management of progressive neuromuscular disorders. Eur Respir J. 2002;19:989-990.
http://erj.ersjournals.com/content/19/6/989.full.pdf+html
http://www.ncbi.nlm.nih.gov/pubmed/12108883?tool=bestpractice.com
It identifies associated obstructive sleep apnea (OSA) in patients with OHS.[2]Mokhlesi B, Tulaimat A, Faibussowitsch I, et al. Obesity hypoventilation syndrome: prevalence and predictors in patients with obstructive sleep apnea. Sleep Breath. 2007;11:117-124.
http://www.ncbi.nlm.nih.gov/pubmed/17187265?tool=bestpractice.com
In addition, it may identify patients with OHS prior to developing awake elevations in PaCO₂.[38]Sivam S, Yee B, Wong K, et al. Obesity hypoventilation syndrome: early detection of nocturnal-only hypercapnia in an obese population. J Clin Sleep Med. 2018 Sep 15;14(9):1477-1484.
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6134235
http://www.ncbi.nlm.nih.gov/pubmed/30176974?tool=bestpractice.com
In CHF with a left ventricular ejection fraction <45% and disturbed sleep, a polysomnogram identifies CSR.[5]Javaheri S, Parker TJ, Liming JD, et al. Sleep apnea in 81 ambulatory male patients with stable heart failure: types and their prevalences, consequences, and presentations. Circulation. 1998;97:2154-2159.
http://circ.ahajournals.org/cgi/content/full/97/21/2154
http://www.ncbi.nlm.nih.gov/pubmed/9626176?tool=bestpractice.com
[6]Sin DD, Fitzgerald F, Parker JD, et al. Risk factors for central and obstructive sleep apnea in 450 men and women with congestive heart failure. Am J Respir Crit Care Med. 1999;160:1101-1106.
http://www.atsjournals.org/doi/full/10.1164/ajrccm.160.4.9903020
http://www.ncbi.nlm.nih.gov/pubmed/10508793?tool=bestpractice.com
[7]Oldenburg O, Lamp B, Faber L, et al. Sleep-disordered breathing in patients with symptomatic heart failure: a contemporary study of prevalence in and characteristics of 700 patients. Eur J Heart Fail. 2007;9:251-257.
http://www.ncbi.nlm.nih.gov/pubmed/17027333?tool=bestpractice.com
[8]Krachman SL, D'Alonzo GE, Berger TJ, et al. Comparison of oxygen therapy with nasal continuous positive airway pressure on Cheyne-Stokes respiration during sleep in congestive heart failure. Chest. 1999;116:1550-1557.
http://journal.publications.chestnet.org/article.aspx?articleid=1078359
http://www.ncbi.nlm.nih.gov/pubmed/10593775?tool=bestpractice.com
A polysomnogram is used in patients with COPD who have suspected overlap syndrome (associated OSA), but use to identify REM-associated hypoventilation is undefined.
An echocardiogram may be performed to evaluate for cor pulmonale and/or the presence of left-sided CHF. It documents the development of pulmonary hypertension in patients with OHS, neuromuscular disease, and COPD. In patients with CSR, an echocardiogram documents the severity of left ventricular dysfunction.[3]Kessler R, Chaouat A, Schinkewitch P, et al. The obesity-hypoventilation syndrome revisited: a prospective study of 34 consecutive cases. Chest. 2001;120:369-376.
http://journal.publications.chestnet.org/article.aspx?articleid=1079891
http://www.ncbi.nlm.nih.gov/pubmed/11502631?tool=bestpractice.com
[5]Javaheri S, Parker TJ, Liming JD, et al. Sleep apnea in 81 ambulatory male patients with stable heart failure: types and their prevalences, consequences, and presentations. Circulation. 1998;97:2154-2159.
http://circ.ahajournals.org/cgi/content/full/97/21/2154
http://www.ncbi.nlm.nih.gov/pubmed/9626176?tool=bestpractice.com
[6]Sin DD, Fitzgerald F, Parker JD, et al. Risk factors for central and obstructive sleep apnea in 450 men and women with congestive heart failure. Am J Respir Crit Care Med. 1999;160:1101-1106.
http://www.atsjournals.org/doi/full/10.1164/ajrccm.160.4.9903020
http://www.ncbi.nlm.nih.gov/pubmed/10508793?tool=bestpractice.com
[7]Oldenburg O, Lamp B, Faber L, et al. Sleep-disordered breathing in patients with symptomatic heart failure: a contemporary study of prevalence in and characteristics of 700 patients. Eur J Heart Fail. 2007;9:251-257.
http://www.ncbi.nlm.nih.gov/pubmed/17027333?tool=bestpractice.com
[8]Krachman SL, D'Alonzo GE, Berger TJ, et al. Comparison of oxygen therapy with nasal continuous positive airway pressure on Cheyne-Stokes respiration during sleep in congestive heart failure. Chest. 1999;116:1550-1557.
http://journal.publications.chestnet.org/article.aspx?articleid=1078359
http://www.ncbi.nlm.nih.gov/pubmed/10593775?tool=bestpractice.com
[39]Sugerman HJ, Fairman RP, Baron PL, et al. Gastric surgery for respiratory insufficiency of obesity. Chest. 1986;90:81-86.
http://www.ncbi.nlm.nih.gov/pubmed/3720390?tool=bestpractice.com
A chest x-ray should be performed to exclude other causes for hypoxemia.
Other investigations
While an elevation of serum bicarbonate, as well as a low oxygen saturation on pulse oximetry, may suggest the presence of alveolar hypoventilation, they are not recommended as diagnostic tests. Measurement of serum bicarbonate may be used to screen for the presence of alveolar hypoventilation, but does not confirm the diagnosis.[2]Mokhlesi B, Tulaimat A, Faibussowitsch I, et al. Obesity hypoventilation syndrome: prevalence and predictors in patients with obstructive sleep apnea. Sleep Breath. 2007;11:117-124.
http://www.ncbi.nlm.nih.gov/pubmed/17187265?tool=bestpractice.com
[40]Mokhlesi B, Masa JF, Brozek JL, et al. Evaluation and management of obesity hypoventilation syndrome. An official American Thoracic Society clinical practice guideline. Am J Respir Crit Care Med. 2019 Aug 1;200(3):e6-e24.
https://www.atsjournals.org/doi/full/10.1164/rccm.201905-1071ST
http://www.ncbi.nlm.nih.gov/pubmed/31368798?tool=bestpractice.com
Pulse oximetry suggests the presence of alveolar hypoventilation, but does not confirm the diagnosis and should not be used to decide when to measure PaCO₂.[40]Mokhlesi B, Masa JF, Brozek JL, et al. Evaluation and management of obesity hypoventilation syndrome. An official American Thoracic Society clinical practice guideline. Am J Respir Crit Care Med. 2019 Aug 1;200(3):e6-e24.
https://www.atsjournals.org/doi/full/10.1164/rccm.201905-1071ST
http://www.ncbi.nlm.nih.gov/pubmed/31368798?tool=bestpractice.com
In the right clinical setting, other laboratory tests may be indicated. In patients with hypercapnia who demonstrate signs of hypothyroidism, measurement of thyroid-stimulating hormone levels may be indicated. Measurement of hematocrit is indicated in all patients with suspected or documented daytime and/or nocturnal hypoxemia. If there is a clinical suspicion for congenital central alveolar hypoventilation, mutations in the paired-like homeobox 2B (PHOX2B) gene should be evaluated, as mutations are noted in up to 91% of these patients.[24]Trang H, Dehan M, Beaufils F, et al; French CCHS Working Group. The French Congenital Central Hypoventilation Syndrome Registry: general data, phenotype, and genotype. Chest. 2005;127:72-79.
http://journal.publications.chestnet.org/article.aspx?articleid=1083054
http://www.ncbi.nlm.nih.gov/pubmed/15653965?tool=bestpractice.com
Patients with PHOX2B mutations exhibit respiratory-related cortical activity on electroencephalograms at rest.[41]Laveneziana P, Albuquerque A, Aliverti A, et al. ERS statement on respiratory muscle testing at rest and during exercise. Eur Respir J. 2019 Jun 13;53(6):1801214.
https://erj.ersjournals.com/content/53/6/1801214.long
http://www.ncbi.nlm.nih.gov/pubmed/30956204?tool=bestpractice.com