Nocturnal enuresis in children with upper airway obstruction DUDLEY J. WEIDER, MD. FACS, MICHAEL J. SATEIA, MD. and RUTH P. WEST, RN, Hanover, New Hampshire
This study presents the results of our experience with 115 children between the ages of 3 and 19 years who have had symptoms of upper airway obstruction and nocturnal enuresis. Twelve children had secondary enuresis, and 103 children had primary enuresis. Surgical removal of upper airway obstruction led to a significant decrease In or complete cure of nocturnal enuresis In 87 (76%) of the children studied. Eleven children were also studied with polysomnographlc tracings In an attempt to determine a relationship between their sleep patterns and nocturnal enuresis. (OTOlARYNGOL HEAD NECK SURG 1991;105:427,)
HISTORICAL REVIEW Although much has been written about nocturnal enuresis, little has been reported about its relationship to upper airway obstruction. Simmons et al. I noted that children having episodes of sleep apnea of 10 seconds or longer occurring repeatedly during the night can become enuretic and may report nightmares. They presented case reports of an 8-year-old boy and a IS-yearold girl whose nocturnal enuresis stopped immediately after upper airway obstruction was surgically eliminated (by tonsillectomy and adenoidectomy and by tracheostomy, respectively). Guilleminault and Dement' studied children with sleep apnea syndrome. Additionally, they examined two adults with intermittent nocturnal enuresis for whom urologic evaluations were sought before their evaluation for sleep apnea. In both cases, the enuresis ceased after successful surgical management of their sleep apnea. Others':" also mention the relationship between nocturnal enuresis and sleep apnea. In 1978, an adenoidectomy was performed by one of the authors (D.J.W.) on a 7-year-old boy with chronic recurrent otitis media with effusion (OME),
From the Section of Otolaryngology and Audiology, Department of Surgery (Dr. Weider and Ms. West) and Department of Psychiatry (Dr. Sateia), Dartmouth-Hitchcock Medical Center. Funded (in part) by the Hitchcock Foundation. Received for publication Sept. 13, 1990; revision received Feb. 12. 1991; accepted Feb. 20, 1991. Reprint requests: Dudley J, Weider, MD, Section of Otolaryngology and Audiology. Dartmouth-Hitchcock Medical Center. 2 Maynard St., Hanover. NH 03756.
23/1/29051
clinically obstructive adenoids, and a severe snoring problem. At his 6-month follow-up visit, this child's mother reported that he had had no further episodes of OME, that his breathing was significantly improved, and that his snoring had stopped. In addition, she asked why he had stopped bedwetting after his adenoidectomy. This child had been a primary enuretic (never having had a significant "dry period'T.l" who experienced nightly enuresis despite parental attempts to limit fluid intake before bed and to take him to the bathroom during the night. After the adenoidectomy, he stopped bedwetting immediately, with virtually no breakthrough episodes. It was this case that provided the impetus for our study. Since that observation, members of our section have routinely asked parents of children manifesting large tonsils, adenoids, or other symptoms of severe upper airway obstruction if their child has a bedwetting problem. We now have collected about 115 cases over a 12-year period. We have previously reported our results with 35 patients.' We would like to stress that upper airway obstruction is not the primary cause of nocturnal enureses in the majority of cases. However, we would like to present the subject of upper airway obstruction as one etiologic factor of nocturnal enuresis.
Purpose The purpose of this article is to demonstrate the effect of tonsillectomy and adenoidectomy and related upper airway surgery on the frequency of nocturnal enuresis in a clinically selected pediatric population. Secondly, it is our hope to heighten the awareness of physicians about the association between sleep disorders caused
427
Downloaded from oto.sagepub.com at University of Warwick on December 10, 2016
OtolaryngologyHead and Neck Surgery
428 WEIDER at 01.
All Patients 5.6
Table 1. Initial clinic visit (N = 115)* Reason for visit
No.
Tonsillitis Ear disease Chronic mouth breathing Allergy Dental malocclusion Sieep apnea
22 46
115 children; age range 3-19 98 males; 17 females
32 14
20 16
'Several patients had more than one presenting complaint.
Preop
1 Month
6 Months 12 Months
Fig. 1. Average number of enuretic nights per week preoperatively and at 1, 6. and 12 months postoperatively for the entire series of 115 patients (98 maies and 17 females. ages 3 to 19 years).
by upper airway obstruction and nocturnal enuresis in the pediatric population. This study corroborates the important findings from our earlier smaller series."
METHODS Patients Our study included 115 children (98 boys and 17 girls) between the ages of 3 and 19 years who were referred to us for various otolaryngologic problems, but who all had symptoms of upper airway obstruction. All children selected were obligate nighttime mouth breathers who additionally had nocturnal enuresis. All snored and many seemed to obstruct for varying periods of time. Each was described as either an extremely restless or an extremely sound sleeper. When taken to the bathroom during the night. most children were difficult to arouse and did not remember the event. Each patient's urologic history was reviewed for the presence of urinary tract infections and other problems relating to the genitourinary system. In one case, a 15-year-old girl was referred to the urology department; no pathology was found. Table 1 shows the types of pathology our patients manifested. The study consists of 103 children with primary enuresis (children who had had enuresis since birth) and 12 children with secondary enuresis (children who had had an interval of dryness lasting longer than 6 months). '.6 Procedures
All parents who brought children to our clinic between July 1978 and October 1987 with symptoms of significant upper airway obstruction were asked if their child had any symptoms of nocturnal enuresis. If so,
the child was included in the study. A careful history was taken of snoring. nighttime obstruction, daytime and nighttime mouth breathing, chronic rhinorrhea. allergy, and excessive daytime sleepiness (EDS). All children in the study snored and some experienced actual obstructive apnea. All children had some type of surgery to eliminate upper airway obstruction. The most common procedure performed was a tonsillectomy, adenoidectomy. or both. All surgery was done to provide a more functional upper airway in a population of children who snored and who had been observed to have frequent episodes of obstruction. In no case was surgery performed to provide a cure for nocturnal enuresis nor were any promises made that such a cure would result. Each patient's history of enuresis was simply observed preoperatively and postoperatively and recorded as described. Six children were studied preoperatively with polysomnography in our sleep laboratory by one author (M.J.S.), Five additional children from other institutions were also studied polysomnographically (Table 2). All patients reported in this article underwent surgery for relief of upper airway obstruction. One patient had a submucous resection, and one had a combination submucous resection and intranasal polypectomy and ethmoidectomy (8-year-old boy with cystic fibrosis and nasal polyposis). Two patients (both male) had adenotonsillectomy in combination with bilateral inferior turbinoplasties (as described by Mabry") and partial uvulectomies. Both presented with what was considered to be allergically related turbinate hypertrophy in addition to tonsil and adenoid hypertrophy. The remaining III children had either tonsillectomy or adenoidectomy or combined adenotonsillectomy, The modalities measured by polysomnography were electroencephalogram (EEG), electro-oculogram (EOG), breathing from the nose and/or mouth (by way of thermisters attached under the nose and mouth), electrocardiogram (ECG), and intercostal electromyogram (EMG) of the thoracic cage. A special mattress was used to register an interruption in electrical current when the child wet the bed. Our ear oximeter was not
Downloaded from oto.sagepub.com at University of Warwick on December 10, 2016
Volume 105 Number 3 September 1991
Table 2. Sleep Patient no./age lvears)
1/12 2/13 3t/5 4t/12 5/6 6t:j:/6 7t/10 8/7 9t/8 10/6
11/6
Nocturnal enuresis in children with upper airway obstruction
429
study (polysomnography) results "10 Sleep Tvpe of enuresis·
1° 1° ?
? 1° 2° 1° 1° 2° 1° 1°
Slage 1
8.8 5.5 10.7 50.9 13.8 10.2 3,7 13.8 4.2 12.6 3,7
Slage 2
Slage 3/4
REM
RDI
40.0 49.9 584 193 42.8 51,6 514 51.8 49.7 46,0 555
27.6 21.9 12,8 21.0 224 26,8 26,1 20.7 307 20,0 26.2
22.4 23.1 17.2
6.0 1.3 06
20.3 11.3 18,6 13.5 14.9 21.0 13,8
17.0 16.0 0 3.5 2.6 3.8 18
Surgical response
No No
No No
Yes Yes surgery surgery Yes Yes surgery No surgery No Yes
ROt, Respiratory disturbance index (combined apnea and hypopnea per hour 01 sleep): REM. rapid eye movement. '1 ° = Primary noctural enuresis; 2° = secondary nocturnal enuresis, tReferred to DHMC for sleep studies, but not included in our operative series, :j:Studied post-adenoldectorny.
used in the first five patients studied because it was too bulky to permit comfortable sleep in the age group being studied. (More recent technologic advances have eliminated this problem.) Information contributing to our database was acquired by interviewing patients and their parents 1 month, 6 months, and 12 months after surgery. A questionnaire was also sent to all parents of patients. Clinical data for our entire population are presented in Fig. 1; data for the six subgroups (classified by symptom categories and patterns of enuresis) are presented in Figs. 2 through 7.
RESULTS For the group as a whole, we observed a 66% reduction in the number of enuretic nights per week 1 month after surgery. By 6 months, a 77% reduction was observed, which has remained constant (Fig. 1). Twelve children, ages 4 to 12 years, manifested secondary nocturnal enuresis (Fig. 2). In this group, actual dry periods ranged from 1 to 4 years. Onset of secondary enuresis invariably coincided with the onset of symptoms of upper airway obstruction. By the end of the first postoperative month there was an 89% reduction in the number of enuretic nights per week. Six months after surgery, all twelve patients had stopped bedwetting (0 enuretic nights per week), a result that was maintained until at least 1 year after the original surgery. We then analyzed data from our group of children who exhibited irregular nocturnal enuresis (Fig. 3). Children in this group exhibited typical dry periods of I week to I month, after which they resumed enuresis on an almost nightly basis. One month after surgery,
this group experienced a 68% reduction in enuretic nights per week and almost no further reduction thereafter. Five children stopped bedwetting completely by the end of the first month and maintained dryness. One patient was unaffected by the surgery and maintained a pattern of four enuretic nights per week. Another child, age 6 years, went from seven enuretic nights per week to 0 enuretic nights per week for 3 weeks (the second to fourth postoperative weeks) and then resumed a pattern of five enuretic nights per week for the remainder of the year. Figure 4 depicts a group of eight boys, ages four to seven years, who exhibited EDS. All but one had primary enuresis. One had secondary enuresis (a 7-yearold with a dry period between the ages of 2 and 7) (Fig. 2). By the end of the first postoperative month. this group experienced a 91% reduction in the number of enuretic nights per week and a complete resolution of EDS. By 6 months, all children had experienced complete cessation of enuresis, and all maintained dryness until at least I year after surgery. Figure 5 depicts our largest group: children ages 3 to 19 years with primary nocturnal enuresis without EDS. These patients typically had a history of nighttime mouth breathing or snoring in combination with a history of enuresis. One month after surgery, there was a 59% reduction in enuretic nights per week; at 6 months this increased to a 71% reduction, with slight additional improvement by I year. This group was further subdivided into children who experienced a reduction in frequency of their nocturnal enuresis (Fig. 6) and those who did not (Fig. 7). The "responders" (Fig. 6) had an 81% reduction in the number of enuretic nights per week at 1 month, which
Downloaded from oto.sagepub.com at University of Warwick on December 10, 2016
430
OtolaryngologyHead and Neck Surgery
WEIDER et 01.
Secondary Nocturnal Enuresis C ill
'';: l':l
7
6.0
6
ill ill
.~
12 children; age range 4-12 10 male; 2 female
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1 Month
6 Months 12 Months
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5.0
eo
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6.0 8 children; age range 4-9 8 male; 0 female
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This study presents the results of our experience with 115 children between the ages of 3 and 19 years who have had symptoms of upper airway obstruction and nocturnal enuresis. Twelve children had secondary enuresis, and 103 children had primary enuresis. Surgical removal of upper airway obstruction led to a significant decrease In or complete cure of nocturnal enuresis In 87 (76%) of the children studied. Eleven children were also studied with polysomnographlc tracings In an attempt to determine a relationship between their sleep patterns and nocturnal enuresis. (OTOlARYNGOL HEAD NECK SURG 1991;105:427,)
HISTORICAL REVIEW Although much has been written about nocturnal enuresis, little has been reported about its relationship to upper airway obstruction. Simmons et al. I noted that children having episodes of sleep apnea of 10 seconds or longer occurring repeatedly during the night can become enuretic and may report nightmares. They presented case reports of an 8-year-old boy and a IS-yearold girl whose nocturnal enuresis stopped immediately after upper airway obstruction was surgically eliminated (by tonsillectomy and adenoidectomy and by tracheostomy, respectively). Guilleminault and Dement' studied children with sleep apnea syndrome. Additionally, they examined two adults with intermittent nocturnal enuresis for whom urologic evaluations were sought before their evaluation for sleep apnea. In both cases, the enuresis ceased after successful surgical management of their sleep apnea. Others':" also mention the relationship between nocturnal enuresis and sleep apnea. In 1978, an adenoidectomy was performed by one of the authors (D.J.W.) on a 7-year-old boy with chronic recurrent otitis media with effusion (OME),
From the Section of Otolaryngology and Audiology, Department of Surgery (Dr. Weider and Ms. West) and Department of Psychiatry (Dr. Sateia), Dartmouth-Hitchcock Medical Center. Funded (in part) by the Hitchcock Foundation. Received for publication Sept. 13, 1990; revision received Feb. 12. 1991; accepted Feb. 20, 1991. Reprint requests: Dudley J, Weider, MD, Section of Otolaryngology and Audiology. Dartmouth-Hitchcock Medical Center. 2 Maynard St., Hanover. NH 03756.
23/1/29051
clinically obstructive adenoids, and a severe snoring problem. At his 6-month follow-up visit, this child's mother reported that he had had no further episodes of OME, that his breathing was significantly improved, and that his snoring had stopped. In addition, she asked why he had stopped bedwetting after his adenoidectomy. This child had been a primary enuretic (never having had a significant "dry period'T.l" who experienced nightly enuresis despite parental attempts to limit fluid intake before bed and to take him to the bathroom during the night. After the adenoidectomy, he stopped bedwetting immediately, with virtually no breakthrough episodes. It was this case that provided the impetus for our study. Since that observation, members of our section have routinely asked parents of children manifesting large tonsils, adenoids, or other symptoms of severe upper airway obstruction if their child has a bedwetting problem. We now have collected about 115 cases over a 12-year period. We have previously reported our results with 35 patients.' We would like to stress that upper airway obstruction is not the primary cause of nocturnal enureses in the majority of cases. However, we would like to present the subject of upper airway obstruction as one etiologic factor of nocturnal enuresis.
Purpose The purpose of this article is to demonstrate the effect of tonsillectomy and adenoidectomy and related upper airway surgery on the frequency of nocturnal enuresis in a clinically selected pediatric population. Secondly, it is our hope to heighten the awareness of physicians about the association between sleep disorders caused
427
Downloaded from oto.sagepub.com at University of Warwick on December 10, 2016
OtolaryngologyHead and Neck Surgery
428 WEIDER at 01.
All Patients 5.6
Table 1. Initial clinic visit (N = 115)* Reason for visit
No.
Tonsillitis Ear disease Chronic mouth breathing Allergy Dental malocclusion Sieep apnea
22 46
115 children; age range 3-19 98 males; 17 females
32 14
20 16
'Several patients had more than one presenting complaint.
Preop
1 Month
6 Months 12 Months
Fig. 1. Average number of enuretic nights per week preoperatively and at 1, 6. and 12 months postoperatively for the entire series of 115 patients (98 maies and 17 females. ages 3 to 19 years).
by upper airway obstruction and nocturnal enuresis in the pediatric population. This study corroborates the important findings from our earlier smaller series."
METHODS Patients Our study included 115 children (98 boys and 17 girls) between the ages of 3 and 19 years who were referred to us for various otolaryngologic problems, but who all had symptoms of upper airway obstruction. All children selected were obligate nighttime mouth breathers who additionally had nocturnal enuresis. All snored and many seemed to obstruct for varying periods of time. Each was described as either an extremely restless or an extremely sound sleeper. When taken to the bathroom during the night. most children were difficult to arouse and did not remember the event. Each patient's urologic history was reviewed for the presence of urinary tract infections and other problems relating to the genitourinary system. In one case, a 15-year-old girl was referred to the urology department; no pathology was found. Table 1 shows the types of pathology our patients manifested. The study consists of 103 children with primary enuresis (children who had had enuresis since birth) and 12 children with secondary enuresis (children who had had an interval of dryness lasting longer than 6 months). '.6 Procedures
All parents who brought children to our clinic between July 1978 and October 1987 with symptoms of significant upper airway obstruction were asked if their child had any symptoms of nocturnal enuresis. If so,
the child was included in the study. A careful history was taken of snoring. nighttime obstruction, daytime and nighttime mouth breathing, chronic rhinorrhea. allergy, and excessive daytime sleepiness (EDS). All children in the study snored and some experienced actual obstructive apnea. All children had some type of surgery to eliminate upper airway obstruction. The most common procedure performed was a tonsillectomy, adenoidectomy. or both. All surgery was done to provide a more functional upper airway in a population of children who snored and who had been observed to have frequent episodes of obstruction. In no case was surgery performed to provide a cure for nocturnal enuresis nor were any promises made that such a cure would result. Each patient's history of enuresis was simply observed preoperatively and postoperatively and recorded as described. Six children were studied preoperatively with polysomnography in our sleep laboratory by one author (M.J.S.), Five additional children from other institutions were also studied polysomnographically (Table 2). All patients reported in this article underwent surgery for relief of upper airway obstruction. One patient had a submucous resection, and one had a combination submucous resection and intranasal polypectomy and ethmoidectomy (8-year-old boy with cystic fibrosis and nasal polyposis). Two patients (both male) had adenotonsillectomy in combination with bilateral inferior turbinoplasties (as described by Mabry") and partial uvulectomies. Both presented with what was considered to be allergically related turbinate hypertrophy in addition to tonsil and adenoid hypertrophy. The remaining III children had either tonsillectomy or adenoidectomy or combined adenotonsillectomy, The modalities measured by polysomnography were electroencephalogram (EEG), electro-oculogram (EOG), breathing from the nose and/or mouth (by way of thermisters attached under the nose and mouth), electrocardiogram (ECG), and intercostal electromyogram (EMG) of the thoracic cage. A special mattress was used to register an interruption in electrical current when the child wet the bed. Our ear oximeter was not
Downloaded from oto.sagepub.com at University of Warwick on December 10, 2016
Volume 105 Number 3 September 1991
Table 2. Sleep Patient no./age lvears)
1/12 2/13 3t/5 4t/12 5/6 6t:j:/6 7t/10 8/7 9t/8 10/6
11/6
Nocturnal enuresis in children with upper airway obstruction
429
study (polysomnography) results "10 Sleep Tvpe of enuresis·
1° 1° ?
? 1° 2° 1° 1° 2° 1° 1°
Slage 1
8.8 5.5 10.7 50.9 13.8 10.2 3,7 13.8 4.2 12.6 3,7
Slage 2
Slage 3/4
REM
RDI
40.0 49.9 584 193 42.8 51,6 514 51.8 49.7 46,0 555
27.6 21.9 12,8 21.0 224 26,8 26,1 20.7 307 20,0 26.2
22.4 23.1 17.2
6.0 1.3 06
20.3 11.3 18,6 13.5 14.9 21.0 13,8
17.0 16.0 0 3.5 2.6 3.8 18
Surgical response
No No
No No
Yes Yes surgery surgery Yes Yes surgery No surgery No Yes
ROt, Respiratory disturbance index (combined apnea and hypopnea per hour 01 sleep): REM. rapid eye movement. '1 ° = Primary noctural enuresis; 2° = secondary nocturnal enuresis, tReferred to DHMC for sleep studies, but not included in our operative series, :j:Studied post-adenoldectorny.
used in the first five patients studied because it was too bulky to permit comfortable sleep in the age group being studied. (More recent technologic advances have eliminated this problem.) Information contributing to our database was acquired by interviewing patients and their parents 1 month, 6 months, and 12 months after surgery. A questionnaire was also sent to all parents of patients. Clinical data for our entire population are presented in Fig. 1; data for the six subgroups (classified by symptom categories and patterns of enuresis) are presented in Figs. 2 through 7.
RESULTS For the group as a whole, we observed a 66% reduction in the number of enuretic nights per week 1 month after surgery. By 6 months, a 77% reduction was observed, which has remained constant (Fig. 1). Twelve children, ages 4 to 12 years, manifested secondary nocturnal enuresis (Fig. 2). In this group, actual dry periods ranged from 1 to 4 years. Onset of secondary enuresis invariably coincided with the onset of symptoms of upper airway obstruction. By the end of the first postoperative month there was an 89% reduction in the number of enuretic nights per week. Six months after surgery, all twelve patients had stopped bedwetting (0 enuretic nights per week), a result that was maintained until at least 1 year after the original surgery. We then analyzed data from our group of children who exhibited irregular nocturnal enuresis (Fig. 3). Children in this group exhibited typical dry periods of I week to I month, after which they resumed enuresis on an almost nightly basis. One month after surgery,
this group experienced a 68% reduction in enuretic nights per week and almost no further reduction thereafter. Five children stopped bedwetting completely by the end of the first month and maintained dryness. One patient was unaffected by the surgery and maintained a pattern of four enuretic nights per week. Another child, age 6 years, went from seven enuretic nights per week to 0 enuretic nights per week for 3 weeks (the second to fourth postoperative weeks) and then resumed a pattern of five enuretic nights per week for the remainder of the year. Figure 4 depicts a group of eight boys, ages four to seven years, who exhibited EDS. All but one had primary enuresis. One had secondary enuresis (a 7-yearold with a dry period between the ages of 2 and 7) (Fig. 2). By the end of the first postoperative month. this group experienced a 91% reduction in the number of enuretic nights per week and a complete resolution of EDS. By 6 months, all children had experienced complete cessation of enuresis, and all maintained dryness until at least I year after surgery. Figure 5 depicts our largest group: children ages 3 to 19 years with primary nocturnal enuresis without EDS. These patients typically had a history of nighttime mouth breathing or snoring in combination with a history of enuresis. One month after surgery, there was a 59% reduction in enuretic nights per week; at 6 months this increased to a 71% reduction, with slight additional improvement by I year. This group was further subdivided into children who experienced a reduction in frequency of their nocturnal enuresis (Fig. 6) and those who did not (Fig. 7). The "responders" (Fig. 6) had an 81% reduction in the number of enuretic nights per week at 1 month, which
Downloaded from oto.sagepub.com at University of Warwick on December 10, 2016
430
OtolaryngologyHead and Neck Surgery
WEIDER et 01.
Secondary Nocturnal Enuresis C ill
'';: l':l
7
6.0
6
ill ill
.~
12 children; age range 4-12 10 male; 2 female
c,
~
5
.l!l
4
'r:
2
0
7
c .~ -:; 6
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1 Month
6 Months 12 Months
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Irregular Nocturnal Enuresis 8 children; age range 4-12 7 male; 1 female
5.0
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6.0 8 children; age range 4-9 8 male; 0 female
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