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 Oral Maxillofacial Surg Clin N Am 14 (2002) xi – xii

Preface
Current medical and surgical management of sleep
related breathing disorders

N. Ray Lee, DDS

Guest Editor

Since the time of early man, we have gathered in- brovascular and gastrointestinal complications is an
formation on the endless psychologic and emotional all too real fact of life in the world of sleep depriva-
observations of the mysterious state of sleep. Con- tion. The gentle prod to ‘‘sleep tight’’ is obviously not
current with the onset of the millennium is a renewed granted automatically. We are simply not biologically
zeal in our quest for increased illumination on the prepared to handle interruptions in sleep.
matter of sleep. Thanks to sleep research pioneers So, with increased awareness of the importance of
such as Dr. William Dement, sleep medicine is now sufficient sleep, more Americans than ever are seek-
recognized as a specialty by the American Medical ing treatment for sleep disorders. As a surgeon, the
Association (1996). Through the avenues of scientific gratification of treating patients with sleep-related
investigation and dramatic advances in technology, breathing disorders is incomparable to any other
our knowledge and understanding of the dynamics of aspect of my practice. To witness the restoration of
sleep is rapidly increasing. a patient’s quality of life is a gift to a surgeon; as I
Even so, Americans continue to move in the listen to a patient’s expression of gratitude I am struck
wrong direction. We have reduced our average sleep by how significant a role medicine has played in that
time by 30% since Thomas Edison invented the person’s literal reawakening to the full spectrum of
light bulb. We have increased our annual working life enjoyment.
and commuting time by more than150 hours. Yet, Thus, the term ‘‘sleep surgeon’’ gives added
the ideal amount of sleep remains the same: one- dimension to a microsubspecialty that is indeed a
third of the average life span or approximately multidisciplinary culling from numerous surgical spe-
24 years. cialties. The evolution of the sleep-related breathing
Americans are paying a price for insufficient sleep disorder surgeon is still in progress. It is clear that
both financially and otherwise. Tens of billions of future data collection is imperative to continued
dollars are expended every year in lost productivity, surgical success. As surgical techniques evolve, it is
accidents, and other byproducts of sleep deprivation. incumbent upon each surgeon to share and dissem-
Too little sleep delivers a devastating impact on the inate knowledge to perfect the interdisciplinary
quality of human life. The immeasurable misery of expertise that is necessary to advance the field.
excessive daytime drowsiness, family dysfunction, This publication coalesces a multidisciplinary ap-
loss of life and property, disabilities secondary to proach in the treatment of sleep-disordered breathing
psychologic and behavioral malfunction, and cere- (SDB). The commitment of the authors to unify their

1042-3699/02/$ – see front matter D 2002, Elsevier Science (USA). All rights reserved.
PII: S 1 0 4 2 - 3 6 9 9 ( 0 2 ) 0 0 0 3 1 - 6
xii Preface / Oral Maxillofacial Surg Clin N Am 14 (2002) xi–xii

diverse expertise brings forth a unified successful duction staff of Elsevier Science for making this
treatment of sleep-related breathing disorders. Al- publication possible.
though there is no universally accepted treatment
protocol, site specific surgical reconstruction of the N. Ray Lee, DDS
upper airway is generally accepted. Let it be our goal Private Practice, 716 Denbigh Boulevard, Suite C-1
to continue to bring the knowledge of science, experi- Newport News, VA 23608
ence, and treatment of complications to publication Assistant Clinical Professor, Department of Oral and
to advance our specialty in the successful treatment Maxillofacial Surgery, Medical College of Virginia
of SDB. Virginia Commonwealth University
Working with friends, colleagues, and contrib- 520 North 12th Street, Richmond, VA 23298
utors on this publication has been a valuable and Assistant Clinical Professor, Department of
interesting learning experience, and I thank them Otolaryngology—Head and Neck Surgery
all for their dedication and contributions to the Eastern Virginia Medical School
Oral and Maxillofacial Surgery Clinics of North P.O. Box 1980 Norfolk, VA 23501
America. I also thank John Vassallo and the pro- E-mail address: [email protected]
 Oral Maxillofacial Surg Clin N Am 14 (2002) 273 – 283

History and epidemiology of sleep-related

breathing disorders
Robert D. Vorona, MDa,b,*, J. Catesby Ware, PhDc,d
a
Division of Sleep Medicine, Department of Internal Medicine, Eastern Virginia Medical School, USA
b
Sleep Disorders Center, Sentara Norfolk General Hospital, 600 Gresham Drive, Norfolk, VA 23507, USA
c
Departments of Internal Medicine and Psychiatry, and Division of Sleep Medicine, Eastern Virginia Medical School, USA
d
Sleep Disorders Center, Sentara Norfolk General Hospital, 600 Gresham Drive, Norfolk, VA 23507, USA

By the late twentieth century, the medical com- William Osler. In The Principles and Practice of
munity recognized that snoring and daytime sleepi- Medicine, Osler wrote in his chapter on obesity, ‘‘A
ness were signs of obstructive sleep apnea syndrome remarkable phenomenon associated with excessive
(OSAS). Parts of the sleep apnea syndrome complex fat in young persons is an uncontrollable tendency to
were, however, known many years earlier by an sleep like the fat boy in Pickwick’’ [8]. But because
insightful few. The 1965 polysomnographic study of the lack of understanding of the various conditions
that described obstructive, central, and mixed sleep that could cause sleepiness and the absence of tech-
apnea events during sleep was the beginning of the niques to study sleep, the term Pickwickian described
objective study of what we now recognize as sleep a heterogeneous group of patients with little regard to
apnea syndrome [1]. Less well known are some specific etiology.
earlier descriptions of the problem. Symptoms of After Burwell’s work, the term Pickwickian typ-
heavy snoring and excessive daytime sleepiness were ically indicated obesity accompanied by somnolence
reported in a patient with acromegaly in 1896 [2]. and lethargy, hypoventilation, hypoxia, and second-
Lavie [3] identified what may have been the first ary polycythemia, but not necessarily repetitive sleep
reported case of sleep apnea in a patient who had apnea events. By including hypoventilation and
components of both obstructive and central apnea polycythemia as part of the syndrome, most of the
events [4]. Lavie also described two other 1889 cases sleep apnea patients seen in sleep disorder centers
with daytime sleepiness and failed respiratory today do not have Pickwickian syndrome. The
attempts during sleep [5,6]. The description of these development of basic polysomnographic tools and
patients leaves no doubt that the phenomenon of procedures in the 1950s and 1960s provided a meth-
obstructive sleep apnea, although unnamed and not od to study causes of daytime sleepiness. Polysom-
understood, was recognized well before the advent nography led to the understanding that daytime
of polysomnography. sleepiness often originated from intrinsic sleep dis-
C.S. Burwell is often credited with first using the turbances in the patients’ sleep. Prior to polysom-
name Pickwickian syndrome when describing an nography, only secondary characteristics of OSAS
obese patient with respiratory acidosis, heart failure, were recognized and treated.
and sleepiness [7]. The term Pickwickian had actu-
ally been used, however, several times earlier to
describe sleepy obese patients including the use by The growth spurt of the 1970s and 1980s

Perhaps the number of publications in the field

* Corresponding author. best reflects the explosion in interest in OSAS.
E-mail address: [email protected] (R.D. Vorona). Publications addressing sleep apnea in some form

1042-3699/02/$ – see front matter D 2002, Elsevier Science (USA). All rights reserved.
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274 R. Vorona, J.C. Ware / Oral Maxillofacial Surg Clin N Am 14 (2002) 273–283

increased by nearly tenfold in the 1980s when events per hour of sleep lasting at least 10 seconds
compared with the 1970s (Fig. 1). At the same each [9]. They extended the investigation of apnea to
time, the reports on Pickwickian syndrome de- children [10 – 13]. Early on, they also hypothesized
creased. This decrease in part was from recognition that sleep apnea might be related to sudden infant
of the heterogeneous population that the term Pick- death syndrome [14]. The recognition of children and
wickian encompassed. infants as possible at-risk populations along with his
An early champion of OSAS in the United States other work considerably increased the interest in and
was a young Frenchman, Christian Guilleminault, at study of sleep apnea.
Stanford University. Despite a focus on insomnia and Researchers employed a variety of techniques to
narcolepsy by the small community of those studying better understand what occurs during sleep apnea
sleep and sleep disorders in the United States, Guil- events. Lateral imaging of the upper airway using
leminault forged ahead with his interest in sleep Xerography in a small number of severe OSAS
apnea, motivated by the idea that differences in the patients revealed no specific pathology when awake
control of vital functions during sleep contributed to a but a clear collapse of airway space at the base of the
number of medical disorders. Returning to Europe tongue during sleep [15]. Compared with controls,
where there was considerable interest in the Pick- fluoroscopy and computed tomography indicated a
wickian syndrome, Guilleminault recorded several more narrow section of the airway in patients in the
hundred patients at a sleep medicine clinic at La region posterior to the soft palate [16]. Direct obser-
Salpetriere Hospital in Paris. He realized that breath- vation with fiberoptic endoscopy of the sudden dra-
ing irregularities and apnea occurred in a variety of matic closure of the airway suggested the possibility
patients, not necessarily obese ones (personal com- of an active process; an alternative explanation was
munications, March 2001). Later after his return to that muscle relaxation and a negative pharyngeal
Stanford University, Guilleminault and associates airway pressure accounted for the rapid airway col-
helped to demonstrate that OSAS caused excessive lapse and apnea events [17,18].
daytime sleepiness more often than narcolepsy. They In the late 1970s, the recognition that OSAS can
developed an objective definition of OSAS as five occur in families added another dimension to the

Fig. 1. The number of publications listed in PubMed (National Library of Medicine, Rockville Pike, MD) generated by the
search terms ‘‘sleep apnea’’ and ‘‘Pickwickian’’ for 5-year blocks, 1965 – 1999.
 R. Vorona, J.C. Ware / Oral Maxillofacial Surg Clin N Am 14 (2002) 273–283 275

problem and further emphasized the complexity tomy was used primarily for end-stage OSAS
without clarifying the etiology [19,20]. For example, patients, the development of a surgical technique
the apnea-potentiating trait of a small or retrognathic with the potential of treating less severe cases was
mandible can have both genetic and environmental welcomed. The UPPP allowed some patients to avoid
contributing factors. Analysis of lateral cephalometric a tracheostomy. In addition, the UPPP fit well into the
radiographs did help identify those with mandibular conceptualization of the etiology of OSAS. Specific
deficiencies and a shallow posterior airway space upper airway obstructions, for example, large tonsils
(PAS), although no imaging technique or other test and adenoids [33,34], nasal obstruction [35], and
when patients were awake was able to identify all supraglottic edema [36], were understood to cause
sleep apnea cases [21]. the obstruction and apnea. Evidence suggested, how-
ever, that other factors also played a role.
A second UPPP benefit was its high failure rate.
Treatment Because the UPPP was far from 100% successful and
was not free of morbidity and mortality, documenta-
Surgery tion of both the presence and severity of OSAS was
necessary before performing an UPPP. Therefore,
The difficulty in treating OSAS patients tempered the availability of the UPPP as a treatment option
the excitement of recognizing the problem of OSAS resulted in the consistent polysomnographic study of
as a primary cause of excessive daytime sleepiness. a large number of patients with symptoms of OSAS.
The standard treatment of tracheostomy was trau- These clinical studies raised the awareness of the
matic but successful in relieving OSAS [22,23]. A prevalence of OSAS and allowed the development of
tracheostomy with its accompanying improvement in an appreciation of the range of severities of OSAS
areas other than daytime sleepiness gave hints of the (from 0 to 100+ events per hour of sleep). This
complexity of the sleep apnea problem. For example, quantification of the frequency of apnea events also
this surgical procedure not only relieved the apnea provided a baseline for comparing different tech-
events but also reduced cardiac arrhythmias [24] and niques for treating OSAS patients. Pre- and post-
improved the ventilatory response to CO2 [25]. This operative polysomnographic studies of OSAS
helped foster the realization that intermittent obstruc- patients also led to an early awareness that subjective
tion during sleep had pervasive effects on neuro- reporting of improvement by the patient often was not
logical, cardiac, and respiratory functioning. It also congruent with polysomnographic findings [37]. The
helped to emphasize the importance of normal sleep approximate 50% success of the UPPP [38], recog-
and raised the problematic question of ‘‘How many nized early on from those doing postsurgical sleep
apnea events during sleep are too many?’’ studies, spurred the search for other treatments.
Even with the threat of a tracheostomy, most Treatment failures also reinforced the idea that for
obese apnea patients could not lose weight by dieting. some OSAS patients, more than a mechanical
Treating OSAS patients with a weight loss prescrip- obstruction of the upper airway was involved. The
tion, although often beneficial when a patient could differences between men and women [39], the effects
lose weight [26], succeeded only in a minority of of aging [40], and the effects of sleep stage on apnea
patients. In addition, there was a high recidivism rate frequency [41] indicated that OSAS involved both
for those who initially lost weight. Therefore, bari- physiological and pathophysiological (as well as
atric surgery to induce weight loss and treat obstruc- anatomical) factors. A number of nonanatomical
tive sleep apnea was introduced in the late 1970s [27] factors may play a role in OSAS. The tone of the
and continues to be used successfully in selected pharyngeal dilating muscles, pharyngeal extramural
cases [28]. pressure, and pharyngeal compliance all may con-
Following the recognition that retrognathia could tribute to OSAS [42], and treatment without regard to
contribute to apnea events, mandibular surgery was the specific etiology is likely to have a significant
used successfully to treat retrognathic OSAS patients failure rate.
[29,30]. In addition, mandibular surgery was success-
fully used to treat OSAS in an obese patient [31]. Oral appliances
The treatment with the greatest impact on OSAS,
although unfortunately not the greatest success rate, Despite the logic of pulling the mandible forward
was that of the uvulopalatopharyngoplasty (UPPP), to open the airway, the sleep community greeted the
[32]. The UPPP was a less drastic but less effective first reports of using an oral appliance to treat OSAS
procedure than tracheostomy. Because a tracheos- with considerable skepticism. As is usually the case
276 R. Vorona, J.C. Ware / Oral Maxillofacial Surg Clin N Am 14 (2002) 273–283

for new procedures, the early reports were not con- and humidification. Once the most common treat-
trolled trials [43,44]. But persistence on the part of ments for OSAS was nonsurgical; patients with milder
the dental community, more rigorous studies showing symptoms of OSAS were evaluated and treated. This
polysomnographic efficacy (rather than self report), led to an explosion in the number of sleep disorder
and the developing need to treat patients intolerant or facilities and patients studied. This growth also rein-
nonresponsive to other treatments gained the oral forced the need for a credentialing body for clinicians
appliances wider use and the opportunity to improve (American Board of Sleep Medicine) and an accred-
and become an important treatment option. itation body for sleep disorders centers (American
Academy of Sleep Medicine). The availability of
Pharmacological treatment CPAP allowed for a change in location to nonhospital
based centers. Treating less severe patients also
Early pharmacological treatment attempts in- opened the possibility for the investigation and use
cluded use of medroxyprogesterone [45,46] and even of portable diagnostic equipment and self-titrating
strychnine in a ‘‘Don’t do this at home study’’ [47]. CPAP devices. Thus, moving some of the diagnostic
The motivation to use strychnine was to correct upper and treatment procedures to the home is now un-
airway hypotonicity. More recently, researchers have der investigation.
attempted to stimulate electrically the upper airway
musculature [48]. One of the problems inherent in
this technique is the arousal from sleep produced Epidemiology of OSAS
by the electrical stimulation. Thus, it is difficult
to separate the specific effects of stimulating the The problem of OSAS spans all age groups and
muscle from the more general effects of an arousal both sexes and is found throughout the globe. Obesity
from sleep. is often but not always a critical determinant of
One of the successful early pharmacological treat- OSAS. An enlarging body of work reveals the car-
ments was the use of protriptyline [49,50]. Although diovascular and cerebrovascular links and consequen-
protriptyline, an alerting tricyclic antidepressant, may ces of OSAS.
have helped because of improved alertness and mood, Classically, OSAS was thought to be a syndrome
it also reduced apnea events possibly by increasing of the middle-aged. All age groups may have apneic
muscle tone. One of the most pronounced effects of events during sleep, however. In over 1000 healthy
protriptyline is the suppression of REM sleep, the full-term infants who ranged in age from 2 to 28
sleep stage accompanied by loss of antigravity weeks, the absolute numbers of obstructive or mixed
muscle tone. The stage of REM sleep can have the apneas in these infants were quite low [54]. Of
longest apnea events with the most severe oxygen interest, males between 8 and 11 weeks of age were
desaturations. To what degree REM sleep and apnea more apt to have obstructive apneic events and more
would return after long-term use is unknown. One events per hour than females. The apnea events tended
study indicated that apnea returns after a year, but to decline in length with age. Apneas do not always
patients continued to report feeling better and had connote disease in infants. Central apneas (apneas
slightly higher O2 saturation baseline [51]. without respiratory effort) may occur in normal
infants, and even protracted central apneas with desat-
Continuous positive airway pressure uration may not be of import [55,56].
Older children are not exempt from OSAS. Red-
The technique of using continuous positive airway line and associates found 1.6% of their children or
pressure (CPAP) in the upper airway essentially teens (2 – 18 years old) had sleep disordered breathing
became the nonsurgical tracheostomy [52] and trans- (SDB) as defined by a respiratory disturbance index
formed the field of sleep disorders medicine by pro- of greater than or equal to 10 events per hour [57].
viding a low morbidity treatment with a high success Between the ages of 2 and 8 years, children are most
rate. If used by the patient, it keeps the upper airway apt to have OSAS [58]. The tonsils and adenoids are
patent. A significant number of patients ( > 30%), of great importance in putting these children at risk
however, do not continue to use CPAP over the long for sleep disordered breathing events. In contrast with
term [53]. Practical problems of administering positive adults in whom tonsillectomy and adenoidectomy are
air pressure to the upper airway comfortably were rarely curative for OSAS, in children the same
difficult to overcome. But FDA approval in the United operative procedure is often but not always effective
States in the mid-1980s and product commercializa- [59]. Frank anatomic abnormalities in children are
tion led to continued refinements in mask fit, material, not, however, the only reason for obstruction of the
 R. Vorona, J.C. Ware / Oral Maxillofacial Surg Clin N Am 14 (2002) 273–283 277

upper airway [58]. Neuromuscular function alteration Young et al performed a critical study to assess the
can also be of import. Cerebral palsy and muscular impact of sleep disordered breathing in middle-aged
dystrophy put children at risk for OSAS [60,61]. adults [69]. In this study of middle-aged Wisconsin
Skeletal abnormalities also put children at risk for state employees aged 30 – 60 years, some 9% of
sleep-disordered breathing [59]. Children with OSAS women and 24% of men had a Respiratory Disturb-
may like their adult counterparts be obese. Redline et ance Index (RDI) of greater than or equal to 5 per
al found what they described as a moderate linkage hour. When coupled with complaints of excessive
with an odds ratio for obesity and SDB of 4.6 [57]. daytime sleepiness, 2% of women and 4% of men
They also linked respiratory disease of both the upper manifested OSAS. This study is perhaps the best
and lower tract. In contrast with data in adults, they measure of the prevalence of OSAS in the US adult
did not, however, find a clear relationship between population to date.
sex and SDB in children. A study in Spain examining the prevalence of
Work from Spain has investigated sleep disor- SDB and OSAS in a 50 – 70-year-old population
dered breathing in children 12 – 16 years old. This found that 29% of the patients (28% men, 30%
study of 101 teens, buttressed by the use of limited women) had an RDI of greater than or equal to
polysomnography, found that 29% snored and 17.8% 5 per hour. Although there was no sex difference
had a respiratory disturbance index of greater than or for number of sleep disordered breathing events, only
equal to 10 [62]. Only 1.9%, however, also had men were symptomatic, and therefore only the men
symptoms indicative of the diagnosis of OSAS. The were diagnosed with OSAS as defined both by sleep
authors noted that this frequency was akin to that disordered breathing and symptoms [70]. Another
found in younger children. study used oxygen desaturation events of greater than
Middle school children with poorer performance or equal to 4% as a surrogate to screen for OSAS in a
are more likely to snore [63]. It appears that even 40 – 64-year-old group; the authors projected from
medical students who snore are more likely to fail their sample that an apnea occurrence of more than
examinations [64]. Potential complications of un- or equal to 15 per hour occurred in 20.3% of the men
treated OSAS in children include failure to thrive, and 7.6% of the women [71].
pulmonary hypertension, cor pulmonale, and arterial Some data suggest even higher rates of sleep
hypertension [13,59,65]. As OSAS usually has been disordered breathing in the elderly. In one study
associated with older patients, the clinician may not investigating the frequency of respiratory disturb-
be as quick to think of OSAS in the child as in the ances in those greater than or equal to 65 years of
adult. Nevertheless, the potential complications, age, 24% of their sample had greater than or equal to
though serious, are remediable [59,66] and there- 5 apneas per hour, and 62% had an RDI greater than
fore warrant that clinicians be cognizant of OSAS or equal to 10 per hour [72]. Over the next 8.5 years,
in children. however, there was no progression in sleep related
OSAS, with its complications including daytime respiratory events [73].
sleepiness and difficulties with memory and concen- The importance of these respiratory events in the
tration, obviously can impair school performance. elderly is controversial. For example, Ancoli-Israel et
Teens already are at risk for difficulties functioning al in 1989 showed that elderly women with sleep
in the morning because of a tendency to delayed sleep disordered breathing had an increased mortality [74].
phase (they tend to go to sleep later and wake up Mant et al [75] in a study of the elderly did not find
later), coupled with high school hours that prod them such increased mortality associated with OSAS as
to start the day early. Concomitant OSAS could only defined by an RDI greater than or equal to 15.
exacerbate this situation and hence deserves consid- Additionally, when Phillips et al. looked at elderly
eration. In addition, sleep loss suffered by teens may subjects who were ostensibly healthy, those with an
exacerbate existing sleep apnea [67]. RDI over or equal to 5 per hour showed no alteration
A study at the Chinese University of Hong Kong in daytime performance [76].
used a questionnaire to study some 1910 students Early evidence indicated that OSAS was over-
followed by limited polysomnographic recordings in whelmingly a male phenomenon [39]. The more
some [68]. They found by questionnaire that some recent information above reveals, however, the male
25.7% had snoring. In the small subgroup who to female ratio more closely approximates 2 – 3: 1.
underwent a limited sleep study, only 2.3% had a Bixler et al [40] showed that 3.9% of men and 1.2%
respiratory disturbance index of greater than 5. of women had OSAS. But premenopausal women
Hence, once again the prevalence of OSAS was and postmenopausal women on hormone replacement
relatively low. therapy had much lower prevalences (0.6% and 0.5%,
278 R. Vorona, J.C. Ware / Oral Maxillofacial Surg Clin N Am 14 (2002) 273–283

respectively) than postmenopausal women (2.7%). breathing when compared with Caucasians [83].
They therefore postulated that the premenopausal Brachycephaly appears to put Caucasians at risk,
status and hormone replacement therapy were pro- whereas soft tissue abnormalities may be of more
tective. Those apneic females who were premeno- importance in African Americans [84].
pausal or postmenopausal and taking hormones were Differences in OSAS between Caucasians and Far
all obese, thus defining the essential role of obesity in East Asians also occur. Ip et al [85] reported that
these subgroups. In addition, Pickett et al found that 4.1% of middle-aged Chinese men have OSAS
the combination of estrogens and progesterone (roughly comparable with Young’s data). Obesity,
decreased respiratory events in nine women posto- however, was a less important exacerbating factor
variectomy and hysterectomy [77]. for OSAS in Chinese than in Caucasians as found by
In a large population of Italian women aged 40 – Young. Caucasian apneics are more obese than Asian
65 years, 19.7% ‘‘always snored’’ and 10.7% had a apneics [86]. If matched for BMI, the Asians with
respiratory disturbance index from 5 to 9 per hour OSAS had more severe disease than Caucasians with
[78]. Nearly 8% had more severe SDB with a OSAS. The Far East Asians had ‘‘a significantly
respiratory index between 10 and 19. Interestingly, shorter anterior cranial base and more acute cranial
in contrast with Bixler et al, this study could not base flexure’’(58). Caucasians and Asians with OSAS
correlate menopausal status and sleep disordered also differed on some soft tissue measures (eg, PAS
breathing. In a study comparing Body Mass Index and mandibular plane to hyoid distances). Liu et al
(BMI) matched men and women presenting to a sleep compared cephalometrics in Chinese and Caucasians
disorders center, men had significantly more apnea in with OSAS and found skeletal differences that they
the young and middle-aged groups but were similar described as ‘‘steeper and shorter anterior cranial
to women in the older age group, presumably when bases’’ [87]. Many but not all of the soft tissue
the women were postmenopausal [79]. structures were similar between Chinese and Cauca-
Sleep disturbances during pregnancy have been sians. These differences may have import in planning
frequently noted with snoring and nocturnal choking either treatment approaches with surgery or mandib-
among the complaints possibly linked to OSAS. Of ular repositioning devices. Further, OSAS should not
127 pregnant women in an outpatient setting, it was be approached as a monolithic syndrome in these
determined that 29.8% snored during pregnancy [80]. different groups.
The majority of these women did not snore prior to Although anatomic differences may exist between
pregnancy. By the last quarter of pregnancy, approx- races with OSAS, obesity is prevalent in its role as a
imately 31% reported awakening choking, a symp- risk factor. Some 60 – 70% of patients with OSAS are
tom the authors attempted to relate to OSAS. This obese [88]. Alternatively, more than 50% of obese
information achieves greater import when one patients with a BMI of greater than 40 kg/m2 have
reviews the data from Franklin et al [81]. In their OSAS [89]. Obesity, although a risk factor for SDB
study of 502 pregnant women, there was a 23% in children and adults, may not be as important in
incidence of nightly snoring in the last week of children [57]. Young’s study of the middle-aged
pregnancy. Those who snored were more than two noted, ‘‘An increase of 1 SD in any measure of body
times as likely to develop hypertension during preg- habitus was related to a threefold increase in the risk
nancy and preeclampsia, and to deliver children with of an apnea-hypopnea score of 5 or higher’’ [69].
growth retardation. A low Apgar score also occurred Grunstein et al showed that increasing central obesity
more frequently in children born to mothers who correlated with worsened sleep apnea [90]. As would
snored habitually. Treating preeclamptic women with be expected, weight loss does reduce OSAS. In a
CPAP demonstrated improved upper airway flow study of 690 patients, a 10% weight gain lead to a
mechanics and blood pressure control [82]. 32% worsening in RDI and a 10% weight loss lead to
Racial differences also occur in OSAS patients. a 26% improvement [91]. Of interest, obesity may
Much of the above information is related to Cauca- have differential effects by gender. Upper body
sians from North America and Western Europe. obesity worsened OSAS in men more than in women
Kripke et al [71] found that 16.3% of the Hispanics [92]. Although there is controversy, some evidence
and other minority patients had 20 or more events per suggests that BMI may be the best predictor of RDI
hour. This was in contrast with only 4.9% of their in women, whereas neck circumference may be the
Caucasian patients. In children, Redline et al [57] best predictor in men [89].
showed that African-Americans were at increased In addition to the above demographics, the oral
risk for OSAS. Elderly African-Americans appear surgeon interested in OSAS must appreciate the
to have twice the risk of severe sleep disordered importance of family history. Pillar and Lavie [93]
 R. Vorona, J.C. Ware / Oral Maxillofacial Surg Clin N Am 14 (2002) 273–283 279

studied the adult children of 45 patients diagnosed risk of coronary heart disease. The Sleep Heart
with OSAS. Remarkably, 47% of these children had Health Study revealed what the authors called ‘‘mod-
OSAS. They also found that an additional 21.9% of est to moderate effects of sleep-disordered breathing
the remaining patients studied had simple snoring. on heterogeneous manifestations of CVD’’ [103].
Finally, no discussion of the epidemiology of They found a more marked relationship for OSAS
OSAS would be complete without discussing some and congestive heart failure than coronary heart
of the recent important information concerning mor- disease. One daunting aspect of the study was that
tality and cardiovascular and cerebrovascular compli- seemingly rather trivial numbers of respiratory dis-
cations. He et al [94] noted that an apnea index of turbances seemed to put one at risk for cardiovascu-
greater than 20 per hour was associated with 0.63 lar consequences. A study in Sweden prospectively
eight-year survival. This is compared with 0.96 eight- tracked patients with coronary heart disease and
year survival in those patients with an index less than OSAS and found that untreated OSAS was linked
20 per hour. As opposed to tracheostomy and CPAP, with an increased death rate from cardiovascular
uvulopalatopharyngoplasty had no impact in decreas- causes [104].
ing this increased mortality rate. Interestingly, there OSAS and stroke have also been associated in the
was a large range in causes of death, a number of sleep literature. The Sleep Heart Health Study not
which were not obviously related to OSAS. Lavie only linked OSAS and heart disease but also linked
et al [95] also found an increased death rate in OSAS with stroke. In fact, the association with stroke
individuals with OSAS. This increased risk of death in this large study was stronger than with coronary
was in those in the fourth and fifth decades of life. heart disease. A study of transient ischemic attack
They postulated that the major risk factor for death (TIA) and stroke with OSAS found a high frequency
was hypertension. of OSAS in both patients with TIA and stroke [105].
Recent data clearly associate OSAS and hyper- The authors pointed out the interesting fact that TIA
tension. The Sleep Heart Health Study evaluated also was clearly linked to OSAS, making it less
sleep disordered breathing and hypertension in likely that the OSAS was a consequence of the
6132 patients who were either middle-aged or elderly cerebral event.
[96]. Despite aggressive attempts at controlling for A number of reasons have been postulated for the
confounding factors, a moderate relationship occurred increased risk of cardiovascular and cerebrovascular
between SDB and hypertension. This relationship events. Several investigators have demonstrated
between SDB and hypertension was dose-dependent increased sympathetic activity in OSAS patients.
(although not linear), thus giving further credence to Somers et al [106] showed that patients with OSAS
the relationship. Peppard et al [97] in the Wisconsin had increased sympathetic nervous system activity
Sleep Cohort also discovered a dose-dependent rela- both awake and asleep. In contrast with the norm,
tionship between sleep disordered breathing and these patients showed elevations in blood pressure
blood pressure level four years later. A large study during sleep. When CPAP was applied to these
by Ohayon et al [98] comprising 13,057 subjects also patients, both sympathetic activity and blood pressure
linked OSAS and hypertension. An earlier study by were reduced. Hedner et al [107] interrogated the
Carlson et al linked OSAS and hypertension as well as response of sympathetic activity in OSAS treated by
age and BMI [99]. These associations between OSAS CPAP by measuring norepinephrine, vanilmandelic
and hypertension are not solely limited to the adult acid, and metanephrines. They found decreases in
population. Marcus et al [100] have compared blood catecholamines but change neither in cardiac struc-
pressure readings in children with OSAS and children ture nor in blood pressure.
with primary snoring. Diastolic blood pressure was
more elevated in the group with OSAS. Conversely,
the presence of hypertension in a patient should make Directions in the twenty first century
the clinician think of OSAS. Worsnop et al [101]
found that 34% of untreated and 38% of treated In addition to refinements in surgical techniques
hypertensive patients had OSAS. for treating OSAS, we expect that there will be
Hypertension is a major risk factor for coronary considerable advances in the understanding of sleepi-
heart disease and cerebrovascular disease. OSAS and ness and other OSAS symptomatology. Although
these two major killers have been investigated. Hung some details are missing, we now know that repeated
et al [102] used polysomnography to study patients disturbances in respiration during sleep play a major
shortly postmyocardial infarction but outside the role in the typical symptoms of OSAS. In addition,
hospital. OSAS was associated with an increased we are beginning to understand how sleep arousals,
280 R. Vorona, J.C. Ware / Oral Maxillofacial Surg Clin N Am 14 (2002) 273–283

hypoxic and hypercapnic insults, and accompanying structive sleep apnea and near miss for SIDS: I. Re-
autonomic liability contribute to OSAS symptomol- port of an infant with sudden death. Pediatrics
ogy. These events may not account, however, for the 1979;63(6):837 – 43.
[12] Guilleminault C, Ariagno RL, Korobkin R, et al.
complete picture. Obesity itself may contribute to the
Mixed and obstructive sleep apnea and near miss
sleepiness [89,108]. The question ‘‘Are obese apnea
for sudden infant death syndrome: 2. Comparison
patients of similar-frequency apnea more sleepy than of near miss and normal control infants by age. Pe-
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[17] Borowiecki B, Pollak CP, Weitzman ED, et al. Fibro-
Acknowledgments optic study of pharyngeal airway during sleep in
patients with hypersomnia obstructive sleep-apnea
The authors were supported in part by NIH award syndrome. Laryngoscope 1978;88(8, Pt 1):1310 – 3.
[18] Guilleminault C, Hill MW, Simmons FB, et al. Ob-
# HL03652-01A1 to JCW.
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 Oral Maxillofacial Surg Clin N Am 14 (2002) 285 – 292

Pathophysiology of obstructive sleep apnea

M. Safwan Badr, MD
Medical Service, Detroit Veterans Affairs Medical Center, Detroit, MI, USA
Division of Pulmonary Critical Care and Sleep Medicine, Department of Medicine, Wayne State University School of Medicine,
3990 John R, Detroit, MI 48201, USA

Obstructive sleep apnea (OSA) is a fairly common deep sleep. The electroencephalogram (EEG) shows
disorder with significant adverse health consequen- decreased frequency and increased amplitude as sleep
ces; however, the pathogenetic mechanisms remain progresses from stages 1 through 4.
incompletely understood. Upper airway (UA) patency
is determined by several neuromuscular and non- Rapid eye movement sleep
neuromuscular factors including the following: (1)
UA-dilating muscle activity, (2) the collapsing trans- REM sleep is the stage when most dreaming
mural pressure generated during inspiration, (3) occurs. While all antigravity muscles are paralyzed;
changes in caudal traction, (4) vasomotor tone, and there is increased activity of the central nervous
(5) mucosal adhesive forces. This article addresses system (CNS), and the EEG is fast with low ampli-
the effect of sleep on UA function and how these tude waves (resembling an ‘‘awake’’ EEG). Thus,
factors conspire to cause UA obstruction. REM sleep is described as ‘‘paradoxical’’ sleep,
The occurrence of UA obstruction during sleep showing an active CNS and paralyzed periphery.
and not wakefulness implicates the removal of the REM sleep occurs in cycles every 90 to 110 minutes.
wakefulness stimulus to breathe as a key factor Its duration is often reduced in the laboratory en-
underlying UA obstruction during sleep. Most of vironment, especially if complex instrumentation
the data on sleep effect are derived from studies is used.
during nonrapid eye movement (NREM) sleep, given
the difficulty in achieving REM during invasive
studies in the laboratory environment. Effect of sleep on ventilation

Although sleep is viewed as a quiet resting period,

Physiology of sleep judging by the ‘‘passive’’ appearance of a sleeping
subject, this is far from true. The sleep state repre-
The sleep state is classified into two distinct broad sents a challenge rather than rest period for the
states: NREM sleep and REM sleep. ventilatory system. The effects of sleep on ventilation
set the stage for the development of sleep apnea and
Nonrapid eye movement sleep may provide the mechanistic link in susceptible
individuals. Loss of the wakefulness stimulus to
NREM sleep is classified into four stages by breathe is the key factor driving changes in breathing
increasing depth from 1 through 4. Stage 1 is light during sleep. Thus, ventilation becomes dependent
sleep, slightly beyond drowsiness; stage 4 represents on chemoreceptor and mechanoreceptor stimuli.
Consequences of loss of wakefulness include reduced
tidal volume, reduced activity and UA dilators,
E-mail address: [email protected] reduced UA caliber and loss of load compensation
(M.S. Badr). [1,2] (Fig. 1).

1042-3699/02/$ – see front matter D 2002, Elsevier Science (USA). All rights reserved.
PII: S 1 0 4 2 - 3 6 9 9 ( 0 2 ) 0 0 0 3 6 - 5
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 CHAPTER XVII
Some Real Investigating

Moselle called from the telephone in the back hall:

“Oh, Miss Sim! It’s the gentleman again—Mr. Pangborn!”

Sim hurried to the instrument while the other girls

looked at one another, laughter in their eyes and with
hearts beating faster.

“Our old friend of the orchard masquerade,” said Arden.

“Do you suppose he’s going to vanish again—take

another name and get into some other mystery?” asked
Terry.

“I hope he’s coming here to spend Christmas!” Dot was

very frank in her desires. “It would be a change from
ghosts and musty old houses.”

“Hush!” warned Arden. “The phone is open—he’ll hear

us.”

They were chattering loudly near where Sim was [149]

speaking and listening over the telephone. They heard
her say:

“Oh, but how nice! Of course!—Come right over. We’ll

have dinner in a little while, and there’ll be a place for
you.—Oh, yes, we have been very busy.—What?—I’ll tell
you when you come over. But what are you doing in this
part of the country?—We thought you were enjoying
your millions.—Oh, getting even with me, I see—you’ll
tell us when you get here.—Yes, this place is easy to
find. All the taxi men know it. See you later!”

Sim danced back through the hall to where her friends

waited with anxiety to hear the other half of the
conversation.

“Was it really Harry Pangborn?” demanded Arden.

“Of course it was and is! He’s coming over!” Sim

laughed merrily.

“But why?”

“How?”

“What for?”

“Wait! Wait!” begged Sim, holding her hands up to ward [150]

off her importunate chums. “He’s going to explain it all
when he comes over. It seems he just arrived in
Pentville this afternoon. He was nice enough to say he
remembered that we all lived here, and he’s lonesome,
so he’s been keeping our line busy. He almost gave up
finding us in.”

“But what’s he out here for?” asked Terry.

“Came especially to see you, my dear,” laughed Sim.

“Oh, be serious!” begged Arden.

“Well, I can safely wager he didn’t come to see me,” Dot

put in. “I really hardly met him. You three monopolized
him at Cedar Ridge and then got his thousand dollars’
reward.”

“We didn’t get the thousand dollars,” Sim said. “It was
really the college swimming pool.”

“And Arden solved that mystery,” added Terry, referring

to one told of in The Orchard Secret.

“If I can only solve this one of Jockey Hollow I’ll go in

for mystery solving as a profession,” Arden laughed. “I
might major in it at Cedar Ridge.”

“Perhaps,” suggested Dot, “now that Harry Pangborn is

here, he can help you.”

Arden looked at the visitor. Was there anything sarcastic

in the remark? Hardly, for Dot smiled brightly.

“I still can’t guess why he has come here,” said Terry.

“You shall know very soon, child,” mocked Sim. “Now [151]
we must get busy and wash our faces. And, oh, I
wonder what sort of a dinner Moselle can give us? I
must have a talk with her. Run along, girls, get painted
and powdered, and I’ll follow as soon as I can.”

Shortly after this, Harry Pangborn drove up to the

Westover home in a “small but expensive car,” as Dot
remarked, catching a glimpse of its gleaming lamps out
on the drive. The young man came in, bronzed as to
complexion, smiling charmingly, and showing his white
even teeth, and greeted the girls with the comradeship
of a co-ed.

“So glad to see you again,” he told them. “And now, as I

heard Sim say she wondered why I was here, I’ll tell
you. I’m here in this particular place because I am
lonesome for such company as yours.” (That was being
gallant.) “And I’m in Pentville because I have a mission
to perform in Jockey Hollow.”

“Jockey Hollow!” cried the four girls together.

“Do you mean you are going to try to rid Sycamore Hall
of its ghosts?” asked Arden a moment later.

“Ghosts!” exclaimed young Mr. Pangborn. “I don’t know

anything about ghosts and less about Sycamore Hall.
What’s the joke?”

“Ever since they got me here,” supplied Dot, who [152]

seemed rather “taken” by the young fellow, “these girls
have done nothing but discover ghosts—ghostly noises,
dead women on a bed, a man mysteriously missing and
found in a cellar—and it all happened at Sycamore Hall,
an old Revolutionary mansion in Jockey Hollow that is
going to be torn down to make room for a new road.”

“This is news to me,” said Harry Pangborn. “I didn’t

count on this when I was asked to come to Jockey
Hollow. But it’s—grand!”

“Just why were you asked?” Sim wanted to know.

“Well, you are familiar with the fact that I fell heir to my
grandfather’s estate on Long Island,” was the answer.
“On it is a big wooded park, and as I happen to be a
nature lover, and a wild bird enthusiast in a small way, I
carried out some ideas started by my late grandfather
and have built up quite a bird sanctuary, as they are
called—a place for the conservation of all wild life; you
know, of course. I put some new ideas into my
experiments. Word of it got around, and I was asked by
Dr. Max Thandu, the State Park Commissioner here in
your part of the country, to make a sort of survey of
Jockey Hollow and lay out a bird sanctuary there. I
agreed, for I thoroughly believe in this sort of thing.”

“You mean you are going to work around here?” [153]

Dorothy asked.

“Work,” echoed Arden. “What Harry does is never just—

work.” She had called him “Harry,” and a self-conscious
flush made her look even prettier.

“I understand Jockey Hollow, with its Revolutionary

associations, is to be made a state or national park,”
Harry went on, smiling kindly at Arden. “The bird
sanctuary will only be incidental to its historic value. But
I am glad to do my little part there. So, having some
leisure time, and the Christmas season being rather a
hectic time down our way, and being fond of the woods
in winter and solitude—in a way—I decided to use my
Christmas vacation by coming to Jockey Hollow and
getting some first-hand information.”

“What could be nicer for us?” Sim complimented.

“Are you going to stay until after Christmas?” Arden

inquired.

“I hope to. I understand Jockey Hollow is rather a big

place, and it will take me several days to survey it,
locate proper places for feeding stations, and arrange
for a water supply for the birds. When I told Dr. Thandu
I would come here, I suddenly happened to remember
that you Cedar Ridge girls lived out this way, and so I’m
afraid I kept the operator rather busy this afternoon
giving her your number, Sim.”

“Oh, that, too, would have been kind of you. Central [154]
isn’t ever very busy here. I’m sure she rather enjoyed it.
The girls listen in, you know.”

“She hasn’t anything on me!” he laughed. “Well, now

you know why I’m here.” They had all settled down
comfortably, and it seemed, with Harry there, their party
was complete.

“But I thought you said,” remarked Dot, “that you

wanted solitude for Christmas,” her eyes were
mischievous.

“Oh, well, there is solitude—and solitude!” he

countered, his gaze sweeping them all in turn, but
lingering upon Arden. “But tell me about the ghosts. Are
they just too—too divine?”

They told him at dinner, which was a success in every

way, Moselle and her daughter doing themselves proud
in the viands and the serving thereof. Moselle simply
loved company, especially young men company.

“Now, what do you think of it all?” Arden asked when

the various phases of the happenings at the Hall had
been recounted.

Harry Pangborn was silent for a moment as he crushed [155]

the ashes of his cigarette on the plate.

The girls waited, not a little anxiously, for his opinion. It

was good to have a man around—especially such a
delightful young man as Harry Pangborn—one whom
they knew and could trust.

“Well?” asked Sim, at length.

“Well,” he blew out a cloud of smoke, “it sounds to me

like either one of two things,” came the answer, slowly
given. “It’s either a trick of some mischievous person or
persons, as you have hinted, perhaps engineered by a
rival contractor. Or—” again a pause—“there may be
something in it.”

“Do you really mean—ghosts?” gasped Terry.

“Well, perhaps what some persons call ghosts,” the

young man answered. “Let us say natural
manifestations that take on a weird meaning or
significance because they are not understood. I now
have a double duty here. I’m going to lay out the Jockey
Hollow bird sanctuary and——”

He lighted a fresh cigarette.

“If you’ll leave this to me,” he continued as he inhaled

the aromatic smoke, “I’ll do some real investigating, if
you want me to.”

“It really ought to be done,” said Arden gladly. “We want [156]
to help Granny Howe if we can, to put her in a position
where she can claim this property; though it seems
hopeless after all these years. And we also want to help
this Jim Danton. We’ll be so grateful for your help,
Harry, and we are so relieved to have you here—just
now.”

“Such as it is, you shall have it!” promised Mr. Pangborn.

 [157]

CHAPTER XVIII
The Figure in Red

Arden Blake fairly jumped into her bedside slippers,

drew on a dressing gown, and in an instant was at the
window.

“What’s the matter?” sleepily inquired Terry, who was in

the other twin bed. “Has anything happened?”

“It’s snowing again,” Arden answered. “I awoke a little

while ago and I heard tiny tappings against the window.
I wondered what it was and I waited a decent time, so I
shouldn’t awaken you, to find out.”

“Nothing to do with the mystery, has it?” yawned Terry.

“No, silly! It’s just snowing. It’s going to be a glorious

storm, much better than the other little fairy we had, I
believe, and oh, don’t you just love snow for
Christmas?”

“That’s so, Christmas is coming,” Terry admitted as she

sat up in her bed and watched Arden, still at the
window. “What time is it?”
“Nearly eight. Too sleepy still to see the faithful clock [158]
right before you,” teased Arden.

“Sim and Dot up yet?”

“I haven’t heard them moving.” Arden inclined an ear

toward the room across the hall where their hostess and
the other girl slept.

“Well, then, come on back to bed,” urged Terry. “No use

getting up until Sim does. And we stayed up so late last
night, talking to Harry Pangborn, that I’m sleepy yet.”

“I’m not, and I’m going to dress. I have something to

do,” declared Arden with a purposeful look on her face.

“What? Going to see Harry? I think he’s awfully nice.”

“He is, but I’m not going to see him. I’m going to the
woods to get some holly branches. I noticed a lovely lot
of bushes some distance back of the old Hall when I
was wandering around by the cellar door that time Betty
Howe popped up out of it.”

“With horror on her face, as they say in books,” drawled

Terry.

“Yes, she was terrified all right,” admitted Arden. “Who

wouldn’t be, coming upon what looked like a dead man?
And that’s another thing we must do.”

“My, aren’t we the busy girls!” laughed Terry. “What [159]

else, for goodness’ sakes? I might as well get up and
dress, I suppose. There’ll be no sleep for me now with
you barging around.”
“Another thing we must do,” said Arden as she began to
dress, “is to see to it that Jim Danton’s poor little family
gets some relief from Mr. Callahan or somebody. He was
hurt while working for the contractor, and the contractor
should pay. That’s the law.”

“It wasn’t exactly his fault, though,” Terry argued. “Mr.

Callahan might claim, as they say they do in some
insurance policies, that it was an act of God, an
unforeseen calamity, and so get out of it—I mean he
might say it was the ghost of Jockey Hollow.”

“I hardly believe he would do that,” remarked Arden,

brushing her hair vigorously. “But it surely is puzzling.
Well, we’ll see what Harry Pangborn can figure out of it,
though I think, since we sort of promised, in a way we
should try and do something for the Danton family.
There is no social service agency around here.”

“Yes, somebody must help them, and they seem nice

folks, too. But about this holly, what are you going to do
with it specially?”

“Decorate this place for Christmas, of course. Coming [160]

with me?”

“I suppose so. Dot and Sim will, I imagine.”

“Yes, we’ll make a little party of it. Oh, I do love to walk

in the snow, and it’s coming down beautifully!” raved
Arden. “Do come and look, Terry!”

“Wait until I get this shoe on. Though if we’re going to

tramp in the snow I suppose I’d better wear heavier
ones.”
“You won’t need them with arctics. But isn’t it a glorious
storm!”

Terry agreed that it was. The two chums finished

dressing and went out in the hall to go down for
breakfast, which was evidently being prepared by
Moselle and her dark daughter, as testified to by the
rattling of dishes and the aroma of bacon and coffee
floating up.

As Terry and Arden were walking toward the stairs, they

heard the door of Sim’s room open, and Dot came out,
wearing a robe. She held her finger on her lips as a
signal for silence.

“What’s the matter?” whispered Arden.

“She has a bad headache,” Dot replied. “She was awake

a good part of the night, and she’s just fallen asleep. I
thought I’d slip down and tell Moselle not to make any
more noise than she can help. Sim needs quiet.”

“Oh, that’s too bad!” murmured Terry. “I wonder if [161]

there’s anything we can do?”

“No, I gave her some aspirin. She’ll be all right. If you’re

going down, would you mind having that little slave
bring me up some coffee? That’s all I want. I’ll be
waiting out in the hall so I won’t disturb Sim by opening
the door too often.”

“It’s too bad,” murmured Terry again. “Can’t you come

down and have some breakfast with us?”

“No, coffee is all I’ll take. Some storm, isn’t it?”

“Terry and I were going out for a walk in it,” whispered
Arden, “and to gather some holly branches to decorate
the place here for Christmas. We hoped you and Sim
would come, but if she has a headache I guess we’ll
postpone the trip.”

“No reason why you should,” Dorothy argued, walking

to the head of the stairs with the others to avoid
whispering so much outside Sim’s door. “I’ll stay here
with her. I don’t feel much like walking in the snow,
though I love fresh-grown holly. Get all you can, and by
the time you come back I’ll be ready to help decorate,
and perhaps Sim’s head will be better.”

“All right,” agreed Arden. “I have my mind set on it, and [162]
I don’t like to change. You’ll come, Terry?”

“Oh, yes.”

Dot had her coffee, the other girls making a more

substantial breakfast, and then, leaving Sim still asleep
and Dot on guard, Terry and Arden set out into the
storm. The flakes were coming down rapidly now, dry,
small flakes that seemed to presage a heavy fall. It was
not yet deep, but would be, as none was melting.

“Oh, it’s so lovely!” murmured Arden raising her face to

let the snowflakes melt on it.

“You seem to have quite a yen on for storms,” remarked

Terry, laughing.

“I always have had. Now we must step out. It’s quite a

distance to the old Hall, and it’s slow walking in the
snow.”
“I’m equal to it,” declared Terry, bracing up and dashing
forward.

They trudged along, laughing and talking—talking

principally of the advent of Harry Pangborn and his
declaration that he would do some real investigating of
the mysterious happenings in Jockey Hollow.

“I wonder if he’ll really discover anything,” said Terry as

they neared the place.

“He might,” was Arden’s opinion. “He has a good head, I [163]
believe.”

“He has nice teeth, anyhow.”

“To bite ghosts with, I suppose!” laughed Arden.

“Yep! Well, I can see the place now,” remarked Terry as

they topped a little rise. “There doesn’t seem to be any
men working there, though—no plaster dust floating out
of the windows as usual when men are tearing down an
old building.”

“It is quiet,” Arden admitted as they walked in front of

the Hall. “I suppose Mr. Callahan is wondering what sort
of workmen to get next, since his white-collar class has
left, apparently.”

“Look!” Terry suddenly exclaimed, pointing. “Footprints

in the snow. At least one man has gone in there!”

“That is very evident, Robinson Crusoe,” laughed Arden.

“As your man Friday, I agree with you. Someone has
gone in, and one man only, judging by the footprints.
And as these are plain footprints and not little scratchy
marks in the snow I think we may safely argue that it is
no ghost.”

“Who said it was?” countered Terry. “But what can one

workman do in tearing down such a big house?”

At that moment a head was thrust out of an upper and [164]

partly demolished window and a voice cheerily called:

“Good-morning, girls!”

“Oh, it’s Harry Pangborn!” exclaimed Arden.

“Hello, Harry!” greeted Terry. Since the episode at Cedar

Ridge, the friends had begun to call one another by
their first names.

“What are you doing in there?” Arden called back.

“Investigating ghosts, as I promised. Want to help me?”

“We’re after holly,” said Terry, “in the back woods.”

“Well, you have time for both ghosts and holly too,
perhaps.”

“No, thank you,” Arden decided, shaking some of the

snow off her hat. “I think you can do your investigating
alone. I mean, you come to it with an open mind. Terry
or I might suggest something to you, in our eagerness,
and that would throw you off the track.” They were so
near the Hall they could talk easily to the young man at
the window above.

“There is something in what you say,” admitted Harry

with an assumed judicial air. “I shall take it under
consideration. Well, then, I’ll go on investigating by
myself, reserving the right to call at Sim’s house to see
you all, later, and report.”

“Yes, do!” invited Terry. [165]

“Have you found anything yet?” Arden wanted to know.

“I only arrived a few minutes ago. Well, on with the

ghost hunt! Stop in if you come past this way, and I’ll
help you carry the holly branches home.”

“Oh, that will be fine!” called Terry. “I was wondering

how we could carry enough to make really satisfactory
decorations.”

“But I draw the line at a Yule Log!” stipulated the young

millionaire, whose car, the girls now noticed, was parked
near a big clump of lilac bushes that nearly concealed it.
He had driven in from a direction opposite that which
they had traversed and so they had not seen the tire
marks.

“Did you come here this morning just to investigate?”

pressed Arden as young Pangborn started away from
the window and she and Terry were about to walk on.

“Well, I came to look into the matter of bird-feeding

stations for the sanctuary Dr. Thandu wants to establish
here, and so I decided I might also take in the Hall. It’s
quite a place.”

“Killing two birds with one stone,” quoted Terry tritely. [166]

“Exactly! See you later!”

He waved a hand to them and disappeared back into

the strange old house.
It was a little farther to the small grove, where the holly
trees and bushes grew, than Arden realized and it was
perhaps ten minutes after their good-bye to the ghost-
hunter that the two girls found a thicket sufficiently
large to ensure a good supply of branches with their
lovely red berries and dark, prickly, glossy leaves. Holly
is always just holly; hard, sharp, but magnificent on its
trees.

They had good pocket knives and soon cut off a

quantity—more, Arden suggested, than they could carry
even with the help of Mr. Pangborn, when Terry,
glancing off toward a little clearing, suddenly cried:

“Look!”

There was something in the tone of her voice that

startled Arden. But she managed to ask, as she whirled
quickly around:

“What is it?”

“A figure in red!” whispered Terry, pointing. “There— [167]

through the trees—someone in red—moving. Oh,
perhaps it’s the ghost of Patience Howe! She is always
seen wearing a red cloak, you know!”

Arden dropped the holly branches from her hand as she

looked toward where Terry pointed.

Something was moving! Red, in all that deep, dark

clump of evergreens!

[168]
 CHAPTER XIX
Santa Claus

Terry and Arden drew closer together, instinctively, for

mutual protection. It was uncanny to see this strange,
scarlet figure capering about in the little clearing, seen
through a screen of fir trees and against a background
of gleaming white snow.

“The ghost of Patience Howe,” murmured Arden,

recalling the story Granny had told—recalling what the
men had said about seeing an apparently dead woman,
in a red cloak, on a bed in the old Hall. And that figure
had mysteriously vanished.

Now it was in sight again—at least, some figure was

there. There was no mistaking it, for it was too plain to
be anything else but a moving elfin thing.

“Oh,” whispered Terry, “do you think, Arden, that Harry

could have disturbed it?”

“Disturbed what?”

“This ghost—I mean, perhaps he came upon the place [169]

where it hides in the house and it ran out—no, ghosts
don’t run, they sort of float, like smoke, don’t they? Oh,
Arden, I’m frightened!”

Then, fascinated, they watched and saw the red-clad

figure seemingly capering about, doing a strange dance
in the snow. And suddenly it started toward where they
were half hidden by bushes and trees. Coming toward
them!

“Oh!” screamed Terry. “Come on, Arden!” She turned to

run, uttered a sudden cry of pain as she clutched her
right ankle and sank down helplessly in the snow.

“Terry! What is it?” begged Arden, dropping to her side.

“My ankle! I twisted it when I turned to run! Oh, how it

hurts! I hope I haven’t broken it!”

“I don’t believe you did, my dear! Ankles don’t break as

easily as that. Oh, I’m so sorry!” She took some snow
up in her hand and pressed it on Terry’s forehead, now
wrinkled with pain. It flashed into Arden’s mind that she
was going to have trouble getting Terry back to Sim’s
house—walking with even a slightly sprained ankle was
out of the question. Then, with a feeling of relief, she
thought of Harry in the ghost house. She would have to
leave Terry there in the snow, however, to go get him to
come to the rescue.

“I’m so sorry,” Arden murmured. “Poor Terry!” [170]

“It was silly of me—making so much trouble. But, oh,

Arden—the red ghost! Look, it’s coming right for us!”
She was facing in the direction of the strange red figure;
Arden had her back toward it. But at Terry’s cry Arden
looked around, and then she had to laugh, even with all
the trouble they seemed to be in. And a moment later
Terry also laughed, in spite of her pain.

For it was no red-cloaked ghost of Patience Howe that

was bouncing over the snow toward the two girls. It
was—Santa Claus!

A rotund figure of a jolly little man with a real beard of

lovely white hair—no cotton whiskers on this St.
Nicholas—came prancing through the underbrush,
scattering snow. He was no ghost, the girls were
assured of that in a moment, for he addressed them in
very human accents. But even with all this reality it was
a puzzle.

“Well, well, young ladies! I thought I heard somebody

scream!” began the little man. “I was over in that
clearing, practising, and I saw you behind the trees, and
I sort of thought you’d think it queer, and I turned to
come and explain. Then I heard a scream and——”

“My friend turned suddenly and sprained her ankle,” [171]

Arden interposed. “It is very painful—I’m afraid she
can’t walk.”

“Luckily I can take care of that,” said Santa Claus. “It

was partly my fault, I reckon. Gave her a start, naturally
—seeing me in this rig. That’s why I came out here to
try it on. I knew it would look sort of silly to anybody
who didn’t understand. I’m terrible sorry.”

“But why are you dressed up this way?” asked Arden.

Terry was just about able to stand and, resting with her
head on her chum’s shoulder, her face showed she was
suffering. Really the ankle was very painful.
“It’s easy explained,” said the little man, pulling at his
luxuriant beard, a thing he never would have dared to
do had he been wearing a masquerade whiskers. “My
name is Janson Henshot, I live over at Bayley Corners,
and I’m superintendent of the Sunday-school there. Up
to this year we always had, for the Sunday-school
children, the little ones, you know, a Santa Claus with a
false beard. The part was played, off and on, by Jake
Heller or Sam Bendon.

“But last year one of the little boys gave the beard of [172]
Santa Claus a pull when he was handing out the
presents, and the beard came off, and it sort of spoiled
things. So, when Christmas was talked of this year,
somebody said I’d do fine for Santa Claus, as my
beard’s real and it’ll stand a lot of pulling and won’t
come off!” He demonstrated, laughing.

Even Terry smiled now, for she was listening and had
opened her eyes. This, truly, was a comical experience,
to find a real Santa Claus in a real wood.

“So I said I’d be Santa Claus,” went on Mr. Henshot. “All

I needed was the uniform, and my wife made this one.
Not bad,” and he looked proudly at his red coat and
trousers, trimmed with real white rabbit fur, and at his
glossy black boots.

“It’s perfect!” declared Arden.

“Glad you like it! Well, after I got the uniform and I
didn’t have to raise any beard, I decided I needed some
practice to act right as Santa Claus, me never having
played the part before, though I’ve watched the others.
So I put the uniform in my old flivver and came out here
in the woods to rehearse, as you might say. This is the
second time I’ve done it. I act like I think the old fellow
would act with a lot of happy children around him—sort
of skipping and prancing. Am I keeping you too long? I
wanted to get it down right before I went out into that
Sunday-school crowd. And that’s what I was doing—
rehearsing—when you saw me. Guess you must have
thought it sort of odd.”

“We—we thought you were a ghost!” murmured Terry. [173]

“Ghost! My stars!”

“The ghost of Patience Howe, on account of the red,”

explained Arden.

“Oh—Patience Howe—I see—her as is supposed to have

been around Sycamore Hall in the Revolution and hid
her horse from the soldiers. Yes, that’s a story around
here, but I don’t know—ghosts—no such animals if you
ask me!” He laughed heartily.

“I suppose you have heard,” suggested Arden, “that the

ghost of Patience, in her red cloak, is said to wander
around the old Hall at times.”

“Oh, yes, I’ve heard that story, but nobody I know ever
saw any ghost like that. Though, now you speak of it, I
did hear that the contractor who’s tearing down the Hall
has been having trouble with his men on account of
queer happenings. But I don’t take any stock in ’em.
Just rantings of the Negro and Italian laborers, I
reckon.”

“Some queer things have happened there,” said Arden. [174]

“But now what are we going to do? I must get Terry
home as soon as possible—a doctor must look at her
ankle at once!”
“I know—sprained ankles can kick up quite a fuss. But
as I’m sort of to blame for this, I’ll do my best to
remedy the trouble. I shouldn’t have kept you here so
long talking, by golly! I’ve got my flivver parked over
near where I was rehearsing. I can run it here—no
trouble at all—my flivver’ll go up the side of a barn. And
we’ll put your friend in and I’ll run her home in a jiffy, if
you want me to.”

“I think that will be the best thing to do,” said Arden.

“We have a friend in Sycamore Hall——”

“You have!” cried Mr. Henshot. “Why, I was told Granny

Howe couldn’t prove title to the place and she had to
get out and it’s being torn down.”

“That’s right,” Arden assented. “But the friend I speak of

is just in there temporarily, looking for ghosts.”

“My stars!” exclaimed Santa Claus. “Well, I’ll go get my

flivver. Be back right quick. Don’t let her step on her
ankle. I’m mighty, mighty sorry this happened!”

He ran away with surprising speed for such an elderly

man, his white beard flying in the wind, and almost
before Arden could shift Terry to a little easier position
on her shoulder Mr. Henshot was back with his creaking
roadster.

To Arden’s surprise he still wore his Santa Claus suit. [175]

“Aren’t you going to take that off?” she asked, for she
knew he had it on over his other clothes.

“Got no time!” he said briskly. “We got to get this young

lady to a doctor right away. I’ll drive you just as I am. I
don’t mind,” he said quickly. “It’s in Pentville, and
nobody’ll know me there. I wouldn’t want to drive
through Bayley Corners like this, for it would sort of
spoil things for the youngsters if they see me ahead of
time. But it’s all right in Pentville. Drive you just the way
I am!”

Terry was feeling too miserable to object, and Arden

realized it would be useless. Besides, she knew Terry
must have her injured ankle looked to as soon as
possible. After all, perhaps no one the girls knew would
see them.

Terry managed to hobble on one foot and, assisted by

Arden and Santa Claus, was placed on the rear seat of
the car with her chum to hold her against the rough
riding. For it would be rough getting out of the stretch
of woods and clearing.

“Might as well take this holly you picked,” said Mr. [176]
Henshot. “It’ll look right pretty in the car with me
dressed like Santa Claus and all this snow coming down.
A regular white Christmas!” he chuckled. “Right pretty!”
He piled the branches in with the girls, putting some in
the empty seat beside him, and slid under the wheel.

Then he started the car, driving carefully, after Terry

gave a little moan of pain at a sudden jolt.

“I’ll have to take a short cut,” he explained, “so we can’t

go past the Hall and pick up your ghost-hunting friend.
Sorry, but I can’t go that way.”

“It’s all right,” said Arden. “He has a car.”

She wondered what those who saw the strange outfit

would say, but this held only a moment’s interest.
Terry’s injury might mean a curtailment of some of the
Christmas festivities, besides all poor Terry’s suffering.

They were out of the woods at last and on a smoother

road, not having passed either Granny’s cottage or the
Hall. In a short time they were on the outskirts of
Pentville and entered the town by a back road. So not
many saw them, and those who did, while they smiled
and laughed and pointed, put it down to an advertising
stunt. Arden saw no one she knew, Terry saw nothing
but Arden’s kind shoulder which she leaned against.

But when the auto of the modern Santa Claus drew up [177]
at Sim’s house and Moselle answered Mr. Henshot’s ring
at the door, she jumped back with fright.

“Mercy sakes alive! Whatever is this? A real live——”

Moselle was most eloquent when silence seized her.

[178]
 CHAPTER XX
Harry Hears Something

Moselle’s involuntary shout of surprise and alarm

brought Dorothy on a run to the front door. She gave
one look at Terry and Arden seated in the flivver,
surrounded by holly branches, another look at Santa
Claus, and then laughingly demanded:

“Where do you play the next performance?”

“It isn’t any play, Dot!” called Arden. “Terry’s hurt!”

“Hurt!” She was serious in a moment.

“It’s only a sprained ankle,” said Terry, trying to speak

with vigor. “All my own fault.”

“No, it’s my fault,” insisted Santa Claus.

Moselle, her eyes almost popping from her head, had

retired to the back hall, but was still peeking and
listening.

“This is Christmas and then some,” said Dorothy. “But

whatever happened?”
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