Oral SURGERY OralMEDICINE AND&d
PATHOLOGY
VOLUME
19
NUMBER
3
MARCH,
1965
Operative oral surgery
Experience with thirty-six dental extractions in patients with hemophilia and Christmas disease D. X. Middleton, F.R.C.S.E., P.D.S.,* S. H. Davies, M.B., F.R.C.P.E., Ph.C.,** R. A. Cumming, U.B.E., M.B., Ch.B., F.C.Path.,*e* K. Kamel, and Alexis Cameron, A.I.M.L.T.,L**“” D.M.Sc., M.D., Ph.D.,**** Edinburgh, Scotland
T
he necessity for regular, expert dental conservation in the hemophilic patient as stressed by Rubin, Levine, and Rosenthal’ cannot be overemphasized. Such conservation allows nontraumatic outpatient procedures to be done for which anesthesia usually need not be used, and it avoids hospitalization for premature extraction with its associated traumatic risks and expense. When extraction is unavoidable, various practices have been advocated for the operative management of these paGents. While some workers have advised general anesthesia,*g 3 others have used local injections.4, 5 All agree, however, that operative trauma should be reduced to a minimum. The number of teeth removed at one time has ranged from one” or two* to total extraction.? Some surgeons have *Consultant in Charge, Department of Oral Surgery, The Royal Infirmary of Edinburgh. **Regional Consultant Hematologist and Honorary Consultant to the Edinburgh and South-East Scotland Blood Transfusion Service. ***Regional Director, Edinburgh and South-East Scotland Blood Transfusion Service. ****Lecturer, Clinical Pathology Department, Ain-Shams University Medical School, Cairo, Egypt; on a study fellowship to the U.K. *“***Senior Technician, Edinburgh and South-East Scotland Blood Transfusion Service.
283
O.S., 0.M: 8.i OP. March, 1965
sutured the socket immediately sft!cr estraction.7-9 Others, because they believe that this increases trauma, can later be a cause of bleeding when the suture is removed, and can produce pressure necrosis through tension, have condemned it.3-” There is general agreement that a previously prepared dental splint should be fitted at the time of extraction in order to protect the socket.“? ~11 Local hemostatics, such as thrombin, Russell viper venom, alginates, gelatin sponge, etc., have been used, and all have their advocates.“, 8 The general management of the hemophilic patient has varied from hypnosis aloneI’ to the attainment of an adequate hemostatic level of Factor VIII by pre-extraction transfusion of blood or blood products and in some cases the maintenance of this level by postextraction transfusion. Inpatient stay has varied from 3 to 23 days.4, l3 This article reports our experience in the management of twenty-three patients with hemophilia and Christmas disease who underwent thirty-six separate dental extraction operations, INVESTIGATIVE
MATERIAL
Eighteen patients with classic hemophilia (Factor VIII deficiency) and five with Christmas disease (Factor IX deficiency), whose ages ranged from 5 to 58 years, had twenty-eight and eight dental operations, respectively (Table I). The largest number of operations in one patient was three, and the number of teeth extracted on any one occasion varied from one to thirteen. The diagnosis of the bleeding disorder had been based on the history, the clinical and hematologic findings, and the blood assay levels of Factors VIII or IX.l*? I5 By this means patients were classified as severely, moderately, or mildly affected. Generally, the clinical state correlated well with the laboratory findings. Severely affected patients with a blood factor level of less than 1 per cent (normal, 60 to 160 per cent) suffered from spontaneous bruising and bleeding; moderately affected patients with 1 to 10 per cent levels bled excessively following minor trauma; and mildly affected patients with 10 to 60 per cent levels bled abnormally only when subjected to moderate trauma. TECHNIQUES Preparation
of the patient
After the number and position of the carious teeth had been surveyed, a roentgenogram of the whole mouth was taken. In inverse proportion to the severity of the disorder, the number of teeth to be extracted at one session varied from one to thirteen. This number was determined jointly by the dental surgeon and the hematologist at the outpatient clinic. Generally, extractions at. any one sessionwere limited to teeth in one jaw. At later operations more teeth might be extracted if the first procedure had been satisfactory. At the outpatient, clinic, once the extraction policy was decided, a Zelex” impression was taken and an acrylic dental splint was made, The splint was designed for fixation to adjacent teeth by bands so that rocking movement and undue pressure on the socket or adjacent mucosa would be avoided. A minimum of 3 months was *Amalgamated
Dental
Trade
Distributors,
Ltd.,
London.
England.
Volume 19 Number 3
Dentul extractions awl hewwphili~
285
allowed to elapse between extraction sessionsso as to minimize the risk of a circulating anticoagulant developing from prolonged and frequent transfusions. The patient was admitted to the hospital on the day before the operation, checked again clinically and hematologically, and rejected if he was bleeding or was otherwise unfit to undergo the contemplated surgical procedure. TWO Standard units of A.C.D. blood were freshly drawn into siliconized containers and cross-matched. A prophylactic transfusion of fresh frozen plasma or of human antihemophilic fraction16 was begun immediately before the operation in ordci to raise the Factor VIII level to at least 10 per cent and preferably to more t,han 60 per cent. This was given as rapidly as possible, within 1 to 2 hours, and timed so as to end as the operation began. Patients with Christmas disease were given either fresh frozen plasma or fresh dried plasma on the same basis. A stick-in needle (hypodermic, 19 S.W.G.) * was always used, usually in a vein on the dorsum of the hand, A strict aseptic technique, together with careful skin sterilization, was adopted and the needle was rigidly fixed by 3 inch wooden splints and adhesive plaster and left in situ for the planned total tra.nsfusion time. Between plasma transfusions the drip was kept open by minimal saline infusion. An endeavor was made to keep the blood factor level above 10 per cent for up to 7 days postoperatively by giving, where necessary, 6 hourly plasma transfusions. MTe hoped that, in this way, we could ensure hemostasis and assist healing. Because of difficulties in preparing antihemophilic fraction, fresh froxcn plasma was commonly used. It was decided that blood would not be given U~CSS the hemoglobin level ha,d fallen to less than 6 Gm. per cent so that the chance of sensitization to red cell antigens would be reduced to a minimum. Anesthesia
For conservation procedures, local infiltration anesthesia may be justified in patients with a low pain threshold if the work cannot bc completed otherwise Mandibular block and injections into the pterygoid fossa are contraindicated because of the risk of hematomata formation in these areas. Hematomas forming further forward in the jaws are contained by the muscle attachments and are of less significance. For extraction procedures, oral or intravenous atropine sulfate and pethidine hydrochloride was used as premeditation. Anesthesia was induced with int,rnvenous thiopentone, and a muscle relaxant was given. The patient was then intubated orally and anesthesia was maintained with a nitrous oxide-oxygpn mixt,ure, Trilene being added in some cases. Two patients (Cases 17B and 1.9A, Table I) were treated experimentally under local anesthesia. Operative
procedures
Before a tooth was removed, its gingival attachment was divided from its neck with a sharp periosteum elevator. The tooth was then extracted with a root forceps and, if the buecal alveolar plate was unavoidably fractured, the loose bone was carefully dissected out. All granulomatous tissue which was not *Internal
diameter
of needle, 1.016 mm.
286
illiddleton
Table
I. Data on thirty-six
Case
OS., O.M.& 0.L’.
et al.
Diagnosis
March,
dental extractions
Age
in patients with
1965
hemophilia and
Extractions
(years)
Number
Teeth
So&et
1
Mild
hemophilia
33
6
2
Mild
hemophilia
10
1
TT2i-p $3
3
Mild
hemophilia
34
9
721 1 1237
4A
Mild
hemophilia
39
11
4B
Mild
hemophilia
40
9
59
Xld
hemophilia
7
1
5B
Mild
hemophilia
8
2
5C
AIild
hemophilia
9
1
i--
Yes
6h
Moderate
hemophilia
42
4
g&
Yes
6B
Moderate
hemophilia
44
9
(6
Yes
6C
Moderate
hemophilia
44
13
( 1234568
NO
7
Moderate
hemophilia
35
3
6541
8
Moderate
hemophilia
29
2
“i
sutured
NO NO NO
84 I 754321
1 12345
No
7321 1 12347
No SO
Al
Yes
m
87654321 864321
for
6J
No Secondary
sutures
18 Sh
Severe
hemophilia
23
2
87)
Yes
9B
Severe
hemophilia
28
3
43)8
x0
SC
Severe
hemophilia
Z!)
2
81 --q-R
Upper
Severe
hemophilia
14
2
70
1 1:
socket
-1 E socket
sutured
sutured
16
t!i
11
Severe
hemophilia
19
1
No
12A
Severe
hemophilia
17
2
12H
Severe
hemophilia
18
2
12C
Severe
hemophilia
18
1
13
Severe
hemophilia
5
1
14
Severe
hemophilia
26
2
15
Severe
hemophilia
24
5
76 / 567
No
16
Severe
hemophilia
77
2
616
SO
17A
Severe
hemophilia
34
2
17B
Severe
hemophilia
35
2
18
Severe
hemophilia
15
1
Yes
4 ‘516
IL m I 57
j-z
65 - I
m
socket
sutured
socket
sutured
Yes No j-?
Secondary
sutures
Secondary
sutures
Yes
Volume Number
Christmas Last day of postoperative transfusion
Dental
19 3
extractions
ad
hemophilia
disease Cessation of bleeding (postopcrati~w2 day)
Healing (postoperative day)
Inpatient stay (days)
Remarks
15
19
23
26
Infected
1
2
9
11
Jnitial
7
7
9
15
Pyuria,
8
12
10
15
8
7
9
15
None
1
6
8
None
1
3
3
hematoma alveolar
abscess
purulent
cavity
sputum
4
4
8
10
2
13
12
14
10
11
13
15
6
10
10
15
5
5
9
11
16
16
7
27
3
1
10
12
5
2
7
9
6
6
10
13
4
6
6
11
2
7
9
12
6
7
11
13
Traumatic hemarthrosis hospitalization
1
14
18
20
Developed
mild
17
24
26
Arm
thrombophlebitis
7
11
14
18
9
10
10
13
7
12
7
9
9
16
16
18
27
9
13
32
Emergency hematuria,
19
12
26
28
Local anesthesia, circulating anticoagulant, hematuria, pyuria, and palatal hematoma
28
25
29
38
Occult
IS
287
Diabetes
difficult
Initial
slight
Upper
respiratory
Initial
septic
vein
to control
gingivitis
tract
sockets
required
hemarthrosis
Ooze occurred after by local measures
blood
infection
extraction, pyuria,
in stools
discharge
further
for
2 days
and was controlled
circulating and infected
anticoagulant, hematoma
288
Middleton
Table
I (Cont’d)
as., U.M. & 0.1’. Marc~h11965 *
et d.
I cnse 19A
Diagnosis Mild
Christmas
I 4Je
disease
I Extmction~
(years)
Number
17
4
1
Teeth
J’ocket
sutwed
NV
61 EqT
19l3
Mild
Christmas
disease
23
2
$17
2OA
Mild
Christmas
disease
56
1
B/
2OR
Mild
Christmas
disease
58
3
87
21
Mild
Christmas
disease
13
2%~
Mild
Christmas
disease
40
22B
Mild
Christmas
disease
40
2:;
Rnverc
Christmas
disease
11
No YW j4
NV
2
p16
-1 6 socket
8
71 / 135678
11 2
843211 61&j%
-
123458
sutured
xv
Socket
of q
1/ b socket
sutured
sutured
adherent and which was not removed with the apex of the tooth was curetted. The dried socket was then packed gently with thrombin, the protective splint was fitted, and the accuracy of fit was carefully checked. In a few cases the splint was lined with gutta-percha over the socket area and was later trimmed if the tissues became edematous and swollen. In those cases in which buccal bone had been removed, an atraumatic silk suture was inserted to approximate the mucosa. In three cases (Cases 8 and 17A and B) it was necessary to use a secondary suture in the maxillary tuberosity when calculated adequate transfusiontherapy had failed to control bleeding. These sutures were inserted under palatine block anesthesia and were entirely satisfactory. All sutures were left in position for 12 to 14 days, and no bleeding followed their removal. Postoperative
care
The patient was maintained on a fluid intake/output chart; blood loss was carefully estimated and no aspirin-conta.ining drugs were given. Twenty-four hours postoperatively the patient began breathing exercises and quadriceps drill. At the end of the planned course of intravenous therapy the patient was gra.dually mobilized and treated on an ad hoc basis if any further bleeding occurred. At this time he was started on oral iron therapy. The splint was removed after 48 hours (earlier if necessary) and checked to make sure that no pressure points had developed, after which it was washed, adjusted when needed, and replaced. This procedure was carried out immediately following a transfusion when the blood factor level was at its maximum. It was repeated on the fourth day and thereafter as required, and the splint was finally discarded when healing was almost complete, usually between the tenth and fourteenth days.
Volume Numiwr
Last day of postoperative transfusion
Dentnl
19 3
Cessation of bteeding (postoperative day)
Healing (postoperative da?l)
InpaGent stay (days)
6
7
9
11
5
7
6
8
None
None
3
3
2
None
7
9
None
1
5
7
2
None
5
2
None
3
1
12
13
eztrnctions
r~,wl,
hemophik
289
Remarks Local
anesthesia
Bleeding occurred 72 hours after was controlled by local measures
discharge
and
12 15
RESULTS AND DISCUSSION The details of these operations and the results obtained are shown in Tables I and II. All patients made a satisfactory recovery. Each of the two patients whose extractions had been performed under local anesthesia developed a hematoma at the injection site within 48 hours after completion of the transfusion therapy. Except for three patients who required secondary sutures, none bled abnormally during transfusion therapy, but some began to ooze within 48 hours after completing it. Bleeding was intermittent and ceased on an average, on the seventh postoperative day in cases of mild hemophilia, on the eleventh day in the moderate and severe cases of hemophilia, and on the second day in the mild cases of Christmas disease. The one patient with severe Christmas disease stopped bleeding after 12 days. Bleeding was more marked in the patients with severe hemophilia and in those who had undergone multiple extractions, some
Table II. Results of thirty-six and Christmas disease-average
dental extractions and range
in patients
with
hemophilia
Diagnosis Mild hemophilia Moderate hemophilia Severe hemophilia Mild Christmas disease Severe Christmas disease *Delayed
healing
occurred
8 1: 7 1
5 8 10 2 1
(O-15) (2-16) (l-28) (O-6)
in complicated
7 11 11 2 12 cases.
(1-19) (5-16) (l-25) (O-7)
10 (3-23)” 10 (7-13) ‘5” i$:;y, 13
13 16 18 7 15
(3-26) (11-27) (9-38) (3-11)
290
Xiddeton
et (11.
of whom required further plasma transfusions. In no case was bleeding sevcrc enough to require whole-blood transfusion. Usually the protective splint needed adjustment after 48 hours, and sometimes adjustment was required again later. Three patients (Cases 1, 3, and 17A, Table I) developed local evidence of infected sockets. Specific oral. antibiotic therapy was used only where indicated. The average time for healing was 10 days in the mild and moderate cases of hemophilia (range : 3 to 23 and 7 to 13 days, respectively) and 14 days in the severe casesof hemophilia (range : 6 to Xl), it was 5 days in the mild cases of Christmas disease (range: 3 to 9). Healing was complete on the thirteenth day in the patient with severe Christmas disease. The inpatient stay averaged 13 days for mild (range : 3 to 26)) 16 days for motlcratc (range : 11 to 27), and 18 days for severe cases of hemophilia (range : 9 to 38) and 7 days for mild (range : 3 to 11) and 15 days for severe cases of Christmas disease. Cencrally, therefore, the more scvcrely affected patients took longer to heal and vvcrc kept in the hospital longer. One patient (Case 17) developed a circulating anticoagulant directed against Factor VIII on the twenty-fifth postoperative day after his first extraction procedure. The anticoagulant soon waned on cessation of transfusion but recurred within 10 days after transfusions were recommenced following a second extraction 8 months later. Both times the patient had septic sockets and he also developed pyuria and hematuria. On both occasions hemostasis was successfully achieved over the few days following the appearance of the anticoagulant by the use of large dosesof antihemophilic fraction. At the end of this time, healing was sufficient,ly far advanced that the transfusion could be stopped and reliance placed on local measures. The titer of the anticoagulant then gradua,lly fell, and it was undetectable 3 months later. There were no ot,her transfusion complications. COMMENTS
It is significant that many of the patients with severe hemophilia and some of the moderately affected patients began bleeding on the seventh and ninth postextraction days, respectively ; that is, within 48 hours following the cessation of transfusion. In some casesloca,l hemostatic action proved inadequate to arrest this hemorrhage and further transfusion was needed. This is in a.ccord with the fact that the blood level of factor VIII tends to fall to the basic level within 24 hours after cessation of transfusion and, unless healing is sufficiently advanced, bleeding may then recur. The cost of hospitalization and of the blood products used during dental extractions is high. When these costs are added to the technical problems associated with preparation of the blood products, it seemsreasonable to stress the importance of prophylactic dental care in these patients. Until some new approach to dental extraction in patients with hemophilia is established we believe that it is best done as an elective procedure under general anesthesia and transfusion cover. We also emphasize the need for reducing trauma to a minimum, the use of local hemostatic agents, and the in-
Volume Number
19 3
sertion of a Nell-fitting protective splint. Generally we avoid using sutures. The whole problem should be treated as a “combined operation,” where the oral surgeon and the hematologist work together. The recent article in which Lucas a.nd his co-workers’2 report that a combination of hypnosis, continuous local hemostasis, and protection of the traumatized area will avoid postoperative bleeding merits further investigation and may offer a new approach to the probIcm of dental extraction in patients with hemophilia. We are grateful to Professor cases to us, and Eve are particularly
R. H. Girdwood and other indebted to Dr. D. Ellis
medical colleagues who referred who prepared the blood products
USd.
REFERENCES
1. Rubin, B., Levine, P., and Rosenthal, M. C.: Complete Dental Care of the Hemophiliac, ORAL SURG., ORAL MED. & ORAL PATH. 12: 665-675, 1959. 2. Leatherdale, R. A.: Anaesthesia for Dental Extraction in Haemophilics, Anaesthesia 13: 27-32, 1958. I. A., and Nicholl, B.: Tooth Extraction in Patients With Hemophilia, ORAL 3. Findlay, SURG.. ORAL MED. & ORAL PATH. 13: 1167-1180. 1960. A. S.: Dental Extract& in Haemophilia and Christmas Disease, 4. Orr, j. A., and Douglas, Brit. M. J. 1: 1035-1039, 1957. 5. McIntyre, H., Nour-Eldin, F., Isrdls, M. C. G., and Wilkinson, J. F.: Dental Extractions in Patients With Haemophilia and Christmas Disease, Lancet 2: 642-646, 1959. 6. White, P. H., and Mallet, S. P.: Management of Hemophilia in Dental Extractions, J. Oral burg. 71 237-246, 19i9. 7. Winstock, D., and Ingram, G. I. C.: Dental Extractions in Haemophilia : Plasma Therapy Without Dental Splints, Brit. M. J. 1: 719-721, 1961. 8. Nichols, C. F., and Baldridge, 0. L.: Multiple Extractions for the Hemophiliac, J. Oral. Surg. 12: 31-42, 1954. 9. Ingram, G. I. C., and Winstock, D.: Oral Surgical and Prosthetic Management of a Severe Haemophiliac, Proc. Roy. Sot. Med. 53: 475-478, 1960. 10. Matheson, W. S.: Dental Extractions in Cases of Haemophilia, Brit. D. J. 87: 312-321, 1949. C., Smith, C. A., Honey, G. E., and Taylor, K. B.: Dental Extraction in 11. Wishart, Haemophilia, Lancet 2: 363-366, 1957. R. T., Finkelmann, A., and Tocantins, L. M.: Tooth Extraction in 12. Lucas, 0. N., Carroll, Hemophilia; Control of Bleeding Without Use of Blood, Plasma or Plasma Fractions! Thromb. Diath. Haemorrh. 8: 209-220, 1962. B., and Findlay, I. A.: 13. Nicholl, Dental Treatment in Haemophilia, Ulster M. J. 28: 55-60. 1959. J., and Richards, G.: The Assay of Antihaemophilic Globulin Activity, 14. Biggg, R., Eveling, Brit. J. Haemat. 1: 20-34, 1955. 15. Bolton, F. G., and Clarke, J. E. : A Method of Assaying Christmas Factor; Its Application to the Study of Christmas Disease (Factor IX Deficiency), Brit. J. Haemat. 5: 396-412, 19!iF)
16. Cumming, Fraction
R. A., Davies, Production. (In
S. H., press.)
and
Ellis,
D.:
Red
Cell
Banking
and
Antihaemophilic