Angioneurotic edema

Angioneurotic edema Report of a case of the allergic mepivacaine hydrochloride

Reynold J. Baumstark, Brooklyn, N. Y. KINGS

COUNTY

HOSPITAL

type

caused

by sensitivity

to

A.B., D.D.S.,” and Reuben Seldin, D.D.S.,“” MEDICAL

CENTER

A

ngioneurotic edema, known by such synonyms as Quincke’s disease, giant urticaria, wandering edema, acute essential edema, acute circumscribed edema, urticaria edematosa, and Bannister’s disease, was described initially by Milton in 1878, Bannister in 1880, and Quincke in 1882. Clinically, angioneurotic edema may have no prodromal period or it may be preceded by an itching or burning sensation. The disease is characterized by the sudden appearance of single or multiple, circumscribed, painless, edematous swellings of the skin and mucous membranes. These lesions, which may develop over a period of 5 minutes to 6 hours, usually affect the face, genitals, and extremities and may persist for 2 to 14 days. Differential diagnosis includes swellings of infectious, traumatic, and neoplastic origin. The sudden onset, absence of pain, inflammation and hyperpyrexia., the possible presence of itchiness, and a history elf previous urticaria, serum sickness, sst.hma, hay fever, or atopic eczema all contribute to the diagnosis.1-3 The term angioneurotic edema is used in a broad, nonspecific sense today. This leads to confusion among clinicia,ns, as there are four basic forms of the disease : ” (1) hereditary angioneurotic edema, (2) hereditary allergic angioneurotic edema, (3) nonhereditary allergic angioneurotic edema, and (4) nonhereditary angioneurotic edema of unknown etiology.“4 The distinction as to the type of disease with which one is dealing is important. For practical purposes, the clinician may distinguish the first (hereditary angioneurotic edema) from the remaining three types. In the present article only these two clinical entities will be considered. “Former Intern, Department of Oral Columbia University School of Dentistry. **Director of Department of Dentistry.

Surgery;

Instructor,

Division

of

Oral

Surgery,

433

O.S., O.AT. 650.1’. ;\pril,

Hereditary

1967

angioneurotic edema is iJrI oJnin(Jm c&as(~, wit]1 a llig]l nl()J*t;llitg from episodes of ache (!denla, of the klryrtS. It iJ~l~)(!iJW to h inherited as a Mendelian dominant, with 414 cases reported in fifty-five families (all Caucasia.ns). Oral surgical procedures in patients with this, form of angioneurotic edema may be dangerous and life threatening. (‘rocker” reported that death has occurred following exodontia, in patients afflict.ed with this form of t,he disease. Richa,rds a,nd CrombsieGreported the case of a %-year-old man who died, 14 hours after exodontia, of occlusion of the airway caused by edema of the vocal cords; this patient had a history of hereditary a,ngioneurotic edema,. Another possible case of this affliction was reported by Scher.’ The remaining three t.ypes of angioneurotic edema, are benign and, because of their similarit.y, will be discussed as a single clinical entity. After studying a series of 500 cases of urticaria a,nd angioneurotic edema of this benign type, SteinhardF concludes that these lesions are most common in women between the ages of 20 and 40 years. The clinical manifestations are the same as those seen in patients with heredit.ary form of the disease. Steinhardt believes tha,t a history of allergic phenomena has diagnostic value but that skin tests and eosinophil counts are not reliable. He re-emphasizes the importance of a deta,iled history in clarifying the problem. Siegel and Bergeron,” in a series of 115 cases involving children and young adults, and Steinhardt, in a similar study, reached the same conclusions regarding the ebiology of the benign form of this condition. They found the most common etiologic factor to be drugs (especially penicillin j, followed by foods, infections, psychosomatic factors, insect. bites, physical causes, contactants, inhalants, and endocrine factors. The pathogenesis of a,ngioneurotic edema is believed to be the result of dilatation of small blood vessels and transudation of fluid through the capillaries into the subcutaneous or submucosaJ tissues. The role of histamine in effecting this vascular change is not clear. There is evidence that there is a direct relationship, but it is not conclusive. Van Arsdel and Beall,l” in a review of the metabolism and function of hista.mine, state: “. . . even the relatively clearcut examples of histamine effects are supported mainly by observations on the exaggerated response t.o exogenous histamine administrat.ion.” Westgate, Schultz, and Va,n Bergen I1 have reported a case in which administration of d-tubocura.rine was followed! by urtica~ria and angioneurotic edema, with a8bnorma.lly high blood a,nd plasma histamine, levels but. a normal eosinophil count. The patient had a history of previous reactions from “green onions, strawberries, and emotional upset.” Cerqua12 reported elevation of blood histamine levels in six patients during the acute phase, with return to normal in 6 hours. Ros@ observed that physical stimuli, such as heat, cold, aad light, evoked initial rises in blood histamine levels in eight of fifteen patients during the acute phase of skin eruption, and in four of these pat,ients the blood histamine levels fell below normal within 30 minutes. Clinically, antihistaminic drugs are beneficial, but they do not produce an isolated action on histamine. There is always the possibility that the sedative rate

resulting

Volume 23 Number 4

Angioneurotic

edema

435

side effects may affect the clinical response significantly. Tripelennamine (Pyribenzamine) or diphenhydramine (Benadryl), in doses of 50 to 100 mg. every 4 hours, may be used. Epinephrine, .o,ne or two doses of the l:l,OoO a,queous r;olution given intramuscularly, may be indicated, particularly if the airway is l;hreatened. Prednisone, 10 to 15 mg. every 6 hours, may be used in severe or nonresponsive cases. When the airway is threatened, a.ggressive therapy should be initiated and the patient watched carefully for signs of distress. One should be prepared for a tracheostomy, if necessary. A search of the literature seems to indicate that an allergic angioneurotic edema reaction to local analgesic drugs used in dental and oral surgical practice is rare, and reported cases are limited to reactions to t.he procaine group of comIpounds. Procaine was introduced by Einhorn in 1905, but it was not unt.il 1947 that .Krol114 treated a patient who developed laryngeal edema 45 minutes after in,jection of a 2 per cent procaine-Pontocaine-Cobefrin solution. The edema was successfully treated, and the reaction was attributed to procaine allergy. Adler and Simon15 reported a case in which a 4 per cent solution of procaine with epinephrine used for infiltration analgesia produced facial edema, without hyper.pyrexia, which regressed in 4 days. When a similar analgesic was a,dministered .3 weeks later, a similar swelling and a ve&ular eruption followed but regressed without complications between 8 and 10 days thereafter. Intradermal and1 patch testing were positive for both procaine and tetracaine. RicklesX6 reported ten eases, all with positive 24 hour skin tests. Mitchelll? reported two more cases, also with positive 24 hour skin tests. Siegalls added three cases to the literature and demonstra,ted positive 24 hour skin tests. However, skin tests were negative for lidocaine, and each of the three patients was able to t.olerate lidocaine without difficulty. Although the number of reported cases of this form of allergic angioneurotic edema is limited, it would, seem that this phenomenon is not as rare as one might think, since many colleagues with whom we have conferred have had experience with cases similar to the one to be described here. Because we believe this to be more common than the literature would indica,te, and beca,use there have been no reported cases of this reaction to a drug of the acetanilid group, the following case is presented. CASE REPORT A 30.year-old Negro woman in moderate distress came to the Oral Surgery on July 10, 1963, with the chief complaint of ‘%welling of the face.” History

of

present

The patient

Clinic

at 9 A.M.

illness

ww treated by her family

dentist, on July 8, 1963, at approximately 7 P.M. Because of the patient’s history of previous allergic angioneurotic edema following the administration of local analgesic drugs, a general anesthetic utilizing nitrous oxide and oxygen ~a.3 administered and removal of the maxillary left lateral incisor and third molar was attempted. The patient reacted poorly to the anesthetic, and the dentist found that it would be necessary to perform a surgical procedure in order to remove the third molar. He therefore administered 1.8 C.C. of a 3 per cent solution of mepivacaine without a vasoconstrictor and

436

Baumstark

O.R.,ox

and 8’eldilb

April,

& 02. 1967

completed the surgical procedure with the pa,tient awake. Swelling of the left face began during the next 2 hours and continued throughout the night. The patient returned to the dentist’s office on the morning of July 9. 110 advised her to use warm saline rinses and cold compresses and prescribed Ilosone capsules. The swelling cont,inued to increase in size, and when the patient returned to the dentist’s office that same evening she was referred to the Oral Surgery Clinic for evaluation and trratrnc,nt. Past

medical

history

The patient stated that she was allergic to “Novocaine” and had had two previous~ episodes of swelling after injection of local analgesic agents for restorative dental procedures, but these were not as severe as the present episode. She denied a history of asthma, hay fever, or other allergies, diabetes, tuberculosis, rheumatic fever, cardiovascular disease, hypertension, liver, lung, or kidney disease. There was no history of bleeding abnormalities or any other serious illness. A pilonidal cyst had been removed surgically when the patient was a teen-ager, but there had been no other hospitalizations or surgical procedures. Physical

examination

The patient had a temperature of 98.6” F., and her blood pressure was 130/70. The pulse was 80 and regular. Routine che’st roentgenogram and physical examination findings were within normal limits, except for a marked asymmetry of the face with an edematous, nonindurated, diffuse swelling involving the left infraorbital, maxillary, and mandibular regions but not crossing the midline or the inferior border of the mandible. Intraorally, the mucous membranes were normal, with no evideace ‘of hematoma formation or infection. There was slight trismus, and the extraction sites were healing normally. The swelling was painless, but the patient complained that it itched, which bothered her. Clinical

impression

This appeared to be a case of allergic (Carbocaine) administration. Treatment

and

angioneurotic

edema secondary

to mepivacaine

course

Diphenhydramine (Benadryl), 50 mg. by mouth every 6 hours’, and Orenzyme tablets, two tablets every 6 hours, were prescribed. When the patient was seen on July 11, the swelling had decreased in size about 25 per cent and was more diffuse and very soft to palpation. On July 12 the swelling was 751per cent decreased and the patient showed great improvement subjectively. When the patient was seen again on July 16, recovery was complete.

DISCUSSION

The case just reported illustrates a phenomenon with which every dentist should be familiar. Allergic angioneurotic edema probably is often dismissed as traumatic, infectious, or surgical in origin. Since it may simulate these superficially and is self-limiting, it may be misdiagnosed if its salient features are not known and if a careful history is not taken. The diagnosis is an important one to make, as the patient’s next response to a local analgesic drug might be anaphylactic in nature. Laboratory testing and skin testing were not done in this case, since we adhere to the philosophy of Monheim,ls Steinhardt,s and others that these tests may be of little value and may provoke a serious reaction in the sensitized patient. As seen in this ease and others reported in the literature, the differential diagnosis can be made if a good history is obtained and the characteristic lesions are present. Laboratory and skin testing should be done by a competent allergist who can cope with untoward reactions.

Volume 23 Number 4

Angioneurotic

edema

437

SUMMARY Angioneurotic edema has been discussed, and its symptoms, types, etiology, pathogenesis, and treatment have been delineated. The literature has been reviewed, and a case of the allergic form of the disease, caused by the administraCon of mepivacaine for aa oral surgical procedure, has been presented. REFERENCES L. W.: 1. F;;-yii

Oral Medicine,

ed. 4, Philadelphia,

1961, J. B. Lippincott

Company,

pp.

H., and Goldman, H. M.: Oral Pathology, ed. 5, St. Louis, 1960, The C. V. 2. Thoma,‘K. Mosby Company, pp. 10691070. 3. Ormsby, 0. S., and Montgomery, H.: Diseases of the Skin, ed. 8, Philadelphia, 1954, Lea & Febiger, pp. 195-197. N. S.: J. Allergy 33: 316-329, 1962. 4. Landerman, 5. Cracker, II. R.: Diseases of the Skin, ed. 3, Philadelphia, 1903, P. Blakiston’s Son & Company, p. 155. 6. Richards, R., and Crombie, H. M.: Brit. M. J. 2: 1787, 1960. 7. &her, I.: ORAL SURG.,ORAL MED. & ORAL PATH. 16: 286-290, 1963. M. J.: Ann. Allergy 12: 659-670, 1954. 8. Steinhardt, 9. Siegel, S. C., and Bergeron, J. G.: Ann. Allergy 12: 241-252, 1954. IO. Van Arsdel, P. P., and Beall, G. N.: Arch. Int. Med. 5: ‘714, 1960. 22: 286.290, 1961. ll. Westgate, H. D., Schultz, E. A., and Van Bergen, F. H.: Anesthesiology 12. Cerqua, 5.: Minerva med. 1: 542, 1936. -13. Rose, B.: J. Allergy 12: 327, 1941. .L4. Kroll, R. G.: J. Oral Surg. 9: 17, 1951. :15. Adler, P., and Simon, M.: ORAL SURG.,ORAL MED. & ORAL PATH. 2: 1029, 1949. 16. Rickles, N. H.: ORAL SURG.,ORAL MED. & ORAL PATH. 6: 375, 1953. .L7. Mitchell, H. S.: J. Canad. Dent. A. 19: 127, 1953. .18. Siegal, S.: J. Allergy 29: 329-335, 1958. ‘19. Monheim, L. M.: Local Anesthesia and Pain Control in Dental Practice, ed. 2, St. Louis, 1961, The C. V. Mosby Company, p. 187.