Globulomaxillary cyst invading the maxillary antrum
T
Report
of a case
Il’illianz Jerome
6’. Johr&on, C’. ~toopnck,
UNITED
STATES
iYA\-AL
Liwtcnan t (IX!) 7iS’N,” ad (‘nptain (D(I) USN** HOSPITAL,
JACKSONVILLE,
k-LA.
he case to be presented is one in which a glolrulorrr;rsillrrry cyst at,tained an unusual degree of growth and inyolvcment. This case is also of interest because it demonstrates the importance of certain relevant factors that are sometimes overlooked in the over-all diagnosis and treatment of neoplastic and developmental lesions. These factors arc ( I) maintenance, bs all departments concerned, of a complete record of diagnosis, diagnostic aids, preoperative photographs, iriitia.1 t,rea.tmcnt, follow-up care, and post,opcrative photographs and (2) an adequate program and schedule of postoperative folio~~up examinations. The availability of a complete record of primary diagnosis, diagnostic aids, and treatment is must important in the event of rccurrcnce and compliaations at, a future date. Accurate and complete records arc cspcciallg- important in this clay of rapid travel and frequent changes of address to all pasts of t,ho country and the world. Witbout tlrcqe ~cor~ds, it is often difficult, if not impossible, to d&ermine the extent and nature of the or?ginal I&on and the amount (vi’ i.issucb ~rmovt~d snrgicniiy durinp initial twntmcnt. An adN~ll:ltc‘ prograrrl 01’ ~>OStOJ)C~illiVt’ i~SillllirtiltioI1 aIlt1 folluh-up sllodd 1x3 instituted in all irrstanccbs of pathologic change. l’criodic cx,amination would ccrt.ainly rovca.1 the prcsencc of recurrent. disease 01 a IWV- primary lesion, should rit,htar clt~vclol), arrd rrrak(l it. possible to limit1 i’urther dt~vc*lopment of: thci disease
676
.lohmfon
rind Stoopuck
O.S., O.M. $ O.P. November, 1966
process: b:~ prompt institution of proper thcrapcutic procedures. This, in turn, would prevent a, permanent defect which is virtually impossible to correct cxcept by further extensive reconstructive surgical procedures or maxillofacial prostheses. Neither of these basic principles wcrc followed a.t the time of initial treatment in the case reported here. As a result, diagnosis and treatment planning during the described admission were complicated. PATHOLOGY
REVIEW
The globuloma,xillary cyst forms and develops at the site of fusion of the globular and maxillary processes of the maxilla. It usually develops as a central bone lesion between the roots of the maxillary lateral incisor and the cuspid. Because of its expa,nsile nature, it brings about separation of the roots, forming an inverted droplike radiolucent lesion as seen roentgenographically in its early stages of deve1opment.l Hertz2 stated that “globulomaxillary cysts are rare,” t.here being approximately 135 cases recorded in the available literature, and he reported two cases involving the maxillary antrum. Microscopically, the cyst wall is composed of a thick, firm membrane of connective tissue whose inner surface is usually lined with a layer of stratified squamous epithelium but may have areas consisting o’f ciliated columnar cells. When the lesion extends beyond the apices of the teeth, it may be easily confused with a periodontal or radicular cyst. A positive diagnosis can be established easily by vitality tests of adjacent teeth, which will usually elicit a positive reaction, and by the presence of a well-outlined periodontal space around the apices of these teeth. Both findings will be present if the cyst is fissural in nature but will be absent in the radicular type of cyst.3 Treatment of the condition consists of surgical enucleation or marsupialization, depending on the size of the osseous defect. CASE REPORT A 29-year-old Caucasian man was admitted to the Dental Service of the United Naval Hospital, Jacksonville, Florida, on Sept. 21, 1965, following referral from the Air Station at Cecil Field, Florida, for cvnluation and treatment of what appeared an osteolytic, cystlike lesion surrounding the roots of the maxillary right molars. This was disclosed by dental roentgenograms during an examination which the patient because of “loose, sore upper right molars” (Figs. 1 and 2).
States Naval to be lesion sought
History
The patient stated that the maxillary right lateral incisor, cuspid, first and second premolars, and an associated small lesion, the nature of which he was unable to remember, were removed in 1946 at a hospital in Texas. Physical
examination
General examination revealed a well-developed, well-nourished man in no acute distress. The bridge of the nose was seen to deviate toward the left, while the tip of the nose was turned toward the right of the midline. Some swelling was noted inferior to the right zygomatic arch (Fig. 3). The na+ifln+ l>~rl smll~tnrnc: of IIIWPP rwniratorv infection. The maxillary right molars
Volume Number
22 5
1
Figs. 1 and 8. Dental periapied roentgenograms ostcolytic activity and expansile nature of lesion.
Fig. J. Facial t~twtrtl riglit.
portrait
of patient
showing
of right
deviation
were quite mobile anil wtbre displaced out of the plane of IO percussion, ancl there was a long, narrow (about 2 by gingiva along the c’ourse of the greater palatine vessels, pressible. The external alveolar cortical plate seemed gingivae were tender and inflamefl. Roentganographic
maxillary
of bridge
alveolar
of now
toward
ridge
left
sllowing
a nd tip
occlusion. The tcctti wcrc very tender 3 cm. j area of edema in the bbalatal which was nontender and t>asilJ i comto be expandtd, and the eve dying
findings
The chest- film was essentially negative. Maxillary sinus films revealed that the right maxillary antrum was opacsifird, with the density of soft tissue. The lateral wall of the right antrum was complrtely destroyed, and partial destruction was noted along the media .I and
O.S., O.M. & O.P. November, 1966 inferior aspwts of tlic antrum into the nasal cavity. The lesion extended upward to the floor of the right orbit, at \vhich point some sclwosic of thr floor was seen, especially at the lateral into the malar cminenw and process. The right molars had been disaspwt, \vitlt invasion plawd dow~~ward 11y the mass (Figs. 1 and 5). Laboratory
findings
Complete blood count, urinalysis, limits, and the serologic examination
and biochemistry was negative.
determinations
were
within
normal
course and treatment After a routine admission medical work-up had been completed, a request for information concerning the earlier procedure and treatment was forwarded to the Texas hospital where the patient had bwn treated in 1946. This was returned to us with the statement that the hospital’s records did not date back that far. This unavailability of past records necessitated surgical intervention without the benefit of information concerning the earlier pathologic examination findings and diagnosis for use as an aid to surgical planning, leaving much to be desired from our standpoint. Because of the lack of such records, and in view of the diffuse invasive appearance of the lesion roentgenographioally, a more radical approach was decided upon. Preoperative antibiotic therapy was started and on Sept. 28, 1965, the patient was taken to the oral surgery operating room, where, under l&al xyloeaine second-division block and infiltration anesthesia, the following surgical procedures were perfowled. The maxillary central and left lateral incisors were extracted for access and with a view to possible future radiation therapy to the maxilla. Following this, an elliptical incision was made from the right tuberosity, forward around the necks of the maxillary molars to the right cuspid arca. The maxillary first, second, and third molars, their alveolar process, which included the tuberosity, the entire floor of the right antrum, and the overlying gingiva, were then excised. During this excision about 30 C.C. of serosanguineous cystic fluid escaped, and a spwimen was taken for culture and sensitivity testing. Upon removal of this section, the entire antral cavity was exposed n.nd destruction of the entire posterior, lateral, and anterior antral walls was disclosed. The medial antral wall was also partially eroded into the right nasal cavity, and the lateral portion of the orbital floor was partially destroyed. The site of the previous operation was thus exposed, and the entire area was debrided. The tissues removed were sent along with the antral floor for biopsy and microscopic Iliagnosis. The gingival tissues were trimmed, and the anterior and posterior aspects mere approximated with 3-O black silk. The antral cavity was then packed open orally wit,11 petrolatum strip gauze. The patient left the operating room in good condition. Several tissue specimens were submitted for pathologic diagnosis. The first, which measured 3.5 by 1.5 cm., consisted of a section of alveolar ridge in which mere embedded three molar teeth. The undersurface of the specimen was marked by a smooth-walled defect, measuring 2.5 by 1 cm., into which projected the roots of the three molar teeth. A transverse SWtion, including the bone of the alveolar ridge, was submitted for decalcification. Hepresentative portions of the cyst wall were stripped away and submitted separately for immediate processing. Microscopic examination of the first specimen revealed sections of the segments of soft tissue showing dense and loose fibrous connective tissue and fragments of granulation tissue which mere densely infiltrated with both lymphocytes and plasma cells. Focal areas of hemorrhage were observed. The segments of tissue had the configuration of cyst wall and were covered hew and there by both stratified squamous and columnar epithelium showing cilia and mucous vacuoles. Decalcified portions of the grossly described segment of alveolar ridge on the opposite aspect (the grossly showed the specimen to be partially covered by gingiva; described cystic area) there was a cystic cavity lined by a single layer of columnar cuboidal epithelium which appeared to be ciliated in some areas. The soft tissue surrounding the bone was rather densely infiltrated with large numbers of lymphocytes and plasma cells. The bone itself was quite sclerotic, with evidence of both osteoblastic and ostooclastic activity.
Hospital
Volume Number
2% 5
Pig.
I’iys. diffuse ix and parti
(film reversed) and Waters d 5. T%itd&%~terioT ‘e appearance of lesion. Note complete obliteration rstruetion of surrounding structures.
vie& &owing rstr of right ma xillar
08, O.M. s; O.P. November. 1966 The second spwimen submitted consisted of multiple irwgular Eragnwnts of soft tissue and bone, the largrst of which measured 2.5 by 0.5 cm. The hony fragments were submitted for decalcification, and the specimrn was totally embedded. Microscopic examination of the second specimen revealwl srctions showiug multiple wgmerits of gingiva mixed with fragments of granulation tissue and bone. The gingival segments and granulation tissue were infiltrated by focal aggregations of mononuclear inflammatory cells. The bony fragments showed areas of ost,eoblastic activity. The pathologic report on the submitted specimens indicated that this lesion rcpresentcd either a large destructive dentigerous cyst or a fissural cyst (globulomaxillary cyst). In view of the associated inflammation and extension into the maxillary antrum, it is possible that, the columnar epithelium originated from the lining of the sinus cavity. However, because of the over-all course of the disease, the roentgenographic appearance, the microscopic appearance, and the surgical findings, it was the consensus of all concerned that this lesion was most representative of a globulomaxillary cyst. Microscopic sections and remaining gross tissue, together with a clinical summary and roentgenograms, were submitted to the Dental Section of the Armed Fowes Institute of Pathology for review. The report returned to this facility on Dec. 27, 1965, revealed that, after having reviewed the submitted specimens and roentgenograms, the A.F.I.1’. staff was in essential agreement with our interpretation. Postoperative antibiotic and supportive therapy was continued for 2 weeks. The antral gauze packing was removed, over a period of 10 days, in 12 inch segments in order to prevent closure of the surgical antro-oral opening. Healing and recovery were uncomplicated. On Oct. 13, 1965, the patient was discharged from the hospital to duty. While he was being followed as an outpatient, a maxillary obturator was constructed to rrplare the missing teeth and maintain the antro-oral opening. All outpatient examinations indicated normal healing, without recurrence.
SUMMARY
AND
CONCLUSION
In the foregoing case, an interesting lesion of the maxilla and maxillary sinus had apparently enlarged over a period of 19 years and entirely obliterated t,he right maxilla and sinus without the patient’s knowledge of its existence. Several attempts were made to acquire the past medical records in this ca.se, but they were not available. These records would have made it possible to obtain a more complete picture of the patient’s condition prior to surgical intervention. This case reiterates the importance of maintaining complete records of diagnosis, diagnostic aids, and initial treatment in all cases of pathologic change. The type of surgical procedure selected is usua,lly governed by the nature of the lesion. A more conservative approach for elimination of the cyst might ha,vc been indicated in this case if the past records had been available. Pre- and postoperative photographs a.re especially important in ca.ses of pathologic change in the oral regions or adjacent structures. These photographs are important guidelines for a proper reconstructive program. An adequate program of postoperative follow-up examinations is essential in the over-all treatment of pathologic conditions which may recur. The unusual size of the lesion and the destruction ,of associated timues in this case might possibly have been prevented if these factors had been properly taken into consideration at the time of the earlier surgical procedure. REFERENCES
1. Thoma, K. H.: Oral Surgery, 2. Hertz, J.: Globulomaxillary nnnr
PATW
16.
?fi?,
1963.
ed. 4, St. Louis, Cysts Invading
1963, The C. V. Mosby Company, pp. 886.888. Maxillary Sinus, ORAL SURG., ORAL MED. &
3. Jaffe, H. L.: Tumors aud Tumorous Conditions of the Bones and Joints, Philadelphia, 1961, Lea & Febiger, pp. 430-434. 4. Robinson, H. B. G., Koch, W. E., and Jasper, L. H.: lnftxted Globulomasillnry Cyst, Am. J. Orthodontics R- Oral Surg. 29: 608-611, 1943. 5. Sayer, B., and Scully, J. H.: Fissural Cysts, Am. J. Orthodont,ics 6: Oral Surg. 29: 320-327, 1943. 6. Armed FOIW~ Iustitute of Pathology : Pwsoual Communication.