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An unorthodox intraosseous lesion of the mandible
AN UNORTHODOX
INTRAOSSEOUS
LESION
OF THE MANDIBLE
Re’port of a Case
T HAS heen fairly well substantiated that there should be no surgical interference in casts ol’ fibrous dysplasia until afkr the patients have passed adolescence, since injudicious disturbance of these lesions during this period will cause them t,o prolifrrate more rapidly than if they are left unmolested. As illustrated by the following case, t,his is also true during a period OS pregnanq’.
I
A 19-p3ar-old Piegro woman Came to the Allwt E:instcin Medical Yentcr dental clinic in Drcember, 1956, complaining of swere l~iu of one \vcek ‘Y duration in the lrft mandible. Fire months earlier she had csperienccd a tenllcr swelling of the left submandibular Icgion. This was attributed to pericoronitis of the left third molars, and these teeth Tvere cstracted. The pain persisted, however, and thv patient was referwtl to the x-rap department. where nothing of significance was found (Pig. 1 ). After two weeks of symptomatic treatment, tlltl patient disappeared and was not swn agniu until the pain returned. The patient was a well-developed, well-nourished young woman in her l.hird month 01 pregnancy. Extraoral examination rcvwled a rliRust:, tvndcr welling of the lcfl mandible*. ~~~~~asuri~~g 3 by L’ cm., which !\a$ hard and fiwd and h;ul tlistt~r~~lrrl the inferior lwrrler ( Fig. :! j. No palpably lymph nodes wlt’o diswrni hII-.
and sinw tlv possibility Because the laboratory tests wew not diagnostic, Under block aud infiltratiw existed, an intraosseous biol)sv was performwl. mucoperiostral flap was elevat,ed and retracted. Thrt outer cortical bony plate
of tumefact,ion anesthesia, :t was lifted away.
oxposing the biopsy site. There was much hemorrhage. The interstices were found to be soft, granular in color, and encased in cortical bouy crypts; they were not limited by > reddish memhrant PS or capsules, and they continued from the body of the mandible up the ascending I’amus. T here seemed to be srrrral nidi of origin. AZfter the flap was returned and roaptcd.
F’ip. Fig P’ig
1:--Koent~enonram -.” --lAxembcr,
19j6.
taken Note
w postoperative diagnosis microscopic examination. Xicroscopic:
essentially
the same
Sections pattern.
on
July svvelling
2.
27, 1956, of left
showing mandible.
no txidencvz
of fibrous dj-splasia was suggested The patholoyist ‘s report of .r:tIl, II,
of pnthologir
chanp~.
and the material w:k 1957, nTas 51s fohv3:
of both the soft and the decalcified material show It consists of trabeculae of well-established bone in
zmd brous
Fig. areas dys
Fix:.
:,.-I’trotorrlicrograyll
showing
area
of
new
bone
and
fib~,ous
clt~t~r~~nts
Since the surgical interference did not elicit the type of clinical respunsc that one might expect is a chronic osteomyelitis, such as swelling, clevntion of temperatulc, ;rntl possible collapse of pcriosteal flap, and sime the pathologist’s report flit1 suggest :IIL ” iwr (Fig. 51, it wns our opinion tllat ne were dealing with a fibrous dysplasia of the mandible whicll wts increasing in siw, due to the hormonal influence associatc~d with pregnancy. More cGlcnec was desired, however, and a hone survey was takcu; this proved negative. The patient was referred for a wml)lete twnmtologic work-up to determine either the presence of a sickle-cell or remot,c Coolcy ‘s ancm~i:t. The patient had continually manifested a peripheral anemia in which hemogloljin wrietl from 8.4 to 10.i grams, hcmatocrits from 32 to 34 lw cent, and white 1~100~1 wlln from 8,400 to 17,700. Vitamin and iron therapy caused no dramatic improvrmerrt. A I)lood smrar demonstrated a 2light xcllronli:~, :L moder:Lt(~ l~oikilocytosi~, ~(1 a marked anisocytosis. bickle-cell anemiu was discounted, an11 mc~g:JolJlastic awmia of l>r~gnanq was ruled out. During the ensuing months, tlic lesion grew larger until it occupied an area approxiIt was tl,bcidctl tllxt a sword biopsy might be more rqlrcscntative rn:tteJy 6 cm. in diamotc~r. 0 f the tunm, and 011 k’csl,. 23, 1037, tlw ptient \v:ts opcmtcd on again. As bvfow, Inwnt, gr:~nular, spongy bone w:w found; howwc~r, in an area adjawut lo the inferior alvt~olar ranal there were several crypts of bone which Iwused a grariulntiou type of tissue different from that yet seen. The bone and granulation tissue wre chisclcrl fwe PII lrloc and placed in IO per cent formalin solution for histologic r~samination. Again, healing was uneventful, and the repair took less time than was anticipated. On tlte sixth ~~ostolwrativc day tlw wturos ‘I’110 pathologist’s report follows: \vcre rf!movcvl. .llil:lYlscopic : The esseutinl tissue is a mixture quitts characteristic plasma cells. These are almost, areas. The dispersal :~ppears more ncoplastic than
of fil~row tissue infiltratctl IJ\ sheetlike in tlialril)ution in som(k inflammatory.
Fig. Avig. 6.- -Roentgenogram E‘ig. 7.- -Photograph Ylet Of the mandible
showing soft-tissue taken in December, and greater soft-tissue
7. enlargement 1957. Note reaction.
of greater
left
angle distentio
11 mndi
,f
the
ble. infm
Fig.
R.-Roentgenogram
showing
rarefkd
areas
filled
with
new
bomb
912
DISCUS8IO~ dySpl?LSia with T am of the opinion that this was ;I ('ilij(! OC fj}jlV)llS csisting chronic osteomyelitist iIll<\ I \VOIlld lilic t0 l)OSt\Llat,c!;lS follO\VS:
il
('I)-
A hamartoma was present, in t,lle ma,ndiblc. ‘Phis latent, congenital defthvl was disturbed by the trauma reslllt in,(p front removal of a mandibular third molar. We can further deduce that the l)roliFrrat.ion of this tissue was caust:tl by the stimulus of the hormones associated with the: pregnancy, for we are already acquainted with the Pact, that surgical int,erference in cases of fibrous dysplasia during the adolescent period has cadged sudden expansive spurts of tissue growth with accompanying disfigurement. With the postpartum changes in the hormonal lcvcl, the lesion regressed and filled with new bone. Since the bone in the expanding lesion was poorl)-
A l!+year-old female patient sought treatment of a swollen, painful jaw in July, 1!%6. Because of pericoronitis, the left rnasillar>- and mandibular third molars were removed. The patient returned five lrlonths later with renewed pain and swelling and in a pregnant, state. A review of former roentgenograms disclosed what could be considered a hamartoma at the inferior aspect of the left mandible. Because of the problematic diagnosis, three biopsy specimens were taken from different sites in the mandible. The jaw swelling increased corltinuously unt,il the patient gave birth to a child in Ma>-, 1957. At t.his time, the swelling decreased and the rarefaction of bone filled. Subsequent to t,his, the patient dc~elopcd abscesses in the suhmaxi1lar.v region in November, 1957, and again in October, 195s. These were incised and drained, and they healed after treatrnent with a broad-spcct,rum antibiotic. Three years after the original complaint, the patient conceived and was dtllivered of a second child with no cxncerbat~ion of the orignial lesion. .1t no time during the second pregnancy was any oral surgical procedure performed. Jt is my opinion that this was a case of fibrous d,vsplasia complicated by a chronic osteornyclitis. CONCLUSION A disfiguring rate of growth will most often 1~3 caused by any tampering with a lesion of fibrous dysplasia when the paiient is in a state of hormonal activity, as during adolescence or pregnancy. (This was borne out in the present case by the fact that during the patient,‘s second pt’egz‘nanc-, when thcrtl was no surgical intervention, there was no increased growth in the .jaw.) C’onseyuently. I belie\-c that, difficult as it. is, watchfnl Sll~V~illilIlW without surgical intct*i’ercncc is th(l treat,mcnt of rlioice in illlv Iuidiagnosed bony jaw lesion which t’ol lows a benign co17rsc.
I. Colbv. 2. 0. 4. 3. 6. 7. S.
B.: Fibrous Dysplasia. In National Naval Medical Center: Color r\tl:w of Oral “‘Pathology, Philabeiphia, 1956, J. B. Lippincott Company, p. 126. Falconer, M. A., Cone, C. L.. and Robb-Smith, A.: Fibrous Dvsplasia of Rone With Endocrine ‘Diso?d& and’ Cutaneous Pigmentation, Quart. J. &d. 11: 121, 1942. ORAL IjVKG., @LIT, &hD. & ORAT, Talky, D. B.: Familial Fibrous Dysplasia of the Jaw, PATH. 5: 1012. 1952. Jsffe, .T. IT.: Giani-Cell Reparative Granuloma, Traumatie Bone Cyst, and Fibrous D~splasix of Jaw Bones, ORAL SURG., ORAL MEI). & ORAL PATII. 6: 159, 1953. Lichtenstein, I,.: Polyostotic Fibrous Dysplxsia, Arch. 8urp. 36: 8il, 19.75. Rchlumberger, H. G.: Fibrous Dysplasia of Single Kones, Mil. Surg. 99: 504, 1%46. Hchlumberger, H. G.: Fibrous Dysplasia of the Masilln :d hnntlihlc, Am. .r. Odh~~dontics & Oral Surg. 32: 5i9, 1946. Tiecke, R,.. Stuteville, O., and Calandra, J.: Pathologic~ I’l~ywiolo~~ of Oral l)iw:~sc:, Sl. I,ouis, 1959, The C. V. RIoshy Company~ 1’. X33.
INTRAOSSEOUS
LESION
OF THE MANDIBLE
Re’port of a Case
T HAS heen fairly well substantiated that there should be no surgical interference in casts ol’ fibrous dysplasia until afkr the patients have passed adolescence, since injudicious disturbance of these lesions during this period will cause them t,o prolifrrate more rapidly than if they are left unmolested. As illustrated by the following case, t,his is also true during a period OS pregnanq’.
I
A 19-p3ar-old Piegro woman Came to the Allwt E:instcin Medical Yentcr dental clinic in Drcember, 1956, complaining of swere l~iu of one \vcek ‘Y duration in the lrft mandible. Fire months earlier she had csperienccd a tenllcr swelling of the left submandibular Icgion. This was attributed to pericoronitis of the left third molars, and these teeth Tvere cstracted. The pain persisted, however, and thv patient was referwtl to the x-rap department. where nothing of significance was found (Pig. 1 ). After two weeks of symptomatic treatment, tlltl patient disappeared and was not swn agniu until the pain returned. The patient was a well-developed, well-nourished young woman in her l.hird month 01 pregnancy. Extraoral examination rcvwled a rliRust:, tvndcr welling of the lcfl mandible*. ~~~~~asuri~~g 3 by L’ cm., which !\a$ hard and fiwd and h;ul tlistt~r~~lrrl the inferior lwrrler ( Fig. :! j. No palpably lymph nodes wlt’o diswrni hII-.
and sinw tlv possibility Because the laboratory tests wew not diagnostic, Under block aud infiltratiw existed, an intraosseous biol)sv was performwl. mucoperiostral flap was elevat,ed and retracted. Thrt outer cortical bony plate
of tumefact,ion anesthesia, :t was lifted away.
oxposing the biopsy site. There was much hemorrhage. The interstices were found to be soft, granular in color, and encased in cortical bouy crypts; they were not limited by > reddish memhrant PS or capsules, and they continued from the body of the mandible up the ascending I’amus. T here seemed to be srrrral nidi of origin. AZfter the flap was returned and roaptcd.
F’ip. Fig P’ig
1:--Koent~enonram -.” --lAxembcr,
19j6.
taken Note
w postoperative diagnosis microscopic examination. Xicroscopic:
essentially
the same
Sections pattern.
on
July svvelling
2.
27, 1956, of left
showing mandible.
no txidencvz
of fibrous dj-splasia was suggested The patholoyist ‘s report of .r:tIl, II,
of pnthologir
chanp~.
and the material w:k 1957, nTas 51s fohv3:
of both the soft and the decalcified material show It consists of trabeculae of well-established bone in
zmd brous
Fig. areas dys
Fix:.
:,.-I’trotorrlicrograyll
showing
area
of
new
bone
and
fib~,ous
clt~t~r~~nts
Since the surgical interference did not elicit the type of clinical respunsc that one might expect is a chronic osteomyelitis, such as swelling, clevntion of temperatulc, ;rntl possible collapse of pcriosteal flap, and sime the pathologist’s report flit1 suggest :IIL ” iwr
of fil~row tissue infiltratctl IJ\ sheetlike in tlialril)ution in som(k inflammatory.
Fig. Avig. 6.- -Roentgenogram E‘ig. 7.- -Photograph Ylet Of the mandible
showing soft-tissue taken in December, and greater soft-tissue
7. enlargement 1957. Note reaction.
of greater
left
angle distentio
11 mndi
,f
the
ble. infm
Fig.
R.-Roentgenogram
showing
rarefkd
areas
filled
with
new
bomb
912
DISCUS8IO~ dySpl?LSia with T am of the opinion that this was ;I ('ilij(! OC fj}jlV)llS csisting chronic osteomyelitist iIll<\ I \VOIlld lilic t0 l)OSt\Llat,c!;lS follO\VS:
il
('I)-
A hamartoma was present, in t,lle ma,ndiblc. ‘Phis latent, congenital defthvl was disturbed by the trauma reslllt in,(p front removal of a mandibular third molar. We can further deduce that the l)roliFrrat.ion of this tissue was caust:tl by the stimulus of the hormones associated with the: pregnancy, for we are already acquainted with the Pact, that surgical int,erference in cases of fibrous dysplasia during the adolescent period has cadged sudden expansive spurts of tissue growth with accompanying disfigurement. With the postpartum changes in the hormonal lcvcl, the lesion regressed and filled with new bone. Since the bone in the expanding lesion was poorl)-
A l!+year-old female patient sought treatment of a swollen, painful jaw in July, 1!%6. Because of pericoronitis, the left rnasillar>- and mandibular third molars were removed. The patient returned five lrlonths later with renewed pain and swelling and in a pregnant, state. A review of former roentgenograms disclosed what could be considered a hamartoma at the inferior aspect of the left mandible. Because of the problematic diagnosis, three biopsy specimens were taken from different sites in the mandible. The jaw swelling increased corltinuously unt,il the patient gave birth to a child in Ma>-, 1957. At t.his time, the swelling decreased and the rarefaction of bone filled. Subsequent to t,his, the patient dc~elopcd abscesses in the suhmaxi1lar.v region in November, 1957, and again in October, 195s. These were incised and drained, and they healed after treatrnent with a broad-spcct,rum antibiotic. Three years after the original complaint, the patient conceived and was dtllivered of a second child with no cxncerbat~ion of the orignial lesion. .1t no time during the second pregnancy was any oral surgical procedure performed. Jt is my opinion that this was a case of fibrous d,vsplasia complicated by a chronic osteornyclitis. CONCLUSION A disfiguring rate of growth will most often 1~3 caused by any tampering with a lesion of fibrous dysplasia when the paiient is in a state of hormonal activity, as during adolescence or pregnancy. (This was borne out in the present case by the fact that during the patient,‘s second pt’egz‘nanc-, when thcrtl was no surgical intervention, there was no increased growth in the .jaw.) C’onseyuently. I belie\-c that, difficult as it. is, watchfnl Sll~V~illilIlW without surgical intct*i’ercncc is th(l treat,mcnt of rlioice in illlv Iuidiagnosed bony jaw lesion which t’ol lows a benign co17rsc.
I. Colbv. 2. 0. 4. 3. 6. 7. S.
B.: Fibrous Dysplasia. In National Naval Medical Center: Color r\tl:w of Oral “‘Pathology, Philabeiphia, 1956, J. B. Lippincott Company, p. 126. Falconer, M. A., Cone, C. L.. and Robb-Smith, A.: Fibrous Dvsplasia of Rone With Endocrine ‘Diso?d& and’ Cutaneous Pigmentation, Quart. J. &d. 11: 121, 1942. ORAL IjVKG., @LIT, &hD. & ORAT, Talky, D. B.: Familial Fibrous Dysplasia of the Jaw, PATH. 5: 1012. 1952. Jsffe, .T. IT.: Giani-Cell Reparative Granuloma, Traumatie Bone Cyst, and Fibrous D~splasix of Jaw Bones, ORAL SURG., ORAL MEI). & ORAL PATII. 6: 159, 1953. Lichtenstein, I,.: Polyostotic Fibrous Dysplxsia, Arch. 8urp. 36: 8il, 19.75. Rchlumberger, H. G.: Fibrous Dysplasia of Single Kones, Mil. Surg. 99: 504, 1%46. Hchlumberger, H. G.: Fibrous Dysplasia of the Masilln :d hnntlihlc, Am. .r. Odh~~dontics & Oral Surg. 32: 5i9, 1946. Tiecke, R,.. Stuteville, O., and Calandra, J.: Pathologic~ I’l~ywiolo~~ of Oral l)iw:~sc:, Sl. I,ouis, 1959, The C. V. RIoshy Company~ 1’. X33.