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Hydatid cyst in the maxillofacial region
CASE REPORTS
Hydatid Cyst in the Maxillofacial
Region
SABRI SHUKER, BDS, MMSC, FDSRCS*
Hydatid cysts are caused by the parasite Echinowhich is found all over the world, but is most prevalent in sheep- and cattleraising countries, such as Australia, New Zealand, South America, Central Europe, and the Middle East. In Iran, they cause 1% of admissions -3 the surgical wards. Hydatidosis is a disease well known to the general surgeon but presents diagnostic difficulties to the oral and maxillofacial surgeon because only a few cases have been reported in the head and neck region.
cocc~~s grun~do.ws,
Report of Two Cases CNMJI. A 35year-old man, a laborer, came to the maxillofacial unit outpatient clinic and reported a painless swelling on the left side of the mandibular body. The patient had noticed the swelling for eight months, and it was increasing in size, causing deformity on the left mandibular body, at the anterior border of the masseter muscle (Figs. 1 and 2). Clinical examination revealed a firm, round, domeshaped swelling which did not interfere with movement of the mandible. The mass was mobile. and the covering skin was healthy. Intraoral examination revealed good oral hygiene, no carious teeth, and healthy gingival tissue. A left lateral oblique radiograph of the mandible showed a small. well-demarcated, oval radiolucent area I cm in diameter (Fig. 3). It had no relation to the periapical region of the teeth. The diagnosis was difftcult, since the size of the rarefied area was very small compared with the size of the external swelling. and no sign or symptom of an odontogenic cyst could be seen. The routine blood analysis showed leukocytosis with 6% eosinophils. While the patient was under general anesthesia, an incision 8 cm long was made in the submandibular region two finger breadths below the lower border of the mandible. The exposed mass appeared to be a white-walled cystic lesion. A hydatid cyst was diagnosed. The cystic fluid was aspirated. 10% formalin was injected in the cystic cavity. and gauze soaked with 2% formalin was placed around the cyst. The cyst enucleated easily. and about 15 daughter cysts were found inside the broad capsule. (Fig. 4). A depression 2 mm deep and 2 cm wide was found in the mandibular body with a through-and-through defect of 1 cm in the middle of the depression. This indicated that the cyst probably arose in the bone and increased in size
FIGURE 1 (ahnve). Dome-shaped swelling in front of the massetric muscle. FIGURE 2 (hr/ttrr*). Lateral view of the swelling on the left mandibular
The wound was closed, and the patient recovered uneventfully (Fig. 5). Postoperative complement fixation test for hydatid disease was positive, but the Casoni test was negative. It was noted. however. that the patient had three dogs at home.
Casr 2. A IO-year-old child, who was a shepherd, had a firm swelling on the left side of the face. pushing upward and laterally under the zygomatic arch. The cheek was swollen in the buccal fat pad region anterior to the parotid gland. The mass was the size of a tennis ball and had been present for one year (Fig. 6). On clinical examination, there was a firm immobile mass, with no facial palsy and no lymph gland enlargement. Intraoral examination revealed the mass pressing against the upper molar teeth and occupying all the retromolar region and infratemporal space. Lateral oblique radiographs of the mandible revealed a very large. round, radiolucent space 7 x 7 cm in diameter. The coronoid process and part of the ramus had been destroyed by pressure. (Fig. 7). A Waters’ view revealed an obliterated right maxillary sinus. and the zygomatic arch was elevated and increased in size (Fig. 8). This space-occupying lesion was of long duration. painless.
buccally. * Consultant Oral Surgeon. Received from Basrah Republic Hospital. Basrah. Iraq. Address correspondence and reprint requests to Sabri Shuker. ll?7$-239118U030010171 $00.80 @ American
body.
Association of Oral and Maxillofacial
171
Surgeons
172
MAXILLOFACIAI.
HYI)A’rII>
(‘k’s I
Parasitology
The word “echinococcus” originates from the Greek word meaning a “hedgehog berry,” a term describing the gross pathology of the lesion. Another descriptive Greek word applied to this disease is “hydatid,” “a drop of water.” This disease process probably was known to Hippocrates. who deThe scribed, “liver(s) . . . filled with water.“’ etiologic agent and its characteristics were described during the 17th and 18th centuries, but the complete life cycle was not documented until the 19th century. Man is usually infected by the accidental ingestion of the fertile ova of the minute tapeworm, Eckinococcus granulosus, 4-6 mm long. The embryo passes through the intestinal wall and enters the portal circulation, eventually reaching the liver. It occasionally goes to the lungs but rarely to other parts of the systemic circulation, i.e.. heart, kidney, spleen, bones, brain, orbit, salivary glands, tonsils, and tongue. In these sites, the embryo transforms into a cyst, which develops the germinal epithelium that produces broad capsules, larval forms, and eventually the scolices. The life cycle is completed when offal infected with hydatid cysts is eaten by a dog. Discussion
FIGURE 3. Oval radiolucent area of 1 cm in diameter in the mandibular body. FIGURE 4. Complete hydatid cyst with 14 daughter cysts.
and in a healthy patient: These facts were all in favor of it being a benign lesion. Since the patient had lived with dogs, hydatid disease was among the differential diagnoses. Laboratory investigation revealed a positive Casoni test and complement fixation test; routine blood analysis showed 11% eosinophils and an erythrocyte sedimentation rate (ESR) of 3.5mm/hour. These findings also favored a diagnosis of hydatid cyst. The chest radiograph was clear. While the patient was under general anesthesia, a Weber-Fergusson incision was made, a flap was raised, and the white wall of the hydatid cyst was exposed. A pack moistened with 2% formalin was packed around the exposed cyst, the fluid of the cystic cavity was aspirated, and a 10% formalin solution was injected into the cavity for 10 minutes. An incision was made through the overlying adventitia, the laminated membrane was grasped with forceps and separated from the adventitia, and the adventitia was then excised, leaving a large cavity (Fig. 9). The wound was closed, and the patient recovered uneventfully (Fig. 10). A liver scan showed irregular distribution of liver activity, with a small cold area in the posterior aspect of the right lobe, which indicated a hydatid cyst in the liver (Fig.
Ilj.
The hydatid cyst should not be overlooked in the differential diagnosis of space-occupying lesions in endemic areas.:’ Rupture of a cyst filled with highly antigenic fluid may result in an anaphylactic reaction.’ In the maxillofacial bones, hydatid disease has
FIGURE
5.
Postoperative
view
173
SHUKER
rarely been reported even in endemic areas, and its presence makes diagnosis very difficult. Bader et al.’ presented a case of hydatid cyst of the neck, which was diagnosed for several years as tuberculosis of the lymph nodes. Basryouni et al.” reported a hydatid cyst presenting as a quinsy, which was drained and only later diagnosed to be a hydatid cyst.
FIGURE 6. Facial 4de of the face.
asymmetry
showing
firm swelling
of the left
FIGURE 7 (crhol~). Large radiolucent space cornea1 process and part of the ramus indented by septic pressure. FIGURE 8 thrlo~). Obliterated right maxillary sinus with elebated zygomatic arch.
FIGURE FIGURE
9. Large hydatid IO. Postoperative
cyst after excision. appearance of the patient.
Few other cases have been reported to occur in the head and neck. Fenu in 1968 reported a case of hydatid cyst in the temporal bone, and three cases in the orbit have been reported.+” Two cases of hydatid cyst in the tongue were reported by Perl et al., in 1972, and Gracanin, in 1963.“.“’ The diagnosis was very difficult in this report’s first case, because hydatid cyst is not common in this locale, but in the second case hydatid cyst was the first differential diagnosis because the Casoni test was positive, as was the complement-fixation test;” also there were 11% eosinophils. In an endemic area, patients with masses having no definite diagnosis should have a specific test to exclude hydatid disease. Chest radiography and liver scanning should be carried out in patients proved to have hydatid cysts. In our second case, liver scanning showed a cold area highly suggestive of hydatid cyst. Total excision of the cyst is the treatment of choice in cases of head and neck hydatid cyst. since there is no effective medical treatment.” A broad-spectrum antihelminthic. mebendazole. kills hydatids in mice but not in man.‘::
MAXILLOFACIAL
Summary
3. 4. 5 6:
Two cases of hydatid cyst, one in the mandibular body and the other in the infratemporal fossa, are reported. Treatment by excision was successful in both patients.
7.
8. 9. 10. II.
References
12.
1. Lewis JW, Koss N, Kerstein MD: Ann Surg 181:390, 1975 2. Bailey, Love: Short Practice of Surgery. Ed. 17. H. K. Lewis & Co.. Ltd.. 860
13.
HYDA.TIll
C‘YS I‘
Nourmand A: Am J Trop Med Hyg, 25:845. 1976 Bader G. Rose KG, Fohlmeister I: HNO 27:237. 1979. Barryouni A. Maher A: Laryngol Otol 92:729. 19$$ Clay C, Gramet C. Morax S. et al: Bull Sot Ophthalmol FI 78:843, 1978 Magilnitski SG: Vestn Ophthaln~ol 3:78. 1978 Shukla IM, Sharna BS: Indian J Ophthalmol 26:48. 1979 Per1 P, Per1 T. Golbberg B: Oral Surg 33579. 1972 Gracanin S: J Laryngol 77:624, 1963. Capron A. Vernes A, Biguet J: Le Kyste Hydatique du Fore Journees Lyonnaises D’ Hydatidologic sim. Ed. Lyon. Amir-Jahed AK. Fardin R. Farzad A, et al: Ann Surg 182:541. 1975 Heath DD. Chenis RAF: Lancet 2:218, 1974
.
F
8
.I#
Multiple Progressive t osmopnl//c Granuloma of the Jaws FRANCISCO
M. GRANDA,
DMD,* AND RAYMOND
Eosinophilic granuloma is a benign disorder, which occurs primarily in bone but can involve soft
$01.00
@J American
DDS, MS**
tissue. It is most frequently seen in young people and usually presents as a solitary osteolytic lesion in the calvarium, femur, mandible, or ribs. Although pain or swelling may occur, the lesion is frequently asymptomatic. Microscopic examination may show large numbers of eosinophils intermingled with focal collections of morphologically normal histiocytes. The term “eosinophilic granuloma” was introduced in 1940 by Lichtenstein and Jaffe’ in a report of two cases of solitary lytic lesions of bone that were composed of histiocytes and eosinophils.
* Major, USAF, DC: Former Chief Resident in Oral and Maxillofacial Surgery. Presently Staff Oral Surgeon, USAF Hospital, Torrejon AFB, Spain. ** Colonel, USAF, DC (Retired); Former Chairman, Department of Oral Pathology. Presently Professor, Department of Pathology, P.O. Box 20068, University of Texas Dental Branch, Houston, TX 77025. The opinions contained in this article are those of the authors and are not to be construed as official or as reflecting the view of the Department of Defense or the United States Air Force. Address correspondence and reprint requests to Colonel McDaniel. (1278.239l/8210300/0174
K.McDANIEL,
Association
174
of Oral and
Max~llofacial
Surgeons
Hydatid Cyst in the Maxillofacial
Region
SABRI SHUKER, BDS, MMSC, FDSRCS*
Hydatid cysts are caused by the parasite Echinowhich is found all over the world, but is most prevalent in sheep- and cattleraising countries, such as Australia, New Zealand, South America, Central Europe, and the Middle East. In Iran, they cause 1% of admissions -3 the surgical wards. Hydatidosis is a disease well known to the general surgeon but presents diagnostic difficulties to the oral and maxillofacial surgeon because only a few cases have been reported in the head and neck region.
cocc~~s grun~do.ws,
Report of Two Cases CNMJI. A 35year-old man, a laborer, came to the maxillofacial unit outpatient clinic and reported a painless swelling on the left side of the mandibular body. The patient had noticed the swelling for eight months, and it was increasing in size, causing deformity on the left mandibular body, at the anterior border of the masseter muscle (Figs. 1 and 2). Clinical examination revealed a firm, round, domeshaped swelling which did not interfere with movement of the mandible. The mass was mobile. and the covering skin was healthy. Intraoral examination revealed good oral hygiene, no carious teeth, and healthy gingival tissue. A left lateral oblique radiograph of the mandible showed a small. well-demarcated, oval radiolucent area I cm in diameter (Fig. 3). It had no relation to the periapical region of the teeth. The diagnosis was difftcult, since the size of the rarefied area was very small compared with the size of the external swelling. and no sign or symptom of an odontogenic cyst could be seen. The routine blood analysis showed leukocytosis with 6% eosinophils. While the patient was under general anesthesia, an incision 8 cm long was made in the submandibular region two finger breadths below the lower border of the mandible. The exposed mass appeared to be a white-walled cystic lesion. A hydatid cyst was diagnosed. The cystic fluid was aspirated. 10% formalin was injected in the cystic cavity. and gauze soaked with 2% formalin was placed around the cyst. The cyst enucleated easily. and about 15 daughter cysts were found inside the broad capsule. (Fig. 4). A depression 2 mm deep and 2 cm wide was found in the mandibular body with a through-and-through defect of 1 cm in the middle of the depression. This indicated that the cyst probably arose in the bone and increased in size
FIGURE 1 (ahnve). Dome-shaped swelling in front of the massetric muscle. FIGURE 2 (hr/ttrr*). Lateral view of the swelling on the left mandibular
The wound was closed, and the patient recovered uneventfully (Fig. 5). Postoperative complement fixation test for hydatid disease was positive, but the Casoni test was negative. It was noted. however. that the patient had three dogs at home.
Casr 2. A IO-year-old child, who was a shepherd, had a firm swelling on the left side of the face. pushing upward and laterally under the zygomatic arch. The cheek was swollen in the buccal fat pad region anterior to the parotid gland. The mass was the size of a tennis ball and had been present for one year (Fig. 6). On clinical examination, there was a firm immobile mass, with no facial palsy and no lymph gland enlargement. Intraoral examination revealed the mass pressing against the upper molar teeth and occupying all the retromolar region and infratemporal space. Lateral oblique radiographs of the mandible revealed a very large. round, radiolucent space 7 x 7 cm in diameter. The coronoid process and part of the ramus had been destroyed by pressure. (Fig. 7). A Waters’ view revealed an obliterated right maxillary sinus. and the zygomatic arch was elevated and increased in size (Fig. 8). This space-occupying lesion was of long duration. painless.
buccally. * Consultant Oral Surgeon. Received from Basrah Republic Hospital. Basrah. Iraq. Address correspondence and reprint requests to Sabri Shuker. ll?7$-239118U030010171 $00.80 @ American
body.
Association of Oral and Maxillofacial
171
Surgeons
172
MAXILLOFACIAI.
HYI)A’rII>
(‘k’s I
Parasitology
The word “echinococcus” originates from the Greek word meaning a “hedgehog berry,” a term describing the gross pathology of the lesion. Another descriptive Greek word applied to this disease is “hydatid,” “a drop of water.” This disease process probably was known to Hippocrates. who deThe scribed, “liver(s) . . . filled with water.“’ etiologic agent and its characteristics were described during the 17th and 18th centuries, but the complete life cycle was not documented until the 19th century. Man is usually infected by the accidental ingestion of the fertile ova of the minute tapeworm, Eckinococcus granulosus, 4-6 mm long. The embryo passes through the intestinal wall and enters the portal circulation, eventually reaching the liver. It occasionally goes to the lungs but rarely to other parts of the systemic circulation, i.e.. heart, kidney, spleen, bones, brain, orbit, salivary glands, tonsils, and tongue. In these sites, the embryo transforms into a cyst, which develops the germinal epithelium that produces broad capsules, larval forms, and eventually the scolices. The life cycle is completed when offal infected with hydatid cysts is eaten by a dog. Discussion
FIGURE 3. Oval radiolucent area of 1 cm in diameter in the mandibular body. FIGURE 4. Complete hydatid cyst with 14 daughter cysts.
and in a healthy patient: These facts were all in favor of it being a benign lesion. Since the patient had lived with dogs, hydatid disease was among the differential diagnoses. Laboratory investigation revealed a positive Casoni test and complement fixation test; routine blood analysis showed 11% eosinophils and an erythrocyte sedimentation rate (ESR) of 3.5mm/hour. These findings also favored a diagnosis of hydatid cyst. The chest radiograph was clear. While the patient was under general anesthesia, a Weber-Fergusson incision was made, a flap was raised, and the white wall of the hydatid cyst was exposed. A pack moistened with 2% formalin was packed around the exposed cyst, the fluid of the cystic cavity was aspirated, and a 10% formalin solution was injected into the cavity for 10 minutes. An incision was made through the overlying adventitia, the laminated membrane was grasped with forceps and separated from the adventitia, and the adventitia was then excised, leaving a large cavity (Fig. 9). The wound was closed, and the patient recovered uneventfully (Fig. 10). A liver scan showed irregular distribution of liver activity, with a small cold area in the posterior aspect of the right lobe, which indicated a hydatid cyst in the liver (Fig.
Ilj.
The hydatid cyst should not be overlooked in the differential diagnosis of space-occupying lesions in endemic areas.:’ Rupture of a cyst filled with highly antigenic fluid may result in an anaphylactic reaction.’ In the maxillofacial bones, hydatid disease has
FIGURE
5.
Postoperative
view
173
SHUKER
rarely been reported even in endemic areas, and its presence makes diagnosis very difficult. Bader et al.’ presented a case of hydatid cyst of the neck, which was diagnosed for several years as tuberculosis of the lymph nodes. Basryouni et al.” reported a hydatid cyst presenting as a quinsy, which was drained and only later diagnosed to be a hydatid cyst.
FIGURE 6. Facial 4de of the face.
asymmetry
showing
firm swelling
of the left
FIGURE 7 (crhol~). Large radiolucent space cornea1 process and part of the ramus indented by septic pressure. FIGURE 8 thrlo~). Obliterated right maxillary sinus with elebated zygomatic arch.
FIGURE FIGURE
9. Large hydatid IO. Postoperative
cyst after excision. appearance of the patient.
Few other cases have been reported to occur in the head and neck. Fenu in 1968 reported a case of hydatid cyst in the temporal bone, and three cases in the orbit have been reported.+” Two cases of hydatid cyst in the tongue were reported by Perl et al., in 1972, and Gracanin, in 1963.“.“’ The diagnosis was very difficult in this report’s first case, because hydatid cyst is not common in this locale, but in the second case hydatid cyst was the first differential diagnosis because the Casoni test was positive, as was the complement-fixation test;” also there were 11% eosinophils. In an endemic area, patients with masses having no definite diagnosis should have a specific test to exclude hydatid disease. Chest radiography and liver scanning should be carried out in patients proved to have hydatid cysts. In our second case, liver scanning showed a cold area highly suggestive of hydatid cyst. Total excision of the cyst is the treatment of choice in cases of head and neck hydatid cyst. since there is no effective medical treatment.” A broad-spectrum antihelminthic. mebendazole. kills hydatids in mice but not in man.‘::
MAXILLOFACIAL
Summary
3. 4. 5 6:
Two cases of hydatid cyst, one in the mandibular body and the other in the infratemporal fossa, are reported. Treatment by excision was successful in both patients.
7.
8. 9. 10. II.
References
12.
1. Lewis JW, Koss N, Kerstein MD: Ann Surg 181:390, 1975 2. Bailey, Love: Short Practice of Surgery. Ed. 17. H. K. Lewis & Co.. Ltd.. 860
13.
HYDA.TIll
C‘YS I‘
Nourmand A: Am J Trop Med Hyg, 25:845. 1976 Bader G. Rose KG, Fohlmeister I: HNO 27:237. 1979. Barryouni A. Maher A: Laryngol Otol 92:729. 19$$ Clay C, Gramet C. Morax S. et al: Bull Sot Ophthalmol FI 78:843, 1978 Magilnitski SG: Vestn Ophthaln~ol 3:78. 1978 Shukla IM, Sharna BS: Indian J Ophthalmol 26:48. 1979 Per1 P, Per1 T. Golbberg B: Oral Surg 33579. 1972 Gracanin S: J Laryngol 77:624, 1963. Capron A. Vernes A, Biguet J: Le Kyste Hydatique du Fore Journees Lyonnaises D’ Hydatidologic sim. Ed. Lyon. Amir-Jahed AK. Fardin R. Farzad A, et al: Ann Surg 182:541. 1975 Heath DD. Chenis RAF: Lancet 2:218, 1974
.
F
8
.I#
Multiple Progressive t osmopnl//c Granuloma of the Jaws FRANCISCO
M. GRANDA,
DMD,* AND RAYMOND
Eosinophilic granuloma is a benign disorder, which occurs primarily in bone but can involve soft
$01.00
@J American
DDS, MS**
tissue. It is most frequently seen in young people and usually presents as a solitary osteolytic lesion in the calvarium, femur, mandible, or ribs. Although pain or swelling may occur, the lesion is frequently asymptomatic. Microscopic examination may show large numbers of eosinophils intermingled with focal collections of morphologically normal histiocytes. The term “eosinophilic granuloma” was introduced in 1940 by Lichtenstein and Jaffe’ in a report of two cases of solitary lytic lesions of bone that were composed of histiocytes and eosinophils.
* Major, USAF, DC: Former Chief Resident in Oral and Maxillofacial Surgery. Presently Staff Oral Surgeon, USAF Hospital, Torrejon AFB, Spain. ** Colonel, USAF, DC (Retired); Former Chairman, Department of Oral Pathology. Presently Professor, Department of Pathology, P.O. Box 20068, University of Texas Dental Branch, Houston, TX 77025. The opinions contained in this article are those of the authors and are not to be construed as official or as reflecting the view of the Department of Defense or the United States Air Force. Address correspondence and reprint requests to Colonel McDaniel. (1278.239l/8210300/0174
K.McDANIEL,
Association
174
of Oral and
Max~llofacial
Surgeons