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Rare otolaryngologic presentation of hydatid cyst
Rare otolaryngologic presentation of hydatid cyst SUATTURGUT,MD, SERDARENSARi,MD, HOSEYiN KATIRCl,MD, and SERDAR(~ELiKKANAT,MD, Ankara, Turkey
H y d a t i d
disease in the head and neck region is rare, and only a few case reports are found in the literature] It is a considerable medical and public health problem that results in serious morbidity and even mortality, especially in Mediterranean countries, the Middle East, South America, New Zealand, and Australia. It is endemic to Turkey, where close and continuous contact exists among dogs, the definitive host, and sheep, the intermediate host of the parasite. 2 Echinococcus granulosus is the most common cause of hydatid disease in human beings. The adult tapeworm, a true hermaphrodite, lives in the intestine of the definitive hosts. Its terminal segment, which contains hundreds of eggs, is released into the intestinal tract of the dog and is expelled in its feces. The intermediate host, sheep or human beings, swallows the ovum, and the outer shell of the ovum dissolves in the duodenum, liberating an embryo. 3,4 The embryo, penetrating the mucosa of the intestine of the host, reaches the liver through the portal venous system. 2 The most frequently involved organ is the liver (65%) because most of the embryos are trapped within it. Other involved sites are the lungs (25%) and, less frequently, the spleen, kidneys, heart, bone, and central nervous system. If the uncooked viscera of sheep are eaten by dogs, protoscolices develop into adult tapeworms, and the life cycle of the parasite is completed. 3,4 However, human beings do not participate in the complete cycle because infested human organs are not eaten by the definitive host. The hydatid cyst consists of three layers: one from the host and two of parasitic origin. 5 The inner (germinal) layer and outer (laminated) layer together form the
From the Department of Otolaryngology-Head and Neck Surgery, Ankara Numune Hospital. Reprint requests: Serdar Ensari, MD, Ergin Sokak, 43/1 Mebusevleri, Tando~an, Ankara, 06580, Turkey. Otolaryngol Head Neck Surg 1997;117:418-21. Copyright © 1997 by the American Academy of OtolaryngologyHead and Neck Surgery Foundation, Inc. 0194-5998/97/$5.00 + 0 23•78•82813
418
endocyst, which contains crystal-clear fluid, daughter cysts, and protoscolices. Reactions of the host tissue form the ectocyst, which is made up of chronic inflammatory cellular material and compressed parenchyma. 4
CASE REPORTS C a s e 1. A 22-year-old woman had a progressively increasing swelling in the right preanricular region of 15 years' duration. On examination, she had a 10 x 5 x 5 cm, immobile, painless, hard mass over the right preauricular region. On intraoral examination, displacement of the fight palatine tonsil towards the midline and soft palate anteriorly was noted. Other examination findings were normal. On CT examination, in the deeper lobe of the parotid, a 10 x 6 x 5 cm mass that originated from the fight parotid gland and filled the fight infratemporal fossa, was observed (Fig. 1). Results of other laboratory tests, abdominal ultrasonography, and chest x ray were normal. Total parotidectomy was performed with the patient under general anesthesia. As the cyst wall was separated from the surrounding tissues, crystal-clear liquid spurted out. It was therefore suspected that the lesion was a hydatid cyst. The operation field was then washed several times with 20% hypertonic saline solution, A Hemowac drain was placed. The postoperative period was uneventful. On histologic examination, a laminated cyst membrane was seen, which was compatible with hydatid disease. We could not find the primary site of origin. At 1-year follow-up the patient was free of disease, and no other hydatid cyst had developed. C a s e 9. A 9-year-old boy first seen with swelling on the left side of his face was admitted to the hospital. The patient also had nasal obstruction and insensibility on the same side of the face. On examination he had a 8 x 6 x 5 cm, immobile, painless, diffuse, hard mass on the left zygoma. The mass extended from the zygoma to the anterior border of the sternocleidomastoid muscle laterally and posteriorly and to the left commissure of the lip anteriorly. On intraoral examination, displacement of the soft and hard palates anteriorly and inferiorly was noted on the left side. The left palatine tonsil was displaced downward. There was also a swelling at the gingivobuccal region of the left side, and ping-pong ball crackling was noticed over this part of the mass. Laboratory findings were normal except for an eosinophilia count of 10%. CT revealed a 8 x 6.5 cm cystic mass filling
OtolaryngologyHead and Neck Surgery
TURGUT et al. 419
Volume 117 Number 4
Fig. 1. Preoperative axial CT scan of case 1.
Fig. 2. Preoperative axial CT scan of case 2.
the left pterygopalatine fossa (Fig. 2). Abdominal ultrasonography and chest x-ray findings were normal. With the patient under general anesthesia, the left pterygopalatine fossa was exposed through a Weber-Ferguson incision without the sublabial component. During surgery the
white cyst wall was perforated inadvertently because of adhesions to the sttrrounding tissue, and clear, watery fluid was discharged. The cyst capsule was removed completely (Fig. 3), and the operation field was thoroughly irrigated several times with 20% hypertonic saline solution. Afterwards the
OtolaryngologyHead and Neck Surgery October 1997
420 TURGUTet al.
Fig. 3. Excised hydatid cyst material of case 2.
cavity was obliterated by muscle and adipose tissue. The postoperative period was uneventful. A histologic examination confirmed the diagnosis of hydatid cyst. No medical treatment was applied. At 1.5-year follow-up the patient was free of disease, and no other hydatid cyst had developed. DISCUSSION
Even though in Turkey it is not unusual to see hydatid cysts in other locations, such as the liver and lung, they are rare in the head and neck region. In a literature review, however, we could find only a few cases located in the pterygopalatine fossa, which were reported by Akylldlz et al. 6 and by Gangopadhyay et al. 1 Two cases located in the maxillary sinus were reported by Goldsher et al. 7 and Atef et al. 8 Hydatid cyst occurrence in the parotid region is very rare. Only a few cases of hydatid cyst in the parotid region, reported by Ennouri et al.,9 Morel, lO Shuker,11 and Sennaro~lu et al., 12 described hydatid cyst of the infratemporal fossa. The diagnosis is made by serologic tests, including indirect hemagglutination, latex agglutination, immunoelectrophoresis, and skin tests. 6 Today the diagnosis of hydatid disease has been greatly facilitated with ultrasonography, CT, and MRI, Surgery is the most important treatment for hydatid cyst.
Although long-term medical treatment with mebendazole or albendazole has been used in the last 15 years, the results are still controversial. 13 Medical treatment may be tried if surgery is not possible because of the general condition of the patient; however, the results are unpredictable. Although the traditional choice of the treatment for hydatid disease is surgery, in the recent literature, there are several reports on percutaneous treatment of abdominal and pulmonary hydatid cysts as an alternative to surgical treatment.t3 They reported less morbidity and mortality and less recurrence. 2,13 Percutaneous treatment of head and neck hydatid cysts has not been performed so far. For this reason we prefer a surgical approach.
REFERENCES
1. GangopadhyayK, Abuzeid MO, Kfoury H. Hydatid cyst of the pterygopalatine-infratemporal fossa. J Laryngol Otol 1996;110: 978-80. 2. Akhan O, Dinqer A, G(SkSzA, Sayek [, Havllo~lu S, Abbasoglu O, et al. Percutaneous treatment of abdominal hydatid cysts with hypertonic saline and alcohol. Invest Radiol 1993;28:1217. 3, Sayek I. Hydatid disease of the liver. Hacettepe Med J 1983; 16:84-92. 4. Beggs I. The radiology of hydatid disease. Am J Radiol 1985; 145:639-48. 5. Onerci M, Turan E, Ruacan ~. Submandibular hydatid cyst. J Craniomaxillofac Surg 1991;19:359-61.
OtolaryngologyHead and Neck Surgery Volume 117 Number 4
6. Akylldaz N, 0zbilen S, Gt~ksu N. Hydatid cyst of the pterygopalatine fossa. J Oral Maxillofac Surg 1991;49:87-8. 7. Goldsher M, Eliachar I, Joachims Z, Golz A. Primary hydatid cyst of the maxillary sinus. J Laryngol Otol 1983'..97:869-70. 8. Atef M, Assal M, Arafa S. Hydatid disease of maxilla. J Laryngol Otol 1984;98:1027-9. 9. Ennouri A, Rhouma S, Makni S, Bouzouita KA, Marrekchi H, Atallah M. Cervicofacial hydatidosis. Rev Laryngol Otol Rhinol 1989;110:169-71.
TURGUTet at. 42~
10. Morel CL. Quistes hidatidicos primitivos de hidago, pancreas y paratida. Pren Med Argent 1968;55:1354-5. 11. Schuker S. Hydatid cyst in maxillofaciaI region. J Oral Maxillofac Surg 1982;40:171-4. 12. Sennaro~lu L, IJnerci M, Turan E, Sungur A. Infratemporal hydatid cyst--unusual location of echinococcosis. J Laryngol Otol 1994;108:601-3. 13. Akhan O, Ozmen MN, Dinqer A, G6qmen A, Katyoncu F. Percutaneous treatment of pulmonary hydatid cysts. Cardiovasc Intervent Radiol 1994; 17:271-5.
H y d a t i d
disease in the head and neck region is rare, and only a few case reports are found in the literature] It is a considerable medical and public health problem that results in serious morbidity and even mortality, especially in Mediterranean countries, the Middle East, South America, New Zealand, and Australia. It is endemic to Turkey, where close and continuous contact exists among dogs, the definitive host, and sheep, the intermediate host of the parasite. 2 Echinococcus granulosus is the most common cause of hydatid disease in human beings. The adult tapeworm, a true hermaphrodite, lives in the intestine of the definitive hosts. Its terminal segment, which contains hundreds of eggs, is released into the intestinal tract of the dog and is expelled in its feces. The intermediate host, sheep or human beings, swallows the ovum, and the outer shell of the ovum dissolves in the duodenum, liberating an embryo. 3,4 The embryo, penetrating the mucosa of the intestine of the host, reaches the liver through the portal venous system. 2 The most frequently involved organ is the liver (65%) because most of the embryos are trapped within it. Other involved sites are the lungs (25%) and, less frequently, the spleen, kidneys, heart, bone, and central nervous system. If the uncooked viscera of sheep are eaten by dogs, protoscolices develop into adult tapeworms, and the life cycle of the parasite is completed. 3,4 However, human beings do not participate in the complete cycle because infested human organs are not eaten by the definitive host. The hydatid cyst consists of three layers: one from the host and two of parasitic origin. 5 The inner (germinal) layer and outer (laminated) layer together form the
From the Department of Otolaryngology-Head and Neck Surgery, Ankara Numune Hospital. Reprint requests: Serdar Ensari, MD, Ergin Sokak, 43/1 Mebusevleri, Tando~an, Ankara, 06580, Turkey. Otolaryngol Head Neck Surg 1997;117:418-21. Copyright © 1997 by the American Academy of OtolaryngologyHead and Neck Surgery Foundation, Inc. 0194-5998/97/$5.00 + 0 23•78•82813
418
endocyst, which contains crystal-clear fluid, daughter cysts, and protoscolices. Reactions of the host tissue form the ectocyst, which is made up of chronic inflammatory cellular material and compressed parenchyma. 4
CASE REPORTS C a s e 1. A 22-year-old woman had a progressively increasing swelling in the right preanricular region of 15 years' duration. On examination, she had a 10 x 5 x 5 cm, immobile, painless, hard mass over the right preauricular region. On intraoral examination, displacement of the fight palatine tonsil towards the midline and soft palate anteriorly was noted. Other examination findings were normal. On CT examination, in the deeper lobe of the parotid, a 10 x 6 x 5 cm mass that originated from the fight parotid gland and filled the fight infratemporal fossa, was observed (Fig. 1). Results of other laboratory tests, abdominal ultrasonography, and chest x ray were normal. Total parotidectomy was performed with the patient under general anesthesia. As the cyst wall was separated from the surrounding tissues, crystal-clear liquid spurted out. It was therefore suspected that the lesion was a hydatid cyst. The operation field was then washed several times with 20% hypertonic saline solution, A Hemowac drain was placed. The postoperative period was uneventful. On histologic examination, a laminated cyst membrane was seen, which was compatible with hydatid disease. We could not find the primary site of origin. At 1-year follow-up the patient was free of disease, and no other hydatid cyst had developed. C a s e 9. A 9-year-old boy first seen with swelling on the left side of his face was admitted to the hospital. The patient also had nasal obstruction and insensibility on the same side of the face. On examination he had a 8 x 6 x 5 cm, immobile, painless, diffuse, hard mass on the left zygoma. The mass extended from the zygoma to the anterior border of the sternocleidomastoid muscle laterally and posteriorly and to the left commissure of the lip anteriorly. On intraoral examination, displacement of the soft and hard palates anteriorly and inferiorly was noted on the left side. The left palatine tonsil was displaced downward. There was also a swelling at the gingivobuccal region of the left side, and ping-pong ball crackling was noticed over this part of the mass. Laboratory findings were normal except for an eosinophilia count of 10%. CT revealed a 8 x 6.5 cm cystic mass filling
OtolaryngologyHead and Neck Surgery
TURGUT et al. 419
Volume 117 Number 4
Fig. 1. Preoperative axial CT scan of case 1.
Fig. 2. Preoperative axial CT scan of case 2.
the left pterygopalatine fossa (Fig. 2). Abdominal ultrasonography and chest x-ray findings were normal. With the patient under general anesthesia, the left pterygopalatine fossa was exposed through a Weber-Ferguson incision without the sublabial component. During surgery the
white cyst wall was perforated inadvertently because of adhesions to the sttrrounding tissue, and clear, watery fluid was discharged. The cyst capsule was removed completely (Fig. 3), and the operation field was thoroughly irrigated several times with 20% hypertonic saline solution. Afterwards the
OtolaryngologyHead and Neck Surgery October 1997
420 TURGUTet al.
Fig. 3. Excised hydatid cyst material of case 2.
cavity was obliterated by muscle and adipose tissue. The postoperative period was uneventful. A histologic examination confirmed the diagnosis of hydatid cyst. No medical treatment was applied. At 1.5-year follow-up the patient was free of disease, and no other hydatid cyst had developed. DISCUSSION
Even though in Turkey it is not unusual to see hydatid cysts in other locations, such as the liver and lung, they are rare in the head and neck region. In a literature review, however, we could find only a few cases located in the pterygopalatine fossa, which were reported by Akylldlz et al. 6 and by Gangopadhyay et al. 1 Two cases located in the maxillary sinus were reported by Goldsher et al. 7 and Atef et al. 8 Hydatid cyst occurrence in the parotid region is very rare. Only a few cases of hydatid cyst in the parotid region, reported by Ennouri et al.,9 Morel, lO Shuker,11 and Sennaro~lu et al., 12 described hydatid cyst of the infratemporal fossa. The diagnosis is made by serologic tests, including indirect hemagglutination, latex agglutination, immunoelectrophoresis, and skin tests. 6 Today the diagnosis of hydatid disease has been greatly facilitated with ultrasonography, CT, and MRI, Surgery is the most important treatment for hydatid cyst.
Although long-term medical treatment with mebendazole or albendazole has been used in the last 15 years, the results are still controversial. 13 Medical treatment may be tried if surgery is not possible because of the general condition of the patient; however, the results are unpredictable. Although the traditional choice of the treatment for hydatid disease is surgery, in the recent literature, there are several reports on percutaneous treatment of abdominal and pulmonary hydatid cysts as an alternative to surgical treatment.t3 They reported less morbidity and mortality and less recurrence. 2,13 Percutaneous treatment of head and neck hydatid cysts has not been performed so far. For this reason we prefer a surgical approach.
REFERENCES
1. GangopadhyayK, Abuzeid MO, Kfoury H. Hydatid cyst of the pterygopalatine-infratemporal fossa. J Laryngol Otol 1996;110: 978-80. 2. Akhan O, Dinqer A, G(SkSzA, Sayek [, Havllo~lu S, Abbasoglu O, et al. Percutaneous treatment of abdominal hydatid cysts with hypertonic saline and alcohol. Invest Radiol 1993;28:1217. 3, Sayek I. Hydatid disease of the liver. Hacettepe Med J 1983; 16:84-92. 4. Beggs I. The radiology of hydatid disease. Am J Radiol 1985; 145:639-48. 5. Onerci M, Turan E, Ruacan ~. Submandibular hydatid cyst. J Craniomaxillofac Surg 1991;19:359-61.
OtolaryngologyHead and Neck Surgery Volume 117 Number 4
6. Akylldaz N, 0zbilen S, Gt~ksu N. Hydatid cyst of the pterygopalatine fossa. J Oral Maxillofac Surg 1991;49:87-8. 7. Goldsher M, Eliachar I, Joachims Z, Golz A. Primary hydatid cyst of the maxillary sinus. J Laryngol Otol 1983'..97:869-70. 8. Atef M, Assal M, Arafa S. Hydatid disease of maxilla. J Laryngol Otol 1984;98:1027-9. 9. Ennouri A, Rhouma S, Makni S, Bouzouita KA, Marrekchi H, Atallah M. Cervicofacial hydatidosis. Rev Laryngol Otol Rhinol 1989;110:169-71.
TURGUTet at. 42~
10. Morel CL. Quistes hidatidicos primitivos de hidago, pancreas y paratida. Pren Med Argent 1968;55:1354-5. 11. Schuker S. Hydatid cyst in maxillofaciaI region. J Oral Maxillofac Surg 1982;40:171-4. 12. Sennaro~lu L, IJnerci M, Turan E, Sungur A. Infratemporal hydatid cyst--unusual location of echinococcosis. J Laryngol Otol 1994;108:601-3. 13. Akhan O, Ozmen MN, Dinqer A, G6qmen A, Katyoncu F. Percutaneous treatment of pulmonary hydatid cysts. Cardiovasc Intervent Radiol 1994; 17:271-5.