Decompression of Odontogenic Cystic Lesions: Past, Present, and Future

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Lesions: Past, Present, and Future

Jaime Castro-Nu~nez, DMD

Tumors and cystic lesions of the jawbones have been described since the late 1600s and it took another 200 years for classification systems to appear in the medical, surgical, and dental literatures.

In the late 1800s, Carl Partsch introduced cystostomy, a method by which the cyst is converted into a pouch by suturing its lining to the mucosa of the oral cavity. The purpose of this article is to analyze the history, present, and future of cystic conditions of the jaws and decompression, a modality of treatment that during the past few years has regained the attention of oral and maxillofacial surgeons and pathologists owing to its relative simplicity and effectiveness compared with other conservative options.

Ó 2016 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 74:104.e1-104.e9, 2016

The history of cystic conditions of the jawbones and decompression (also known as marsupialization or exteriorization)¹as a treatment modality is intimately related to the birth of oral and maxillofacial pathology.

Odontogenic cysts and tumors were noted long before oral pathology was recognized as a specialty of dentistry by the American Dental Association in 1950, just 2 years after the founding of the American Academy of Oral Pathology.

Certainly, oral and maxillofacial pathology as currently understood began during the 1930s and 1940s, an epoch when the world was shaken by the Second World War and whose end led to a flour- ishing in virtually all fields of knowledge. Textbooks on oral pathology, such as those written by Russell Welford Bunting² and Kurt Hermann Thoma,³ and the first issues of the Archives of Clinical Oral Pa- thology in 1937 and Oral Surgery, Oral Medicine, Oral Pathology in 1948 greatly contributed to the consolidation of the specialty.

The purpose of this article is to analyze the history, present, and future of cystic conditions of the jaws and decompression, a modality of treatment that during the past few years has regained the attention of oral and maxillofacial surgeons and pathologists owing to

its relative simplicity and effectiveness compared with other conservative options.

Early Descriptions of Odontogenic Entities

The texts by Bunting²and Thoma,³published in 1929 and 1941, respectively, were the epitome of work that had started 3 centuries previously, mostly with French and British doctors. For example, in 1671 Iean Scultet⁴ described cysts of the jaws as ‘‘liquid tumors.’’ The idea that a cyst was capable of exerting enough pressure to cause bony expansion was formed at that time.⁵ In 1746 Pierre Fauchard⁶ provided the first accurate description of an odontoma. In 1774 John Hunter⁷ described what seems to be the case of a patient with an odontogenic cyst. Four years later, Anselm Louis Ber- nard Brechillet Jourdain⁸, on page 19 of his Traite des maladies et des operations reellement chirurgicales de la bouche, et des parties qui y correspondent, described 3 cases that appear to be dentigerous cysts.

During the following decades, dentists, pathologists, and surgeons started to describe with greater regularity cystic lesions and other entities of the maxillary bones. It seems that the first efforts to identify the

Researcher, Research Department, Institucion Universitaria Colegios de Colombia, Colegio Odontologico, Bogota, Colombia;

Oral and Maxillofacial Surgeon, Clınica Nueva, Bogota, Colombia;

Oral Pathologist, Universidad El Bosque School of Dentistry, Bogota, Colombia; and Visiting Professor, Universidad Evangelica de El Salvador School of Dentistry, San Salvador, El Salvador.

Address correspondence and reprint requests to Dr Castro- Nu~nez: Calle 63 #7A-44, Monterıa, Colombia; e-mail: jacastron@

hotmail.com

Received September 1 2015 Accepted September 1 2015

Ó 2016 American Association of Oral and Maxillofacial Surgeons 0278-2391/15/01272-0

http://dx.doi.org/10.1016/j.joms.2015.09.004

104.e1

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many entities of the oral and maxillofacial region started in approximately 1840, thanks to the release of the American Journal of Dental Science (AJDS) in 1839, in which such pathologies were available for the dental community to be studied, debated, and characterized. One must not forget that all this happened during the time that is known in history as the Golden Age of Dentistry (1835 to 1860).

The first volume of the AJDS contained a report of an odontogenic tumor (OT) that currently would be diagnosed as ‘‘cementoblastoma.’’⁹ The ‘‘periapical cyst’’ (sac) was described in 1839 by Brown¹⁰ and the dentigerous cyst (distended capsule, osseous cyst, serous cyst) in 1842, although it seems it had been described in France in 1778. The ‘‘keratocystic odontogenic tumor’’ (KCOT; encysted tumor, cyst, cystic carcinoma) was reported in 1844.⁵ The following years witnessed an explosion of articles related to oral pathology in the AJDS that helped dentists, surgeons, and physicians understand the com- plex pathologic processes that take place in the mouth and jaws.

It was the famous British pathologist James Paget¹¹ who in 1853 coined the term dentigerous cyst to refer to any cystic condition of dental origin. Other pathologists used many other terms to refer to unilocular cysts.

For example, what Emile Magitot called the ‘‘radicular cyst’’ was the same entity Amedee Forget knew as the

‘‘periosteal cyst’’ and Louis-Charles Malassez knew as the ‘‘radiculo-dental cyst.’’

Classifying Odontogenic Tumors

Owing to the increased activity in research and the wide variety of reports on the subject in Europe and the Americas, in 1869 Paul Pierre Broca¹² published Traite des tumeurs, where he suggested the first classification of OTs:

I. Ordinary odontomas 1. Embryoplastic period

Embryoplastic odontomas Fibroplastic

Fibrous

2. Odonto-plastic period Odonto-plastic odontomas

Cemental Bulbar

3. Crown formation period Coronal odontomas

Cemental

Pulpal or dentinal 4. Root formation period

Radicular odontomas Cemental

Dentinal

II. Composed odontomas III. Heterotopic odontomas

Broca used the term odontoma for any tumor arising from the formative dental tissues and classified them ac- cording to the stage of tooth development when abnormal growth started. In 1885 Malassez¹³made slight modifications that did not gain much attention. Three years later, in 1888, John Bland-Sutton¹⁴cleverly classified OTs based on the nature of the cells from which the ma- lignancy arose. In this system, he included odontogenic cysts and fibrous odontogenic entities, but the term odontoma remained as the usual designation for OTs.¹⁵ In 1885 Poulet and Bousquet proposed the term unilocular¹³ to designate single-cavity lesions. The first mention of a ‘‘simple cyst’’ was by the American pathologist Charles Freeman Geschickter. In an article published in 1935, Geschickter¹⁶mentioned the ‘‘simple follicular cyst,’’ which meant that the entity origi- nated from simple (primitive) odontogenic tissues.

Classification of Odontogenic Cysts

At the beginning of the 20th century, a myriad of tooth-related cystic conditions had been described and a classification system was urgently needed.¹⁷In his 1937 oral diagnosis and treatment planning textbook, Thoma¹⁸ subdivided the classification of the follicular cyst in the following manner:

1. Simple (without tooth formation) 2. Dentigerous

3. With odontoma

In 1949 he added a fourth category, the multiple cyst.¹⁹ Also in 1937, Hamilton Robinson,²⁰one of the most influential early oral pathologists in addition to Thoma and William Shafer, subdivided dentigerous cysts into 4 categories:

1. Simple 2. Compound 3. Eruption 4. Heterotropic

Eight years later (in 1945), Robinson²¹ proposed another classification for the cysts of the jawbones.

1. Developmental cysts from odontogenetic tissue 1.1. Periodontal

1.2. Dentigerous 1.3. Primordial

2. Developmental cysts of non-dental origin 2.1. Median

2.2. Globulomaxillary 2.3. Incisive canal cysts 3. Ameloblastomas

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In this classification of 1945, the term primordial was mentioned for the first time and it seems to be the same ‘‘simple cyst’’ described by Ge- schickter in 1935. The new term was favored owing to the researcher’s beliefs that the cyst arose from remnants of the dental lamina or enamel organ (primordial origin) before enamel had formed, and it replaced a tooth.²² During the first half of the 20th century, many classification methods for odontogenic cysts and tumors (odontomas) were proposed,¹⁵ and 4 terms were gener- ally used to describe odontogenic cysts:

dentigerous, simple or primordial, and follicular.

The term follicular was applied to dentigerous and primordial cysts, until Mervyn Shear²³ estab- lished in 1976 that dentigerous and follicular were synonymous.

Rise and Fall of Decompression: the Past

Understanding the behavior of benign and malignant conditions of the maxillofacial region has taken almost 4 centuries, if the work of Scultet⁴is used as a starting point. Four hundred years of research have helped to not only undress these pathologies to the point of knowing them as they are, but also to understand how they are best treated. Long before World War II, investigators knew that odontogenic cysts and tumors had the potential to recur, regardless of the treatment modality used. Factors such as type, size, and nature of the lesion, localization, etiology, and age of the patient play a role in determining the therapy.

During the past 20 years, researchers have been able to give reliable statistics on the chances an entity has to recur after a given treatment plan. It is not the purpose of this article to animate the controversy of whether marsupialization should be used. Rather, this article addresses the historical facts related to this surgical option, which is currently used to treat unicystic ameloblastomas,^1,24 KCOTs,²⁵ and other odontogenic cysts and tumors.²⁶

A cyst is a pathologic cavity in the soft tissue or bone with an outer wall composed of connective tissue and an inner wall composed of epithelium. The cavity has a watery, semisolid, or colloidal content. Cysts gradually enlarge owing to a combination of osmotic pressure²⁷ and release of growth factors and prostaglandins.^28,29 This persistent pressure exerted on the bony walls coupled with biomolecules cause bone resorption while the entity expands. Therefore, mitigating the pressure by making a small window into the cyst and keeping it open guarantees permanent drainage, thus preventing its enlargement. Eventually, bone growth will reduce the space.

This applied physical principle was the ratio- nale that Polish professor Carl Franz Maria Partsch (Fig 1) used to introduce the concept of cystostomy in 1892³⁰ and cystectomy in 1910.³¹ In an article published in German at the end of the 19th century, Partsch reported the conversion of a cyst into a pouch by suturing its lining to the mucosa of the oral cavity (Fig 2). Today, cystostomy is known as Partsch I or marsupialization, a term from the modern Latin marsupialis, from the Latin for the Greek marsupion, meaning ‘‘pouch.’’ Cystec- tomy (Partsch II) is enucleation and primary closure.

The antibiotic era was not in sight when Partsch introduced cystostomy, which, fortunately, was not associated with the development of infection, therefore becoming the treatment of choice during the first decades of the 20th century. In 1932 E.B.

Dowsett³² noted that enucleation often led to wound infection and treatment failure. Expanding on Partsch’s concept, Thomas³³ described decompression in 1947. Thomas pointed out its benefits, which were maintenance of pulp vitality, preserva- tion of the inferior alveolar nerve or maxillary sinus, prevention of fracture of the jaw, and low risk of recurrence.

FIGURE 1. Carl Franz Maria Partsch (1855 to 1932).

Jaime Castro-Nu~nez. Decompression of Odontogenic Cystic Lesions. J Oral Maxillofac Surg 2016.

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The term decompression encompasses marsupialization and is defined as any method used to relieve intracystic pressure by keeping a patent opening into the exterior, which could be the mouth (Fig 3), nose (Fig 4), or maxillary sinus (Fig 5).

Two decades after Dowsett’s article, the panorama for marsupialization and decompression was about to change. The introduction of antibiotics was almost a death knell for marsupialization and decompression and at the same time acted as a life- boat for enucleation and primary closure, which, aided by antibiotics, became the more accepted method of treatment owing to faster results. The real understanding of the many pathologic conditions of the mouth started during the 1950s and

the aggressive behavior of some of them was fully documented during this time.

Another factor that reinforced the idea that marsupialization and decompression should be avoided was the better understanding of what Hans Peter Phillipsen³⁴ called the odontogenic keratocyst in 1956 (ie, KCOT).

During the next 30 years, articles would warn against malignant transformation occurring within odontogenic cysts.^35-37 Although marsupialization was deemed unpredictable at that time, a minority still had faith in it.³⁸What is more, the great surgeon Harry Seldin³⁹reported the usefulness of marsupialization as a presurgi- cal treatment for unicystic ameloblastoma in children.

To combat recurrence, aggressive methods prevailed until the end of the 20th century.^40-44

FIGURE 2. A, Odontogenic cystic condition. B, Partsch I (cystostomy).

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FIGURE 4. Decompression to the nose.

FIGURE 3. Decompression to the mouth.

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Reborn as the Phoenix: the Present

In 1991 an article by Brøndum and Jensen⁴⁵paved the way for the reintroduction of marsupialization and decompression, which recently had resurrected like the Phoenix. In 1996 Marker et al⁴⁶successfully decompressed 23 odontogenic keratocysts using small polyethylene tubes (Fig 6). They concluded that decompression resulted in new bone formation, thick- ening of the cyst wall, and conservation of bone and anatomic structures. Another interesting conclusion was that the keratocyst epithelium was modulated histologically to non-keratocyst after decompression.

Despite such optimistic results, from the literature review it seems that it was not until the early 2000s that marsupialization and decompression regained the attention of surgeons and pathologists, much at the urging of Pogrel.^47,48

The past 10 years have observed how researchers design conduit systems to decompress odontogenic cysts and tumors. Of note, in a systematic review published by Lau and Samman¹ regarding recurrence related to treatment modalities of ameloblastoma, they called attention to the fact that 3 reported cases treated by marsupialization had complete resolution, which is paradoxical because tumors do not regress by exteriorization.

In 2007 Huang et al²⁴described a decompression plug for ameloblastomas. This plug, which is basically

the same as that described by Marker et al,⁴⁶ was effective in decreasing tumor volume and minimizing the extent of surgery. The following year, Tolstunov⁴⁹ used a catheter for the same purpose. One of the drawbacks of decompression is the size of the tube, which could be dislodged over time. This issue was cleverly tackled by Kolokythas et al²⁶ using a 16-gauge needle to create a passage for a 28-gauge

FIGURE 5. Decompression to the nasal sinus.

FIGURE 6. Decompression plug as described by Marker et al.⁴⁶ Jaime Castro-Nu~nez. Decompression of Odontogenic Cystic Lesions. J Oral Maxillofac Surg 2016.

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wire that would be secured to the teeth (Fig 7).

Another stent fixation method, using 1.2-mm screws (Fig 8), was proposed in 2012 by Swantek et al.⁵⁰In 2014 Gao et al⁵¹introduced a thermoplastic resin stent, with and without a clasp. In 2015 Delgado-Rueda et al²² maximized the benefits of decompression by using 2 decompression anesthesia tubes in the same patient (Fig 9).

The Future of Decompression

Decompression, as any other treatment technique, is not a panacea. The benefits of this old method have been described and proved over the years, and as patients and surgeons become less aggressive, this technique will become more popular when indicated.

Cases must be carefully selected and the surgeon has

FIGURE 7. Fixation method using a wire as described by Kolokythas et al.²⁶A, Passing the wire through the needle. B, Fixing the tube to the teeth.

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to weigh many factors, such as patient age, type of lesion and time of evolution, and the patient’s cooperation.

A Closing Remark: Introduction and Acceptance of a Pleonasm

To the unprepared eye, the word pleonasm might be confused with neoplasm, but they are different! A pleonasm is the use of more words than necessary to convey meaning, as a fault of style or for emphasis. This seems to be the case of the expression odontogenic

origin, which was introduced to the dental literature in the late 1970s. In this sense, Shafer’s Textbook of Oral Pa- thology⁵², a book widely used in North American and Latin American dental schools, helped disseminate the pleonasm. In the Spanish version of the book, the expression was translated as origen odontogenico. The word odontogenic comes from Greek odous, odont-, meaning

‘‘tooth,’’ and guenesis, which means ‘‘origin.’’ Therefore, the expression odontogenic origin (and its Spanish trans- lation, origen odontogenico) must be avoided because the term odontogenic already means that the condition being described is related to dental tissue.

FIGURE 8. Fixation method using small screws.

FIGURE 9. Two decompression tubes (double decompression) in the same patient.

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Acknowledgments

The artwork included in this article was done beautifully by Glenna Castro-Nu~nez, to whom the author expresses his most sincere gratitude. A word of gratitude is extended to the American Board of Oral and Maxillofacial Pathology, especially to Clarita Scioscia, for her invaluable assistance during the preparation of this article.

References

1. Lau SL, Samman N: Recurrence related to treatment modalities of unicystic ameloblastoma: A systematic review. Int J Oral Max- illofac Surg 35:681, 2006

2. Bunting RW: A Textbook of Oral Pathology, for Students and Practitioners of Dentistry. Philadelphia, PA, Lea & Febiger, 1929 3. Thoma KH: Oral Pathology. St Louis, MO, CV Mosby, 1941 4. Scultet I: L’Arcenal de Chirurgie. Lyon, France, Antoine Cellier,

1671

5. Bouquot JE, Lense EC: The beginning of oral pathology, part I:

First dental journal reports of odontogenic tumors and cysts, 1839-1860. Oral Surg Oral Med Oral Pathol 78:343, 1994 6. Fauchard P: Le Chirurgien Dentiste, ou Traite des Dents. Tome

1–2. Paris, France, Pierre Jean Mariette, 1746

7. Hunter J: The Natural History of the Human Teeth. London, UK, J Johnson, 1774

8. Jourdain ALB: Traite des maladies et des operations reellement chirurgicales de la bouche et des parties qui y correspondent.

Paris, chez Valleyre l’a^ıne, 1778

9. Philipsen HP, Reichart PA, Slootweg PJ, Slater LJ: Odontogenic tumours, in Barnes L, Eveson JW, Reichart P, et al (eds): Head and Neck Tumours. Pathology and Genetics. WHO Classification of Tu- mours. Lyon, France, IARC Press, 2005, pp 283–327

10. Brown AM: Review of Burdell and Burdell’s observations on the structure, physiology, anatomy and diseases of the teeth. Am J Dent Sci 1:19, 1839

11. Paget J: Lectures on Surgical Pathology. London, UK, Royal Col- lege of Surgeons of England, 1853

12. Broca PP: Traite des Tumeurs, Vol. 2. Paris, Asselin P, Libraire de la Faculte de Medicine, 1869

13. Martınez Castrillo JM: Diccionario General de Odontologıa y de Arte Dental. Madrid, Spain, Bailly-Bailliere, 1915

14. Bland-Sutton J: Odontomes. Trans Odont Soc London 20:32, 1888

15. Philipsen HP, Reichart PA: Classification of odontogenic tumours. A historical review. J Oral Pathol Med 35:525, 2006 16. Geschickter CF: Tumors of jaws. Am J Cancer 24:90, 1935 17. Ivey RH, Churchill HR: The need of a standardized surgical and

pathological classification of tumors and anomalies of dental origin. Am Assoc Dent Sch Trans 7:240, 1930

18. Thoma KH: Oral diagnosis and treatment planning. Philadelphia, WB Saunders, 1937

19. Thoma KH: Oral and Dental Diagnosis. Philadelphia, PA, WB Sa- unders, 1949

20. Robinson HBG: Ameloblastoma. Arch Pathol 23:831, 1937 21. Robinson HBG: Classification of cysts of the jaws. Am J Orthod

Oral Surg 31:370, 1945

22. Pogrel MA: The history of the odontogenic keratocyst. Oral Max- illofac Surg Clin North Am 15:311, 2003

23. Shear M: Cysts of the Oral Regions. Bristol, UK, John Wright &

Sons, 1976

24. Huang IY, Lau ST, Chen CH, et al: Surgical management of ameloblastoma in children. Oral Surg Oral Med Oral Pathol Oral Ra- diol Endod 104:478, 2007

25. Delgado-Rueda J, Ayala-Gomez A, Castro-Nu~nez J: Decompres- sion of an enormous keratocystic odontogenic tumor. J Cranio- fac Surg 26:460, 2015

26.Kolokythas A, Schlieve T, Miloro M: Simple method for securing a decompression tube for odontogenic cysts and tumors: A tech- nical note. J Oral Maxillofac Surg 69:2392, 2011

27.Toller PA: The osmolality of fluids from cysts of the jaws. Br Dent J 129:275, 1970

28.Harris M: Odontogenic cyst growth and prostaglandin induced bone resorption. Ann R Coll Surg Engl 60:85, 1978

29.Ninomlya T, Kubota Y, Koji T, et al: Marsupialization in- hibits interleukin-1a expression and epithelial cell prolifer- ation in odontogenic cysts. J Oral Pathol Med 31:526, 2002

30.Partsch C: Uber kiefercysten. Dtsch Mschr Zahnheilkd 10:271, 1892

31.Partsch C: Zur behandlung der kieferzysten. Dtsch Mschr Zahn- heilkd 28:252, 1910

32.Dowsett EB: Operative procedure for cysts of the jaws. Proc R Soc Med 25:47, 1932

33.Thomas EH: Saving involved vital teeth by tube drainage. J Oral Surg 5:1, 1947

34.Phillipsen HP: OM Keratocyster (kolestea-tomer). I. Kaeberne.

Tandiaegebladet 60:963, 1956

35.Hardman FG: Carcinomata arising in dental cysts. Br J Oral Surg 1:124, 1963

36.Browne RM, Gough NG: Malignant change in the epithelium lining odontogenic cysts. Cancer 29:1199, 1972

37.Areen RG, McClatchey KD, Baker HL: Squamous cell carcinoma developing in an odontogenic keratocyst. Arch Otolaryngol 107:

568, 1981

38.Brady CL, Browne RM, Calverley BC, et al: Symposium on odontogenic epithelium. Br J Oral Surg 8:1, 1970

39.Seldin SD: Ameloblastoma in young patient, report of two cases.

J Oral Surg 19:508, 1961

40.Jensen J, Sindet-Pedersen S, Simonsen EK: A comparative study of treatment of keratocysts by enucleation or enucleation com- bined with cryotherapy. A preliminary report. J Craniomaxillo- fac Surg 16:362, 1988

41.Bramley P: The odontogenic keratocyst: An approach to treatment. Int J Oral Surg 3:337, 1974

42.Voorsmit RA, Stoelinga PJ, van Haelst UJ: The management of keratocysts. Maxillofac Surg 9:228, 1981

43.Irvine GH, Bowerman JE: Mandibular keratocysts: Surgical management. Br J Oral Maxillofac Surg 23:204, 1985

44.Williams TP, Connor FA: Surgical management of the odontogenic keratocyst: Aggressive approach. J Oral Maxillofac Surg 52:964, 1994

45.Brøndum N, Jensen VJ: Recurrence of keratocysts and decompression treatment. A long-term follow up of forty four cases.

Oral Surg Oral Med Oral Pathol 72:265, 1991

46.Marker P, Brøndum N, Clausen PP, et al: Treatment of large odontogenic keratocysts by decompression and later cystectomy: A long-term follow-up and a histologic study of 23 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 82:122, 1996

47.Pogrel MA: Marsupialization as a definitive treatment for the odontogenic keratocyst. J Oral Maxillofac Surg 62:

651, 2004

48.Pogrel MA: Treatment of keratocysts: The case for decompression and marsupialization. J Oral Maxillofac Surg 63:1667, 2005 49.Tolstunov L: Marsupialization catheter. J Oral Maxillofac Surg 66:

1077, 2008

50.Swantek JJ, Reyes MI, Grannum RI, et al: A technique for long term decompression of large mandibular cysts. J Oral Maxillofac Surg 70:856, 2012

51.Gao L, Wang XL, Li SM, et al: Decompression as a treatment for odontogenic cystic lesions of the jaw. J Oral Maxillofac Surg 72:

327, 2014

52.Shafer WG, Hine MK, Levy BM: A Textbook of Oral Pathology.

Philadelphia, WB Saunders, 1974