55
Myospherulosis. Report of a Case M.L. Wallace* and B.W.
Nevillef
A case of myospherulosis, a condition first reported in 1969, is reported following the extraction of mandibular third molars and subsequent placement of Terra-Cortril and Gelfoam into the extraction sites. The lesions were discovered during a periodontal surgical procedure and to the best of our knowledge this is the first such report in the
periodontal
literature. J Perlodontol 1990;61:55-57.
Key Words: Myospherulosis; molar, third; tooth extraction/adverse effects.
Myospherulosis is a recently recognized granulomatous inflammatory condition which was first described in 1969 when McClatchie et al.1 reported seven Kenyan patients with soft tissue swellings of the arms, legs, gluteal, and scapular regions. Because the microscopic features of the lesions resembled an endosporulating fungus, they believed that they were dealing with a previously undescribed fungal
infection. Two years later, Hütt et al.2 reported five similar cases from Uganda, noting that the lesions occurred in areas of the body commonly used by local practitioners for the injection of foreign materials in reference to tribal customs. The condition was first reported in the United States in 1979 when Kyriakos3 described 16 patients with lesions of
the nose, paranasal sinuses, and middle ear. A common feature in all of these patients was a prior history of surgery in the affected area which subsequently was packed with a petrolatum-based antibiotic ointment. He theorized that myospherulosis was an iatrogenic lesion in which the as yet unidentified fungal organism was introduced via the unsterilized petrolatum ointment. In an effort to substantiate the relationship between petrolatum-based antibiotics and myospherulosis, De Schryver-Kecskemeti and Kyriakos4 conducted an animal study whereby 3% Achromycin ointment on gauze was introduced into rats using several methods. The results revealed a tissue reaction similar to that seen in human cases of myospherulosis, thus providing further evidence of a petrolatum-based antibiotic in the etiology of the condition. Histologically, myospherulosis is characterized by multiple cyst-like spaces which contain numerous brown to black staining spherules. Sometimes these spherules form aggregates surrounded by an outer membrane referred to as a parent body, forming structures which resemble a "bag 'Private
practice, Sumter,
of marbles." The cyst-like spaces are surrounded by fibrotic connective tissue containing a granulomatous inflammatory response characterized by macrophages and multinucleated giant cells. Further clarification of the true nature of myospherulosis was presented by Rosai5 in 1978, who reported a case from the maxillary sinus which occurred following surgery in the area that subsequently was packed with gauze impregnated with 3% tetracycline ointment. After consulting with pathologists who reviewed the case and noted that the spherules resembled erythrocytes, Rosai then performed special stains and discovered that the structures stained positive for hemoglobin. Rosai went on to perform a simple experiment in which he incubated packed erythrocytes in test tubes which had been coated with a 3% petrolatum-based tetracycline ointment. Smears from the preparation showed formation of hemoglobin positive bodies similar to those seen in tissue sections of the condition. He concluded that the spherules were erythrocytes altered by the petrolatum and the brownish-black color was due to decomposition of the
hemoglobin. Myospherulosis was first reported in the dental literature in 1980 when Dunlap and Barker6 described two cases in the jaws. More recently, Lynch et al.7 reported six cases from the oral cavity and summarized the literature on myospherulosis. Most of the oral cases have occurred secondary to oral surgical procedures where the surgical site was treated with tetracycline containing petrolatum-based ointment, either Terra-Cortril* or Achromycin.§ The majority of the cases
have occurred in the mandible, while one case occurred in the maxilla and another in the mandibular vestibular soft tissues. The lesions often present as a swelling which may be otherwise asymptomatic, while some cases have been associated with pain or purulent drainage. Several cases have presented as asymptomatic radiolucencies discovered
SC.
tDivision of Oral Pathology, College of Dental Medicine, Medical Uni-
versity of South Carolina, Charleston,
SC.
tPfizcr, Inc., New York, NY. 5Ledcrle Laboratories, Pearl River,
NY.
56
in
J Periodontol 1990
MYOSPHERULOSIS. REPORT OF A CASE
radiographie examination. Upon surgical exploration,
January a
black, greasy, tar-like substance is usually found.
We report here a patient with bilateral myospherulosis of the mandibular third molar regions which was discovered during periodontal surgery. To the best of our knowledge, this is the first report of myospherulosis in the periodontal
literature.
CASE REPORT In February 1985 a 16 year old black female underwent removal of four impacted third molars by the Department of Oral and Maxillofacial Surgery at the Medical University of South Carolina. Surgery was performed while the patient was under intravenous sedation and local anesthesia. Subsequent to removal of the third molars, Gelfoam|| and TerraCortril were placed in the extraction sites. The patient presented 10 days later with a chief complaint of pain confined to the mandibular third molar areas. Examination revealed food debris present in both mandibular third molar extraction sites. The areas were irrigated with saline rinses and the patient was given an irrigating syringe and instructed in its use and told to return to the clinic if the pain continued. The patient did not report to the clinic again until June 1987 with a chief complaint of pain of the mandibular left posterior quadrant of 3 days duration. Examination revealed marked gingival inflammation with purulent drainage. A panorex taken at this time revealed radiolucent areas in the mandibular third molar areas. The radiolucent areas were not pronounced and the pain was thought to be due to a periodontal abscess. The left mandibular third molar area was irrigated and the patient was also instructed to rinse with warm saline rinses. At a follow-up appointment 6 days later, the patient returned complaining that the pain had not subsided. Probing revealed depths of 6 mm to 7 mm distal to the left mandibular second molar with a purulent exúdate. A periodontal consult was obtained at this time and upon examination it was determined that periodontal flap surgery would be required for root evaluation and removal of deposits with reduction of depths to be accomplished by a distal wedge procedure. Similar clinical findings were apparent distal to the mandibular right second molar and although the patient did not complain of pain in that area at this time, she did relate a history of pain in the area subsequent to the third molar extractions which she failed to report. In light of this, it was believed to be in the patient's best interest to treat this area in an identical manner as the mandibular left area. The patient presented to the graduate periodontics clinic in July 1987 to undergo the proposed surgical procedures. Using local anesthesia and nitrous-oxide oxygen sedation, periodontal flaps were raised distal to the mandibular left and right second molars. Upon removal of soft tissue wedges, bony craters were observed in both areas. These craters contained a black, stringy material resembling jelly (Fig.
HThe Upjohn Company, Kalamazoo, MI.
Figure 1. Clinical appearance of the bony crater which contained a black, stringy material.
Figure 2. Low power photomicrograph showing cyst like spaces filled with spherical structures. Original magnification X 80.
1). This material was curetted from the defects and submitted in separate vials for biopsy. The following conditions were considered in the differential diagnosis of the
lesion: a saprophytic fungal infection, foreign body giant cell reaction, blood filled cyst, and myospherulosis. Microscopic examination of both specimens revealed curetted fragments of fibrous connective tissue exhibiting multiple cyst like spaces which contained numerous brownstaining spherules (Figs. 2 and 3). A chronic granulomatous infiltrate was present, with flattened histocytes and occasional multinucleated giant cells noted lining the spaces. A diagnosis of myospherulosis was made. Healing of the surgical sites occurred uneventfully. DISCUSSION The use of Terra-Cortril/Gelfoam in the third molar extraction sites has been shown to significantly reduce the incidence of dry sockets;8,9 however, the use of copious lavage following extraction has also shown significant reductions in incidence.10 Although the authors are not advising against
Volume 61 Number 1
WALLACE,
S-*
,
be noted in the
·
f A.
·
:
c
*¿°„ .^.4:
e'"v^ ·.
,0.
4.
1
»·-.#.
-
Figure 3. High power photomicrograph demonstrating appearance of the spherules. Original magnification x 200.
5. 6. 7.
the prudent use of Terra-Cortril on Gelfoam due to the demonstrated benefits of its use, it is important to have thorough follow-up visits to insure complete healing of the bony crypt. A socket that does not show filling with new bone should be suspected, particularly if accompanied by episodes of pain, swelling, or drainage.3 Since periodontal flaps and distal wedge procedures are often performed by periodontists following third molar extractions, it would be wise to carefully investigate remaining bony crypts for material suggestive of myospherulosis. If cases of myospherulosis are not surgically debrided, they can result in recurrent
area.
REFERENCES 1. McClatchie S, Warambo MW, Bremner AD. Myospherulosis: A previously unreported disease? Am J Clin Pathol 1969; 51:699. 2. Hütt MSR, Fernandes BJJ, Templeton AC. Myospherulosis. Subcutaneous spherulocystic disease. Trans R Soc Trop Med Hyg 1971; 3.
4«,.·
57
episodes of pain and absence of bone fill in a previous surgical site. In addition, persistent abscess formation may
per*
r
NEVILLE
8.
9. 10.
65:182-188. Kyriakos M. Myospherulosis of the paranasal sinuses, nose and middle ear. A possible iatrogenic disease. Am J Clin Pathol 1977; 67:118. De Schryver-Kecskemeti C, Kyriakos M. The induction of human myospherulosis in experimental animals. Am J Pathol 1977; 87:33. Rosai J. The nature of myospherulosis of the upper respiratory tract. Am J Clin Pathol 1978; 69:475. Dunlap CL, Barker BF. Myospherulosis of the jaws. Oral Surg Oral Med Oral Pathol 1980; 50:238. Lynch DP, Newland JR, McClendon JL. Myospherulosis of the oral hard and soft tissues. / Oral Maxillofac Surg 1984; 42:349. Rutledge JL, Marcoot RM. Terra-Cortril/Gelfoam for reduction of the incidence of localized osteitis following mandibular third molar removal. / Oral Med 39:51, 1984; 39:51. Julius LL. Prevention of dry socket with local application of terracortril in gelfoam. / Oral Surg 1982; 40:285. Butler DP, Sweet JB. Effect of lavage on the incidence of localized osteitis in mandibular third molar extraction sites. Oral Surg Oral Med Oral Pathol 1977; 44:14.
Send reprint requests to: Dr. Marshall L.
Sumter, SC 29150. Accepted for publication July 17, 1989.
Wallace, 3 Professional Court,
Myospherulosis. Report of a Case M.L. Wallace* and B.W.
Nevillef
A case of myospherulosis, a condition first reported in 1969, is reported following the extraction of mandibular third molars and subsequent placement of Terra-Cortril and Gelfoam into the extraction sites. The lesions were discovered during a periodontal surgical procedure and to the best of our knowledge this is the first such report in the
periodontal
literature. J Perlodontol 1990;61:55-57.
Key Words: Myospherulosis; molar, third; tooth extraction/adverse effects.
Myospherulosis is a recently recognized granulomatous inflammatory condition which was first described in 1969 when McClatchie et al.1 reported seven Kenyan patients with soft tissue swellings of the arms, legs, gluteal, and scapular regions. Because the microscopic features of the lesions resembled an endosporulating fungus, they believed that they were dealing with a previously undescribed fungal
infection. Two years later, Hütt et al.2 reported five similar cases from Uganda, noting that the lesions occurred in areas of the body commonly used by local practitioners for the injection of foreign materials in reference to tribal customs. The condition was first reported in the United States in 1979 when Kyriakos3 described 16 patients with lesions of
the nose, paranasal sinuses, and middle ear. A common feature in all of these patients was a prior history of surgery in the affected area which subsequently was packed with a petrolatum-based antibiotic ointment. He theorized that myospherulosis was an iatrogenic lesion in which the as yet unidentified fungal organism was introduced via the unsterilized petrolatum ointment. In an effort to substantiate the relationship between petrolatum-based antibiotics and myospherulosis, De Schryver-Kecskemeti and Kyriakos4 conducted an animal study whereby 3% Achromycin ointment on gauze was introduced into rats using several methods. The results revealed a tissue reaction similar to that seen in human cases of myospherulosis, thus providing further evidence of a petrolatum-based antibiotic in the etiology of the condition. Histologically, myospherulosis is characterized by multiple cyst-like spaces which contain numerous brown to black staining spherules. Sometimes these spherules form aggregates surrounded by an outer membrane referred to as a parent body, forming structures which resemble a "bag 'Private
practice, Sumter,
of marbles." The cyst-like spaces are surrounded by fibrotic connective tissue containing a granulomatous inflammatory response characterized by macrophages and multinucleated giant cells. Further clarification of the true nature of myospherulosis was presented by Rosai5 in 1978, who reported a case from the maxillary sinus which occurred following surgery in the area that subsequently was packed with gauze impregnated with 3% tetracycline ointment. After consulting with pathologists who reviewed the case and noted that the spherules resembled erythrocytes, Rosai then performed special stains and discovered that the structures stained positive for hemoglobin. Rosai went on to perform a simple experiment in which he incubated packed erythrocytes in test tubes which had been coated with a 3% petrolatum-based tetracycline ointment. Smears from the preparation showed formation of hemoglobin positive bodies similar to those seen in tissue sections of the condition. He concluded that the spherules were erythrocytes altered by the petrolatum and the brownish-black color was due to decomposition of the
hemoglobin. Myospherulosis was first reported in the dental literature in 1980 when Dunlap and Barker6 described two cases in the jaws. More recently, Lynch et al.7 reported six cases from the oral cavity and summarized the literature on myospherulosis. Most of the oral cases have occurred secondary to oral surgical procedures where the surgical site was treated with tetracycline containing petrolatum-based ointment, either Terra-Cortril* or Achromycin.§ The majority of the cases
have occurred in the mandible, while one case occurred in the maxilla and another in the mandibular vestibular soft tissues. The lesions often present as a swelling which may be otherwise asymptomatic, while some cases have been associated with pain or purulent drainage. Several cases have presented as asymptomatic radiolucencies discovered
SC.
tDivision of Oral Pathology, College of Dental Medicine, Medical Uni-
versity of South Carolina, Charleston,
SC.
tPfizcr, Inc., New York, NY. 5Ledcrle Laboratories, Pearl River,
NY.
56
in
J Periodontol 1990
MYOSPHERULOSIS. REPORT OF A CASE
radiographie examination. Upon surgical exploration,
January a
black, greasy, tar-like substance is usually found.
We report here a patient with bilateral myospherulosis of the mandibular third molar regions which was discovered during periodontal surgery. To the best of our knowledge, this is the first report of myospherulosis in the periodontal
literature.
CASE REPORT In February 1985 a 16 year old black female underwent removal of four impacted third molars by the Department of Oral and Maxillofacial Surgery at the Medical University of South Carolina. Surgery was performed while the patient was under intravenous sedation and local anesthesia. Subsequent to removal of the third molars, Gelfoam|| and TerraCortril were placed in the extraction sites. The patient presented 10 days later with a chief complaint of pain confined to the mandibular third molar areas. Examination revealed food debris present in both mandibular third molar extraction sites. The areas were irrigated with saline rinses and the patient was given an irrigating syringe and instructed in its use and told to return to the clinic if the pain continued. The patient did not report to the clinic again until June 1987 with a chief complaint of pain of the mandibular left posterior quadrant of 3 days duration. Examination revealed marked gingival inflammation with purulent drainage. A panorex taken at this time revealed radiolucent areas in the mandibular third molar areas. The radiolucent areas were not pronounced and the pain was thought to be due to a periodontal abscess. The left mandibular third molar area was irrigated and the patient was also instructed to rinse with warm saline rinses. At a follow-up appointment 6 days later, the patient returned complaining that the pain had not subsided. Probing revealed depths of 6 mm to 7 mm distal to the left mandibular second molar with a purulent exúdate. A periodontal consult was obtained at this time and upon examination it was determined that periodontal flap surgery would be required for root evaluation and removal of deposits with reduction of depths to be accomplished by a distal wedge procedure. Similar clinical findings were apparent distal to the mandibular right second molar and although the patient did not complain of pain in that area at this time, she did relate a history of pain in the area subsequent to the third molar extractions which she failed to report. In light of this, it was believed to be in the patient's best interest to treat this area in an identical manner as the mandibular left area. The patient presented to the graduate periodontics clinic in July 1987 to undergo the proposed surgical procedures. Using local anesthesia and nitrous-oxide oxygen sedation, periodontal flaps were raised distal to the mandibular left and right second molars. Upon removal of soft tissue wedges, bony craters were observed in both areas. These craters contained a black, stringy material resembling jelly (Fig.
HThe Upjohn Company, Kalamazoo, MI.
Figure 1. Clinical appearance of the bony crater which contained a black, stringy material.
Figure 2. Low power photomicrograph showing cyst like spaces filled with spherical structures. Original magnification X 80.
1). This material was curetted from the defects and submitted in separate vials for biopsy. The following conditions were considered in the differential diagnosis of the
lesion: a saprophytic fungal infection, foreign body giant cell reaction, blood filled cyst, and myospherulosis. Microscopic examination of both specimens revealed curetted fragments of fibrous connective tissue exhibiting multiple cyst like spaces which contained numerous brownstaining spherules (Figs. 2 and 3). A chronic granulomatous infiltrate was present, with flattened histocytes and occasional multinucleated giant cells noted lining the spaces. A diagnosis of myospherulosis was made. Healing of the surgical sites occurred uneventfully. DISCUSSION The use of Terra-Cortril/Gelfoam in the third molar extraction sites has been shown to significantly reduce the incidence of dry sockets;8,9 however, the use of copious lavage following extraction has also shown significant reductions in incidence.10 Although the authors are not advising against
Volume 61 Number 1
WALLACE,
S-*
,
be noted in the
·
f A.
·
:
c
*¿°„ .^.4:
e'"v^ ·.
,0.
4.
1
»·-.#.
-
Figure 3. High power photomicrograph demonstrating appearance of the spherules. Original magnification x 200.
5. 6. 7.
the prudent use of Terra-Cortril on Gelfoam due to the demonstrated benefits of its use, it is important to have thorough follow-up visits to insure complete healing of the bony crypt. A socket that does not show filling with new bone should be suspected, particularly if accompanied by episodes of pain, swelling, or drainage.3 Since periodontal flaps and distal wedge procedures are often performed by periodontists following third molar extractions, it would be wise to carefully investigate remaining bony crypts for material suggestive of myospherulosis. If cases of myospherulosis are not surgically debrided, they can result in recurrent
area.
REFERENCES 1. McClatchie S, Warambo MW, Bremner AD. Myospherulosis: A previously unreported disease? Am J Clin Pathol 1969; 51:699. 2. Hütt MSR, Fernandes BJJ, Templeton AC. Myospherulosis. Subcutaneous spherulocystic disease. Trans R Soc Trop Med Hyg 1971; 3.
4«,.·
57
episodes of pain and absence of bone fill in a previous surgical site. In addition, persistent abscess formation may
per*
r
NEVILLE
8.
9. 10.
65:182-188. Kyriakos M. Myospherulosis of the paranasal sinuses, nose and middle ear. A possible iatrogenic disease. Am J Clin Pathol 1977; 67:118. De Schryver-Kecskemeti C, Kyriakos M. The induction of human myospherulosis in experimental animals. Am J Pathol 1977; 87:33. Rosai J. The nature of myospherulosis of the upper respiratory tract. Am J Clin Pathol 1978; 69:475. Dunlap CL, Barker BF. Myospherulosis of the jaws. Oral Surg Oral Med Oral Pathol 1980; 50:238. Lynch DP, Newland JR, McClendon JL. Myospherulosis of the oral hard and soft tissues. / Oral Maxillofac Surg 1984; 42:349. Rutledge JL, Marcoot RM. Terra-Cortril/Gelfoam for reduction of the incidence of localized osteitis following mandibular third molar removal. / Oral Med 39:51, 1984; 39:51. Julius LL. Prevention of dry socket with local application of terracortril in gelfoam. / Oral Surg 1982; 40:285. Butler DP, Sweet JB. Effect of lavage on the incidence of localized osteitis in mandibular third molar extraction sites. Oral Surg Oral Med Oral Pathol 1977; 44:14.
Send reprint requests to: Dr. Marshall L.
Sumter, SC 29150. Accepted for publication July 17, 1989.
Wallace, 3 Professional Court,
