Cryosurgery of Supraglottic Cavernous Hemangioma O'Neill, MD; G. Gordon Snyder III, MD; James M. Toomey, MD, DMD
John V.
\s=b\ Cryosurgery is more
importance
as a
gaining more and therapeutic modal-
ity within the field of head and neck surgery. It has been used effectively in the treatment of cavernous hemangiomas of the skin and oral cavity. Supraglottic cavernous
hemangiomas present a challenge
in management when surgical intervention is indicated. The results of cryosurgery in the treatment of such a lesion are
reported. (Arch Otolaryngol 102:55-57, 1976)
Since
its introduction to clinical 1961, cryosurgery has
usage in
been
employed therapy in
as
primary
or
adju-
wide variety of situations. It has found application in a number of the surgical specialties, and recent advances in basic cryobiology have increased its usefulness considerably. It remains the responsibility of the practicing otorhinolaryngologist with an understanding of the underlying principles to verify appropriate, as well as inappropriate, applications of cryosurgery in head and neck surgery. This paper reviews the treatment of laryngeal hemangiomas and convant
a
Accepted for publication Aug 20, 1975. From the Division of Otorhinolaryngology and Facial Plastic Surgery, University of Connecticut School of Medicine, Farmington, Conn. Reprint requests to Division of Otorhinolaryngology and Facial Plastic Surgery, University of Connecticut Health Center, Farmington, CT 06032 (Dr O'Neill).
firms the role of cryosurgery in the treatment of this disease.
REPORT OF A CASE Clinical Findings A 28-year-old man was first seen for medical evaluation in December 1968, when he experienced the sudden onset of profuse bleeding from the mouth. This episode was brief and self-limited. The patient stated that he felt a "tickle" in his throat and coughed up a copious quantity of blood. There was no associated pain or respiratory distress. He was seen shortly afterward by an otolaryngologist who, on examination with indirect laryngoscopy, found a mass in the left supraglottic area that had the clinical appearance of a cavernous hemangioma. There was no evidence of bleeding at the time of examination. The patient was scheduled for direct laryngoscopy, and this was accomplished without incident with the patient under local anesthesia. At that time a hemangioma was found to involve part of the left aryepiglottic fold. It extended onto the laryngeal surface of the epiglottis, the left false cord, and the medial wall of the left pyriform sinus. It was thought that surgical intervention was not indicated at that time. The patient was instructed to inform future physicians of this lesion before undergoing any general anesthesia. In 1971 the patient did indeed undergo an operation without incident while under general endotracheal anesthesia. The patient remained asymptomatic until October 1974, when he again experienced the sudden onset of hemoptysis that
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brief, of moderate quantity, and selflimited. He was seen at that time by the original otolaryngologist, and results of the indirect laryngoscopic examination were essentially unchanged from those of December 1968. At this point, the patient was referred to our service for evaluation. We believed that cryosurgery would be of benefit in the treatment of this lesion, and the patient entered the hospital in December 1974. Review of the patient's past medical history failed to elicit any episodes suggestive of airway obstruction as a child or an adult other than the two previously mentioned, and the family history was completely negative. A review of systems also gave negative findings. Physical examination disclosed small hemangiomas of the nose and lip. Results of indirect laryngoscopy were unchanged from previous examinations, and the remainder of the general physical was completely within normal limits. was
Laboratory and X-ray Findings Laboratory investigations including
complete blood cell count, prothrombin time, partial thromboplastin time, and blood chemistry studies were all within normal limits. The chest roentgenogram was unremarkable. A laryngogram obtained prior to surgery showed a mass involving the supraglottic area on the left (Fig 1). Mobility of the vocal cords was normal. There was no subglottic involvement.
Hospital Course Following completion of the
preopera-
Fig 1.—Preoperative laryngogram reveals left-sided supraglottic mass involving aryepi¬ glottic fold and medial wall of pyriform sinus. Left, Modified Valsalva maneuver; Right, Quiet breathing. Fig 2.—Laryngogram obtained five months
geal
contours.
postoperatively
Left, Phonation; Right, Reverse Valsalva
demonstrates normal
laryn¬
maneuver.
tive evaluation, a tracheostomy was per¬ formed with the patient under local an¬ esthesia. General anesthesia was then attained, and the lesion was visualized via direct laryngoscopy. A closed, cylindrical cryosurgery probe was positioned first on one side of the aryepiglottic fold and then the other. Each freeze was continued until the iceball involved the entire lesion. Postoperatively the patient did well. He was able to swallow easily by the fourth day, and a slough of the lesion took place over the next two weeks. Indirect laryn¬ goscopy prior to removal of the tracheoto¬ my tube showed mild supraglottic edema. The patient was subsequently decannulated without any difficulty. Follow-up examinations with indirect laryngoscopy have shown normal healing of the mucosa, resolution of the edema, and normal cord function. The patient was last seen in May 1975, and results of indi¬ rect laryngoscopy at that time were nor¬ mal. A laryngogram obtained the same day was interpreted as being within nor¬ mal limits (Fig 2). The patient is presently asymptomatic, continues to lead an active outdoor life, and is pleased with the results of surgery.
COMMENT The first
report of
a case
of he¬
mangioma of the larynx was made by Mackenzie in 1868.1 By the time of his report in 1944, Ferguson2 had found a
total of 123 cases in the literature. He credited Sweetser3 in 1921 as being the first to separate the tumors into two classes, the rarer ones occurring in infants, and the more common type seen in adults. Sweetser noted that the hemangioma was generally located below the level of the vocal cords in infants, while it occurred on or above the vocal cords in adults. Ferguson2 and Kleinsasser4 have pointed out the difference between symptoms characterizing the infan¬ tile and adult forms. Because of the subglottic position of the infantile form, a slight infection of the upper respiratory system is often sufficient to cause symptoms of respiratory dis¬ tress. On the other hand, the adult may have symptoms that are often vague and of extremely long dura¬ tion. Indeed, years may elapse before the patient seeks medical attention.
Hoarseness, dyspnea, hemoptysis,
dysphagia were the mon adult complaints. and
Downloaded From: http://archotol.jamanetwork.com/ by a University of Iowa User on 06/14/2015
most
com¬
Calcaterra5 again acknowledges the contributions of Mackenzie1 and Sweetser.' His paper reports the first known histologie evaluation of irra¬ diated subglottic hemangioma. The case report deals with an infant who had a hemangioma in the subglottic area obstructing half the lumen. This was irradiated with 950 rads through opposing lateral ports over a period of eight days. Two months later, the child was admitted to the hospital with biventricular failure. Endoscopy at that time revealed slight fullness in the subglottic area without notable compromise of the lumen. A few days later the child suffered a cardiopulmonary arrest. Autopsy showed endocardial fibroelastosis, which was presumed to be the cause of death. Histologie examination of the larynx and trachea demonstrated a discrete
hemangioma prominent
1
cm
in diameter with channels and no
cavernous
evidence of irradiation changes. Calcaterra5 believes that the natu¬ ral history of regression of infantile hemangiomas accounts for the suc¬ cess rate previously attributed to ir¬ radiation. In his summary, he states that cryotherapy remains an inter¬ esting but unreported method of treatment for those cases that cannot be managed by tracheostomy alone.
Miller,6·7 Holden,8·9 DeSanto,10 Chandler,11 and others12·13 have re¬ ported the role of cryosurgery in treating both benign and malignant tumors of the head and neck; these authors also reviewed the underlying principles of cryosurgery. Huang,14 Dogo," Henderson,1" Goldwyn,17 and others have reported success in the treatment of cavernous hemangiomas
of the skin and oral surgery. To our stances of
cavity with cryo¬
knowledge, specific in¬ laryngeal hemangiomas
treated with cryosurgery have not been verified. Zacarian18 does men¬ tion in his text one patient with a massive hemangioma of the oral cav¬ ity that had extension onto an aryte¬ noid, but the massiveness of the le¬ sion clouds the potentially successful results that can be expected in a more limited lesion. In the case presented here, cavern¬ ous hemangioma of the larynx was successfully treated by cryosurgery. We believe that these lesions, when of small to moderate size and involving accessible portions of the larynx, can be effectively treated by endoscopie cryosurgery.
References 1. Mackenzie M:
Essay on Growth of the LarPhiladelphia, Linsay & Blakiston Co, 1871. 2. Ferguson GB: Hemangioma of the adult and of the infant larynx: A review of the literature and a report of two cases. Arch Otolaryngol 40:189-195, 1944. 3. Sweetser TH: Hemangioma of the larynx. Laryngoscope 31:797, 1921. 4. Kleinsasser 0: Microlaryngoscopy and Endolaryngeal Microsurgery. Philadelphia, WB Saunders Co, 1968, p 113. 5. Calcaterra TC: An evaluation of the treatment of subglottic hemangioma. Laryngoscope 78:1956-1964, 1968. 6. Miller D: Three years experience with cryosurgery in head and neck tumors. Ann Otol Rhiynx.
nol Laryngol 78:786-791, 1969. 7. Miller D: Cryosurgery for the treatment of neoplasms of the oral cavity. Otolaryngol Clin North Am 5:377-388, 1972. 8. Holden HB: Cryosurgery in ENT practice. J Laryngol Otol 86:821-827, 1972. 9. Holden HB: Cryosurgery in head and neck neoplasia. Br J Surg 59:709-712, 1972. 10. DeSanto LW: Application of cryosurgery to otolaryngology. Minn Med 53:29-32, 1972. 11. Chandler JR: Cryosurgery in the management of tumors of the head and neck. South Med J 64:1440-1445, 1971. 12. Smith MF: Cryosurgical techniques in removal of angiofibromas. Laryngoscope 74:1072\x=req-\ 1080, 1964.
Downloaded From: http://archotol.jamanetwork.com/ by a University of Iowa User on 06/14/2015
13. Barton RS: Cryosurgery of nasopharyngeal neoplasms. Am Surg 32:744-747, 1966. 14. Huang T: Use of cryosurgery in management in intraoral hemangioma. South Med J
65:1123-1127, 1972. 15. Dogo G: Cryosurgery and cryotherapy in plastic surgery. J Cryosurg 2:56, 1969.
16. Henderson RL: Cryosurgical treatment of hemangiomas. Arch Otolaryng 93:511-513, 1971. 17. Goldwyn RM: Cryosurgery forlarge hemangiomas in adults. Plast Reconstr Surg 43:605-611,
1969. 18. Zacarian SA: Cryosurgery of Tumors of the Skin and Oral Cavity. Springfield, Ill, Charles C Thomas Publishers, 1973.
John V.
\s=b\ Cryosurgery is more
importance
as a
gaining more and therapeutic modal-
ity within the field of head and neck surgery. It has been used effectively in the treatment of cavernous hemangiomas of the skin and oral cavity. Supraglottic cavernous
hemangiomas present a challenge
in management when surgical intervention is indicated. The results of cryosurgery in the treatment of such a lesion are
reported. (Arch Otolaryngol 102:55-57, 1976)
Since
its introduction to clinical 1961, cryosurgery has
usage in
been
employed therapy in
as
primary
or
adju-
wide variety of situations. It has found application in a number of the surgical specialties, and recent advances in basic cryobiology have increased its usefulness considerably. It remains the responsibility of the practicing otorhinolaryngologist with an understanding of the underlying principles to verify appropriate, as well as inappropriate, applications of cryosurgery in head and neck surgery. This paper reviews the treatment of laryngeal hemangiomas and convant
a
Accepted for publication Aug 20, 1975. From the Division of Otorhinolaryngology and Facial Plastic Surgery, University of Connecticut School of Medicine, Farmington, Conn. Reprint requests to Division of Otorhinolaryngology and Facial Plastic Surgery, University of Connecticut Health Center, Farmington, CT 06032 (Dr O'Neill).
firms the role of cryosurgery in the treatment of this disease.
REPORT OF A CASE Clinical Findings A 28-year-old man was first seen for medical evaluation in December 1968, when he experienced the sudden onset of profuse bleeding from the mouth. This episode was brief and self-limited. The patient stated that he felt a "tickle" in his throat and coughed up a copious quantity of blood. There was no associated pain or respiratory distress. He was seen shortly afterward by an otolaryngologist who, on examination with indirect laryngoscopy, found a mass in the left supraglottic area that had the clinical appearance of a cavernous hemangioma. There was no evidence of bleeding at the time of examination. The patient was scheduled for direct laryngoscopy, and this was accomplished without incident with the patient under local anesthesia. At that time a hemangioma was found to involve part of the left aryepiglottic fold. It extended onto the laryngeal surface of the epiglottis, the left false cord, and the medial wall of the left pyriform sinus. It was thought that surgical intervention was not indicated at that time. The patient was instructed to inform future physicians of this lesion before undergoing any general anesthesia. In 1971 the patient did indeed undergo an operation without incident while under general endotracheal anesthesia. The patient remained asymptomatic until October 1974, when he again experienced the sudden onset of hemoptysis that
Downloaded From: http://archotol.jamanetwork.com/ by a University of Iowa User on 06/14/2015
brief, of moderate quantity, and selflimited. He was seen at that time by the original otolaryngologist, and results of the indirect laryngoscopic examination were essentially unchanged from those of December 1968. At this point, the patient was referred to our service for evaluation. We believed that cryosurgery would be of benefit in the treatment of this lesion, and the patient entered the hospital in December 1974. Review of the patient's past medical history failed to elicit any episodes suggestive of airway obstruction as a child or an adult other than the two previously mentioned, and the family history was completely negative. A review of systems also gave negative findings. Physical examination disclosed small hemangiomas of the nose and lip. Results of indirect laryngoscopy were unchanged from previous examinations, and the remainder of the general physical was completely within normal limits. was
Laboratory and X-ray Findings Laboratory investigations including
complete blood cell count, prothrombin time, partial thromboplastin time, and blood chemistry studies were all within normal limits. The chest roentgenogram was unremarkable. A laryngogram obtained prior to surgery showed a mass involving the supraglottic area on the left (Fig 1). Mobility of the vocal cords was normal. There was no subglottic involvement.
Hospital Course Following completion of the
preopera-
Fig 1.—Preoperative laryngogram reveals left-sided supraglottic mass involving aryepi¬ glottic fold and medial wall of pyriform sinus. Left, Modified Valsalva maneuver; Right, Quiet breathing. Fig 2.—Laryngogram obtained five months
geal
contours.
postoperatively
Left, Phonation; Right, Reverse Valsalva
demonstrates normal
laryn¬
maneuver.
tive evaluation, a tracheostomy was per¬ formed with the patient under local an¬ esthesia. General anesthesia was then attained, and the lesion was visualized via direct laryngoscopy. A closed, cylindrical cryosurgery probe was positioned first on one side of the aryepiglottic fold and then the other. Each freeze was continued until the iceball involved the entire lesion. Postoperatively the patient did well. He was able to swallow easily by the fourth day, and a slough of the lesion took place over the next two weeks. Indirect laryn¬ goscopy prior to removal of the tracheoto¬ my tube showed mild supraglottic edema. The patient was subsequently decannulated without any difficulty. Follow-up examinations with indirect laryngoscopy have shown normal healing of the mucosa, resolution of the edema, and normal cord function. The patient was last seen in May 1975, and results of indi¬ rect laryngoscopy at that time were nor¬ mal. A laryngogram obtained the same day was interpreted as being within nor¬ mal limits (Fig 2). The patient is presently asymptomatic, continues to lead an active outdoor life, and is pleased with the results of surgery.
COMMENT The first
report of
a case
of he¬
mangioma of the larynx was made by Mackenzie in 1868.1 By the time of his report in 1944, Ferguson2 had found a
total of 123 cases in the literature. He credited Sweetser3 in 1921 as being the first to separate the tumors into two classes, the rarer ones occurring in infants, and the more common type seen in adults. Sweetser noted that the hemangioma was generally located below the level of the vocal cords in infants, while it occurred on or above the vocal cords in adults. Ferguson2 and Kleinsasser4 have pointed out the difference between symptoms characterizing the infan¬ tile and adult forms. Because of the subglottic position of the infantile form, a slight infection of the upper respiratory system is often sufficient to cause symptoms of respiratory dis¬ tress. On the other hand, the adult may have symptoms that are often vague and of extremely long dura¬ tion. Indeed, years may elapse before the patient seeks medical attention.
Hoarseness, dyspnea, hemoptysis,
dysphagia were the mon adult complaints. and
Downloaded From: http://archotol.jamanetwork.com/ by a University of Iowa User on 06/14/2015
most
com¬
Calcaterra5 again acknowledges the contributions of Mackenzie1 and Sweetser.' His paper reports the first known histologie evaluation of irra¬ diated subglottic hemangioma. The case report deals with an infant who had a hemangioma in the subglottic area obstructing half the lumen. This was irradiated with 950 rads through opposing lateral ports over a period of eight days. Two months later, the child was admitted to the hospital with biventricular failure. Endoscopy at that time revealed slight fullness in the subglottic area without notable compromise of the lumen. A few days later the child suffered a cardiopulmonary arrest. Autopsy showed endocardial fibroelastosis, which was presumed to be the cause of death. Histologie examination of the larynx and trachea demonstrated a discrete
hemangioma prominent
1
cm
in diameter with channels and no
cavernous
evidence of irradiation changes. Calcaterra5 believes that the natu¬ ral history of regression of infantile hemangiomas accounts for the suc¬ cess rate previously attributed to ir¬ radiation. In his summary, he states that cryotherapy remains an inter¬ esting but unreported method of treatment for those cases that cannot be managed by tracheostomy alone.
Miller,6·7 Holden,8·9 DeSanto,10 Chandler,11 and others12·13 have re¬ ported the role of cryosurgery in treating both benign and malignant tumors of the head and neck; these authors also reviewed the underlying principles of cryosurgery. Huang,14 Dogo," Henderson,1" Goldwyn,17 and others have reported success in the treatment of cavernous hemangiomas
of the skin and oral surgery. To our stances of
cavity with cryo¬
knowledge, specific in¬ laryngeal hemangiomas
treated with cryosurgery have not been verified. Zacarian18 does men¬ tion in his text one patient with a massive hemangioma of the oral cav¬ ity that had extension onto an aryte¬ noid, but the massiveness of the le¬ sion clouds the potentially successful results that can be expected in a more limited lesion. In the case presented here, cavern¬ ous hemangioma of the larynx was successfully treated by cryosurgery. We believe that these lesions, when of small to moderate size and involving accessible portions of the larynx, can be effectively treated by endoscopie cryosurgery.
References 1. Mackenzie M:
Essay on Growth of the LarPhiladelphia, Linsay & Blakiston Co, 1871. 2. Ferguson GB: Hemangioma of the adult and of the infant larynx: A review of the literature and a report of two cases. Arch Otolaryngol 40:189-195, 1944. 3. Sweetser TH: Hemangioma of the larynx. Laryngoscope 31:797, 1921. 4. Kleinsasser 0: Microlaryngoscopy and Endolaryngeal Microsurgery. Philadelphia, WB Saunders Co, 1968, p 113. 5. Calcaterra TC: An evaluation of the treatment of subglottic hemangioma. Laryngoscope 78:1956-1964, 1968. 6. Miller D: Three years experience with cryosurgery in head and neck tumors. Ann Otol Rhiynx.
nol Laryngol 78:786-791, 1969. 7. Miller D: Cryosurgery for the treatment of neoplasms of the oral cavity. Otolaryngol Clin North Am 5:377-388, 1972. 8. Holden HB: Cryosurgery in ENT practice. J Laryngol Otol 86:821-827, 1972. 9. Holden HB: Cryosurgery in head and neck neoplasia. Br J Surg 59:709-712, 1972. 10. DeSanto LW: Application of cryosurgery to otolaryngology. Minn Med 53:29-32, 1972. 11. Chandler JR: Cryosurgery in the management of tumors of the head and neck. South Med J 64:1440-1445, 1971. 12. Smith MF: Cryosurgical techniques in removal of angiofibromas. Laryngoscope 74:1072\x=req-\ 1080, 1964.
Downloaded From: http://archotol.jamanetwork.com/ by a University of Iowa User on 06/14/2015
13. Barton RS: Cryosurgery of nasopharyngeal neoplasms. Am Surg 32:744-747, 1966. 14. Huang T: Use of cryosurgery in management in intraoral hemangioma. South Med J
65:1123-1127, 1972. 15. Dogo G: Cryosurgery and cryotherapy in plastic surgery. J Cryosurg 2:56, 1969.
16. Henderson RL: Cryosurgical treatment of hemangiomas. Arch Otolaryng 93:511-513, 1971. 17. Goldwyn RM: Cryosurgery forlarge hemangiomas in adults. Plast Reconstr Surg 43:605-611,
1969. 18. Zacarian SA: Cryosurgery of Tumors of the Skin and Oral Cavity. Springfield, Ill, Charles C Thomas Publishers, 1973.
