Subglottic hemangiomas in infants: treatment with intralesional corticosteroid injection and intubation

Subglottic hemangiomas in infants: treatment with intralesional corticosteroid injection and intubation

International Journal of Pediatric Otorhinolatyngologv, 19 (1990) 145-150 145 Elsevier PEDOT 00644 Subglottic hemangiomas in infants: treatment w...

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International Journal of Pediatric Otorhinolatyngologv, 19 (1990) 145-150

145

Elsevier

PEDOT

00644

Subglottic hemangiomas in infants: treatment with intralesional corticosteroid injection and intubation J. Meeuwis

‘, C.E. Bos ‘, L.J. Hoeve

’ and E. van der Voort

*

’ Department of ENT and 2 Pediatric Intensive Care Unit, Sophia Children’s Hospital, Rotterdam (The Netherlands) (Received 8 August 1989) (Revised version received 24 January (Accepted 5 February 1990)

Key words: Subglottic

hemangioma;

Corticosteroid

1990)

therapy

Abstract Six children with a subglottic hemangioma were treated in the Sophia Children’s Hospital in the period 1982-1987 by means of intralesional corticosteroid injection, followed by intubation. After treatment all children were symptomfree. In 3 patients this result was attained after 1 injection, in 2 after 3 injections. One patient needed 5 injections. The average duration of intubation was 19 days (7-36). Three months (i-7;) after the onset of therapy all patients were free of symptoms. No serious complications were observed. No patient needed a tracheotomy. The average follow-up period was 3.3 years (14-5:). The authors feel that not only the effect of intralesional corticosteroids, but also local gentle pressure by the tube is of therapeutic importance. The above-mentioned treatment of subglottic hemangioma in children is now the treatment of choice in our clinic.

Introduction Subglottic hemangiomas in children are rare lesions associated with a significant degree of morbidity and mortality. The patients usually present with recurrent episodes of progressive laryngotracheitis. The symptoms appear after a few weeks or months after birth and vary with the size of the tumor. It is found that there is a large female preponderance. The diagnosis is based on the clinical history and laryngoscopic findings. A sufficiently deep biopsy from the submucosal lesion shows on histological examination a capillary type of hemangioma (Fig. 1).

Correspondence: J. Meeuwis, Academic Molewaterplein 40, 3015 GD Rotterdam, 0165-5876/90/$03.50

Hospital Rotterdam, The Netherlands.

0 1990 Elsevier Science Publishers

Department

B.V. (Biomedical

of Otorhinolaryngology,

Division)

Dr.

146

Fig. 1. Capillary hemangioma

(H.E. Stain

x

19).

In about 50% of the cases patients display associated cutaneous hemangiomas, which are often localized in the head and neck region. Thus the presence of a cutaneous hemangioma in an infant with inspiratory stridor is suggestive of a similar lesion involving the airway. Hemangiomas tend to regress spontaneously after the age of one year. This makes the evaluation of treatment difficult: ‘post or propter’ is the question. Over the years multiple treatment modalities have been advocated. Several review articles on this subject have recently been published [4,8,13]. The use of sclerosing agents and cryosurgery is associated with a high complication rate. Irradiation carries the risk of a malignancy of the thyroid at an older age. In recent literature there is a tendency towards treatment with the CO,-laser and various authors have reported good results [10,11,15,19]. Sebastian and Kleinsasser advocate a tracheotomy and wait for spontaneous regression to occur [18]. Mawson in the early sixties and recently Mulder and van den Broek excised the lesion using a midline cricotracheotomy [14,16]. Treatment with systemic corticosteroids is effective [5,9]. A disadvantage is that therapy may be necessary for a rather long period of time. Possible side effects are a Cushing syndrome, growth retardation or an increased susceptibility to infection. We wanted to have an effective therapy, that respects the integrity of the mucosa of the infant larynx in its most vulnerable part, i.e. the subglottic space. In our clinic we already gained experience in treating subglottic

147

granulations of the larynx by means of intralesional methylprednisolon injections [3]. In ophthalmology Kushner reported encouraging results with such a treatment of hemangiomas of the eye adnexa [12]. Also cutaneous hemangiomas can be treated this way [7]. The procedure and results of the treatment of subglottic hemangioma by intralesional corticosteroid injection followed by intubation are presented in this paper.

Patients and methods In the period 1982-1987, 6 patients with a subglottic hemangioma were treated in the Sophia Children’s Hospital. The data regarding these 6 patients are summarized in Table I. The average age at the beginning of the therapy was 4 months (2-6:). The endoscopic diagnosis was confirmed in 3 cases by a biopsy. The diagnosis in the other 3 patients was based on the typical endoscopic findings together with the presence of a cutaneous hemangioma. All patients had symptoms of a slowly progressive episode of dyspnea and initially the diagnosis ‘pseudocroup’ was made in 4 patients on admission. In these 4 patients the progressive respiratory distress necessitated immediate laryngoscopy. Two patients were transferred from other centers, where extubation failed after treatment with systemic corticosteroids. The procedure of treatment is as follows: at laryngoscopy with a rigid pediatric

TABLE

I

Cure history overview Patienl No.

Age at onset rherapy (months)

Sex

Previous therapy

% airway obstruction at endoscopy

Biopsy

1

2

f

_

90

capillary hemangioma

2

5;

f

systemic

50-60

capillary

Other heman giomas

hemangioma

corticosteroids 3

6:

f

-

60-70

_

4

3

m

-

50

capillary hemangioma

5

2

f

intubation and systemic corticosteroids

not mentioned

face, cheek, tongue, gingiva

6

4

f

intubation and high doses of systemic corticosteroids

50

lip, neck (beard-like hemangioma)

back, clavicle

148

Fig. 2. Rigid bronchoscope

with ‘Optic needle’.

bronchoscope (Doesel-Huxley type, manufactured by Storz #10338F) the hemangioma is visualized and injected with 20-40 mg methylprednisolon (Depomedrol) (t-1 ml), depending upon the size of the tumor. Injection is performed with a so-called ‘optic needle’ (a Hopkins type endoscope Storz #28115A with a 0.5-mm long needle mounted on a Storz #11338LO shaft) as illustrated in Fig. 2. The patient is ventilated via the bronchoscope. After injection, because of increased tumor size, the patient had to be intubated to provide an adequate airway. The ‘loose fit’ nasotracheal tube remained in place for an average period of a week (3-10 days). Hereafter extubation followed when possible. Only when symptoms of respiratory distress reappeared the procedure was repeated. No ‘second look’ laryngoscopy was done if patients remained free of symptoms after the first treatment.

Results Table II shows the results of the therapy. All patients were symptom-free at the end of treatment. This result was attained in 3 patients after one injection, in 2 patients after 3 injections and 1 patient needed 5 injections. The average duration of intubation was 19 days (7-36). Three months (i-74) after the onset of therapy all patients were free of symptoms. No patient needed a tracheotomy. In the first patient a pneumonia could adequately be treated with antibiotics. The third patient was at the same time treated for two large cutaneous haemangiomas and showed a temporary fluffy hairgrowth and acne. A pneumonia as a complication of intubation could also be treated without problems. The sixth patient showed a slight growth retardation as reported by his general practitioner, probably due to previously administered high doses of systemic corticosteroids.

149

TABLE

II

Therapy ouerotew Patient No.

Number of wjectlons + intubation

Start-End of therapy (months)

Results

bation time (days)

Total intu-

1

3

34

5

no symptoms

Follow-up period

Complications/ Remarks

(years) pneumonia

at 7 months age 2

1

7

1 i

no symptoms

5

no complications

3

pneumonia,

at 6 months age 3

3

27

If

no symptoms age

4

5

5

1

36

8

5;

I i

no symptoms

transient

acne and fluffy hairgrowth cutaneous hemangiomas treated simultaneously

at 14 months

3

no complications

at 8: months

patient’s

age

an angiofibroma of the nasopharynx

father had

no symptoms

no complications

at 2; months age 6

1

Discussion

10

1 i

no symptoms

1

z

slight growth

at 4: months

retardation

age

to previous high doses of systemic corticosteroids

due

and conclusions

Treatment of subglottic hemangioma by means of intralesional injection with methylprednisolon (Depomedrol) followed by intubation proved to be effective and relatively simple. The authors feel that not only the effect of intralesional corticosteroid, but also local gentle pressure by the tube on the tumor is of therapeutic importance. No serious complications from this treatment were observed. No subglottic scarring or stenosis was seen. Side effects were only seen in the third patient who simultaneously received corticosteroid injections into two large cutaneous hemangiomas. There is no risk of radiation-induced malignancy with this treatment [2]. Although mortality is not as high as it has been previously, a tracheotomy at this age is still associated with significant morbidity. Plugging of the cannula, accidental decannulation, difficulties with feeding and speech development retardation are possible complications [l]. None of our patients needed a tracheo-

150

tomy. A shorter hospitalisation-time, less nosocomial infections and less interference with the normal development of the child are other advantages of this treatment. Cotton recently reported 3 cases of severe laryngeal stenosis following carbon dioxide laser treatment of large subglottic hemangiomas [6]. With our treatment the parents should be advised what to do when symptoms reappear. When the patient lives at a great distance from a hospital (which is never the case in the Netherlands) the time necessary to reach the hospital must be considered. Our patients were symptom-free at an average age of 7 months. This is 10 months earlier than Sebastian and Kleinsasser calculated for the time needed for a spontaneous regression to occur after a tracheotomy. The above-mentioned treatment of subglottic hemangioma in children is now the treatment of choice in our clinic. References 1 Arcand, P. and Granger, J., Pediatric tracheotomies: changing trends, J. Otolaryngol., 17 (1988) 121-124. 2 Benjamin, B. and Carter, P., Congenital laryngeal hemangioma, Ann. Otol. Rhinol. Laryngol., 92 (1983) 448-455. 3 Berkovits, R.N.P., Bos, C.E. et al., Congenital cricoid stenosis pathogenesis, diagnosis and method of treatment, J. Laryngol. Otol., 92 (1978) 1083-1100. 4 Brodsky, L., Yoshpe, N. and Ruben, R.J., Clinical-pathological correlates of congenital subglottic hemangiomas, Ann. Otol. Rhinol. Laryngol., 92 Suppl 105 (1983) 4-18. 5 Cohen, S.R. and Wang, C., Steroid treatment of hemangioma of the head and neck in children, Ann. Otol. Rhinol. Laryngol., 81 (1972) 584-590. 6 Cotton, R.T. and Tewfik, T.L., Laryngeal stenosis following carbon laser in subglottic hemangioma, Ann. Otol. Rhinol. Laryngol., 94 (1985) 494-497. 7 van Essen-Zandvliet, E.E.M., et al., Behandeling van hemangiomen van de huid bij kinderen, Ned. Tijdschr. Geneeskd., 131 (1987) 185-187. 8 Feuerstein, S.S., Subglottic hemangioma in infants, Laryngoscope, 83 (1973) 466-475. 9 Hawkins, D.B., Crockett, D.M. et al., Corticosteroid management of airway hemangiomas: long-term follow-up, Laryngoscope, 94 (1984) 633-637. 10 Healy, G.B., Fearon, B. et al., Treatment of subglottic hemangioma with the carbon dioxide laser, Laryngoscope, 90 (1980) 809-813. 11 Healy G.B., McGill, T. and Friedman, E.M., Carbon dioxide laser in subglottic hemangioma-an update, Ann. Otol. Rhinol. Laryngol., 93 (1984) 370-373. 12 Kushner, B.J., Intralesional corticosteroid injection for infantile adnexal hemangioma, Am. J. Ophtalmol., 93 (1982) 496-506. 13 Leikensohn, J.R., Benton C. and Cotton, R., Subglottic hemangioma, J. Otolaryngol., 5 (1976) 487-492. 14 Mawson, S., Subglottic haemangioma of the larynx treated by excision, J. Laryngol. Otol., 75 (1961) 1076-1081. 15 Mizono, G., and Dedo, H.H., Subglottic hemangiomas in infants: treatment with CO1 laser, Laryngoscope, 94 (1984) 638-641. 16 Mulder, J.J.S. and van den Broek, P., Surgical treatment of infantile subglottic hemangioma, Int. J. Pediatric Otorhinolaryngol., 17 (1989) 57-63, 17 Overcash, K.E., Putney, F.J., Subglottic hemangioma of the larynx treated with steroid therapy, Laryngoscope, 83 (1973) 679-682. 18 Sebastian, B. and Kleinsasser, O., Zur Behandlung der Kehlkopfhlmangiome bei Kindem, Laryngol. Rhinol. Otol., 63 (1984) 403-407. 19 Wenig, B.L. and Abramson, A.L., Congenital subglottic hemangiomas: a treatment update, Laryngoscope, 98 (1988) 190-192.