EXPERIENCE AND REASON-BRIEFLY RECORDED 795
Pilonidal
Sinus
in
Infancy
Infection of a pilonidal sinus in the neonatal
period has not apparently been reported in
the literature. This paper presents the case of
a newborn infant in whom a severe cellulitis developed from a pibonidal sinus; in addition, cases are reviewed of pilonidal sinus occurring
in infants under 1 year of age admitted to Louisville Children’s Hospital during a
ten-year period.
CASE REPORT
KM., a white female infant, was born on June
22, 1964, by caesarian section. Her birth weight
was 7 lb. 3 oz. (1.3 kg). The infant appeared nor-mal on initial physical examination; no pilonidal
sinus was noted at this time. She made normal
progress until the third day of life, when a raised,
red nodule was noted in the midline in the
coccygeal area. This nodule increased in size
un-til the fifth day of life when, on her back an area of cellulitis developed, triangular in shape with the apex in the midline at the superior margin of the natal cleft; her temperature was 104 F#{176}(40#{176}C)
per rectum and there were no other abnormal findings. The I)lOOd count showed hemoglobin
14.4 gm/l00 ml, white blood cell count 4,218/
mm. A diagnosis of erysipelas was made, and
treatment was started with aqueous penicillin
50,000 units every 6 hr intramuscularly.
The following day the area of cellulitis had
in-creased, and there was a purulent discharge from
a midline sinus overlying the coccygeal area at the site of the original swelling. The sinus was
opened to permit free drainage; biopsy of the lesion was not attempted. Chloramphenicol 25
mg every 8 hr and streptomycin 17.5 mg every
8 hr were administered intravenously in addition to the penicillin.
On the fifth day of the illness, the seventh day of life, the area of infection was unchanged. There was some abdominal distension, but the infant
was feeding well and passing normal stools. X-rays of the chest, abdomen, and spine were
nor-mal. The white cell count was 10,200/mm’
(polymorph. 22%, lymphocytes 66%, monocytes
7).
The next day there was evidence of further spread of the infection locally. Cultures taken from the sinus grew Staphylococcus aureus, coagulase positive, sensitive only to
chlorampheni-col and methicillin. Penicillin and streptomycin
were discontinued and chloramphenicol 25 mg
every 6 hr and methicillin 90 mg every 6 hr
were given intravenously; the pilonidal sinus
area was treated with warm soaks.
In the ensuing 4 days there was progressive worsening of the color of the skin of the back,
and on the eleventh day of the illness there was necrosis of a substantial area. Pyrexia continued but the infant’s general condition remained good. The intravenous infusion was replaced by intra-muscular injections of chloramphenicol 50 mg/6 hr and methicillin 90 mg every 6 hr; one injec-tion of gamma-globulin 2 cc was given. In the next 48 hours there was further necrosis and sloughing of the skin across the back. The hemo-globin which had fallen to 11.8 gm/iOO ml was restored by a transfusion of 80 cc of whole blood. On the thirteenth day of illness the blood count showed hemoglobin 15.8 gm/ 100 ml, white blood cell count 24,092 mm’. Urinalysis was normal. Blood
cultures were reported negative. Cultures from the
necrotic area of the back grew E. coli and
Klebsiella-aerobacter group.
Two days later, under general anesthesia,
de-bridement of the skin and subcutaneous tissues was performed. The skin edges were found to be undermined for 5 cm beyond the necrotic area; these were folded back and sutured. The wound was protected by a peroxide dressing through which oxygen was passed to minimize the risk of
infection by anerobic organisms.
The wound was subsequently treated as a burn; the area of skin loss was estimated at 13% of the
body surface. Thirteen days after the first
opera-tion patch-grafting was carried out after removal of the sutures. The infant’s subsequent progress
was satisfactory; healing was almost complete
when she was discharged from the hospital, age 7 weeks.
Although the question may be raised of
whether there is adequate evidence of the
na-ture of this patient’s lesion, the only certain proof,
by biopsy, was not available because of the dam-age by infection. The clinical and anatomical
find-ings strongly suggested pilonidal sinus as the primary condition.
REVIEW
From 1954 to 1963, 38 cases of pilonidal sinus in children tinder 1 year of age were
seen in Louisville Children’s Hospital; 23 were
male (60.5%) and 15 were female (39.5%). In
1 1 of these patients the diagnosis was made
as an incidental finding at autopsy; of the
remaining 27 infants, 18 (66.6%) were noted
to have had a pilonidal sinus at birth.
The diagnosis in these 27 cases was based
on the presence of one or more of the
follow-ing physical signs: coccygeal dimple, 8
(29.6%); cystic swelling, 13 (48.1%); and mid-line sinus 16 (59.2%).
Of the 38 patients there were associated
congenital anomalies in 15 (39.2%). These
796 PILONIDAL SINUS
inguinal hernia (2 cases), cutaneous
hemangi-oma (2 cases), and cleft palate, spina bifida,
Hirschsprung’s disease, fibrocystic disease,
dermoid cyst, renal anomaly, and mental
re-tardation.
Twenty-two (81.4%) of the 27 cases
diag-nosed during life underwent surgical excision of the sinus and primary closure of the wound;
there were no postoperative complications. In
all of these patients the diagnosis was con-firmed histologically. Four cases were not
treated, and in one 10-week infant a pilonidal
cyst ruptured and healed spontaneously.
COMMENT
Huberl first described pibonidal sinus in the
newborn, although the condition had earlier
been noted in older infants by Dunbop.2 There
have since been several reports of the disease
in infancy.36 In the present series, the pro-portion of cases in whom the disease was noted at birth (66.6%) would appear to
con-firm the opinion#{176} that pibonidal sinus is
con-genital in origin. The occurrence in the
pres-ent series of associated congenital defects in
39.2% of the cases may support the idea of a
congenital origin of the pilonidal sinus in these patients. However, Warren11 and more re-cently Patey and Scarffl2 have upheld the theory that the disease in adults is acquired
and often provoked by trauma. Coodalll3
proposes the existence of both congenital and
acquired types, pointing out that pilonidal
sinus as seen in infancy has a different natural history from the adult variety.
In the adult, infection is the commonest
presenting feature, but in infancy it is rare.
Kleckner5 described one case, and Coodalll3
has described two cases in older infants. Of
the present series, two cases were described
histologically, but not clinically, as showing
“chronic inflammation.”
No reports have been found in the
litera-ture of infection in the newborn spreading
beyond the confines of the sinus track, as
occurred in the patient recorded here. Ripley
and Thompsont4 described the case of a 33-month-old infant who developed
Staphy-present case is not established; there were no
cases of Staphylococcal infection in the
nurs-ery for at least 4 weeks before this infant was
born and there were no subsequent cases for
over 1 month. There was no pre- or
post-operative infection in the mother.
It
would appear that the congenital type ofpilonidal sinus is a potential danger to the
patient; Swenson16 recommends excision and
primary closure in these cases, and this
pro-cedure was undertaken in 22 patients (8 1.4%)
of the present series without complication.
SUMMARY
The case presented is of a newborn infant
with a fulminating infection with Staph ylococ-cus aureus arising from a pibonidal sinus. The
patients admitted over a 10-year period to Louisville Children’s Hospital with a diagnosis
of pilonidal sinus are reviewed. The disease
appears to be of congenital origin in this age
group. The potential danger of this condition
in infants is emphasized.
RICHARD
A.
LEWIN,MB.,
B.Chir. (Cantab.)University of Louisvifle, Department of Pediatrics;
Children’s Hospital 226 East Chestnut Street Louistille, Kentucky 40202
I
wish to thank Dr. F. Falkner, Professor andChairman, Department of Pediatrics, for his help
and encouragement.
I
am grateful to Dr. B. F. Andrews, Director of New-Born Services, and Dr. C. K. Sergeant, Chiefof Section, Department of Pediatric Surgery for
permission to publish their case and for their ad-vice and criticism in the preparation of this paper.
REFERENCES
1. Huber, F. : Sacro-coccygeal tumor in a child three weeks old. Archiv. Pediat., 9:891, 1892.
2. Dunlop, A. : Abnormal coccyx. Nature, 18:94, 1878.
3. Breidenbach, L., and Wilson, H. L. : Pilonidal cysts and sinuses. Ann Surg., 102:455, 1935.
EXPERIENCE
AND
REASON-BRIEFLY
RECORDED
7978. Stone, H. B.: Origin of pilonidal sinus. Ann.
Surg., 94:317, 1931.
9. Beigeleisen, H. I.: Scherotherapy for pilonidal
Cysts. Amer. J. Surg., 44:622, 1939.
10. Hopping, R. A.: Pilonidal disease. Amer. J. Surg., 88:780, 1954.
11. Warren,
J.
M.: Fistulous opening near thebase of the coccyx containing hair. Boston
Med. Surg. J., 96:328, 1877.
12. Patey, D. H., and Scarf, R. W.: The hair of
the pilonidal sinus. Lancet, 1:772, 1955.
13. Coodall,
P.
: Aetiology and treatment ofpilonidal sinus. Brit. J. Surg., 49:212, 1961.
14. Ripley, W., and Thompson, D. C.: Pilonidal
sinus as a route of infection in a case of
staphylococcal meningitis. Amer. J. Dis.
Child., 36:785, 1928.
15. Moise, T. S.: Staphylococcal meningitis
see-ondary to congenital sacral sinus. Surg.
Gynaec. Obstet., 42:394, 1926.
