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Case Report

Ovarian Dermoid Cyst Super-infected with Methicillin-sensitive


Staphylococcus Aureus Leading to the Misdiagnosis of Appendicitis
in an Adolescent
Ryan J. Spencer MD 1, Kyle C. Kurek MD 2, Marc R. Laufer MD 1,3,*
1
2
3

Department of Obstetrics, Gynecology, and Reproductive Medicine, Brigham and Womens Hospital, Boston, Massachusetts, USA
Department of Pathology, Childrens Hospital Boston, Harvard Medical School, Boston, Massachusetts, USA
Division of Gynecology, Childrens Hospital Boston, Harvard Medical School, Boston, Massachusetts, USA

a b s t r a c t
Background: Mature cystic teratomas (dermoid cysts) are the most common ovarian tumor in adolescents. Super-infection of a dermoid
cyst is a rare event usually associated with a concomitant infection.
Case: A 14-year-old female was transferred to our institution with ve days of fevers and abdominal pain. CT of the abdomen/pelvis was
read as acute appendicitis with a 7.6 cm right adnexal dermoid cyst. The patient was treated for appendicitis but later found to have an
infected dermoid cyst primarily infected with methicillin-sensitive staphylococcus aureus, which led the misdiagnosis of appendicitis.
Summary and Conclusion: Super-infection of an ovarian dermoid cyst is an extremely rare event. We recommend that previously described
evaluation, surgical management, and ovarian conservation be employed in all cases of ovarian dermoid cysts.
Key Words: Adolescent, Appendicitis, Dermoid cyst, Staphylococcus aureus

Introduction

Case

Mature cystic teratomas (dermoid cysts) are the most


common ovarian tumor in adolescents.1 Although dermoid
cysts can contain structures derived from any germ cell
layer (e.g., hair, bone, teeth, cartilage, sebaceous glands, or
elements of any organ system), rarely do these tumors
become infected even though they can contain foreign
bodies.
Each of the ve case reports of infected dermoid cysts
found in the English literature had a concomitant pathologic condition that likely directly or indirectly contributed
to the infection. These cases involved extra-intestinal
Salmonella, disseminated brucellosis, disseminated schistosomiasis, a torsed dermoid with concurrent ectopic
pregnancy, and a tubo-ovarian abscess (TOA) with positive
Chlamydia cervical culture after pregnancy termination and
subsequent dilation and curettage for retained products of
conception and have been reviewed previously by Luk
et al.2 This case reports on a dermoid cyst primarily superinfected with methicillin-sensitive staphylococcus aureus
(MSSA) and multiple anaerobes that led to the misdiagnosis
of appendicitis both radiographically and on initial pathologic examination. We will review current best-practice
management strategies for ovarian dermoid cysts in the
adolescent population. Additionally, we review suggestions
for care of these patients in difcult clinical circumstances
and with rare complications by drawing from the Pediatric
Surgery literature.

A 14-year-old female with no previous medical or


gynecologic history was transferred to our institution from
an outside hospital with ve days of abdominal pain,
temperature 40 C, systolic blood pressure in the 80s with
tachycardia to 104 beats per minute (bpm) and a concern
for septic shock of unknown source. She was started on
intravenous ceftriaxone.
Upon arrival at our institution, initial vital signs showed
temperature of 40.3 C, heart rate 112 bpm, blood pressure
110/50, respiratory rate 24, and oxygen saturation of 97% on
room air. Initial labs showed white blood cell (WBC) count
of 21,650/mL. CT scan of the abdomen and pelvis was
interpreted as acute appendicitis with no evidence of
perforation or abscess and right adnexal mass with features
of a dermoid cyst. Pediatric surgery and Gynecology
consults were called and the decision was made to proceed
to the operating room for laparoscopic appendectomy.
An uncomplicated appendectomy was performed with
the surgeon noting evidence of acute inammation of the
appendix. Intra-operative gynecology consultation was
requested regarding the ovarian mass. Using recently published data on the surgical management of large ovarian
neoplasms in adolescents,3 and the potential adverse risks
of spillage of the neoplasms contents, the decision was
made to return at a later time to resect the ovarian mass so
that the proper preoperative evaluation (e.g., AFP, LDH, CA125, Inhibin-A, and hCG levels) and the appropriate
procedure (i.e. laparotomy and cystectomy with ovarian
preservation) could be performed. The patient had an
uncomplicated postoperative course and was discharged
home without further antibiotics on postoperative day 1.

* Address correspondence to: Marc R. Laufer, MD, 300 Longwood Ave, Boston, MA
02115
E-mail address: Marc.laufer@childrens.harvard.edu (M.R. Laufer).

1083-3188/$ - see front matter 2011 North American Society for Pediatric and Adolescent Gynecology. Published by Elsevier Inc.
doi:10.1016/j.jpag.2010.08.014

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R.J. Spencer et al. / J Pediatr Adolesc Gynecol 24 (2011) e25ee28

During the interim, the initial pathology report of the


appendix was reported as acute appendicitis; however,
subsequent pathologic examination did not reveal acute
appendicitis, but rather acute serositis of the appendix
(Fig. 1). This indicated an intra-abdominal source of
inammation outside of the appendix itself.
Evaluation of the ovarian mass and surgical planning of
the ovarian mass included the serum tumor markers listed
above. All values returned within normal limits (LDH 162,
AFP 1, CA-125 34, Inhibin-A 14.4, hCG ! 0.1). Follow-up
ultrasound revealed an 8.2  7.0  6.8 cm heterogeneous
right adnexal mass with fatty and cystic components in the
region of the right adnexa consistent with a dermoid cyst.
Throughout the time after the appendectomy, the patient
remained afebrile and asymptomatic.
Approximately two months after the appendectomy was
performed, the patient returned electively to the operating
room to undergo laparotomy and ovarian cystectomy as
previously described.3 Upon entrance into the peritoneal
cavity, there was a foul odor indicative of purulent material.
We were concerned that the appendiceal stump might have
become infected. With the aid of Pediatric Surgery, the
small bowel, ileocecal junction, appendiceal stump, and
right colon were inspected and found to be unremarkable.
We then proceeded to ovarian cystectomy.
The ovary was brought outside the peritoneum and
moist laparotomy pads were placed to decrease risk of
spillage of the cyst contents into the abdominal cavity. A
small tuft of hair was noted to be extruding from the cyst
indicating cyst rupture prior to the operative procedure
(Fig. 2). A small defect in the cyst wall was noted and
caseous material was draining. Cultures were obtained.
Levooxacin and metronidazole were started in the OR for
empiric, broad-spectrum antibiotic coverage. A cystectomy
was performed as previously described.3 The remainder of
the cystectomy was uncomplicated and the patients
abdomen was irrigated with copious amounts of warm
normal saline. Since all of the infected tissue (the dermoid)
was removed, the remaining ovarian tissue was preserved.

Fig. 1. The mucosa of the appendix was normal in appearance, although rare
neutrophils were present in the crypts. This likely reects the on-going inammatory
process of the serosal surface shown towards the bottom of the picture.

Postoperative Course

pelvis, suggestive of postsurgical inammatory changes, but


there was no denite focal collection that could be
described as an abscess. The Infectious Disease service was
consulted and agreed with the patient work-up and recommended switching antibiotics to pipercillin/tazobactam.
Over the following ve days, through POD #9, the patient
met all appropriate postoperative milestones with WBC
that trended from 16,960/mL (90% neutrophils) down to
12,970/mL (76% neutrophils). In order to continue IV pipercillin/tazobactam, a PICC line was placed for at least a total
of 4e6 weeks planned duration and the patient was
discharged home.
Three days later, the patient returned to the ED with
temperature 38.9 C. CT scan of the abdomen/pelvis was
performed and showed multiple small pelvic abscesses, the
largest of which measured 5.9  2.7  2.0 cm. Interventional
Radiology (IR) service was consulted to place a drain into
the largest collection. An uncomplicated drain placement
was performed with aspiration of only 2 cc of material with
the appearance of old blood. This material was cultured and
returned with 4 polymorphonuclear leukocytes but no
organisms were seen. In consultation with the Infectious
Disease team, IV pipercillin/tazobactam was continued.

On POD #2 the cultures revealed multiple anaerobes and


moderate amount of staphylococcus aureus and in the
afternoon, the patient had a temperature of 38.1 C. WBC
was 21,640/mL (89% neutrophils) and abdominal exam was
unremarkable. Blood and urine cultures were drawn and
were ultimately negative and antibiotics were changed to
ampicillin, gentamicin, and metronidazole. On POD #4, the
patient had a temperature 39.1 C. Abdominal exam was
unremarkable, blood and urine cultures were sent, and
chest radiography was done. All these studies were negative. WBC count was 20,780/mL (95% neutrophils). At this
point, the cyst cultures had returned as MSSA that was
resistant to penicillin so the patients antibiotic regimen was
changed to vancomycin, gentamicin, and metronidazole.
On POD #5, the patient had a temperature 39.2 C. Blood
cultures and urine cultures were again taken and were
negative. A CT scan of the abdomen/pelvis with contrast
showed moderate amount of uid in the abdomen and

Fig. 2. Opened gross specimen with classic features of a dermoid cyst including
numerous hairs and keratin debris. The cyst had a putrid odor and the contents have
a yellow, mucoid appearance of purulent material rather than the usual chalky-white
color of a classic dermoid.

R.J. Spencer et al. / J Pediatr Adolesc Gynecol 24 (2011) e25ee28

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On hospital day #10, the IR drain was removed and the


patient was transitioned from IV antibiotics to PO moxioxacin and metronidazole. She was discharged home on
hospital day #11 (POD # 22) to follow-up with both the
Gynecology and Infectious Disease services as an outpatient. The patient ultimately had difculty with nausea
using oral antibiotics, so a PICC line was placed to complete
an 8-week total course of antibiotics.
At 3-month follow-up the patient was completely
asymptomatic and a pelvic ultrasound revealed a normalsized right ovary with no residual abnormalities.
Pathology

By the time of discharge, the pathology of the ovarian


cyst returned as mature cystic teratoma. While this was the
expected outcome, there were comments that have not
been seen by these authors after previous dermoid resections. Grossly, the pathologist noted a mucoid appearance
of purulent material rather than the usual chalky-white of
a classic dermoid.[with] a putrid odor. (Fig. 2) Microscopically, there was extensive chronic inammation with
foreign-body giant cell reaction that extended to the surface
of the dermoid suggestive of previous perforation. Areas of
the cyst wall contained acute full-thickness inammation
with brin depositionthe histological characteristics for
abscess (Fig. 3).
Summary

Although mature cystic teratomas are the most common


ovarian tumor in adolescents, they rarely become infected.
A review of the literature reveals only ve previous case
reports in English; all were associated with a concomitant
pathology or systemic infection which lead to the subsequent infection of the dermoid. We are unaware of any case
report of an ovarian dermoid infected with MSSA. Additionally, we are unaware of any report of an infected
dermoid leading to the misdiagnosis of another intraabdominal pathology such as appendicitis, as described
here.
In examining the facts of the case, it appears that the
dermoid led to the appendiceal inammation and not vice
versa. If appendicitis had been the primary diagnosis which
led to the super-infection of the ovarian dermoid, then
pathologic examination of the appendix would have
revealed full thickness inammatory changes. Only the
serosal lining of the appendix was infected, indicating an
inammatory cause extrinsic to the appendix. While the
case is interesting because of its novelty, there are many
important points to be taken from the difcult clinical
decisions that had to be made while this case unfolded.
When the patients entire clinical picture was reviewed,
the decision to take the patient to the operating room for an
appendectomy seemed straightforward. She presented with
high fevers, nausea and vomiting, right lower quadrant
pain, elevated WBC and CT evidence of inammation at the
ileocecal junction. The initial surgical plan by the General
Surgeons was to remove the appendix laparoscopically and
then proceed to laparoscopic right oophorectomy. The

Fig. 3. The surface lining of the dermoid is shown here (top). It consists mostly of acute
inammation and brin both of which are histological characteristics of abscess.

consulting Gynecology service recommended against


oophorectomy based on the prevailing evidence that: (1)
ovarian masses in adolescents should be managed as
conservatively as possible4; (2) cystectomy is preferable to
oophorectomy for ovarian masses in adolescents5; (3)
patients with unilateral oophorectomy more frequently
seek infertility consultation.6
A difcult discussion was then made pertaining to
whether the ovarian cystectomy should be performed at the
time of appendectomy to save the patient from another
surgical procedure, or if it was prudent to proceed only with
appendectomy at that time and perform an interval evaluation of the ovarian mass prior to its removal. While it is
difcult to consign a 14-year-old girl to a second exposure
to anesthesia and another operative procedure, an adolescent that has an ovarian mass may have as much as a 33%
chance of having a malignant neoplasm.7 Since the risk of
ovarian malignancy in an adolescent with an ovarian mass
is signicant, appropriate preoperative evaluation should
consist of ultrasound exam and serum tumor markers,
specically CA-125, bHCG, AFP, and LDH.3 Any evidence of
malignancy should prompt the surgeon to make arrangements for a possible staging procedure.
The preferred method of ovarian cystectomy in adolescents is a topic of debate. While exploratory laparotomy is
performed at our institution, there are proponents of the
laparoscopic approach. Retrospective studies and case
series have shown that that laparoscopy results in less
intra-operative blood loss, shorter hospital stay, decreased
analgesia, earlier return to activities and improved
cosmetics when compared to laparotomy.8 All of these
advantages are important for both individual patients and
on a systems level in an era when cost savings and costeffectiveness are concepts that are increasingly viewed as
essential components of medical decision-making.
In this case, we employed a safe and effective laparotomy
technique described previously for adolescents which
allows for complete resolution of the ovarian abnormality
and return to normal ovarian tissue volume.3 We favor
laparotomy due to evidence that the recurrence rate of
dermoids after laparoscopic removal has been published at
7.6% compared to 0% with open surgery5 and the rate of
spillage of dermoid material during laparoscopy has been

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R.J. Spencer et al. / J Pediatr Adolesc Gynecol 24 (2011) e25ee28

reported as high as 100%.8 While the rates of chemical


peritonitis are low in adult series that studied laparoscopic
intervention for dermoid cysts,9 it is not known denitively
if the pediatric and adolescent populations are as resilient.
And even though there is some evidence that cyst spill does
little harm in the short term,8 long-term sequelae and
potential effects on future fertility are unknown. Another
concern with spillage is that even in patients with normal
tumor markers, there can be immature cells that could lead
to spread of malignant disease if present.10
In retrospect, one may conclude that laparoscopic cystectomy at the time of appendectomy could have been
benecial for this patient, and potentially have avoided
febrile morbidity, a prolonged hospital course, readmission,
and long-term IV antibiotic administration. Additionally,
the immediate laparoscopic approach would have spared
the patient from having a Pfannenstiel incision and a second
round of anesthesia in addition to the benets of laparoscopy noted previously. However, laparoscopy does result in
cyst rupture and spillage of dermoid contents in a large
percentage of cases. Since the infected dermoid contents led
to the difculties experienced in this patients post-op
course, we will never be able to say for certain if the
immediate laparoscopic approach could have avoided those
complications. Additionally, infected dermoids are such
a rare entity that the decision-making did not include the
possibility that the dermoid was the central cause in this
patients presentation.
Another recurrent concern that has surfaced was the
decision to not place an intrabdominal drain at the time of
exploratory laparotomy when rupture and spillage had
occurred and the suspicion for infection within the dermoid
was high. While each side has its proponents and detractors, it appears that the prevailing sentimentboth in the
literature and in our institutionis that placing an intraperitoneal drain is of little benet when no abscess can be
identied at the time of surgery.11,12 In the case presented,
the infected dermoid was removed and no other intraabdominal pathology was either visualized or palpated
during the procedure.
Initial theories for the etiology of the infection in this
case were that a transient bacteremia or direct inoculation
of skin ora during the appendectomy may have been to
blame. This seems unlikely since it is clear that the dermoid
was affecting the appendix before the appendectomy (evidenced by the appendiceal serositis) and therefore the
infection must have occurred previous to the appendectomy. It is possible that a transient MSSA bacteremia
occurred during a prior, distinct break in the skin which
found its way to the epidermal elements within the
dermoid and established colonies there.
We have engaged numerous colleagues regarding
possible reasons to why the patient remained afebrile in the
interval between the appendectomy and the cystectomy if
the dermoid was the source of the infection. The initial fever
was likely a combination of initial infection in the dermoid
coupled with an inamed appendixso inamed that both
the surgeon and the initial pathology reading felt acute
appendicitis was the diagnosis. Inammation manifested by
fever is a phenomenon that is seen in Gynecology after

myomectomy, the immediate postoperative state in


general, atelectasis, and postoperative, non-infectious
phlegmon. Our hypothesis is that the dermoid became
infected, developed a central area of abscess with inammatory cyst wall/ovarian cortex surrounding that central
area much like would occur in a true appendicitis.13 The
periphery of inamed cyst/ovarian tissue with central
abscess would have been lying out of the pelvis due to its
size and offered the opportunity to abut the appendix
leading to the inammatory response seen in the histology
of appendiceal serosa on pathology as well as in the ileocecal region radiographically. We further hypothesize that
the antibiotics given to the patient during her initial diagnostic evaluation were able to control the infection inside of
the dermoid to a degree that allowed the abscess to remain
walled off until it ruptured at a later time proximate to the
second surgery.
For presumed benign ovarian teratomas in adolescents,
the authors advocate for conservation of the ovary via
exploratory laparotomy even in the case of an infected
dermoid in order to preserve future fertility. Evidencebased preoperative evaluation and surgical management
should be followed. While no direct evidence exists as to
how to treat an infected dermoid cyst postoperatively, we
have drawn upon the literature from ruptured appendices
to advocate for long-term antibiotic administration (i.e.,
6e8 weeks) and close follow-up using both subjective
symptoms and objective measures such as WBC countand
imaging studies when clinically relevantto monitor for
complete resolution of the infection. While infected dermoid cysts are rare, any concomitant intra-abdominal
pathology should raise suspicion and prompt appropriate
action by the gynecologist to remove the dermoid, avoid
spilling cyst contents, obtain cultures, and consider empiric
broad-spectrum antibiotics.
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