Keywords
mucormycosis - rhino-orbital cutaneous mucormycosis - ROCM - orbital exenteration
- orbital reconstruction - opercular approach
Introduction
Rhino-orbital cutaneous mucormycosis (ROCM) is an opportunistic infection that occurs
in immunosuppressed patients. ROCM can result in high mortality or dysfunction and
disfigurement. The COVID-19 wave saw a large number of patients of ROCM who underwent
orbital exenteration.[1]
The conventional approach to reconstruction has been filling of the orbital volume
defect by various methods including local, regional, and free flaps.[2] Elements of reconstruction included obliteration of the orbital mucosa, which is
often unstable, with ulcers, and filling of the orbital volume defect with soft tissue
and a transverse oval of skin representing the eyelids. The other goal in reconstruction
has been readying the orbit to receive a bone anchored eye prosthesis.[3]
As an original idea, the senior author has adopted the “opercular” approach in orbital
reconstruction, which closes off the orbital volume defect externally and still retains
the ability to accommodate a future prosthesis. The residual volume defect becomes
part of a larger rhino-antro-orbital space and drains into the nasal or oral cavity.
Treated patients have expressed satisfaction with the procedure and chosen not to
opt for an ocular prosthesis—bone integrated or external.[4]
Materials and Methods
Fourteen patients with exenterated orbits (13 with type IIIc and 1 with type IVa ROCM[5]) and associated palatal defects, partial maxillectomy or septal defects, were referred
to our institution between July 2022 and January 2024.
Eight of 14 patients with orbital exenteration due to ROCM, without palatal defect
(in 2 cases, the palatal defect was repaired at an earlier stage), local disease free,
and medically fit to undergo surgery, who underwent reconstruction of the orbital
defect via the opercular approach, were included in the study. Patients with existing
palatal defects were excluded from the study.
Eight of the 14 patients underwent reconstruction of the exenterated orbit via the
opercular approach.
Each patient was taken up for surgery with a clear disease-free interval of 6 months
or more and in cases of unstable orbital mucosa (ulcerated or crusted), frequent saline
dressings were done for at least 15 days or till complete mucosal healing.
An orbital inlet perimeter incision was made and hinge flaps developed, based posteriorly,
closed with inverting mattress sutures of 3–0 polyglactin (Vicryl, Ethicon India),
to form an inner layer. In seven of eight cases, the ipsilateral nasofacial flap was
advanced to close the external defect, with 4–0 polyamide (Ethilon, Ethicon India),
with a suction or corrugated drain kept between the two layers ([Figs. 1]
[2]
[3]
[4]
[5]
[6]
[7]). In one case with unstable orbital mucosa and sequestrated infraorbital rim, after
sequestrectomy, the median forehead flap was used to form the lining instead of the
hinge flaps (([Figs. 8]
[9]
[10]
[11]
[12]).
Fig. 1 Case 1: Preoperative.
Fig. 2 Case 1: Preoperative marking.
Fig. 3 Case 1: Intraoperative view of the inner layer.
Fig. 4 Case 1: Intraoperative view of the nasofacial flap advanced.
Fig. 5 Case 1: Intraoperative view after closure.
Fig. 6 Case 1: Review after 1 year and 5 months. Oblique view.
Fig. 7 Case 1: Review after 1 year and 5 mo. Frontal view.
Fig. 8 Case 2: Preoperative frontal view.
Fig. 9 Case 2: Preoperative oblique view.
Fig. 10 Case 2: Intraoperative view showing the forehead flap for lining and nasofacial flap
as cover.
Fig. 11 Case 2: Intraoperative view after closure.
Fig. 12 Case 2: Review. Short term.
The suction or corrugated drain was removed after 5 to 6 days and sutures were removed
after the 10th day.
All patients were on follow-up for at least 6 months and, where indicated, nasoendoscopy
was done to view the inner layer and examine the ipsilateral naso-antral-orbital space
for crusting or ulceration. Early crusts that developed in two cases were removed
endoscopically; further follow-up endoscopy was done after 10 days' interval to monitor
healing ([Figs. 13]
[14]
[15]
[16]
[17]
[18]).
Fig. 13 Case 3: Intraoperative view of the inner layer.
Fig. 14 Case 3: Preoperative.
Fig. 15 Case 3: Intraoperative view of the hinge flap.
Fig. 16 Case 3: Intraoperative view of the nasofacial flap advanced.
Fig. 17 Case 3: Review.
Fig. 18 Case 3: Appearance of the hinge flap endoscopically.
At follow-up, all patients were asked to rate their overall satisfaction of facial
appearance on a visual analog scale of 1 to 10 and score the sensory apperception
of the treated area in comparison to the opposite side on a scale of 1 to 5.
Results
The eight patients who underwent orbital exenteration for ROCM developed during Covid
2021 were aged 47.2 years on an average (25–58 years; 7 of the 8 patients were in
their 50s), predominantly male (7 males and 1 female), with diabetes mellites type
II in six of eight patients, hypertension in three patients, and history of transient
hemiplegia (magnetic resonance imaging showed thrombosis of intracranial segment of
internal carotid artery on left side) in one patient. The average disease-free interval
was 8.8 months ([Table 1]). Four patients required saline dressings for at least 2 weeks where the orbital
mucosa was deemed unstable.
Table 1
Patient particulars
|
Sl. no.
|
Age/sex
|
Cause of mucormycosis
|
Comorbidity
|
Date of orbital exenteration
|
Duration since cessation of antifungal treatment
|
|
1
|
55/M
|
COVID: 2021
|
T2 DM, HTN
|
June 2021
|
6 mo
|
|
2
|
37/M
|
COVID: 2021
|
T2 DM, HTN
|
May 2021
|
8 mo
|
|
3
|
50/M
|
COVID: 2021
|
T2 DM
|
May 2021
|
9 mo
|
|
4
|
50/M
|
COVID: 2021
|
Hemiplegia, T2 DM
|
June 2021
|
9 mo
|
|
5
|
58/M
|
COVID: 2021
|
HTN
|
May 2021
|
12 mo
|
|
s6
|
52/M
|
COVID: 2021
|
T2 DM, HTN, >creatinine levels
|
June 2021
|
8 mo
|
|
7
|
51/M
|
COVID: 2021
|
T2 DM, HTN
|
May 2021
|
1 y
|
|
8
|
25/F
|
COVID: May 2021
|
|
May 2021
|
2 y and 3 mo
|
Abbreviations: DM, diabetes mellitus; HTN, hypertension.
The seven patients who underwent nasofacial flap cover needed an average surgical
time of 2 hours and 8 minutes (including anesthesia) compared with the single case
with radial forearm flap cover, which need 6 hours ([Table 2]).
Table 2
Reconstructive surgery details
|
Sl. no.
|
Preoperative saline dressings
|
Lining
|
Cover
|
Duration of the surgery
|
Complications/undesirable outcomes
|
Further orbital prosthesis
|
Follow-up
|
Patient satisfaction
|
Nerve sensation
|
|
|
Hinge flap/others
|
Nasolabial flap/radial forearm flap
|
|
|
|
|
|
|
|
1
|
None
|
Yes
|
NL
|
135 min
|
Minor wound dehiscence
|
No
|
2 y
|
8
|
5
|
|
2
|
Saline dressings (15D)
|
|
RFF
|
6 h
|
None
|
No
|
2 y
|
8
|
1
|
|
3
|
Saline dressings (1M)
|
Yes
|
NL
|
150 min
|
None
|
No
|
1 y and 10 mo
|
10
|
5
|
|
4
|
None
|
Yes
|
NL
|
50 min
|
None
|
No
|
1 y and 7 mo
|
10
|
5
|
|
5
|
Saline dressings (1 M)
|
Yes
|
NL
|
135 min
|
None
|
No
|
6 mo
|
9
|
5
|
|
6
|
None
|
Median forehead flap
|
NL
|
180 min
|
None
|
No
|
3 mo
|
8.5
|
5
|
|
7
|
Saline dressings (2M)
|
Yes
|
NL
|
130 min
|
None
|
No
|
3 mo
|
9
|
5
|
|
8
|
None
|
Yes
|
NL
|
120 min
|
None
|
|
2 mo
|
8
|
5
|
Abbreviations: D, days; M, months; NL, Naso-labial flap; RFF, radial forearm flap.
All patients healed well, except one patient who had a minor wound dehiscence, which
was repaired and went on to heal uneventfully. In one patient with a significant medial
orbital wall defect and partial maxillectomy, nasoendoscopic removal of crusts was
done once, postoperatively, which went on to mucosalize well ([Table 2]).
The four patients who expressed satisfaction with their appearance at long-term review
(>1 year) did not have any specific complaints with regard to the residual orbital
volume defect and did not opt for ocular prosthesis at the time of review. The remaining
four of the 8 patients expressed satisfaction at this point in time and will be reviewed
for at least 1 year. All patients felt relieved at not having to cover the hollow
orbits with dressings.
The overall patient satisfaction on a visual analog score after surgery range between
8 and 10.
All patients rated their facial sensation as 5/5, except the one patient who had radial
forearm flap cover who scored 1/5 ([Table 2])
Discussion
The COVID-19 pandemic affected the entire world on an unprecedented scale. In India,
the impact of the second wave in 2021 was huge, with 19.29 million confirmed cases
and 242,211 confirmed deaths reported between March 1 and June 30, 2021.[6] Particularly during the second wave, a dangerous complication followed in the form
of COVID-19-associated mucormycosis.[7] The Indian Union Health Minister stated that 28,252 cases of mucormycosis were reported
from 28 states/union territories in the country, of which 86% cases had a history
of COVID-19 infection and 62.3% had a history of diabetes.[8] In total, 2,826 cases of ROCM were reported in a multicenter study[5] in India, of which 78% were diabetics.
After the initial medical management and radical debridement, there was a cohort of
cases that had a gaping orbital defect sometimes associated with a maxillary, palatal,
or septal defect. The disfigurement, regurgitation of fluids, and crusting led the
patients to seek a surgical solution.
The conventional approach in type III ROCM cases involves filling of the orbital volume
defect with local flaps, using the temporalis muscle with a skin graft, or using the
lateral arm, radial forearm, and anterolateral thigh flap as free tissue transfer.[4]
[5] Local skin flaps like the forehead flap and cheek rotation flap have been used along
with the temporalis muscle[9]; the defacement, scarring, and hollowing make it an undesirable procedure. The temporalis
muscle flap used along with skin graft can have limitations in reaching the medial
aspect of the orbital cavity and is used for separating intracranial communication
with the orbit.[10] Radial forearm, lateral arm, and anterolateral thigh flaps used as free tissue transfers
have the advantage of acting as fillers as well as covering adjacent cheek defects;
their use is more common in orbital or cheek malignancy.[11]
In our series of 8 of 14 cases of ROCM, which were reconstructed, we developed the
original idea of closure of the orbital aperture as an operculum, using the orbital
mucosa as a hinge flap to form the inner lining and nasofacial skin as cover in 7
cases; in 1 case, a free radial forearm flap was used as cover and in another the
forehead flap was used as lining. This opercular approach simplified the entire procedure
of orbital reconstruction, making it quicker and much less expensive and reducing
the chances of flap failure. The treated area had a natural “feel” in the cases done
with a nasofacial flap cover. Our series demonstrates that filling the orbital volume
gap may not be necessary.
On review, patients were quite satisfied with their appearance and the social inclusion
gained as they did not have to wear an eye patch all the time. None of the reviewed
patients wished to have an ocular prosthesis. All patients achieved social inclusion
after the procedure including a temple priest and a church priest, both of whom have
gone back to performing their priestly duties.
The drawback of the study is that this approach may not be feasible in cases of extensive
periorbital scarring or presence of an oroantral fistula.
This study is limited in terms of persons treated and is an ongoing evolution of technique.
However, the ease of execution, simplicity, and cost-effectiveness of the opercular
approach make it a desirable option.
