Volume 7 Issue 1, January - June 2022

P-ISSN: 2460-9757, E-ISSN: 2597-7288

Nationally Accredited Journal, Decree No. 36/E/KPT/2019.

Case Report

Malignant Melanoma of Anorectal: A Report of

Two Cases
Muhammad Ihwan Kusuma1, Samuel Sampetoding1, Ferdinandes1, Muhammad
Faruk2

1
Division of Digestive, Department of Surgery, Faculty of Medicine, Hasanuddin
University, Makassar, Indonesia
2
Department of Surgery, Faculty of Medicine, Hasanuddin University, Makassar,
Indonesia

Corresponding Author:
Name: M. Ihwan Kusuma
Email: [email protected]

ARTICLE INFO ABSTRACT

Keywords: Introduction and importance: Occurring in only 1% of

Malignant melanoma; cases of anorectal malignancy, malignant melanoma
Anorectal malignancy; generally appears in the fifth and sixth decades of life, with
Abdominoperineal
complaints of anorectal bleeding or pain. The prognosis is
resection; late-stage;
Case report; generally poor. Although anorectal melanoma currently
lacks a recommended treatment, surgery alone remains
How to cite: the primary modality of treatment, the role of adjuvant
Kusuma MI., therapy is generally minimal, and the survival rate
Sampetoding S., improves with early diagnosis.
Ferdinandes., Faruk Presentation of case : Here, we report two cases of a rare
M. (2022). Malignant
Melanoma of malignant melanoma in the rectum treated with
Anorectal: A Report of abdominoperineal resection and local excision. The first
Two Cases. case is a 60-year-old man with a history of defecating
Nusantara Medical bloody stool and the appearance of a lump on the anal
Science Journal, 7(1), opening. The abdominoperineal resection was performed
12-20 followed by adjuvant chemoradiotherapy. The second
case is a 51-year-old woman with a similar complaint and
DOI:
10.20956/nmsj.v7i1.1 clinical finding to the first case. The second case was
7951 treated with a tumor excision procedure followed by
adjuvant chemoradiotherapy. However, the lump was
recurrence after 7 months of prior tumor excision.
Discussion: Surgical resection of anorectal melanoma
is debatable. While surgical resection is still the most
common treatment option, there is no agreement on

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 Nusantara Medical Science J. 7(1): 12-20

whether abdominoperineal resection or local excision is

the best option. Conclusions: The abdominoperineal
resection may offer a lower recurrence probability than
that of tumor excision alone. However, further study is
required to provide evidence-based surgical treatment.
Copyright © 2022 NMSJ. All rights reserved.

1. INTRODUCTION
Anorectal melanoma is a malignant tumor arising from melanocyte cells that
develops in the anal canal.1,2 Metastasis from malignant melanoma (MM) is known to
spread by local extension, by the lymphatics, or by the bloodstream.3 Most commonly
appearing on the skin, followed by the meninges, eyes, mucosae, anorectal region, and
the bone4, melanoma occurs with the same incidence in men as in women, especially
ones who have experienced frequent exposure to sunlight.2 Among men, melanoma
most often appears on the upper back, whereas women most often experience it on the
lower legs.5 Regardless of gender, adults and especially older adults contribute most to
melanoma’s incidence, with a peak among people in the sixth decade of life.6
Extremely rare, anorectal melanoma is responsible for only 1% of cases of
anorectal malignancy, which is almost always caused by adenocarcinoma and various
squamous cell cancers. Predominantly occurring among women, anorectal melanoma is
most common in the fifth and sixth decades of life, and patients with the disease usually
present with rectal bleeding and an altered defecation pattern. The prognosis for
anorectal melanoma is generally poor, with a median survival of 24 months and a 5-year
survival rate of 10%.2 Most people with melanoma of the rectum die from metastases.
Although no consensus currently exists regarding the most appropriate surgical
approach for anorectal melanoma, possible procedures range from local excision to
abdominoperineal resection (APR), either with or without adjuvant radiotherapy.2,7 Here,
we report two cases of a rare malignant melanoma in the rectum treated in our institution.

2. CASE PRESENTATION
Case 1
A 60-year-old man with a 6-month history of bloody stool was admitted to the
hospital with complaints of painful lumps in the anal opening 2 days prior to admission.
On physical examination, the patient looked pale, no abnormality was detected by
abdominal examination, and indicated no pain but also presented a brittle mass in the
anus measuring 7 × 7 cm, with the consistency of dense rubber and that bled easily
(Figure 1). Digital rectal examination suggested that the mass had originated on the anal
verge.
Beyond that, laboratory tests revealed a hemoglobin (Hb) count of 7.4 g/dL and
a Carcinoembryonic antigen (CEA) count of 8.5 ng/ml, and the results of a biopsy
indicated malignant melanoma. Next, a computerized tomography (CT) scan showed a
mass in the rectum but no lymph nodes (Figure 2) or metastases in the liver, and the
chest X-ray did not reveal any metastatic nodules in the lungs. Considering the
symptoms, physical examination, radiological examination, and histopathology results,

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we classified the first patient as cT3N0M0 (stage IIB) according to the American Joint
Committee on Cancer (AJCC) 8th edition (AJCC 8).

Fig. 1 Mass of fragile anorectal tumor that bled easily

The patient was fully informed about the treatment modality and choice. The
abdominoperineal resection (APR) was explained as a procedure that completely
removes the distal colon, rectum, and anal sphincter complex using both anterior
abdominal and perineal incisions, resulting in a permanent colostomy. The tumor
excision procedure was also explained as an alternative surgery. We had also informed
the complications of tumor excision procedure which were the higher risk of bleeding,
infection, chronic wound, recurrence probability, and fecal incontinence. We are
concerned about the quality of life and the autonomy of the patient. The patient
understood completely the treatment and agreed to APR. In consideration of those
results, the patient underwent APR with a good outcome and was discharged on the
eighth day after surgery. Once the pathology result of malignant melanoma was
confirmed, the patient was scheduled for adjuvant chemoradiotherapy (Figure 3A dan
3B).

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Fig.2. A CT scan of the abdomen and mass in the rectum (arrow).

Fig. 3. a) Tumor mass after abdominoperineal resection; b) Histopathological results of 40× enlargement
showing atypical cells with spindle nuclei contained melanin.

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Case 2
A 51-year-old woman with a 1-year history of anorectal bleeding presented at the
hospital with complaints of lumps in the anus for the past month. A year prior, the patient
had received an endoscopy that revealed a polyp mass in the rectum indicating
pathology of chronic inflammation (Figure 4). On physical examination, the patient looked
pale, with no distention of the patient’s abdomen, but also an anal mass measuring 10 ×
15 cm that was fragile and bled easily. Laboratory tests revealed a Hb count of 6.6 g/dL,
while the chest X-ray showed no signs of pulmonary metastatic nodules. We classified
the second patient as cT3N0M0 (stage IIB) according to AJCC 8. The patient was well
informed about the surgical treatment modality (APR and tumor excision) and the
complication of the surgery. The patient chose the tumor excision procedure.

Fig. 4. Anorectal mass that is fragile and bleeds easily .

The tumor was excised, and its examination confirmed the pathology of malignant
melanoma (Figure 5). The patient was discharged 6 days postoperative in good
condition. The patient was completely informed about the adjuvant chemoradiotherapy,
including the benefit and the complication. The chemoradiotherapy session had been
scheduled from the outpatient clinic. However, the patient refused to have
chemoradiotherapy immediately with the consideration of waiting for the surgical wound
to heal. The patient went back to her hometown which was far from our institution.

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Fig.5. Histopathological results of 40× enlargement show atypical cells with spindle nuclei containing
melanin.

Seven months later, the patient returned with new complaints of anal bleeding
without any prior adjuvant chemoradiotherapy, which general conditions Karnofsky
grade 60%. A physical examination of the patient revealed an anemic, flat, but painless
abdomen, after which a digital examination of the rectum revealed a mass measuring 2
× 5 cm at the base of the dentate line that bled easily. A CT scan of the abdomen showed
a rectal mass with multiple metastatic nodules on the liver, and a chest x-ray showed
metastases in the lungs. A re-excision of the tumor was performed to stop the bleeding,
and the patient was discharged on the fourth day after surgery.

3. DISCUSSION
First reported by Moore in 1957, anorectal melanoma is a highly rare disease.
Melanoma of the rectum, which accounts for just 1% of anorectal malignancies and
occurs predominantly in women in their fifth and sixth decades of life, is difficult to detect
and is sometimes misdiagnosed as rectal polyps, hemorrhoids, or ulcer lesions that co-
occur with rectum prolapse.8
Anorectal melanoma appears on the dentate line, where it develops from
melanocytes in the dentate area. From a macroscopic standpoint, the tumors are polyp-
shaped, and if microscopically pigmented with immunostaining features for melanosomal
proteins, the cells appear to be arranged in nests. The histological markers S-100, HMB-
45, and vimentin can help to clarify the diagnosis. If the biopsy shows a suspicious
specimen for a sarcoma (e.g., leiomyosarcoma), then S-100 staining should be
performed, and if the result is positive, then the tumor is most likely melanoma. 9 Tumor
histology includes epithelial cells or spindles; however, as in our second case, histology
results can be a mixed tumor, round oval to the spindle. Tumors with pure epithelioid
histology are less likely to recur.2

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The rarity of malignant melanoma of the rectum stems from the observation of
tubulovillous polyps in the tumor pedicles. Melanoma occurring at the anal verge can
turn into a polyp and become an invasive melanoma in the rectal mucosa, after which
infiltration into the dysplastic polyp occurs. For that reason, such melanoma is also
sometimes called a “collision tumor”. 9
Wide local excision (WLE), abdominoperineal resection (APR), and endoscopic
mucosal resection (EMR) are some of the surgical options. In rare circumstances, EMR
can resect the melanoma and provide long-term survival. WLE has low morbidity and
does not impair local function while preserving the anal sphincter. APR is frequently
linked to a high risk of morbidity and functional impairment. When the tumor margins
were macroscopically and microscopically negative, the operation was classed as R0,
R1 when the margins were positive on microscopy, and R2 when the resection was
macroscopically incomplete. 1
Surgical resection of anorectal melanoma is debatable. While surgical resection
is still the most common treatment option, there is no agreement on whether APR or
local excision is the best option. Because it can restrict lymphatic dissemination and
provide a bigger negative margin for local control, APR is considered the standard
procedure for treating anorectal melanoma. 9
Maliha Khan et al reported a case of a 71-year-old female with anorectal
melanoma. Their patient was treated with APR prior to chemotherapy. However, the
long-term outcome was unknown.10 In 2019, Binh Van Pham et al reported two cases of
anorectal melanoma. One of their cases was treated with APR and another with local
excision of the tumor followed by ultra-low anterior resection (ULAR). The patient who
underwent the APR procedure had no complications from surgery and no sign of
metastases or recurrence 6 months after surgery, whereas the patient who received the
ULAR experienced low anterior resection syndrome and evidence of recurrence was
found 16 months after the surgery. 11
Despite the lack of consensus on whether rectal melanoma should be treated
with APR or wide local excision,12 several recent studies have suggested that, if possible,
sphincter-sparing local excision and adjuvant radiation can effectively control loco-
regional disease while preventing functional morbidity due to APR. At the same time,
mesorectal lymph nodes play a role in the appearance of inguinal lymph nodes, which
differ in incidence from squamous cell carcinoma in the anus. Thus, if an APR is
performed, then mesorectal lymph node resection can contribute to better staging,
whereas prophylactic inguinal lymph node resection will have no benefit. As for the
outcomes, patients without lymph node metastases had a 5-year survival rate of 20%
versus 0% among patients with lymph node metastases. Meanwhile, the survival rate of
patients with recurrent or metastatic rectal melanoma is less than 10 months.9 Poor
prognosis in tumors is influenced by tumor thickness, tumor necrosis, and perineural
invasion, and amelanotic lesions typically receive worse prognoses. 13

4. CONCLUSION
Malignant melanoma of the rectum, a rare anorectal malignancy, is a highly
aggressive disease that is difficult to diagnose. Although anorectal melanoma currently
lacks a recommended treatment, surgery alone remains the primary modality of

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treatment, the role of adjuvant therapy is generally minimal, and the survival rate
improves with early diagnosis.

INFORMED CONSENT
Written informed consent was obtained from the patients for participation in our
study.

ACKNOWLEDGMENTS
None

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Conflict of Interest Statement:

The author declares that the case report was conducted in
the absence of any commercial or financial relationships
that could be construed as a potential conflict of interest.
Copyright © 2022 NMSJ. All rights reserved.

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