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International Journal of Surgery Case Reports 109 (2023) 108599

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International Journal of Surgery Case Reports


journal homepage: www.elsevier.com/locate/ijscr

Case report

Small intestinal perforation secondary to metastasis from skin squamous


cell carcinoma: A case report and literature review
Maxence Emmanuel Reynard, Titika-Marina Strati, Bernhard Egger *
Department of Surgery, HFR Fribourg – Cantonal Hospital, Chemin des Pensionnats 2-6, 1752 Villars-sur-Glâne, Switzerland

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction and importance: Primary and metastatic carcinoma of the small intestine are rare. While most of these
Intestinal perforation malignancies are adenocarcinomas, squamous cell carcinoma (SCC) of the gastrointestinal tract is uncommon.
Metastatic squamous cell carcinoma We present a case report of a rare occurrence of skin SCC metastasizing to the ileum, highlighting diagnostic
Diagnostic challenges
challenges and clinical implications.
Clinical implications
Case presentation: An 83-year-old female had a history of cutaneous SCC excision in the right temporal region two
Literature review
Case report years prior to the current emergency department visit, followed by metastatic recurrence in a right intra-parotid
lymph node treated with radiotherapy. The patient exhibited septic shock and an acute abdomen, and an
abdominal computed tomography scan revealed signs of intestinal perforation. Emergency exploratory lapa­
rotomy confirmed purulent peritonitis and perforation of the terminal ileum. Subsequently, a 20 cm intestinal
resection was performed. Histopathological examination of the resected specimen revealed a 4 cm perforated
SCC of the small intestine (pT4 pN0 L0 V1 Pn0 R0).
Clinical discussion: Metastases of the small intestine are rare. The primary sites for these metastases are typically
the uterus, cervix, colon, lung, breast, or melanoma. SCC of the small intestine is particularly rare and poses
challenges in diagnosis owing to non-specific symptoms. The prognosis for SCC of the small intestine is generally
poor, and the potentially aggressive behavior of some skin SCC emphasizes the need for increased awareness and
vigilance in managing such cases.
Conclusion: This case report underscores the importance of considering metastatic disease in the small bowel of
patients with a history of skin SCC who present with new-onset abdominal symptoms.

1. Introduction tumor (GIST), adenocarcinoma, and metastatic carcinomas of the hy­


popharynx, cervix, and lung [2].
Intestinal perforation is a life-threatening condition that is Primary and metastatic squamous cell carcinomas (SCC) of the small
commonly encountered in visceral surgery. The primary treatment intestine are extremely rare. Fewer than 30 cases of primary SCC of the
approach involves emergency surgery, including damage control sur­ small intestine have been reported in the literature. Although there are
gery or intestinal resection with definitive repair. The leading causes of few references of SCC metastasis in the small intestine, only in four case
perforation in industrialized countries are bowel obstruction and in­ reports, the skin is the primary site [3–6].
flammatory bowel disease whereas infectious causes of intestinal per­ Cutaneous squamous cell carcinoma (cSCC) is the second most
forations are more common in developing countries [1]. Early prevalent form of skin cancer after basal cell carcinoma (BCC) [7]. Early
recognition and prompt treatment are crucial to prevent the high mor­ diagnosis and treatment of cSCC generally lead to an excellent prognosis
tality rates associated with peritonitis. The most common malignancies [8]. A recently published epidemiologic cohort study suggested that the
found after histopathologic examination of perforated small bowel incidence rate of cSCC continues to increase, especially among female
specimens include lymphoma, leiomyosarcoma, gastrointestinal stromal patients. Furthermore, the occurrence of multiple cSCCs in a single

Abbreviation: cSCC, cutaneous squamous cell carcinoma; BCC, basal cell carcinoma; INR, International Normalized Ratio; aPTT, Activated Partial Thromboplastin
Time; KDIGO, Kidney Disease Improving Global Outcomes; SCARE, Surgical Case Report; GIST, Gastrointestinal Stromal Tumor; ENT, Ear, Nose, and Throat; PET,
Positron emission tomography; PPI, Proton pump inhibitors; P16, cyclin-dependent kinase inhibitor 2A.
* Corresponding author at: HFR Fribourg – Cantonal Hospital, Chemin des Pensionnats 2-6, 1752 Villars-sur-Glâne, Switzerland.
E-mail address: bernhard.egger@h-fr.ch (B. Egger).

https://doi.org/10.1016/j.ijscr.2023.108599
Received 23 June 2023; Received in revised form 25 July 2023; Accepted 27 July 2023
Available online 2 August 2023
2210-2612/© 2023 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
M.E. Reynard et al. International Journal of Surgery Case Reports 109 (2023) 108599

patient significantly contributes to the current and future burden on intraoperative hemodynamic instability and considering the patient’s
dermatologic healthcare, which requires additional resources for diag­ comorbidities, medication, and suspected malignancy, a damage control
nosis and treatment [9]. surgical approach was decided, and the two intestinal stumps were left
Herein, we present an extremely rare case of a patient admitted to closed intra-abdominal. A second-look laparotomy was scheduled
Cantonal Swiss Hospital in an emergency setting with ileum perforation, within 24–48 h.
identified as a metastasis of cSCC through histopathological examina­ Small bowel tissue was sent for histological analysis and reported an
tion. The case report was conducted in accordance with the SCARE approximately 4 cm (~ 1.5 in) outbreak of a non-keratinizing squamous
(Surgical CAse REport) criteria [10]. cell carcinoma, acantholytic variant, P16[− ], in the small intestinal
wall, which was perforated and associated with fibrinoleukocytic peri­
2. Case presentation tonitis. Other findings included the presence of venous invasion and one
lymph node identified in the mesenteric adipose tissue without
An 83-year-old woman was transported to the Emergency Depart­ malignancy.
ment by ambulance because of worsening abdominal pain, hema­ Blood cultures obtained before surgery were positive for Klebsiella
tochezia, and asthenia that had been present for several weeks. The pneumoniae. The peritoneal fluid collected during the operation showed
detailed history revealed one episode of hematochezia two days before positive results for Klebsiella pneumoniae, Escherichia coli and some spe­
admission and abdominal pain for the last 5 days. cies of Bacteroides, such as Bacteroides sp., Bacteroides thetaiotaomicron,
Her personal medical history was marked 2 years prior to the current and Bacteroides vulgatus.
emergency department visit by a total excision of cutaneous squamous The patient was admitted postoperatively to the Intensive Care Unit.
cell carcinoma (cSCC) in the right frontotemporal region, staged as pT1 She suffered from severe septic shock and underwent aggressive fluid
uN0 cM0 G2. The tumor extended into the subcutis, the greatest vertical resuscitation, continuous norepinephrine support up to 0.5 micrograms/
tumor thickness was 5 mm, the lateral extension <2 cm and the excision kg/min., broad-spectrum antibiotic (Piperacillin-Tazobactam followed
margin >5 mm (R0). One year later, she presented with a recurrence of by Meropenem) and corticoid administration. Despite this treatment,
squamous cell carcinoma in the metastatic form of the right parotid the patient remained in shock, requiring high norepinephrine doses.
gland. Radiation therapy was initiated and was well tolerated. Subse­ Because of the severity of the illness and the expressed wishes of the
quent sonography and CT follow-up revealed a persistent heterogeneous patient, as reported by her relatives, it was decided not to pursue further
lesion within the right lobe of the right parotid gland, which was sus­ treatment, and a second-look laparotomy was not performed. The pa­
pected of either recurrence or post-radiation alterations. Furthermore, a tient peacefully died shortly thereafter.
rounded hypodense structure of 3 cm in the pelvic cavity and in contact
with the small intestine showed in the CT scan was difficult to interpret 3. Discussion
because of artifacts due to prosthetic material of the right hip. No further
investigations were conducted regarding these findings, and a clinical Primary and metastatic carcinomas of the small bowel are rare, with
and radiological follow-up of 3 months was decided. Chronological an incidence of approximately 2 in 100,000 individuals [11]. Metastases
events associated with this disease are summarized in Fig. 1. account for approximately 10 % of cases, most often arising from pri­
The patient also had a history of insulin-dependent type 2 diabetes, mary tumors in the uterus, cervix, colon, breast, lung, or melanoma. In
hypertension, rhythmic and valvular heart disease, chronic renal failure most cases, primary malignancies are adenocarcinomas [12]. Squamous
(KDIGO stage G3a), and polymyalgia rheumatica. Her surgical history cell carcinoma (SCC) of the small bowel, either primary or metastatic, is
included cholecystectomy for symptomatic cholelithiasis, hysterectomy rare. Interestingly, only four case reports have documented skin as the
for heavy monthly bleeding, oophorectomy for a benign ovarian cyst, primary site of secondary tumors of the small intestine [3–6]. They are
and minor orthopedic surgeries. summarized in Table 1.
Her current medication consisted of metoprolol, apixaban, and Cutaneous squamous cell carcinoma (cSCC) is characterized by the
prednisone 5 mg once daily. abnormal and accelerated growth of squamous cells [7,8]. Although in
In the clinical setting, the patient’s initial vital signs revealed a low the greater part it has a good prognosis and low rates of metastasis, there
blood pressure of 85/35 mmHg in both arms, and a regular pulse rate of are certain risk factors and features associated with more aggressive
66 beats per minute. Physical examination revealed guarding and behavior [7]. Gastrointestinal involvement in cSCC is extremely rare
rebound tenderness of the lower abdomen. The digital rectal examina­ and metastasis is believed to occur primarily via lymphatic spread.
tion was unremarkable, with an empty rectal ampulla, and no blood was Secondary lesions found in the small bowel indicate an advanced stage
found. and poor prognosis. Furthermore, when they manifest with a compli­
Laboratory tests showed a plasma creatinine level of 240 umol/l, cated course such as perforation, mortality is even higher [13,14].
white blood cell count of 15 G/l, hemoglobin level of 86 g/l, C-reactive Overall, small-bowel malignancies have a poor prognosis, partly due
protein level of 68 mg/l and hyperlactatemia of 4.5 mmol/l. to frequent delays in diagnosis, as symptoms are mainly vague and
An abdominal CT scan without contrast revealed numerous extra­ nonspecific. Acute presentations vary, including obstruction, intussus­
luminal air bubbles, fat stranding opposite the right colonic angle, and ception, bleeding, or perforation. Furthermore, even under the suspicion
intra-abdominal free fluid, compatible with intestinal perforation of a secondary small-bowel lesion, radiologic detection is difficult.
(Fig. 2). The location of this lesion corresponded to that identified on the Different modalities, such as small-bowel endoscopy, contrast studies,
CT scan performed three months earlier after the completion of radio­ and CT scans, can be helpful. However, only 18F-2-fluoro-2-deoxy-D-
therapy (Fig. 3). glucose-positron emission tomography (FDG PET)/CT and capsule
The patient was promptly resuscitated with fluids and received high- endoscopy have shown some early success in the diagnosis of small-
dose proton pump inhibitor (PPI) therapy (80 mg bolus followed by 8 bowel metastatic tumors [4,15].
mg/h) and antibiotic treatment (piperacillin-tazobactam). Blood cul­ In cases of primary or secondary SCC bowel lesions, surgical treat­
tures were collected for further analyses. ment is a validated approach and may be unavoidable in emergencies.
An emergency exploratory laparotomy revealed generalized puru­ Wide local surgical resection of the tumor and lymph node-bearing
lent peritonitis. Approximately 20 cm from the ileocecal valve, a short mesentery could be considered the surgical treatment of choice. How­
loop of the ileum with a 4 cm perforation in the middle was identified, ever, in the case of secondary lesions or in an emergency setting, which
surrounded by local fecal peritonitis (Fig. 4). Abdominal exploration corresponds to an advanced stage disease, surgical treatment has a
revealed no macroscopic peritoneal or hepatic metastases. Segmental palliative role, and complex resections are not recommended. In both
resection of the affected ileum (20 cm) was performed. Due to cases, the outcome was poor.

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M.E. Reynard et al. International Journal of Surgery Case Reports 109 (2023) 108599

Fig. 1. Course of events related to cSCC.

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M.E. Reynard et al. International Journal of Surgery Case Reports 109 (2023) 108599

Fig. 2. Subject to hardening artifacts of the right hip prosthesis, presence of a pelvic mass, with foamy content and suspicion of fistula with a small bowel loop (white
arrow) on CT-Scan.

Although the majority of patients with cSCC present an excellent screening and investigation, even in the presence of nonspecific and rare
prognosis after adequate surgical resection, a subset of patients carry an findings in otherwise nonaggressive tumors that rarely metastasize to
increased risk of lymph node metastasis, local recurrence, and disease- the abdominal cavity.
specific death [7,9,14]. High-risk features include tumor diameter ≥ There are only four case reports in the literature concerning sec­
2 cm, tumor thickness ≥ 2 mm, anatomic tumor site (face, ear, pre/post ondary cutaneous SCC of the small bowel [3–6]. Known as a disease of
auricular area, genitalia, hands, and feet), poor histological differenti­ late adulthood, with the exception of appearance at a younger age in
ation, certain histological subtypes, evidence of perineural invasion, immunocompromised patients, in these four case reports, the youngest
immunosuppression, and the presence of parotid metastases [3,7,16]. patient was 53 years old and the oldest was 71 years old. It is of great
In our case and in concordance with the literature, our patient pre­ interest that, as in our case, three of these four patients were treated
sented with an aggressive form of cSCC, the acantholytic variant, which surgically in an emergency setting without their metastatic small bowel
had already led to parotid and level II lymph node metastases soon after SCC being already known. Bowel perforation was the cause in two cases
the primary diagnosis and surgical treatment. Furthermore, immuno­ and obstruction in the one. In the last case, the small bowel lesion was
suppression, with great mention to transplant patients, should always be incidentally found preoperatively during elective sigmoid resection
considered, presenting a higher risk of both cSCC development and a because of an already proven cSCC metastasis in a colonic diverticulum.
worse disease course [2,10]. Our patient was in long-term medical However, in one of these four cases, perforated metastatic small bowel
treatment with corticoids because of rheumatic polymyalgia, probably SCC was the first acute presentation of the disease, resulting in further
contributing to a more aggressive tumor behavior but also influencing to investigations and subsequent detection of the primary cutaneous site.
a certain degree, along with the patient’s hemodynamic instability, Of the four cases, only one small bowel metastasis was located in the
emergency surgical treatment, and the decision to perform a damage ileum, while all three others were identified in the jejunum. All patients
control surgery without proceeding to anastomosis creation. were treated surgically and small bowel resection was performed. In
Finally, we would like to outline the difficulty in detecting these three case reports, the outcome was poor, with death occurring 8 weeks
secondary small bowel lesions, firstly because of their rarity and because to 7 months post-operatively. In only one case, the last follow-up, six
they lack specific characteristics, they often remain unidentifiable until months after the operation, did not show any other metastatic disease or
they reach an advanced stage or become complicated [12]. In our case, recurrence. All four case reports stress the rarity of metastatic small
the diagnosis of metastatic cSCC was confirmed only after pathological bowel cSCC and their presentation usually in an emergency setting with
examination results were obtained. Additionally, it is noteworthy that a poor prognosis.
previous follow-up CT scan described a round hypodense structure of
approximately 3 cm in the pelvic area, in contact with the small bowel, 4. Conclusion
but it was not interpreted due to artifacts from the prosthetic material in
the right hip (Fig. 3). The examination was carried out in a patient Most cases of cSCC are limited to the skin and are successfully
believed to have a total response to radiotherapy. These facts, together managed with local therapy. However, distant metastases occur with an
with the rarity of a small intestinal metastasis from a cSCC, did not lead annual incidence in up to 4 %, leading to increased mortality [7].
to further investigations. It further highlights the challenges in diag­ Therefore, early diagnosis and detection are important. However, non-
nosing these lesions and raises questions about the need for further specific symptoms and imaging findings, as well as the lack of

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M.E. Reynard et al. International Journal of Surgery Case Reports 109 (2023) 108599

Fig. 3. CT-Scan of a round hypodense structure (~3 cm) in the pelvic area (white arrow), in contact with the small bowel, subject to artifacts from the pros­
thetic material.

awareness of this very low but existing proportion of tumors, result in a Ethics Committee(s) or Institutional Board(s).
considerable delay in diagnosis and a poor outcome. Surgeons should be Our institution does not require ethical approval for reporting indi­
alerted in cases of acute abdominal pain or no specific abdominal vidual cases or case series.
symptoms when there is a history of cancer, even in cases where the
primary tumor rarely metastasizes in the gastrointestinal tract. Funding
However, as the incidence of cSCC continues to increase [8,9], these
rare metastatic sites might also become more apparent, making aware­ All authors have not declared a specific grant for this research from
ness of this entity even more important. any funding agency in the public, commercial or not-for-profit sectors.
Increased awareness, vigilance, and comprehensive evaluation are
key to identifying this subset of cases and providing timely interventions Guarantor
for improved patient management, in light of the growing prevalence of
SCC. The guarantor for this statement is Dr. med. Maxence Emmanuel
Reynard. He can be reached at any time under reynard.maxence@gmail.
Consent com.

Written informed consent was obtained from the patient for publi­ Registration of research studies
cation of this case report and accompanying images. A copy of the
written consent is available for review by the Editor-in-Chief of this This study is not a “First in Man” study.
journal on request.
CRediT authorship contribution statement
Ethical approval
All authors contributed to the design, data collection, analysis and
This study involves human participants but was not approved by an interpretation of this protocol. All authors contributed read and

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M.E. Reynard et al. International Journal of Surgery Case Reports 109 (2023) 108599

Fig. 4. Ileal perforation.

Table 1
Overview of the reviewed sources.
Author Year Patient demography Primary localisation Metastasis localisation Complication Outcome

N. Hitchen et al. 2016 70-year-old woman Back Jejunum Perforation with peritonitis Death 8 weeks after surgery
R. Li et al. 2014 71-year-old man Hand Ileum Small bowel obstructions Death 4 months after surgery
B. Schwartz et al. 2016 65-year-old male Chest Jejunum None n.e.
H. Zheng et al. 2010 53-year-old man Scalp Jejunum Perforation with peritonitis Death 7 months after surgery

approved the final manuscript. identification is low.

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