A rare cause of intestinal obstruction in children: signet-ring cell adenocarcinoma of the colon

Erginel, Basak; Mustafayeva, Naila; Karada�, �etin ali; Yanar, Fatih; Kebudi, Rejin; Tanyildiz, Hikmet Gulsah; Tugcu, Deniz; Berker, Neslihan; Ilhan, Burak; G�n Soysal, Feryal

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A rare cause of intestinal obstruction in children: signet-ring cell adenocarcinoma of the colon [Ulus Travma Acil Cerrahi Derg]

Ulus Travma Acil Cerrahi Derg. 2023; 29(7): 798-805 | DOI: 10.14744/tjtes.2023.64257

A rare cause of intestinal obstruction in children: signet-ring cell adenocarcinoma of the colon

Basak Erginel¹, Naila Mustafayeva¹, �etin ali Karada�², Fatih Yanar³, Rejin Kebudi⁴, Hikmet Gulsah Tanyildiz⁵, Deniz Tugcu⁵, Neslihan Berker⁶, Burak Ilhan³, Feryal G�n Soysal¹
¹Department of Pediatric Surgery, Istanbul University, Istanbul Medical Faculty, �stanbul-T�rkiye
²Department of Pediatric Surgery, Sisli Hamidiye Etfal Training and Research Hospital, �stanbul-T�rkiye
³Department of General Surgery, Istanbul University, Istanbul Medical Faculty, �stanbul-T�rkiye
⁴Department of Pediatric Hematology-Oncology, �stanbul University, Oncology Institute, �stanbul-T�rkiye
⁵Department of Pediatric Hematology-Oncology, �stanbul University, �stanbul-T�rkiye
⁶Department of Pathology, �stanbul University, �stanbul Medical Faculty, �stanbul-T�rkiye

BACKGROUND: Signet-ring cell adenocarcinoma of the colon is well-recognized in adult patients who are extremely rare and not well-documented in children. Our study aims to raise awareness about this rare disease and its long-term outcomes.
METHODS: We retrospectively evaluated patients with signet-ring cell colon adenocarcinoma.
RESULTS: Six patients, three boys and three girls, with a mean age of 14.83 (range, 13�17 years), presented with signs of intesti-nal obstruction and were diagnosed with signet-ring cell colon adenocarcinoma. All patients had air-fluid levels on abdominal X-ray. Abdominal ultrasonography of all patients revealed subileus. Abdominal computed tomography was performed in five patients, and pre-operative colonoscopy was conducted in two patients before the emergency intervention. All of the patients underwent emergent exploratory laparotomy with the preliminary diagnosis of acute abdomen. In two patients, debulking surgery followed by a stoma was performed. The remaining four patients were treated with anastomosis following intestinal resection. All girls had metastases on the ovary. One of the patients died due to the burden of multiple metastases in the early period, and three died in the sixth post-operative year. We have been following the remaining two patients since then.
CONCLUSION: Although signet-ring cell carcinomas (SRCCs) are rare, they should be considered in the differential diagnosis of acute abdomen and intestinal obstruction in pediatric patients. Despite early diagnosis and treatment, SRCC has a poor prognosis in the pediatric population.

Keywords: Acute abdomen, pediatric surgery; signet-ring cell adenocarcinoma.

�ocuklarda nadir bir ba��rsak t�kan�kl�� nedeni: Kolon ta�l� y�z�k h�creli adenokarsinom

Basak Erginel¹, Naila Mustafayeva¹, �etin ali Karada�², Fatih Yanar³, Rejin Kebudi⁴, Hikmet Gulsah Tanyildiz⁵, Deniz Tugcu⁵, Neslihan Berker⁶, Burak Ilhan³, Feryal G�n Soysal¹
¹�stanbul �niversitesi, �stanbul T�p Fak�ltesi, �ocuk Cerrahisi Anabilim Dal�, �stanbul, T�rkiye
²�i�li Hamidiye Etfal E�itim ve Ara�t�rma Hastanesi, �ocuk Cerrahisi Anabilim Dal�, �stanbul, T�rkiye
³�stanbul �niversitesi, �stanbul T�p Fak�ltesi, Genel Cerrahi Anabilim Dal�, �stanbul, T�rkiye
⁴�stanbul �niversitesi, �stanbul T�p Fak�ltesi Onkoloji Enstit�s�, Pediatrik Hematoloji-Onkoloji B�l�m�, �stanbul, T�rkiye
⁵�stanbul �niversitesi, �stanbul T�p Fak�ltesi, Pediatrik Hematoloji-Onkoloji Anabilim Dal�, �stanbul, T�rkiye
⁶�stanbul �niversitesi, �stanbul T�p Fak�ltesi, Patoloji Anabilim Dal�, �stanbul, T�rkiye

AMA�: Kolonun ta�l� y�z�k h�creli adenokarsinomu yeti�kin hastalarda iyi tan�nmaktad�r, ancak son derece nadirdir ve �ocuklarda o kadar iyi belgelenmemi�tir. �al��mam�z bu nadir hastal�k ve uzun vadeli sonu�lar� hakk�nda fark�ndal�k yaratmay� ama�lamaktad�r.
GERE� VE Y�NTEM: Ta�l� y�z�k h�creli kolon adenokarsinomlu hastalar�m�z� retrospektif olarak inceledik.
BULGULAR: Barsak obstr�ksiyonu bulgular� ile ba�vuran ve ta�l� y�z�k h�creli kolon adenokarsinomu tan�s� alan ortalama ya�lar� 14.83 (da��l�m 13-17) olan �� erkek ve �� k�z olmak �zere alt� hastam�z vard�. T�m hastalar�n kar�n grafisinde hava-s�v� seviyeleri mevcuttu. Abdominal ultrasonog-rafide t�m hastalar�n subileus saptand�. Acil giri�im �ncesinde be� hastaya kar�n bilgisayarl� tomografisi, iki hastaya preoperatif kolonoskopi yap�ld�. Hastalar�n tamam�na akut kar�n �n tan�s� ile acil eksploratuvar laparotomi uyguland�. �ki hastada kitle k��ltme ameliyat� ard�ndan stoma a��ld�. Kalan d�rt hasta intestinal rezeksiyon sonras� anastomoz ile tedavi edildi. T�m k�zlar�n yumurtal�klar�nda metastaz vard�. Hastalardan biri erken d�nemde multipl metastaz y�k� nedeniyle, �� ise postoperatif alt�nc� y�lda kaybedildi. O zamandan beri kalan iki hastay� takip etmekteyiz.
TARTI�MA: Ta�l� y�z�k h�creli karsinomlar nadir g�r�lmekle birlikte akut kar�n ve barsak obstr�ksiyonu olan pediatrik hastalar�n ay�r�c� tan�s�nda ak�lda tutulmal�d�r. Erken tan� ve tedaviye ra�men pediatrik pop�lasyonda prognozu k�t�d�r.

Anahtar Kelimeler: Akut kar�n, �ocuk cerrahisi; ta�l� y�z�k h�creli karsinom.

Corresponding Author: Basak Erginel, T�rkiye
Manuscript Language: English

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A rare cause of intestinal obstruction in children: signet-ring cell adenocarcinoma of the colon

�ocuklarda nadir bir ba��rsak t�kan�kl��� nedeni: Kolon ta�l� y�z�k h�creli adenokarsinom

�ocuklarda nadir bir ba��rsak t�kan�kl�� nedeni: Kolon ta�l� y�z�k h�creli adenokarsinom