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Appendiceal Endometriosis As a Rare Cause of Abdominal Pain – a Case Report and Literature Review

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Appendiceal Endometriosis As a Rare Cause of Abdominal Pain – a Case Report and Literature Review Case Report Appendiceal endometriosis as a rare cause of abdominal pain – a case report and literature review Rafael Denadai1, Rafael Aliceda Ferraz2, Ricardo de Álvares Goulart3, Rogério Saad-Hossne4, Fábio Vieira Teixeira5 1Resident, General Surgery, Department of Surgery at the Hospital Municipal Dr. Mário Gatti – Campinas (SP), Brazil. 2Resident, General Surgery at the Santa Casa de Ourinhos – Ourinhos (SP), Brazil. 3Physician at GASTROSAUDE Clinic – Marília (SP), Brazil. 4Full Member of the Sociedade Brasileira de Coloproctologia; Assistant Professor, Doctor, Department of Surgery and Orthopedics, School of Medicine at the Universidade Estadual Paulista “Júlio de Mesquita Filho” (UNESP) Botucatu – Botucatu (SP), Brazil. 5Guest Professor, Doctor, Department of Surgery and Orthopedics, School of Medicine at the UNESP Botucatu – Botucatu (SP); Physicina at GASTROSAUDE Clinic – Marília (SP), Brazil. Denadai R, Ferraz RA, Goulart RDÁ, Saad-Hossne R, Teixeira FV. Appendiceal endometriosis as a rare cause of abdominal pain – a case report and literature review. J Coloproctol, 2012;32(3): 324-328. ABSTRACT: Endometriosis is an estrogen-dependent inflammatory disease, common in young women, characterized by the presence of endo- metrial tissue outside the uterine cavity. This ectopic endometrial tissue is most commonly found in the ovaries, peritoneum, uterosacral ligaments and rectovaginal cul-de-sac, with extremely rare involvement of the appendix. The main symptom is chronic abdominal pain, and the diagnosis is often made later, after the result of the histopathological examination. This study reports a 34-year-old patient complaining of chronic pelvic pain refractory to medical treatment, having undergone diagnostic laparotomy. During the surgery, we observed the presence of endometrioma fixed to the uterine wall, and the appendix was enlarged, but without evidence of inflammation. Endometrioma resection and appendectomy were performed, with good postoperative recovery. The anatomopathological exam showed endometriosis in the cecal appendix. Keywords: endometriosis; gastrointestinal tract; appendix; abdominal pain; appendectomy. RESUMO: Endometriose é uma doença inflamatória estrogênio-dependente frequente em mulheres jovens, caracterizada pela presença de te- cido endometrial fora da cavidade uterina. Esse tecido ectópico de endométrio é mais comumente encontrado nos ovários, peritônio, ligamentos uterossacros e fundo de saco retovaginal, sendo o acometimento do apêndice cecal extremamente raro. O quadro clínico predominante é o de dor abdominal crônica, sendo muitas vezes o diagnóstico feito posteriormente, após o resultado do anatomopatológico. Relatamos o caso de uma pa- ciente de 34 anos com queixa de dor pélvica crônica, refratária ao tratamento clínico, tendo sido submetida à laparotomia exploradora diagnóstica. Durante o ato cirúrgico, observamos a presença de endometrioma fixo à parede uterina, bem como apêndice cecal aumentado de volume, porém sem evidência de sinais flogísticos. Procedeu-se à ressecção do endometrioma e apendicectomia, com boa evolução pós-operatória. O resultado do exame anatomopatológico revelou a presença de endometriose no apêndice cecal. Palavras-chave: endometriose; trato gastrointestinal; apêndice; dor abdominal; apendicectomia. INTRODUCTION It affects the gastrointestinal tract (GIT) around 12% of the cases 4, but the involvement of the cecal appen- Endometriosis is an estrogen-dependent inflam- dix is rare,4,6,7-10. matory disease that affects 5 to 10% of women at re- When the appendix is affected, symptoms may productive age1, characterized by the presence of en- vary from acute abdominal pain (simulating appendi- dometrial tissue outside the uterine cavity2-6. It is most citis)3,4, to chronic (significant reduction in the quality commonly found in the peritoneum and the ovaries5,7. of life)2,5,7,9-11 or even asymptomatic abdominal pain3,5. Study carried out at the Service of Coloproctology and Digestive System Surgery, at GASTROSAUDE Clinic – Marília (SP), Brazil. Financing source: none. Conflict of interest: nothing to declare. Submitted on 02/12/2010 Accepted on 03/24/2010 324 J Coloproctol Appendiceal endometriosis as a rare cause of abdominal pain – a case report and literature review Vol. 32 July/September, 2012 Rafael Denadai et al. Nº 3 Regarding the diagnostic investigation, en- At the physical examination, she had regular dorectal ultrasound and laparoscopy can add im- general health, normal vital signs, no fever, flat ab- portant information when patients present with un- domen, presence of bowel sounds, pain at deep pal- explainable abdominal pain6,9,12. Typical treatment pation of the hypogastrium and right iliac fossa and consists of suppressed ovulation and, if required, absence of peritoneal signs. surgical excision1,7. At vaginal exam, the patient had anteverted uter- In cases of appendix involvement, intraoper- us, fibroelastic colon, intense pain at uterine mobiliza- ative inspection may not show any alteration that tion and at palpation of posterior and right lateral cul- suggests endometriosis3,9,11. Then, the definitive di- de-sac; non-palpable attachments. She was submitted agnosis is confirmed through anatomopathological to abdominal ultrasound, which showed normal at- analysis3,6,7,9,11,12. tachments, normal size of the uterus, with a small Some authors describe patients with abdominal quantity of free fluid in the cavity. pain who, after undergoing appendectomy, presented Based on that, the treatment choice was hos- improvements and their abdominal symptoms disap- pitalization and conservative treatment with anal- peared3,4,6,9-11,13. However, incidental appendectomy gesics, anti-inflammatory medication and intrave- during the surgical treatment of pelvic endometriosis nous hydration. After two days, the pain increased is a controversial theme9. and, although the patient had no peritoneal signs, Based on these considerations, this study reports exploratory laparotomy was performed. The find- a rare case of appendiceal endometriosis in a woman ings were: presence of a small amount of blood in with chronic pelvic pain. the pelvic region, endometrioma measuring around 1.0 cm in diameter fixed to the lateral wall of the CASE REPORT uterus and enlarged cecal appendix, but no signs of inflammation. Endometrioma resection and appen- A 34-year-old female patient, with history of dectomy were performed. chronic pelvic pain related to the menstrual period The patient did well postoperatively, and was for one year, with worsening of symptons in the last discharged from the hospital without symptoms on the four months. She came to our service with intense second day after the surgery. The anatomopathologi- pain in hypogastric region and right iliac fossa for cal exam concluded that there was an endometrioma seven days. of the uterus and appendiceal endometriosis (Figure). A B Figure. Photomicrograph of cecal appendix. (A) Mucosa of the appendix without alterations. (B) Presence of endometrial tissue in the middle of the tunica muscularis of the appendicular wall (hematoxylin and eosin staining, enlarged 40x). 325 J Coloproctol Appendiceal endometriosis as a rare cause of abdominal pain – a case report and literature review Vol. 32 July/September, 2012 Rafael Denadai et al. Nº 3 DISCUSSION peritoneal pseudomyxoma10, intussusception5, perfo- ration4,10 and intestinal bleeding3. Endometriosis is defined as the ectopic pres- The patients rarely present with acute appendici- ence of endometrial tissue outside the uterine cav- tis4,7. Appendiceal endometriosis corresponds to less ity2-6. There is no consensus regarding its histological than 1% of the causes of acute appendicitis7. In a study origin1,4,5. It may be inherited, as the disease preva- that analyzed the cecal appendix histology of 1,225 lence in women related to affected females is seven patients submitted to surgical treatment with clinical times greater if compared to women without family diagnosis of acute appendicitis, only 0.25% of the an- history1. Histologically, endometriosis is character- alyzed specimens presented microscopic diagnosis of ized by the ectopic presence of an endometrial stroma, endometriosis17. chronic hemorrhage and signs of inflammation, and it Thus, these findings agree with results of other may occur separately or in combination with other af- studies that appendiceal endometriosis is more related fections1,7. This inflammation may cause pelvic nerve to the diagnosis of chronic pelvic pain2,3,5-7,9-11,15 usu- damage and consequent pain1. It is usually found dur- ally associated with pelvic endometriosis3,11, and not ing explorations in the abdominal cavity due to pelvic to acute pain4,7. pain, pelvic mass or infertility7. Therefore, the appendix is an important organ Prevalence rates vary according to the inves- in the evaluation of non-diagnosed chronic pelvic tigated population, ranging from 0.7 to 45% in as- pain7,6,11. However, only 41% of the patients with ap- ymptomatic women; 20 to 40% in infertile women; pendiceal endometriosis complain of intermittent pain 6 to 18% in women submitted to sterilization and 15 in the right lower quadrant7, which may – or may not – to 70% in patients with chronic abdominal pain14. In be related to menstruation2,3. general, the disease affects around 5 to 10% of wom- Preoperative diagnosis of appendiceal endome- en, with annual incidence of 1.9 cases among 1,000 triosis is uncommon3,18. It is frequently found during women, and aged between 15 and
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