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Deciduosis of the Appendix Manifesting as Acute Abdomen in Pregnancy

Deciduosis of the Appendix Manifesting as Acute Abdomen in Pregnancy 10.2478/chilat-2014-0110 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) CASE REPORT Deciduosis of the Appendix Manifesting as Acute Abdomen in Pregnancy Anita Balta, Maija Lubgane, Ineta Orube, Guna Ziemele, Andrejs Vanags, Janis Gardovskis, Ilze Strumfa Riga Stradins University, Riga, Latvia SUMMARY Acute appendicitis is one of the most common surgical diseases during pregnancy. In rare cases, ectopic decidual tissues (deciduosis) can develop in the appendix and cause inflammation. Here we report a well-documented case of appendicular deciduosis presenting with acute abdominal pain and resulting in gangrenous appendicitis. Key words: appendicitis, pregnancy, deciduosis preterm delivery, cancer, acute pyelonephritis or AIM OF THE DEMONSTRATION In order to increase the awareness of rare appendicular rupture of ovarian cyst. However, considering the intraoperative surgical findings as well as morphological diseases and the peculiar differential diagnosis of appendicitis in pregnancy, here we present a rare case and IHC data, the final diagnosis was appendicular of appendicular deciduosis causing acute appendicitis in deciduosis, complicated by gangrenous appendicitis and a pregnant lady. phlegmonous periappendicitis and mesenteriolitis. CASE REPORT DISCUSSION A 33-year-old primigravida woman entered the hospital Development of decidual cells outside the endometrium, in 28/29 weeks of gestation. The patient reported right first described by Walker in 1887, is named ectopic lower abdominal pain lasting for 11 hours. Initially, decidua or deciduosis (Walker, 1887; Bolat et al., 2012). preterm delivery was suspected. The body temperature Such changes most commonly affect ovaries, uterine was 37.2 C0. The white blood cell count was 16.7x10 serosa (Kondoh et al., 2012), fallopian tubes and cervix /L (laboratory reference interval (LRI) 4 – 10). The level (Bolat et al., 2012). Deciduosis is less frequently seen in of C-reactive protein (CRP) reached 27.4 mg/L (LRI 0 – the appendix (Adhikari and Shen, 2013), diaphragm, 5). The abdominal ultrasonography showed picture of liver, spleen, paraaortic and pelvic lymph nodes or renal acute appendicitis as the appendix had thickened wall pelvis (Bolat et al., 2012). The involvement of omentum and was surrounded by a small amount of liquid. Gross is considered rare by some authors (Adhikari and Shen, intraoperative findings were suggestive of gangrenous 2013), while others suggest that it could be disclosed appendicitis. Conventional appendectomy was frequently, if careful sampling would be possible performed. The postoperative period was uneventful. (Buttner et al., 1993; Rodriguez et al., 2006). Deciduosis The patient received analgetic and antibiotic therapy. can be found incidentally in tissues removed or Fetal movements were monitored and sensed well. biopsied during a caesarean section, treatment of tubal Uterus was normotonic. There was no abdominal pregnancy, elective tubal ligation and appendectomy pain except sensitivity around the surgical wound by (Bolat et al., 2012). palpation. By pathologic examination, the removed The pathogenesis of deciduosis is not yet fully appendix grossly measured 6 x 1.2 x 0.8 cm and understood (Kondoh et al., 2012) and the physiologic showed uneven surface. Microscopically, gangrenous nature of this reaction is considered controversial appendicitis was revealed along with perforation and (Bolat et al., 2012). The most frequently suggested wide areas of deciduous ectopic reaction s. deciduosis explanations include de novo development from (Fig.1), characterised by nodules of discohesive submesothelial stroma, or decidual transformation of large polygonal cells with widespread degenerative pre-existing endometriosis (Bolat et al., 2012; Kondoh cytoplasmic vacuolisation resulting in signet ring cell et al., 2012; Adhikari and Shen, 2013). The de novo like appearance. Acute inflammation with fibrinous and pathway would involve progesterone-related subserosal purulent component extended to the periapendicular stromal metaplasia. Confirming the role of hormonal and mesenteriolar tissues. By immunohistochemical influences, deciduosis regresses within 4 – 6 weeks after investigation (IHC), the large polygonal and vacuolated pregnancy along with decidual involution (Bolat et al., cells in pathologic foci expressed progesterone receptors 2012). High level of progesterone in twin gestations and vimentin but lacked pan-cytokeratin and calretinin. has been attributed to diffuse peritoneal deciduosis in Thus, IHC confirmed deciduosis and ruled out such patients (Adhikari and Shen, 2013). Occasionally, malignant tumour. The differential diagnosis over the deciduosis in non-pregnant ladies has been explained by whole course of illness comprised acute appendicitis, adrenal progesterone secretion acting on submesothelial 43 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) stroma sensitized by inflammation or trauma (Buttner 0.13% (Kirshtein et al., 2009; Chung et al., 2013). et al., 1993; Rodriguez et al., 2006). Although deciduosis Thus, appendicitis must be considered as the cause of has been identified in patients lacking any history of acute abdomen during pregnancy. Cases of deciduosis endometriosis (Bolat et al., 2012), increased awareness of manifesting by abdominal pain, leukocytosis and this condition is recommended managing pregnancy in elevated CRP level necessitate even more careful women with history of dysmenorrhea or endometriosis consideration of appendicitis in the differential diagnosis (Kondoh et al., 2012). (Kondoh et al., 2012). The history of the described case Albeit deciduosis frequently lacks clinical symptoms, shows an additional novel, clinically highly important it can manifest by abdominal pain and leukocytosis fact – the appendicitis can also be true complication of that has been related to the production of granulocyte deciduosis. colony-stimulating factor (Kondoh et al., 2012). In conclusion, deciduosis is a rare, pregnancy-related Occasionally, intestinal obstruction (Rodriguez et al., process that in rare cases can affect appendix. In 2006) or significant abdominal or gastrointestinal our patient, gangrenous inflammation supervened haemorrhage can develop (Bashir et al., 1995; Kondoh necessitating urgent appendectomy. Thus, deciduosis et al., 2012). Lethal outcome of deciduosis has been should be considered in the differential diagnosis of previously reported underlining the necessity of correct acute abdomen in pregnancy. diagnosis and timely surgical treatment (Theissig et al., 1988). Conflict of interest: None In contrast to frequently mentioned asymptomatic course, our patient had pain and leukocytosis. In the REFERENCES presented case, these manifestations can be explained 1. Adhikari LJ, Shen R. Florid diffuse peritoneal by severe acute inflammation related to rich presence deciduosis mimicking carcinomatosis in a of neutrophilic leukocytes. The gangrenous tissues were primigravida patient: a case report and review of widely haemorrhagic. However, there was no evidence the literature // Int J Clin Exp Pathol, 2013; 6: of prior primary haemorrhage within the tissues. 2615 – 2619 The clinical diagnosis of deciduosis is difficult even 2. Bashir RM, Motgomery EA, Gupta PK, Nauta RM, in the presence of widespread intraabdominal Crockett SA, Collea JV, al-Kawas FH. Massive involvement (Kondoh et al., 2012). The intraoperative gastrointestinal hemorrhage during pregnancy findings can yield broad differential diagnosis including caused by ectopic decidua of the terminal ileum and primary or metastatic malignant tumours as deciduoid colon // Am J Gastroenterol, 1995; 90:1325 – 1327 mesothelioma, abdominal carcinomatosis or metastatic 3. Bolat F, Canpolat T, Tarim E. Pregnancy- abdominal spread of malignant melanoma (Ustun related peritoneal ectopic decidua (deciduosis): et al., 2011; Adhikari and Shen, 2013). Regarding morphological and clinical evaluation // Turk the differential diagnosis with malignant tumours, Patoloji Derg, 2012; 20:56 – 60 morphological examination of the removed tissues is the 4. Buttner A, Bassler R, Theele C. Pregnancy- “gold standard”. Deciduosis is characterised by nodular associated ectopic decidua (deciduosis) of the architecture. The nodules are composed of large cells with greater omentum. An analysis of 60 biopsies with well-defined cell borders and eosinophilic cytoplasm. cases of fibrosing deciduosis and leiomyomatosis Degenerative changes lead to cytoplasmic vacuolisation, peritonealis disseminata // Pathol Res Pract, 1993; physaliphorous, signet ring or lipoblastic appearance 189:352 – 359 of cells and myxoid stroma. Mitotic activity and/ or 5. Chung JC, Cho GS, Shin EJ, Kim HC, Song OP. nuclear atypia are absent (Rodriguez et al., 2006; Bolat et Clinical outcomes compared between laparoscopic al., 2012). However, even by light microscopy decidual and open appendectomy in pregnant women // change may be mistaken for metastatic carcinoma or Can J Surg, 2013; 56:341 – 346 malignant mesothelioma (Adhikari and Shen, 2013). 6. Kirshtein B, Perry ZH, Avinoach E, Mizrahi S, In such cases, IHC is advisable. Cytoplasmic positivity Lantsberg L. Safety of laparoscopic appendectomy of vimentin and nuclear presence of progesterone during pregnancy // World J Surg, 2009; 33: receptors along with lack of pan-cytokeratin, HMB-45 475 – 480 and calretinin is characteristic for deciduosis. Although 7. Kondoh E, Shimizu M, Kakui K, Mikami Y, Tatsumi the lesions typically are negative for cytokeratin, few K, Konishi I. Deciduosis can cause remarkable positive cells can be present (Bolat et al., 2012; Adhikari leukocytosis and obscure abdominal pain // J Obstet and Shen, 2013). Metastatic carcinoma is characterised Gynaecol Res, 2012; 38:1376 – 1378 by cytokeratin expression, while mesothelioma – by 8. Theissig F, Kemmer C, Kunze KD. Fatal course of calretinin along with pan-cytokeratin, cytokeratin 5/6 mesenteric deciduosis in a gravida I // Pathologe, and vimentin. HMB-45 expression is almost diagnostic 1988; 9:50 – 54 of melanoma (Ustun et al., 2011; Bolat et al., 2012; 9. Ustun H, Astarci HM, Sungu N, Ozdemir A, Adhikari and Shen, 2013). Ekinci C. Primary malignant deciduoid peritoneal Appendectomy is the most common non-obstetric mesothelioma: a report of the cytohistological surgical intervention in pregnant patients. The incidence and immunohistochemical appearances // Diagn of acute appendicitis in pregnancy is estimated as 0.05 – Cytopathol, 2011; 39:402 – 408 44 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) 10. Walker A. Der bau der eihaeute bei graviditatis abdominalis // Virchows Arch Path Anat, 1887; 197:72 – 99 Address: Andrejs Vanags Department of Surgery, Riga Stradins University, Pilsonu Street 13, LV1002, Riga, Latvia E-mail: Andrejs.Vanags@rsu.lv Fig. 1. Appendicular deciduosis. A, Overview of the affected tissue. Note the nodular foci of deciduosis. Haematoxylin-eosin (HE), original magnification (OM) 50x. B, Cell morphology of the decidual foci. Note the intact deciduoid cell (yellow arrow) and the degenerative changes resulting in signet ring cell like appearance (green arrow). HE, OM 400x. C, Expression of progesterone receptors (PR) in deciduoid cells. Immunoperoxidase, anti-PR, OM 100x. D. Purulent inflammation (arrow) surrounding a deciduoid cell (yellow arrow). HE, OM 400x. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Acta Chirurgica Latviensis de Gruyter

Deciduosis of the Appendix Manifesting as Acute Abdomen in Pregnancy

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Abstract

10.2478/chilat-2014-0110 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) CASE REPORT Deciduosis of the Appendix Manifesting as Acute Abdomen in Pregnancy Anita Balta, Maija Lubgane, Ineta Orube, Guna Ziemele, Andrejs Vanags, Janis Gardovskis, Ilze Strumfa Riga Stradins University, Riga, Latvia SUMMARY Acute appendicitis is one of the most common surgical diseases during pregnancy. In rare cases, ectopic decidual tissues (deciduosis) can develop in the appendix and cause inflammation. Here we report a well-documented case of appendicular deciduosis presenting with acute abdominal pain and resulting in gangrenous appendicitis. Key words: appendicitis, pregnancy, deciduosis preterm delivery, cancer, acute pyelonephritis or AIM OF THE DEMONSTRATION In order to increase the awareness of rare appendicular rupture of ovarian cyst. However, considering the intraoperative surgical findings as well as morphological diseases and the peculiar differential diagnosis of appendicitis in pregnancy, here we present a rare case and IHC data, the final diagnosis was appendicular of appendicular deciduosis causing acute appendicitis in deciduosis, complicated by gangrenous appendicitis and a pregnant lady. phlegmonous periappendicitis and mesenteriolitis. CASE REPORT DISCUSSION A 33-year-old primigravida woman entered the hospital Development of decidual cells outside the endometrium, in 28/29 weeks of gestation. The patient reported right first described by Walker in 1887, is named ectopic lower abdominal pain lasting for 11 hours. Initially, decidua or deciduosis (Walker, 1887; Bolat et al., 2012). preterm delivery was suspected. The body temperature Such changes most commonly affect ovaries, uterine was 37.2 C0. The white blood cell count was 16.7x10 serosa (Kondoh et al., 2012), fallopian tubes and cervix /L (laboratory reference interval (LRI) 4 – 10). The level (Bolat et al., 2012). Deciduosis is less frequently seen in of C-reactive protein (CRP) reached 27.4 mg/L (LRI 0 – the appendix (Adhikari and Shen, 2013), diaphragm, 5). The abdominal ultrasonography showed picture of liver, spleen, paraaortic and pelvic lymph nodes or renal acute appendicitis as the appendix had thickened wall pelvis (Bolat et al., 2012). The involvement of omentum and was surrounded by a small amount of liquid. Gross is considered rare by some authors (Adhikari and Shen, intraoperative findings were suggestive of gangrenous 2013), while others suggest that it could be disclosed appendicitis. Conventional appendectomy was frequently, if careful sampling would be possible performed. The postoperative period was uneventful. (Buttner et al., 1993; Rodriguez et al., 2006). Deciduosis The patient received analgetic and antibiotic therapy. can be found incidentally in tissues removed or Fetal movements were monitored and sensed well. biopsied during a caesarean section, treatment of tubal Uterus was normotonic. There was no abdominal pregnancy, elective tubal ligation and appendectomy pain except sensitivity around the surgical wound by (Bolat et al., 2012). palpation. By pathologic examination, the removed The pathogenesis of deciduosis is not yet fully appendix grossly measured 6 x 1.2 x 0.8 cm and understood (Kondoh et al., 2012) and the physiologic showed uneven surface. Microscopically, gangrenous nature of this reaction is considered controversial appendicitis was revealed along with perforation and (Bolat et al., 2012). The most frequently suggested wide areas of deciduous ectopic reaction s. deciduosis explanations include de novo development from (Fig.1), characterised by nodules of discohesive submesothelial stroma, or decidual transformation of large polygonal cells with widespread degenerative pre-existing endometriosis (Bolat et al., 2012; Kondoh cytoplasmic vacuolisation resulting in signet ring cell et al., 2012; Adhikari and Shen, 2013). The de novo like appearance. Acute inflammation with fibrinous and pathway would involve progesterone-related subserosal purulent component extended to the periapendicular stromal metaplasia. Confirming the role of hormonal and mesenteriolar tissues. By immunohistochemical influences, deciduosis regresses within 4 – 6 weeks after investigation (IHC), the large polygonal and vacuolated pregnancy along with decidual involution (Bolat et al., cells in pathologic foci expressed progesterone receptors 2012). High level of progesterone in twin gestations and vimentin but lacked pan-cytokeratin and calretinin. has been attributed to diffuse peritoneal deciduosis in Thus, IHC confirmed deciduosis and ruled out such patients (Adhikari and Shen, 2013). Occasionally, malignant tumour. The differential diagnosis over the deciduosis in non-pregnant ladies has been explained by whole course of illness comprised acute appendicitis, adrenal progesterone secretion acting on submesothelial 43 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) stroma sensitized by inflammation or trauma (Buttner 0.13% (Kirshtein et al., 2009; Chung et al., 2013). et al., 1993; Rodriguez et al., 2006). Although deciduosis Thus, appendicitis must be considered as the cause of has been identified in patients lacking any history of acute abdomen during pregnancy. Cases of deciduosis endometriosis (Bolat et al., 2012), increased awareness of manifesting by abdominal pain, leukocytosis and this condition is recommended managing pregnancy in elevated CRP level necessitate even more careful women with history of dysmenorrhea or endometriosis consideration of appendicitis in the differential diagnosis (Kondoh et al., 2012). (Kondoh et al., 2012). The history of the described case Albeit deciduosis frequently lacks clinical symptoms, shows an additional novel, clinically highly important it can manifest by abdominal pain and leukocytosis fact – the appendicitis can also be true complication of that has been related to the production of granulocyte deciduosis. colony-stimulating factor (Kondoh et al., 2012). In conclusion, deciduosis is a rare, pregnancy-related Occasionally, intestinal obstruction (Rodriguez et al., process that in rare cases can affect appendix. In 2006) or significant abdominal or gastrointestinal our patient, gangrenous inflammation supervened haemorrhage can develop (Bashir et al., 1995; Kondoh necessitating urgent appendectomy. Thus, deciduosis et al., 2012). Lethal outcome of deciduosis has been should be considered in the differential diagnosis of previously reported underlining the necessity of correct acute abdomen in pregnancy. diagnosis and timely surgical treatment (Theissig et al., 1988). Conflict of interest: None In contrast to frequently mentioned asymptomatic course, our patient had pain and leukocytosis. In the REFERENCES presented case, these manifestations can be explained 1. Adhikari LJ, Shen R. Florid diffuse peritoneal by severe acute inflammation related to rich presence deciduosis mimicking carcinomatosis in a of neutrophilic leukocytes. The gangrenous tissues were primigravida patient: a case report and review of widely haemorrhagic. However, there was no evidence the literature // Int J Clin Exp Pathol, 2013; 6: of prior primary haemorrhage within the tissues. 2615 – 2619 The clinical diagnosis of deciduosis is difficult even 2. Bashir RM, Motgomery EA, Gupta PK, Nauta RM, in the presence of widespread intraabdominal Crockett SA, Collea JV, al-Kawas FH. Massive involvement (Kondoh et al., 2012). The intraoperative gastrointestinal hemorrhage during pregnancy findings can yield broad differential diagnosis including caused by ectopic decidua of the terminal ileum and primary or metastatic malignant tumours as deciduoid colon // Am J Gastroenterol, 1995; 90:1325 – 1327 mesothelioma, abdominal carcinomatosis or metastatic 3. Bolat F, Canpolat T, Tarim E. Pregnancy- abdominal spread of malignant melanoma (Ustun related peritoneal ectopic decidua (deciduosis): et al., 2011; Adhikari and Shen, 2013). Regarding morphological and clinical evaluation // Turk the differential diagnosis with malignant tumours, Patoloji Derg, 2012; 20:56 – 60 morphological examination of the removed tissues is the 4. Buttner A, Bassler R, Theele C. Pregnancy- “gold standard”. Deciduosis is characterised by nodular associated ectopic decidua (deciduosis) of the architecture. The nodules are composed of large cells with greater omentum. An analysis of 60 biopsies with well-defined cell borders and eosinophilic cytoplasm. cases of fibrosing deciduosis and leiomyomatosis Degenerative changes lead to cytoplasmic vacuolisation, peritonealis disseminata // Pathol Res Pract, 1993; physaliphorous, signet ring or lipoblastic appearance 189:352 – 359 of cells and myxoid stroma. Mitotic activity and/ or 5. Chung JC, Cho GS, Shin EJ, Kim HC, Song OP. nuclear atypia are absent (Rodriguez et al., 2006; Bolat et Clinical outcomes compared between laparoscopic al., 2012). However, even by light microscopy decidual and open appendectomy in pregnant women // change may be mistaken for metastatic carcinoma or Can J Surg, 2013; 56:341 – 346 malignant mesothelioma (Adhikari and Shen, 2013). 6. Kirshtein B, Perry ZH, Avinoach E, Mizrahi S, In such cases, IHC is advisable. Cytoplasmic positivity Lantsberg L. Safety of laparoscopic appendectomy of vimentin and nuclear presence of progesterone during pregnancy // World J Surg, 2009; 33: receptors along with lack of pan-cytokeratin, HMB-45 475 – 480 and calretinin is characteristic for deciduosis. Although 7. Kondoh E, Shimizu M, Kakui K, Mikami Y, Tatsumi the lesions typically are negative for cytokeratin, few K, Konishi I. Deciduosis can cause remarkable positive cells can be present (Bolat et al., 2012; Adhikari leukocytosis and obscure abdominal pain // J Obstet and Shen, 2013). Metastatic carcinoma is characterised Gynaecol Res, 2012; 38:1376 – 1378 by cytokeratin expression, while mesothelioma – by 8. Theissig F, Kemmer C, Kunze KD. Fatal course of calretinin along with pan-cytokeratin, cytokeratin 5/6 mesenteric deciduosis in a gravida I // Pathologe, and vimentin. HMB-45 expression is almost diagnostic 1988; 9:50 – 54 of melanoma (Ustun et al., 2011; Bolat et al., 2012; 9. Ustun H, Astarci HM, Sungu N, Ozdemir A, Adhikari and Shen, 2013). Ekinci C. Primary malignant deciduoid peritoneal Appendectomy is the most common non-obstetric mesothelioma: a report of the cytohistological surgical intervention in pregnant patients. The incidence and immunohistochemical appearances // Diagn of acute appendicitis in pregnancy is estimated as 0.05 – Cytopathol, 2011; 39:402 – 408 44 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) 10. Walker A. Der bau der eihaeute bei graviditatis abdominalis // Virchows Arch Path Anat, 1887; 197:72 – 99 Address: Andrejs Vanags Department of Surgery, Riga Stradins University, Pilsonu Street 13, LV1002, Riga, Latvia E-mail: Andrejs.Vanags@rsu.lv Fig. 1. Appendicular deciduosis. A, Overview of the affected tissue. Note the nodular foci of deciduosis. Haematoxylin-eosin (HE), original magnification (OM) 50x. B, Cell morphology of the decidual foci. Note the intact deciduoid cell (yellow arrow) and the degenerative changes resulting in signet ring cell like appearance (green arrow). HE, OM 400x. C, Expression of progesterone receptors (PR) in deciduoid cells. Immunoperoxidase, anti-PR, OM 100x. D. Purulent inflammation (arrow) surrounding a deciduoid cell (yellow arrow). HE, OM 400x.

Journal

Acta Chirurgica Latviensisde Gruyter

Published: Nov 24, 2014

Keywords: Medicine; Clinical Medicine; Surgery; Surgery, other

References