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approach with appropriate broad management. Splenic aspiration cell-hemoglobin C disease: angiographic findings. Am J Roentgenol 1977; 129:927-928. spectrum antibiotics, and specific may be warranted in some of these 8. Adekile AD, Tuli M, Haider MZ, Al-Zaabi K, Mohanna-d treatment initiated based on the cases. These patients can be man-a di S, Owunwanne A. Influence of alpha thalassaemia trait on function in sickle cell anemia patients culture report, followed by splenec-t aged initially by conservative meas- with high HbF. Am J Hematol 1996 ; 53: 1-5. tomy after appropriate vaccinations. sures followed by . The 9. Al-Salem AH, Al-Aithan S, Bhamidipati P, Al- Jama A, AlDabbous I. Sonographic assessment of Since the functional residual splenic predominant indication for splenec-t spleen size in Saudi patients with sickle cell dis-e tissue in massive splenic tomy would be persistent pain, press- ease. Ann Saudi Med 1998; 18: 217-20. 10. Moores DC, McKee MA, Wang H, Fischer JD, is minimal, splenectomy is not ex-p sure effects and the risk of Smith JW, Andrews HG. Pediatric laparoscopic pected to worsen the hyopsplenic of the infarcted splenic tissue. splenectomy. J Pediatric Surg. 1995; 30: 1201-5. state postoperatively. The recomm- mended indications for splenectom- Salam Alkindi, Norman Machado, my in patients with massive splenic Pradeep Chopra, Mohammed Uterine sarcoma: a rare infarction are persistent upper ab-d Al-Huneini, Khalil AlFarsi, Anil cause of uterine inversion dominal pain, pressure symptoms Pathare of a large spleen, accidental rupture To the Editor: We describe a case and the concern of potential risk of From the Sultan Qaboos of uterine inversion associated with infection, or the presence of an ab-s University Hospital, Muscat, endometrial sarcoma. Initially, the scess of the infarcted splenic tissue. Oman patient was thought to have a cer-v Splenectomy could, however, pose vical mass, as she developed severe special technical problems due to Correspondence: and the mass protruded the size of the spleen and surroundi- Salam Alkindi, MD outside the vagina. A CT scan was ing perisplenic inflammation. Dense Department of Haematology done before examination under adhesions are usually seen between Sultan Qaboos University anesthesia, with findings of a large the spleen, diaphragm, stomach Hospital, uterine mass. We performed a to-t and perinephric region. This makes P.O. Box 35, PC 123, Al Khod, tal abdominal hysterectomy and laparascopic splenctomy a relative Muscat 000123 Oman bilateral salpingo-oophorectomy. contraindication.10 Dissection could T: +96899353188 A completely inverted uterus was be partly facilitated by decompressi- F: +96824144887 found during surgery. Histology ing the cystic mass intraoperatively [email protected] showed adenosarcoma of the en-d by aspiration, while taking precau-t dometrium. The tumor was limited tion to avoid spillage of splenic tiss- DOI: 10.5144/1658-3876.2011.144 to the endometrium. At the 5-year sue into the peritoneal cavity, as we follow up, there was no clinical or did in our second case. radiological evidence of recurrent In conclusion, massive splenic REFERENCES disease. Uterine sarcoma should be 1. Al Jama AH, Al Salem AH, Al Dabbous IA. Mas-s infarction is rare, and constituted sive splenic infarction in Saudi patients with sickle suspected in uterine inversion diagn- approximately 3.8% of patients who cell anemia-A unique manifestation. Am J Hemat- nosed in a postmenopausal woman. tol 2002 ;69:205-9. required splenectomy for splenic se-q 2. Sears DA, Udden MM. Splenic infarction, splenic Most cases of uterine inversion are questration in this large single insti-t sequestration, and functional hyposplenism in hemo-g obstetric related and encountered globin S-C disease. Am. J. Hematol 1985; 18: 261-8. tution study. All our three patients 3. Al-Salem AH, Indications and complications of during the puerperium. Non-puer-p with massive splenic infarction were splenectomy for children with . peral uterine inversion is estimated J Pediatr Surg. 2006 ; 41: 1909-15. adults with persistent 4. Wali YA, Al-lamki Z, Hussein SS, Bererhi H, to account for just 17% of all cases well into adulthood, making them Kumar D, Wasiuddin S, Zachariah M, Ghosh K. of uterine inversion.1 Benign uteri- Splenic function in Omani children with sickle cell liable to this complication. An el-e disease: correlation with severity index, hemoglo-b ine pathology may present as uteri- evated HbF is an important risk bin phenotype, iron status, and alpha-thalassemia ine inversion (submucosal myoma trait. Pediatr Hematol Oncol 2002; 19:491-500. 2 factor that is significantly associated 5. Alkindi S, Al Zadjali S, Al Madhani A, Daar S, Al- is a good example). Rarely, uterine with massive splenic infarction. A Haddabi H, Al Abri Q, Gravell D, Berbar T, Pravin S, inversion may complicate the pre-s Pathare A, Krishnamoorthy R. Forecasting hemo-g 6-12 potential complication of a massive globinopathy burden through neonatal screening sentation of uterine sarcoma. infarcted spleen is splenic . in Omani neonates. Hemoglobin 2010; 34 :135-44. The differential diagnosis of i-uter 6. Adekile AD, Owunwanne A, Al-Zaabi K, Haider A high index of clinical suspicion MZ, Tuli M, Al-Mohannadi S. Temporal sequence ine inversion in a postmenopausal supported by radiological evidence of splenic dysfunction in sickle cell disease. Am J woman should include uterine pa-t Hematol 2002; 69: 23-7. on ultrasound and CT scan would 7. Fishbone G, Nunez D, Leon R, Paz G, Isturiz P, thology, particularly sarcoma. help early, prompt and appropriate McLaughlin C. Massive splenic infarction in sickle A 76-year-old woman was ad-m

144 Hematol Oncol Stem Cell Ther 4(3) Third Quarter 2011 hemoncstem.edmgr.com letter mitted through the emergency department with postmenopausal bleeding and lower for six weeks prior to her presenta-t tion. She was obese with a BMI of 31. The patient was known to have high blood pressure and cardio-m myopathy. A CT scan suggested a mass involving the upper vagina and surrounding the lower uterine segment and cervix with air also in the endometrial canal that may have represented endometritis, although extension from the endometrial ca-n nal into the cervix could not be exc- cluded. The patient developed prof- fuse vaginal bleeding and a 10-cm mass protruded outside the vagina. The on-call team managed her by applying a suture ligature around the pedicle, which controlled the Figure 1. Uterine inversion at time of laparotomy with uterine dimple at the uterine fundus indicating the inversion. bleeding. Examination under an-e esthesia was done and a huge mass in the vagina was found. The cervix could not be visualized and the uter-u us was enlarged. A biopsy was taken of the mass and histological exami-n nation indicated adenosarcoma of the endometrium. Total abdominal hysterectomy, a bilateral salpingo- oophorectomy and a bilateral pel-v vic lymphadenectomy were done. During laparotomy, a completely inverted uterus was found with a fungating necrotic mass expand-i ing the vagina. The inverted uterus demonstrated the characteristic uterine dimple at the uterine fundus (Figure 1). The final histopathology was adenosarcoma of the endome-t trium with a clear margin (Figure 2). Postoperatively, the patient re-c covered well and follow-up, both Figure 2. Histopathology of endometrial adenosarcoma. clinically and by CT scan, showed no evidence of recurrence. The last CT scan in October 2009 (5 years that the frequency of nonpuerperal version, and the diagnosis is often from the diagnosis) showed no evi-d uterine inversion with endometrial made at time of surgery. Particularly dence of local or distant recurrence. carcinoma was 6.8% (71.6% were in postmenopausal women, uterine Non-puerperal uterine inversion leiomyomas, 13.6% were sarcoma, inversion with corpus is an extremely rare disease, account-i and 8.0% were idiopathic).1 It is can be misdiagnosed as a cervical ing for only one sixth of all cases of extremely difficult to preoperatively malignancy.4 Furthermore, it has inversion.2 Takeno et al. reported diagnose nonpuerperal uterine in-v been described that the symptoms

Hematol Oncol Stem Cell Ther 4(3) Third Quarter 2011 hemoncstem.edmgr.com 145 letter

associated with a nonpuerperal shock, there is enough time to per-f and review of literature. Cent Afr J. Med 1997;43- :268-71. Medline uterine inversion were vaginal bleed-i form tests such as MRI to deliver 7. Ehrlich CE, Nonaventura LM. Nonpuerperal ing, vaginal tumor, lower abdominal an accurate preoperative diagnosis. inversion of the uterus by endometrial stromal sarcoma of the uterine fundus. South Med J pain, menorrhagia and urinary dis-t However, in our case the severe 1977;70:872-3. MedlineWeb of Science turbance. Including our cases, none vaginal bleeding and mass protrude- 8. Silveira Pinheiro L, Ponte JG. Acute nonpu-e erperal inversion. Int Surg 1975;60:559-60 Medl- of the reported cases were in shock, ed outside the vulva, which made lineWeb of Science which is sometimes associated with surgical intervention life saving. At 9. Schulman JM, Stanton JS. Acute nonpuer-p peral uterine inversion. South MJ 1981;74:1142-5. puerperal uterine inversion. present, surgery and/or chemo-t MedlineWeb of Science Uterine inversion is suspected therapy have been recommended 10. Krenning RA, Dorr PJ, de Groot WH, de Goey WB. Non-puerperal uterine inversion. Case report. Br J Ob-s when a tumor is palpable in the va-g for the treatment of uterine inver-s stet Gynaecol 1982;247-9. MedlineWeb of Science gina, but the uterine fundus is not sion caused by sarcoma, but the 11. Wiedswang G, Moen MH. Non-puerperal inv- 5 version of the uterus. Acta Obstet Gynecol Scand palpable by a pelvic examination. standard therapy has not yet been 1989;68:559-60. CrossRefMedlineWeb of Science In the present case, extruded tumor established. Therefore, treatment 12. Lai FM, Tseng P, Yeo SH, Tsakok FH. Non-puer-p peral uterine inversion – a case report. Singapore was observed in the vagina, but a options for this combination should Med J 1993;34:466-8. Medline pelvic examination failed to identify be studied in the future. uterine inversion. MRI has been shown to be a useful diagnostic Ezzeldin Korshid,a Ismail A. Al- tool since it can examine the char-a Badawib Lead intoxication mimick-i acteristic image of the uterine cavity ing a malignancy and a thickened and inverted uter-i From the aKing Fahad Medical ine fundus on a sagital image and a City and the bKing Faisal Specialist To the Editor: A 51-year-old male ‘bulls eye’ configuration on an axial Hospital and Research Centre, presented to the hospital with a 3- image are signs indicative of uterine Riyadh, Saudi Arabia month history of low to moderate inversion.12 Our case was diagnosed grade fever, fatigue, weight loss and by CT scan, but inversion was not Correspondence: upper abdominal pain. Clinical suspected by CT. The diagnosis of Ismail A. Al-Badawi, MD examination was unremarkable uterine inversion was made during Department of Obstetrics except mild pallor. Investigations the surgical procedure. and Gynecology revealed low hemoglobin (9.8 g/ The etiological factors of this King Faisal Specialist Hospital dL) with normal blood counts and combination include (a) sudden ex-t and Research Centre elevated transminase levels (SGOT: trusion of a tumor from the uterus, MBC 52, P.O. Box 3354, Riyadh 345U/L, SGPT: 123 U/L). Other (b) thin uterine wall, (c) dilatation 11211, Saudi Arabia biochemical parameters were nor-m of the uterine cervix, (d) tumor size, T: +966-1-4427392 mal. Blood and urine cultures were (e) thickness of the tumor pedicle F: +966-1-4427393 sterile. CT of the abdomen revealed and (f ) tumor attachment site.3 In [email protected] pericholecystic fluid, favoring a our case, as the tumor with its ped-i diagnosis of chronic cholecystitis icle attached to the uterine muscle DOI: 10.5144/1658-3876.2011.146 with acute exacerbation. He und- became progressively relaxed at the derwent laparoscopic cholecystect- point of pedicle attachment, the cer-v tomy along with a wedge biopsy of vix dilated and the tumor extruded REFERENCES liver. Liver biopsy showed only fatty

into the vagina, resulting in uterine 1. Takano K, Ichikawa Y, Tsuboda H, Nishida M. Uter-i change. In view of no response to inversion. When a rapidly growing ine inversion caused by uterine sarcoma: a case report. broad-spectrum oral antibiotics he Jpn J Chin Oncol 2001 (Jan); 31(1):39-42. tumor exhibits signs indicative of 2. Kopal S, Seckin NC, Turhan NO. Acute uterine was further investigated. Contrast- myoma delivery, patients should be inversion due to a growing submucous myoma in enhanced CT of the chest showed an elderly woman: case report. Eur J Obstet Gyne-c treated as having not only uterine col Reprod Biol 2001 (Nov); 99(1):118-20. bilateral parenchymal infiltrates sarcoma, but also uterine inversion. 3. Das P. Inversion of the uterus. J Gynaecol Br with mediastinal . Emp 1940;47:525-48. Ehrlich et al. reported that the 4. Krenning RA. Nonpuerperal uterine inver-s During the course of investigation abdominal approach is the best sion. Review of literature. Clin Exp Obstet Gynecol he started developing pancytopenia. 1982;9:12-5. Medline and least hazardous treatment for 5. Lascarides E, Cohen M. Surgical management The presence of mediastinal nodes uterine inversion caused by uter-i of nonpuerperal inversion of the uterus. Obstet Gy-n and cytopenias raised a suspicion 5 necol 1968;32:376-81. MedlineWed of Science ine sarcoma. Since patients with 6. Mwinyoglee J, Simelela N, Marivate M. Non- of lymphoma with marrow involve-m this disease are less likely to suffer puerperal uterine inversions. A tow case report ment. A trial of naproxen further

146 Hematol Oncol Stem Cell Ther 4(3) Third Quarter 2011 hemoncstem.edmgr.com