Duct Remnant Research Articles

Thyroglossal cyst with intralaryngeal extension – An unusual case report

Thyroglossal duct cysts are the most prevalent congenital neck masses originating from thyroglossal duct remnants. Here, we present a case of thyroglossal dust cyst with hoarseness and difficulty breathing. A contrast-enhanced computed tomography scan confirmed the intra-laryngeal extension and airway compromise. Sistrunk procedure along with laryngeal reconstruction was done. Histopathology confirmed thyroid follicles in the cyst wall.

Omphalomesenteric Ducts and Urachal Remnants: A Retrospective Study and Case Series.

The management strategies for umbilical disorders remain undefined. This study aims to review our experience and propose a management algorithm for symptomatic urachal and omphalomesenteric duct anomalies. We retrospectively reviewed medical charts between January 2013 and September 2017 of 28 patients with clinical concern for umbilical disorders, out of which 10 were diagnosed with omphalomesenteric duct remnants (OMDR) or urachal remnants (UR). We assessed patients' sex, age at operation, initial presentation, imaging findings, surgical approach, histopathological findings, and prognostic outcome. Among 10 patients with OMDR or UR, initial presentations were omphalitis in four, umbilical discharge in three, abdominal pain in two, and umbilical mass in one. Ultrasonography (US), computed tomography (CT), magnetic resonance imaging (MRI), and voiding cystourethrography were performed in 10, seven, three, and four patients, respectively. Transumbilical extraperitoneal excision from a small expanded umbilical incision and laparoscopic approach combined with transumbilical excision was performed in eight and two patients, respectively. Postoperative wound infection occurred in 10% of patients. Ultrasonography was mostly used as an initial diagnostic modality, and in cases in which there were signs of infection, they were drained adequately; CT/MRI was chosen for further evaluation of suspicious cases for other complications. Thus, we recommended surgical excision in cases with persistent umbilical disorders. The umbilical approach displays good cosmetic results with easy, complete excision, and the laparoscopic approach could be an excellent diagnostic and therapeutic method for the management of complicated conditions.

Laparoscopy versus ultrasonography for the evaluation of Müllerian duct remnants in male patients with disorder of sex differentiation

BackgroundThe diagnosis of male differences of sex development is a challenging multidisciplinary team task, that requires external genital evaluation, karyotyping, hormonal profiling, radiological work up and frequently diagnostic laparoscopy and biopsy, for evaluation of internal duct system and nature of gonads. The debate still persists regarding the best diagnostic modality for accurate visualization of Müllerian duct remnants (MDRs) in those patients.The aim of the study was to compare between laparoscopy (L) and ultrasonography (US) regarding the diagnostic accuracy in detection of Müllerian duct remnants, in addition to describing their anatomical nature and relations with the male duct system, in patients with male DSD, with various karyotypes.MethodsWe prospectively included 20 patients with male DSD, mostly due to 46 XY DSD or chromosomal DSD, over 2 years. The medical and radiological data were collected and analyzed.ResultsThe age at the first diagnostic intervention ranged from 8 to 24 months (mean: 17 months). There were 14 patients with 46XY DSD with variable diagnoses (3 ovotesticular DSD, 3 partial gonadal dysgenesis, 6 persistent Müllerian duct remnants syndrome and 2 mixed gonadal dysgenesis). Two patients with 46XX DSD were included (one XX male, and one patient with ovotesticular DSD). One patient with chimerism (46XY/46XX) and three patients with 46XY/45XO mixed gonadal dysgenesis were also recruited. MDRs were evident in all cases (100%) by laparoscopy, only 25% (n = 5) were visualized by US. There was a statistically significant difference between laparoscopy and US regarding gonadal and MDR visualization, being higher with laparoscopy (p values, 0.0180 and 0.001).ConclusionsUltrasonography failed to visualize Müllerian remnants in 75% of patients with complex DSD. On the other hand, laparoscopy provided optimum visualization of MDRs and gonads in those children.

Various Clinical Manifestation of Omphalomesenteric Duct Remnant in a Child: A Case Report

Omphalomesenteric duct (OMD) remnant is a condition in which the omphalomesenteric duct fails to obliterate completely or partially. Failure in this step can produce various patterns, such as a vitelline cyst, umbilical sinus, polyp, Meckel’s diverticulum, and patent OMD. Various clinical manifestations of OMD rarely occur in one patient. We present the case of omphalomesenteric duct remnant, manifested as anterior wall abdominal mass, intraperitoneal hemorrhage, and obstructive ileus. Case report: A four-year-old girl was referred to our hospital with obstructive ileus and abdominal wall mass on the left lower quadrant. Contrast-enhanced abdominal computed tomography (CT) scan found an irregular cystic mass in the abdominal cavity that adheres to the anterior wall of the abdomen and bowel structure with a fluid-solid level without enhancement. Further medical history was taken and found a history of intermittent watery umbilical discharge that appears three times in 2 years after birth. Omphalomesenteric duct remnant was suspected, then proceed to exploratory laparotomy. We found intraperitoneal hemorrhage and 5x4 centimeters frail mass adhering to the anterior wall of the abdomen on the left side of the umbilicus. Histopathological examination confirmed intestinal tissue, suggesting remnants of the omphalomesenteric duct and pancreatic tissue with a chronic inflammatory pattern. Diagnosing omphalomesenteric duct remnant could be challenging and should be considered a differential diagnosis of obstructive ileus, particularly if persistent umbilical discharges are also present. On the other hand, early diagnosis and appropriate management of persistent umbilical discharge could prevent a greater outcome.

Successful Management of the Omphalomesenteric Fistula: About Three Case Reports

The omphalomesenteric duct is an embryologic connection between the midgut and yolk sac, which typically disappears at the 5th to 7th week of gestation. Failure of the obliteration process can lead to omphalomesenteric duct remnants. It usually has a difficult diagnosis, manifested by a variety of cutaneous signs, such as an umbilical mass, granulation tissue, or discharge. This article reports three cases of total persistence of the OMD and reviews the surgical management of those lesions. The first case reports a 6-day-old boy with a patent omphalomesenteric duct fistula opening to the umbilicus presenting with feces sprouting from the umbilical stump. The second one reports a one-month-old infant brought to the hospital with umbilical granuloma, with no response to topical treatment that later started draining feces from the umbilical cord. The third one reports a three-day-old male newborn that manifested a granuloma-like lesion on the umbilical scar with no improvement with topical treatment. A simple catheterization with a relation catheter was performed in all cases showing drainage of a bilious secretion. they were then diagnosed with a persistence of the Vitelline Duct. All infants had a similar surgical approach, with an abdominal elective incision made around the lesion on the abdominal wall. Identification of the fistula trajectory, followed by resection of the bowel segment that contains the fistulae and a formation of a terminal-terminal anastomosis.

Persistent Paramesonephric (Müllerian) Duct Remnant in Male Patients: Report of 2 Cases Presenting as Pelvic Masses.

Persistent Paramesonephric (Müllerian) Duct Remnant in Male Patients: Report of 2 Cases Presenting as Pelvic Masses.

Clinical characteristics and surgical treatment of children with 45, X/46, XY differences of sex development

This study retrospectively analyzes the clinical data of 18 children with 45,X/46,XY differences of sex development (DSD), summarizes their clinical features and explores gonadal and Müllerian duct remnants surgical treatment methods. The clinical data of 18 children with karyotype 45,X/46,XY diagnosed in the Department of Urology of Hunan Children's Hospital from March 2011 to October 2021 were collected. All children underwent HCG stimulation testing, laparoscopic exploration, urethroscopy and bilateral gonadal biopsy. After DSD multidisciplinary team (MDT) meeting, some children underwent gonadectomy and genitalia reconstructive surgeries. The median age at first diagnosis was 1 year and 4 months (range: 10 months ∼ 16 years and 3 months). 5 children presented with female gender; they all maintained their gender assignment. The external masculinisation score (EMS) of patients raised as female was 1 (0∼3) [median (range)]. 13 children presented with male gender, 10 maintained a male gender, 3 were assigned a neutral gender. The EMS of the children raised as male was 5 (2-8) [median (range)], the EMS of the children raised as neutral gender was 4 (3.5-9.5) [median (range)]. The HCG stimulation test was positive in 11 cases, partially positive in 2 case, and negative in 5 cases. There was no relationship between the percentage of chimerism (45X ratio) and the appearance and severity of genital abnormalities. (t=-1.08, P=0.298). There was 1 case of complete gonadal dysgenesis (CGD), 10 cases of mixed gonadal dysgenesis (MGD), 5 cases of partial gonadal dysgenesis (PGD), 1 case of bilateral normal testes and 1 case of ovotesticular DSD (split-lateral type). No gonadal specimen showed germ cell tumor changes. Five cases selected to maintain the female gender, among which 3 cases underwent bilateral gonadectomy and genitalia reconstructive surgeries. Among the 10 children who chose to maintain the male gender, unilateral streak gonadectomy was performed in 4 (57.1%) with MGD, unilateral dysgenetic orchiectomy in 1 (25%) with PGD, and right ovariectomy in 1 with OTDSD. Nine of them underwent genitalia reconstructive surgeries. Four of them preserved their uterus and vagina did not have any complications during the follow-up period. Hypospadias combined with cryptorchidism and residual Müllerian duct structures is the most common phenotype of children with 45, X/46, XY DSD. Mixed gonadal dysgenesis (MGD) is the most common gonadal type. Gender assignment should be carefully selected after a thorough evaluation, while genitalia reconstructive surgery can be considered in selected patients. In children who choose the male gender, the Müllerian duct can be preserved.

Cystic Duct Remnant–Duodenal Fistula following Laparoscopic Cholecystectomy: A Case Report and Literature Review

Laparoscopic cholecystectomy is the most common procedure performed for the management of symptomatic gallstone disease. This, however, can be complicated by the formation of fistulous communications between the biliary tree and the gastrointestinal tract. This abnormal communication allows for the flow of bile and bowel contents between two systems (biliary system and intestine), which can cause abdominal pain, nausea, vomiting, and biliary sepsis. We would like to present a rare case of fistulous communication between the cystic duct stump and duodenum and outline possible contributing factors. The literature review describes the most common interventions for the management of fistulas with emphasis on ERCP and stent preferences to eliminate transpapillary pressure gradient, which directly contributes to fistula closure.

Carcinoma arising from thyroglossal duct remnants.

Thyroglossal duct cysts (TGDCs) are unusual remnants of the thyroid gland that are rarely observed in association with carcinoma. The present study aimed to showcase the clinical characteristics, diagnosis and management of patients with TGDC carcinoma. It was a single-center study conducted from February 2016 to February 2020. The study involved the retrospective analysis of a series of cases with TGDC carcinoma. A total of 10 patients were included in the study, of whom eight (80%) were females. Their age ranged from 25 to 48 years with a mean age of 39.1 years. A total of five cases (50%) underwent only Sistrunk's procedure, four patients (40%) underwent total thyroidectomy along with Sistrunk's procedure and one patient (10%) was treated by Sistrunk operation and lobectomy. In all of the cases (100%), histopathological examination confirmed papillary thyroid carcinoma in TGDCs. In addition, in the thyroid tissue, 2 cases (20%) also had papillary thyroid carcinoma and 3 cases (30%) had papillary thyroid microcarcinoma. Radioiodine was administered in one patient (10%) with suppressive thyroxine. A three-year follow-up with ultrasound revealed no suspicious lesions in any of the cases. Although rare, carcinoma may develop in the thyroglossal cysts. In this situation, both the thyroid gland and different lymph node compartments should be evaluated for malignancy. Surgical intervention is the cornerstone of management.

An atypical presentation of a patient with a combination of omphalomesenteric duct-related anomalies: A case report

The omphalomesenteric duct is an embryonic structure which connects the yolk sac to the midgut. The omphalomesenteric duct attenuates between the 5th and 9th week of gestation. Failure of the omphalomesenteric duct involution, either partial or complete, results in various omphalomesenteric duct remnants including Meckel's diverticulum, patent vitelline duct, fibrous band, sinus tract, umbilical polyp and cyst. Omphalomesenteric duct remnants are present in 2% of the population but related diseases have seldom been reported in children. We present a case of a 12 years old boy there was coexistence of the previously mentioned anomalies of the omphalomesenteric duct (Meckel's diverticulum, sinus and cyst). To our knowledge, our case is the first pediatric case with the coexistence of the previously mentioned anomalies of the omphalomesenteric duct.

Combination of Urachal and Omphalomesenteric Duct Remnant: A Rare Case Reports

Umbilical problems encountered in the pediatric population are often associated with retained umbilical cord structures or with failure of the umbilical ring to close at birth. This article reported a case of umbilical anomalies in 3-years old male, with a chief complaint of a lump on the umbilical area a urinary discharge from the umbilical since birth. Incidence of patent vitelointestinal duct (omphalomesenteric duct) varies from 1 in 5000-8000 while patent urachus are still rare, ranging from 1-2 per 100000. The combination of both anomalies is very rare, only 12 reported cases since 1898 until 2016. Ultrasound can play an important diagnostic role for these types of anomalies if performed by expert. In our case, the ultrasound was not able to confirm the diagnosis due to a lot of gas in the abdomen. Based on clinical data and histopathologic examination, the patient was diagnosed with combination of urachal and omphalomesenteric duct remnant.

Thyroid Cancer in Thyroglossal Duct Remnants: Case Series and Extensive Review of the Literature

Thyroid Cancer in Thyroglossal Duct Remnants: Case Series and Extensive Review of the Literature

SAT500 Thyroglossal Duct Cyst Cancer: Diagnosis And Management

Abstract Disclosure: A.P. Solis Pazmiño: None. C. Rocha: None. L. Figueroa: None. C. Garcia: None. Background: Thyroglossal duct remnants form during the embryologic development of the thyroid gland. In rare cases, with an incidence of about 1%, carcinoma is present within these cysts, most commonly papillary thyroid carcinoma. Sistrunk procedure (SP) is the main treatment and optimal patient management includes multidisciplinary consultation in order to improve survival. However, there is a debate about whether SP is enough if there is a malignancy in the thyroid or if total thyroidectomy should be done on patients 45 years of age and individuals with aggressive disease [1]. TGDCC harbors an excellent prognosis and low recurrence rate. Clinical Case: We present a case of an elderly woman with a large submental mass that had been gradually enlarging for about 2 years. She had symptoms of dysphagia but denied odynophagia, hoarseness, or shortness of breath. Our patient had high blood pressure and a penicillin allergy but was otherwise healthy without a family history of carcinoma. Physical examination revealed a large submental mass (4.5x4x3.5 cm) that was hard and with smooth edges and did not move with deglutition. There was no extension into the oral cavity on bimanual palpation or palpable masses/nodules in the cervical region. There was a thyroid gland enlargement (right lobe 5x3x1.5, left lobe 4.5x2x1.2 cm). The ultrasound showed enlarged cervical lymph nodes. Fine needle aspiration (PAAF) suggested metastasis of thyroid cancer. Clinical lesson: Although Sistrunk surgery is accepted, performing a total thyroidectomy for TGDCC is controversial. Evidence suggests that it should be reserved for patients older than 45 years, like the patient described in this case. Total thyroidectomy is preferably performed based on the FNAB result (high-risk malignant) or if both thyroid lobes do not consist of healthy tissue anymore, regarding postoperative complication. Since TGDCC is a rare malignancy, a broad differential diagnosis should be considered: metastasis from an unknown primary, paraganglioma, hyalinizing trabecular tumor, and thyroglossal duct cyst, among others. The clinicopathological correlation, as well as the use of the FNA and the immunohistochemical markers, improve the diagnosis accuracy and help to decide the best treatment for the patient. Presentation Date: Saturday, June 17, 2023

Neophallus and Urethroplasty Revisions: Step-by-step Surgical Techniques and Key Reconstructive Maneuvers

Abstract Objective: The aim of this video is to demonstrate step-by-step surgical techniques for neophallus and urethroplasty revisions, emphasizing key reconstructive maneuvers and improving patient outcomes. Materials and Methods: Neophallus and urethroplasty revisions can be challenging procedures associated with complications. We present a 25-year-old male with a history of ambiguous genitalia and micropenis due to 17-hydroxylase deficiency. The patient presented with bothersome penile skin redundancy at the base of the neophallus and a history of intermittent urinary incontinence, necessitating “milking the base of the neophallus” to completely evacuate his urethra. The patient also reported foul-smelling discharge with urine. Imaging evaluation, including retrograde urethrogram and voiding cystogram, revealed a smooth cystic dilatation of the posterior urethra with a normal bladder contour and capacity. Further evaluation with cystoscopy identified a Mullerian duct remnant in the urethra. We recommend surgical excision of the remnant and neophallus skin revision using intraoperative SPY Indocyanine green to carefully delineate the blood supply to the neophallus and mitigate potential risks and complications. Results: This paper demonstrates step-by-step surgical techniques for neophallus and urethroplasty revisions, highlighting key surgical maneuvers such as using intraoperative SPY Indocyanine green to delineate blood supply, excision of the Mullerian duct cystic lesion, and urethroplasty. These techniques aim to improve patient outcomes by addressing penile skin redundancy and urinary incontinence while minimizing potential complications. Conclusion: As neophallus reconstruction becomes increasingly common, familiarity with surgical anatomy, complications management, and outcomes is essential. This video provides a valuable resource for surgical education and may aid in the improvement of patient outcomes.

Cribriform adenocarcinoma of the minor salivary glands: a case report

BackgroundPolymorphous adenocarcinoma is the third most common malignant salivary gland tumor. Within polymorphous adenocarcinoma, cribriform adenocarcinoma of salivary glands is a rare subtype and resembles papillary thyroid carcinoma histopathologically. Diagnostically, cribriform adenocarcinoma of salivary glands is challenging for pathologists and surgeons alike as initial presentation and cytologic nuclear features can be easily confused with papillary thyroid carcinoma arising from a thyroglossal duct remnant or lingual thyroid.Case presentationA healthy 64-year-old Caucasian woman presented to a community otolaryngologist with a 4-year history of progressive postnasal drip, globus sensation, and eventual dysphonia. Flexible fiberoptic laryngoscopy showed a large, smooth, vallecular lesion filling the oropharynx. Computed tomography imaging of the neck showed a rounded heterogeneous mass centered within the right aspect of the oropharynx measuring 4.2 × 4.4 × 4.5 cm. Fine needle aspiration biopsy was suspicious for papillary carcinoma due to microscopic findings of malignant cells, nuclear grooves, and a powdery chromatin pattern. In the operating room, the tumor was resected en bloc using a lateral pharyngotomy approach with partial resection of the right lateral hyoid. A limited cervical lymphadenectomy was performed to facilitate the lateral pharyngotomy approach and two out of three lymph nodes demonstrated regional metastatic disease. Nuclear grooves, nuclear membrane notching, and occasional intranuclear pseudoinclusions were identified, which are overlapping histopathological characteristics of papillary thyroid carcinoma and cribriform adenocarcinoma of salivary glands. It was negative for thyroglobulin and thyroid transcription factor-1, which was in keeping with cribriform adenocarcinoma of salivary glands rather than papillary thyroid carcinoma.ConclusionIt is difficult to distinguish cribriform adenocarcinoma of salivary glands from papillary thyroid carcinoma solely by cytology, and the distinct characteristics of regional lymph node metastasis coupled with nuanced histologic differences should be emphasized in the evaluation of patients presenting with neck lymphadenopathy and an unknown primary or tongue mass. If sufficient fine needle aspiration biopsy material is available, thyroid transcription factor-1, thyroglobulin, or molecular testing may prove useful in differentiating cribriform adenocarcinoma of salivary glands from papillary thyroid carcinoma. A misdiagnosis of papillary thyroid carcinoma may lead to inappropriate treatment including unnecessary thyroidectomy. Therefore, it is critical for both pathologists and surgeons to be aware of this uncommon entity to avoid misdiagnosis and subsequent mismanagement.

Thyroglossal Duct Lipoma: A Case Report and a Systematic Review of the Literature for Its Management.

Thyroglossal duct (TGD) remnants in the form of cysts or fistulas usually present as midline neck masses and they are removed along with the central body of the hyoid bone (Sistrunk's procedure). For other pathologies associated with the TGD tract, the latter operation might be not necessary. In the present report, a case of a TGD lipoma is presented and a systematic review of the pertinent literature was performed. We present the case of a 57-year-old woman with a pathologically confirmed TGD lipoma who underwent transcervical excision without resecting the hyoid bone. Recurrence was not observed after six months of follow-up. The literature search revealed only one other case of TGD lipoma and controversies are addressed. TGD lipoma is an exceedingly rare entity whose management might avoid hyoid bone excision.

Length of the Remnant Cystic Duct and Bile Duct Stone Recurrence: a Case‒Control Study

BackgroundSince the introduction of the Critical View of Safety approach in laparoscopic cholecystectomy, exposure of the common bile duct, and common hepatic duct is not recommended, therefore, the length of the cystic duct remnant is no longer controlled. The aim of this case‒control study is to evaluate the relationship between the length of the cystic duct remnant and the risk for bile duct stone recurrence after cholecystectomy. MethodsAll MRIs with dedicated sequences of the biliary tract taken between 2010 and 2020 from patients who underwent prior cholecystectomy were reviewed. The length of the cystic duct remnant was measured and compared between the patients with and without bile duct stones using multivariate logistic regression analysis. ResultsA total of 362 patients were included in this study, 23.5% of whom had bile duct stones on MRI. The cystic duct remnant was significantly longer in the patients with stones than in the control group (median 31 mm versus 18 mm, P < 0.001). In the MRIs performed > 2 years after cholecystectomy, the cystic duct remnant was also significantly longer in the patients with bile duct stones (median 32 mm versus 21 mm, P < 0.001). A cystic duct remnant ≥ 15 mm in length increased the odds of stones (OR = 2.3, P = 0.001). Overall, the odds of bile duct stones increased with an increasing cystic duct remnant length (≥ 45 mm, OR = 5.0, P < 0.001). ConclusionsAn excessive cystic duct remnant length increases the odds of recurrent bile duct stones after cholecystectomy.

A new connection: management of disconnected segments 5 and 6 bile leak via the cystic duct remnant

Video 1Management of disconnected segments 5 and 6 bile leaks.

Biliary Atresia Remnants Revisited: Myogenesis, Hepatic Duct-Like Structures, and Fate of Peribiliary Glands.

Proximal levels of excised remnants from youngest infants may reveal early features of biliary atresia (BA). A targeted IHC survey was applied to 34 most proximal 2 levels in 17 BA remnants excised at age 10-74 days including 7 = <30 days old and 6 control hepatic ducts (HD). Severity of inflammation and extent of active fibroplasia do not distinguish proximal remnants in younger (n = 7) and older (n = 10) infants. In 27/34 levels of 14/17 remnants, reactive stroma is focally SM-MHC-2 (+), marking smooth muscle myosin, termed reactive myogenesis (RM), that is absent in controls. RM facilitates identification of 3 novel hepatic duct remnants (HDR): an HD-like collagen collar lined by degenerating cholangiocytes (n = 5); erosion defects in loose reactive stroma (n = 14); solitary foci of hyperplastic squamoid epithelium (n = 4). Peribiliary glands are either hyperplastic or atretic and typically lack RM. Minimally inflammed end-stage lesions in BA remnants occur at youngest ages favoring prenatal onset. Three novel HDR are defined. RM, a useful surrogate for HDR, is a prevalent inappropriate stromal reaction in proximal remnants of uncertain biological significance. RM is the source of mature smooth muscle in BA remnants.

Two-centre experience in the treatment of thyroglossal duct remnants using the modified muscle-sparing Sistrunk technique

The Sistrunk procedure has long been the method of choice for treating patients with thyroglossal duct remnants (TDRs). However, the extent of the surgical resection in the suprahyoid segment of the TDR remains controversial, as this anatomical site is the origin of both disease recurrence and surgical morbidity. The aim of this two-centre retrospective cohort study was to investigate the outcomes of a modified muscle-sparing Sistrunk procedure in primary TDRs. The primary predictor was the surgical approach, and the outcome variable was the recurrence rate. A total of 110 consecutive patients (62 (56.4%) male, 48 (43.6%) female) who underwent a modified muscle-sparing Sistrunk procedure were included in the study. Their mean age at presentation was 26.9 ± 18.9 years. A modified muscle-sparing Sistrunk procedure using cold instruments, electrocautery, and a harmonic scalpel was performed in all patients. Recurrence was recorded in six (5.5%) patients. The median hospital stay was 2 days (range 1–7 days), and the median follow-up duration was 7 years (range 2–13 years). There was no significant difference in recurrence rate between the conventional and modified muscle-sparing Sistrunk procedure in primary TDRs. The study findings showed that the modified muscle-sparing Sistrunk procedure had low recurrence and complication rates in the context of primary TDR.